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Health Technology Assessment 2007; Vol. 11: No. 32 School entry hearing screen

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Health Technology Assessment 2007; Vol. 11: No. 32

Current practice, accuracy, effectiveness and cost-effectiveness of the school entry hearing screen J Bamford, H Fortnum, K Bristow, J Smith, G Vamvakas, L Davies, R Taylor, P Watkin, S Fonseca, A Davis and S Hind

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August 2007

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Current practice, accuracy, effectiveness and cost-effectiveness of the school entry hearing screen J Bamford,1* H Fortnum,2 K Bristow,1 J Smith,3 G Vamvakas,4 L Davies,4 R Taylor,3† P Watkin,5 S Fonseca,6 A Davis7 and S Hind8 1

Human Communication and Deafness, University of Manchester, UK Trent Research and Development Support Unit, University of Nottingham, UK 3 Department of Public Health and Epidemiology, University of Birmingham, UK 4 Health Economics Research, University of Manchester, UK 5 Paediatric Audiological Medicine, Whipps Cross Hospital, London, UK 6 Developmental Paediatrics, St George’s Hospital, London, UK 7 MRC Hearing and Communication Group, University of Manchester, UK 8 MRC Institute of Hearing Research, University of Nottingham, UK 2

* Corresponding author † Present address: Peninsula Technology Assessment Group, Universities of Exeter and Plymouth, UK Declared competing interests of authors: A Davis is the Director of the UK National Newborn Hearing Screening Programme

Published August 2007 This report should be referenced as follows: Bamford J, Fortnum H, Bristow K, Smith J, Vamvakas G, Davies L, et al. Current practice, accuracy, effectiveness and cost-effectiveness of the school entry hearing screen. Health Technol Assess 2007;11(32). Health Technology Assessment is indexed and abstracted in Index Medicus/MEDLINE, Excerpta Medica/EMBASE and Science Citation Index Expanded (SciSearch®) and Current Contents®/Clinical Medicine.

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he Health Technology Assessment (HTA) programme, now part of the National Institute for Health Research (NIHR), was set up in 1993. It produces high-quality research information on the costs, effectiveness and broader impact of health technologies for those who use, manage and provide care in the NHS. ‘Health technologies’ are broadly defined to include all interventions used to promote health, prevent and treat disease, and improve rehabilitation and long-term care, rather than settings of care. The research findings from the HTA Programme directly influence decision-making bodies such as the National Institute for Health and Clinical Excellence (NICE) and the National Screening Committee (NSC). HTA findings also help to improve the quality of clinical practice in the NHS indirectly in that they form a key component of the ‘National Knowledge Service’. The HTA Programme is needs-led in that it fills gaps in the evidence needed by the NHS. There are three routes to the start of projects. First is the commissioned route. Suggestions for research are actively sought from people working in the NHS, the public and consumer groups and professional bodies such as royal colleges and NHS trusts. These suggestions are carefully prioritised by panels of independent experts (including NHS service users). The HTA Programme then commissions the research by competitive tender. Secondly, the HTA Programme provides grants for clinical trials for researchers who identify research questions. These are assessed for importance to patients and the NHS, and scientific rigour. Thirdly, through its Technology Assessment Report (TAR) call-off contract, the HTA Programme commissions bespoke reports, principally for NICE, but also for other policy-makers. TARs bring together evidence on the value of specific technologies. Some HTA research projects, including TARs, may take only months, others need several years. They can cost from as little as £40,000 to over £1 million, and may involve synthesising existing evidence, undertaking a trial, or other research collecting new data to answer a research problem. The final reports from HTA projects are peer-reviewed by a number of independent expert referees before publication in the widely read monograph series Health Technology Assessment. Criteria for inclusion in the HTA monograph series Reports are published in the HTA monograph series if (1) they have resulted from work for the HTA Programme, and (2) they are of a sufficiently high scientific quality as assessed by the referees and editors. Reviews in Health Technology Assessment are termed ‘systematic’ when the account of the search, appraisal and synthesis methods (to minimise biases and random errors) would, in theory, permit the replication of the review by others. The research reported in this monograph was commissioned by the HTA Programme as project number 03/05/01. The contractual start date was in October 2004. The draft report began editorial review in August 2006 and was accepted for publication in February 2007. As the funder, by devising a commissioning brief, the HTA Programme specified the research question and study design. The authors have been wholly responsible for all data collection, analysis and interpretation, and for writing up their work. The HTA editors and publisher have tried to ensure the accuracy of the authors’ report and would like to thank the referees for their constructive comments on the draft document. However, they do not accept liability for damages or losses arising from material published in this report. The views expressed in this publication are those of the authors and not necessarily those of the HTA Programme or the Department of Health. Editor-in-Chief: Series Editors: Programme Managers:

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ISSN 1366-5278

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Health Technology Assessment 2007; Vol. 11: No. 32

Abstract Current practice, accuracy, effectiveness and cost-effectiveness of the school entry hearing screen J Bamford,1* H Fortnum,2 K Bristow,1 J Smith,3 G Vamvakas,4 L Davies,4 R Taylor,3† P Watkin,5 S Fonseca,6 A Davis7 and S Hind8 1

Objectives: To describe and analyse in detail current practice of school entry hearing screening (SES) in the UK. Data sources: Main electronic databases were searched up to May 2005. Review methods: A national postal questionnaire survey was addressed to all leads for SES in the UK, considering current practice in terms of implementation, protocols, target population and performance data. Primary data from cohort studies in one area of London were examined. A systematic review of alternative SES tests, test performance and impact on outcomes was carried out. Finally, a review of published studies on costs, plus economic modelling of current and alternative programmes was prepared. Results: The survey suggested that SES is used in most of England, Wales and Scotland; just over 10% of respondents have abandoned the screen; others are awaiting national guidance. Coverage of SES is variable, but is often over 90% for children in state schools. Referral rates are variable, with a median of about 8%. The test used for the screen is the pure tone sweep test but with wide variation in implementation, with differing frequencies, pass criteria and retest protocols; written examples of protocols were often poor and ambiguous. There is no national approach to data collection, audit and quality assurance, and there are variable approaches at local level. The screen is performed in less than ideal test conditions and resources are often limited, which has an impact on the © Queen’s Printer and Controller of HMSO 2007. All rights reserved.

quality of the screen. The primary cohort studies show that the prevalence of permanent childhood hearing loss continues to increase through infancy. Of the 3.47 in 1000 children with a permanent hearing loss at school screen age, 1.89 in 1000 required identification after the newborn screen. Newborn hearing screening is likely to reduce significantly the yield of SES for permanent bilateral and unilateral hearing impairments; yield had fallen from about 1.11 in 1000 before newborn screening to about 0.34 in 1000 for cohorts that had had newborn screening, of which only 0.07 in 1000 were unilateral impairments. Just under 20% of permanent moderate or greater bilateral, mild bilateral and unilateral impairments, known to services as 6year-olds or older, remained to be identified around the time of school entry. No good-quality published comparative trials of alternative screens or tests for SES were identified and studies concerned with the relative accuracy of alternative tests are difficult to compare and often flawed by differing referral criteria and case definitions; with full pure tone audiometry as the reference test, the pure tone sweep test appears to have high sensitivity and high specificity for minimal, mild and greater hearing impairments, better than alternative tests for which evidence was identified. There is insufficient evidence regarding possible harm of the screen. There were no published studies identified that examined the possible effects of SES on longer term outcomes. No good-quality published economic evaluations of SES were identified and a

iii

Abstract

universal SES based on pure tone sweep tests was associated with higher costs and slightly higher qualityadjusted life-years (QALYs) compared with no screen and other screen alternatives; the incremental costeffectiveness ratio for such a screen is around £2500 per QALY gained; the range of expected costs, QALYs and net benefits was broad, indicating a considerable degree of uncertainty. Targeted screening could be more cost-effective than universal school entry screening; however, the lack of primary data and the wide limits for variables in the modelling mean that any conclusions must be considered indicative and exploratory only. A national screening programme for permanent hearing impairment at school entry meets

all but three of the criteria for a screening programme, but at least six criteria are not met for screening for temporary hearing impairment. Conclusions: The lack of good-quality evidence in this area remains a serious problem. Services should improve quality and audit screen performance for identification of previously unknown permanent hearing impairment, pending evidence-based policy decisions based on the research recommendations. Further research is needed into a number of important areas including the evaluation of an agreed national protocol for services delivering SES to make future studies and audits of screen performance more directly comparable.

Health Technology Assessment 2007; Vol. 11: No. 32

Contents List of abbreviations ..................................

vii

Executive summary ....................................

1 Background and main questions ............... Historical background ................................ The nature of childhood hearing impairment ................................................. Main questions and overall design of the study ...........................................................

1 1

2 National survey of current screening practice ...................................................... Introduction ............................................... Methods ...................................................... Results ........................................................ Summary .................................................... 3 Possible effects of newborn hearing screening on the school entry hearing screen: evidence from a series of studies in Waltham Forest ..................................... Introduction ............................................... Changes in the programmes of early screening and identification ....................... Evaluating the SES ..................................... Generalising the study ............................... Changes in the SES .................................... Results ........................................................ Summary ....................................................

6 Summary and conclusions ......................... Introduction: strengths and weaknesses of the study ..................................................... Summary of findings .................................. The OME issue and some further analyses ....................................................... Conclusions ................................................ Implications for practice ............................ Recommendations for future research .......

80 82 84 84

Acknowledgements ....................................

References ..................................................

Appendix 1 Screening at school entry for childhood hearing impairment: an appraisal against National Screening Committee criteria .....................................

75 77

5 5 5 6 16

Appendix 2 Questionnaire used in the survey of national practice ......................... 103 17 17 17 18 19 19 20 28

4 Systematic review of the effectiveness of school entry hearing screening ................. Background ................................................ Hypotheses tested in the review (research questions) ................................................... Methods ...................................................... Studies included in the review ................... Studies excluded from the review .............. Results of review ......................................... Summary of findings ..................................

31 31 33 33 33 48

5 Cost-effectiveness and cost-effectiveness acceptability of the school entry hearing screen ......................................................... Methods ...................................................... Results ........................................................ Summary ....................................................

49 49 57 72

31 31

Appendix 3 Prevalence rates for permanent childhood hearing impairment for three cohorts in Waltham Forest and Redbridge Districts and for one cohort in Trent Region of the UK .................................................... 119 Appendix 4 Search strategies used in the systematic review ........................................ 123 Appendix 5 Quality criteria for systematic reviews ........................................................ 129 Appendix 6 Quality criteria for diagnostic test studies .................................................. 133 Appendix 7 Excluded studies and reasons for exclusions .............................................. 137 Appendix 8 Summary of quality of systematic reviews ....................................... 141 Appendix 9 Quality of primary studies ..... 143 Appendix 10 Two by two tables for sensitivity and specificity where available ...................................................... 147

Contents

Appendix 11 Economic search strategies ..................................................... 151

Appendix 15 Economic data extraction form ............................................................ 159

Appendix 12 Subsequent management intervention search strategies .................... 153

Appendix 16 Description of included papers ......................................................... 167

Appendix 13 Inclusion/exclusion economic data form .................................................... 155

Health Technology Assessment reports published to date ....................................... 169

Appendix 14 Inclusion/exclusion subsequent interventions data form .......... 157

Health Technology Assessment Programme ................................................ 185

Health Technology Assessment 2007; Vol. 11: No. 32

List of abbreviations AABR

automated auditory brainstem response

ABR

auditory brainstem response

AOAE

automated otoacoustic emissions

ASHA

American Speech and Language Hearing Association

BACDA

British Association of Community Doctors in Audiology

BHE

better hearing ear

CASP

Critical Skills Appraisal Programme

CEAC

cost-effectiveness acceptability curve

confidence interval

CRD

Centre for Reviews and Dissemination

MMR

measles, mumps and rubella

MRC

Medical Research Council

not applicable

NHSP

Newborn Hearing Screening Programme

NICE

National Institute for Health and Clinical Excellence

NNHS

no neonatal hearing screen

NPV

negative predictive value

not reported

NZHTA

New Zealand Health Technology Assessment

OME

otitis media with effusion

PCHI

permanent childhood hearing impairment

PCT

primary care trust

degrees of freedom

DNA

did not attend PPV

positive predictive value

DPOAE

distortion product otoacoustic emissions

PSA

probabilistic sensitivity analysis

ENT

ear, nose and throat

pure tone

health board

PTA

pure tone audiometry

Hib

Haemophilus influenzae type b

QALY

quality-adjusted life-year

hearing level

QUADAS Quality Assessment of Studies of Diagnostic Accuracy

ICER

incremental cost-effectiveness ratio

IDT

Infant Distraction Test

IQR

interquartile range

LHSCG

local health and social care group

R&D

research and development

RCT

randomised controlled trial

RICHS

Regional Interactive Child Health System continued

List of abbreviations

List of abbreviations continued ROC

receiver operating characteristic

standard deviation

SEM

school entry medical examination

SES

school entry hearing screen

SES-C

composite school entry screening

SES-PQ

SES using parental questionnaire

SES-PTS SES using pure tone sweep testing

STARD

Standards for Reporting Studies of Diagnostic Accuracy

SVEP

Sweep Visual Evoked Potential

TEOAE

transient evoked otoacoustic emission

TNHS

targeted neonatal hearing screen

UNHS

universal newborn hearing screen

VASC

verbal audiometric screening for children

SES-SW

SES using spoken word tests

SES-T

SES using tympanometry

WHE

worse hearing ear

SHA

strategic health authority

YNHI

year with no hearing impairment

All abbreviations that have been used in this report are listed here unless the abbreviation is well known (e.g. NHS), or it has been used only once, or it is a non-standard abbreviation used only in figures/tables/appendices in which case the abbreviation is defined in the figure legend or at the end of the table.

viii

Health Technology Assessment 2007; Vol. 11: No. 32

Executive summary Background The ability to hear is important, particularly during children’s formal education. Hearing impairment is amenable to intervention and hence a screening programme when children begin their school careers has potential value. School entry hearing screening (SES) has been implemented throughout the UK since the 1950s. There is evidence of mixed practice and uncertainty about the value of the screen. In addition, recent changes in childhood hearing screening policy (abandonment of a screen at 8 months and introduction of universal newborn screening) have implications for identification of children with hearing impairment at school entry.

●

Objectives

●

This report aimed to determine answers to the following three questions:

●

● ●

●

What is current practice for the SES in the UK? What is known about the accuracy of alternative screening tests and the effectiveness of interventions? What is known about costs, and what is the likely cost-effectiveness of the SES?

The evidence from the primary cohort studies is that: ●

●

Methods A national postal questionnaire survey was addressed to all leads for the SES in the UK, considering current practice in terms of implementation, protocols, target population and performance data. Primary data from cohort studies in one area of London were examined. A systematic review of alternative SES tests, test performance and impact on outcomes was carried out. Finally, a review of published studies on costs, plus economic modelling of current and alternative programmes was prepared.

the SES is in place in most areas of England, Wales and Scotland; just over 10% of respondents have abandoned the screen; others are awaiting guidance in the light of the national implementation of newborn hearing screening coverage of the SES is variable, but is often over 90% for children in state schools referral rates are variable, with a median of about 8% the test used for the screen is the pure tone sweep test but with wide variation in implementation, with differing frequencies, pass criteria and retest protocols; written examples of protocols were often poor and ambiguous there is no national approach to data collection, audit and quality assurance, and there are variable approaches at local level the screen is performed in less than ideal test conditions resources are often limited and this has an impact on the quality of the screen.

●

the prevalence of permanent childhood hearing impairment continues to increase through infancy of the 3.47 in 1000 children with a permanent hearing impairment at school screen age, 1.89 in 1000 required identification after the newborn screen the introduction of newborn hearing screening is likely to reduce significantly the yield of SES for permanent bilateral and unilateral hearing impairments; yield had fallen from about 1.11 in 1000 before newborn screening to about 0.34 in 1000 for cohorts that had had newborn screening, of which only 0.07 in 1000 were unilateral impairments just under 20% of permanent moderate or greater bilateral, mild bilateral and unilateral impairments, known to services as 6-year-olds or older, remained to be identified around the time of school entry.

The evidence from the systematic review of the alternative tests and of the effectiveness of interventions is that:

Executive summary

●

no good-quality published comparative trials of alternative screens or tests for school entry hearing screening were identified studies concerned with the relative accuracy of alternative tests are difficult to compare and often flawed by differing referral criteria and case definitions; with full pure tone audiometry as the reference test, the pure tone sweep test appears to have high sensitivity and high specificity for minimal, mild and greater hearing impairments, better than alternative tests for which evidence was identified there is insufficient evidence to draw any conclusions about possible harm of the screen there were no published studies identified that examined the possible effects of SES on longer term outcomes.

Conclusions The lack of good-quality evidence in this area remains a serious problem. Services should improve quality and audit screen performance for identification of previously unknown permanent hearing impairment, pending evidence-based policy decisions based on the research recommendations.

Recommendations for research Further research is highlighted in the following areas: ●

The evidence from the cost-effectiveness study is that: ● ●

●

no good-quality published economic evaluations of SES were identified a universal SES based on pure tone sweep tests was associated with higher costs and slightly higher quality-adjusted life-years (QALYs) compared with no screen and other screen alternatives; the incremental cost-effectiveness ratio for such a screen is around £2500 per QALY gained; the range of expected costs, QALYs and net benefits was broad, indicating a considerable degree of uncertainty targeted screening could be more cost-effective than universal SES lack of primary data and the wide limits for variables in the modelling mean that any conclusions must be considered indicative and exploratory only.

A national screening programme for permanent hearing impairment at school entry meets all but three of the criteria for a screening programme, but at least six criteria are not met for screening for temporary hearing impairment.

●

evaluation of an agreed national protocol for services delivering the SES to make future studies and audits of screen performance more directly comparable development and evaluation of systems for data monitoring so that robust data on screen performance are available determination with greater certainty of the prevalence of congenital unilateral hearing impairment, and permanent mild and minimal hearing impairment at school entry, that could be identified by a suitable quality-assured screen protocol a comparison of the effectiveness, efficacy and efficiency of alternative approaches (reactive services, formal surveillance, targeted screening and universal screening at school entry age) to the identification of permanent hearing impairment postnewborn screen controlled studies of the effectiveness of hearing screening and subsequent interventions for later outcomes in children with permanent mild, minimal and unilateral hearing impairment identified at school entry determination of the distribution of detection thresholds for pure tones in the population at school entry.

Health Technology Assessment 2007; Vol. 11: No. 32

Chapter 1 Background and main questions Historical background There is a long history in the UK of screening for hearing impairment in childhood. By the 1930s hearing screening by various methods was being implemented at school entry, which at that time represented the most obvious point at which the child population was available for mass screening. In 1955, as simple screening audiometers became available, it was recommended that all children undergo school entry hearing screening using the pure tone ‘sweep’ test.1 This test requires the child to indicate that he or she has detected each of a number of tones of different frequencies (pitches) presented to each ear separately at an intensity level indicative of normal hearing. The screen quickly became established across the UK, organised and managed through local authority school health services. In 1976 the Court Report recommended that hearing screens be carried out at least twice in school.2 However, there was no nationally agreed protocol for the screen, and implementation thus varied in small but possibly important details across services. In the 1974 reorganisation of local government the school health service was brought into the NHS, and the school entry hearing screen (SES) has remained the responsibility of the NHS ever since, undertaken in the main by school nurses and community paediatricians. Evidence about the costs and effectiveness of the screen has remained elusive, as has clarity about its aims, despite a number of reviews.3–6 There are anecdotal reports that some services have supplemented the pure tone sweep test with other tests, while others are said to have abandoned the screen in the light of lack of national guidance and unwanted variability from a variety of sources, including screen protocols, test environment (schools can be noisy places), tester competence and equipment calibration. Despite this, there has until recently been a widespread if implicit consensus that the pure tone sweep test has value educationally and provides a safety net to catch any deficiencies of the earlier screening system in the overall public health provision,4 a position broadly endorsed by Hall.7 Haggard’s4 comment on the deficiencies of the earlier screening system refers to the Infant © Queen’s Printer and Controller of HMSO 2007. All rights reserved.

Distraction Test (IDT) screen. From the mid-1950s a hearing screen was performed on all infants in the UK aged 8 months using the IDT.8 This is a behavioural test in which sounds are presented to the infant under controlled conditions and the child’s responses, if any, noted. However, this apparently simple test did not perform well, and there were credible reports of high referral rates, with high false-positive and false-negative rates. During the 1990s, developments in technology made it possible to test the auditory function of newborn babies using otoacoustic emissions and/or auditory evoked responses. A review in 19975 led to a policy decision in England (with Wales, Scotland and Northern Ireland following suit) to phase out the IDT screen and to replace it with a national programme of newborn hearing screening. The newborn screen in England is a contingent two-test screen involving an automated otoacoustic emissions (AOAE) test followed, if either ear fails to show a clear response, by an automated auditory brainstem response (AABR) test, again requiring a clear response on both ears for a pass decision. The Newborn Hearing Screening Programme (NHSP) was fully implemented in England by March 2006; the evidence is that it is highly effective, reducing the age of identification of permanent congenital bilateral hearing impairment of moderate or greater degree from some 80 weeks to 10 weeks of age.9,10 The extent of the beneficial effects of this early identification for children with permanent hearing impairment upon developmental outcomes in general, and communication in particular, has been demonstrated,11 although much detail remains to be added.

The nature of childhood hearing impairment Hearing impairment in childhood can be permanent or temporary. Permanent childhood hearing impairment of a moderate degree or greater [i.e. detection thresholds >40 dB hearing level (HL) averaged across 0.5, 1, 2 and 4 kHz] is present at birth at a rate of about 1.6 per 1000 live births, of which approximately 1.0 in 1000 are bilateral impairments and 0.6 in 1000 are unilateral impairments.5,10 In terms of incidence,

Background and main questions

this means that in the UK about 800 children per year will be born with permanent bilateral hearing impairment of a moderate or greater degree; and about 500 per year will be born with unilateral hearing impairment (i.e. hearing within normal limits in one ear, but hearing impairment of moderate or greater degree in the other ear). There is good evidence that the prevalence of permanent bilateral moderate or greater hearing impairment increases through the first decade of childhood.12,13 The reasons for this are not entirely clear, but include meningitis, measles and other causes of acquired impairment; progression of unilateral to bilateral impairments; and lateonset/progressive impairments linked to prenatal or perinatal infection or to hereditary factors. It is possible that the prevalence of bilateral moderate or greater impairment reaches 2 in 1000 by the age of about 9 years. The evidence on permanent unilateral hearing impairment is more limited. Although it appears from NHSP data that the prevalence at birth is about 0.6 in 1000, it is not known whether there are significant numbers of later onset cases, whether some of the impairments are progressive, and whether there is a tendency for congenital or postnatal unilateral hearing impairment to progress to bilateral impairment. Unilateral hearing impairment would be expected to affect auditory perception in various predictable ways, such as poor localisation of sound sources, and difficulty in noisy or reverberant environments such as schools, and there is some evidence of detrimental effects on academic progress.14 Unlike bilateral permanent childhood hearing impairment however, management of unilateral impairment remains uncertain, and it is not known whether early family advice and support, a hearing aid in the affected ear or other approaches would be helpful. The significance of the increase in prevalence in permanent hearing impairment in the first decade of life is that a newborn hearing screening programme would fail to detect these additional cases and other processes are required, whether based on professional responsiveness to parental observations, structured surveillance, or later screening. In addition, the newborn screen as presently conceived will not identify mild and minimal permanent hearing impairment, whereas a later screen could.

In children, temporary hearing impairment is much more common than permanent hearing impairment. It is linked in the main to colds and

upper respiratory tract infections that lead to otitis media with effusion (OME) – the presence of fluid in the middle ear. There is a huge literature on OME (see Haggard and Hughes 199115 for an early but comprehensive review), which addresses prevalence, pathology, assessment, management options, the time-course, and short-, medium- and long-term effects of the condition. The point prevalence of OME is of the order of 15–25% in the 0–6-year-old age group, with peaks in the first year of life and at school entry.15–17 The period prevalence across that age range may be as high as 80%. Most cases resolve within 2–3 months. Some recur, and some persist for much longer. In those that do, there can be significant short- and medium-term effects not only on hearing, but also on behaviour, socialisation, speech and academic progress. The difficulty for services is to be able to identify those cases, perhaps around 3 or 4%, that are likely to have the condition recurrently, and/or with a persistence and severity likely to cause concern (i.e. to affect significantly development, whether attention, communication, behaviour or other domains). Although case finding is done through hearing impairment, intervention options may be directed at other effects, and include advice for parents and teachers, speech and language therapy, and/or surgery to remove the fluid and decrease the chance of recurrence [myringotomy, ventilation tubes (grommets), adenoidectomy].

Main questions and overall design of the study Despite several attempts, some of which are recent,5,6 to investigate the value and effectiveness of the SES in the UK, there remains a pressing need to understand the pattern of current practice (which has developed ‘bottom–up’) and to evaluate the likely accuracy of alternative tests and costs and effectiveness of the screen to guide policy decisions. Furthermore, the introduction of newborn hearing screening to replace the underperforming 8-month hearing screen has significantly changed the landscape of hearing screening in childhood. Whereas the 8-month IDT screen was very unlikely to identify permanent unilateral hearing impairment, the newborn screening protocol will identify unilateral as well as bilateral permanent hearing impairment; this potentially reduces one of the justifications for the SES. However, the SES might continue to yield significant numbers of mild, high-frequency or late-onset/progressive impairments that would otherwise be missed (or missed until later

Health Technology Assessment 2007; Vol. 11: No. 32

concerns led to identification), and might identify significant numbers of children with persistent middle ear disorders not otherwise known to services at a time when good hearing is of particular importance educationally. These uncertainties lie behind the current research study. The authors were commissioned in 2004 by the HTA Programme of the NHS Research and Development initiative to ●

●

carry out a national survey of current SES practice conduct a systematic review of the accuracy of alternative screening tests and the effectiveness of subsequent interventions assess the costs of the screen and to model costeffectiveness.

The study therefore has three strands: ●

●

Strand 1 (reported in Chapter 2) is a national questionnaire survey of current practice. A survey instrument was designed and piloted, and the final version completed by lead clinicians/managers for the SES across the UK. The questionnaire was designed to collect information on whether the screen was still being implemented, what tests and protocols were being used, what the target population was, who performed the screen and where, with what training and what equipment, and whether any screen performance data were collected and available. Strand 2 (reported in Chapter 4) is a systematic review of the existing research literature which aims to evaluate the accuracy of alternative approaches to a school entry hearing screen, and to summarise the evidence on screen performance (i.e. screen uptake and yield for different screen options and different case definitions) and the impact of the screen on children’s outcomes (language, communication, social and educational). Strand 3 (reported in Chapter 5) is an assessment of the costs, outcomes and associated levels of uncertainty of alternative models of school entry screening using economic modelling techniques.

Throughout the three strands, cognisance is taken of three distinct possible case definitions: first, children with moderate, severe or profound bilateral permanent hearing impairment, for whom the evidence on the consequences of not identifying and intervening appropriately is strong; secondly, children with permanent mild, © Queen’s Printer and Controller of HMSO 2007. All rights reserved.

minimal or unilateral hearing impairment, or hearing impairment affecting only some frequencies, about which the evidence on consequences, intervention (and prevalence) is less clear; and thirdly, children with temporary hearing impairment associated with persistent and/or severe OME, the treatment for and sequelae of which give rise to considerable controversy.18 One reason for this is that while permanent hearing impairment has a variety of causes, the treatment and management are in the main directed at the hearing impairment itself; for children with OME, however, although there is a coherent set of disease processes at its core about which something is known, hearing impairment is by no means the whole story, and interventions aim to treat more than just the hearing impairment. Since the evidence base for the characteristics and yield of screening for the latter two categories is so weak, the study also considers a case definition in terms of a disability measure for which there is some evidence, hearing in noise, or specifically the minimum signal to noise ratio required to score at a given criterion level on a speech perception task. When gathering evidence on the effectiveness and the efficiency of the SES for each of these case definitions, it is important in the cost-effectiveness modelling (strand 3) to take account of the likely incremental yield of the screen: for this one needs to know, or to be able to estimate, the number of cases that remain to be identified by the SES after the identification of these cases by the newborn hearing screen, standard surveillance, parental concern and professional responsiveness. As well as referring to the published evidence on prevalence of cases and yields of screens and systems before school entry, the researchers examined primary data from cohort studies to which they have direct access through authorship (Chapter 3). One of these (from Watkin) was a series of studies undertaken in Waltham Forest where universal newborn screening was introduced some 10 years ago (so the outcomes from recent SES cohorts would be expected to reflect the effect of newborn screening on cases left to be found), and the other has been the use of a large database of children with persistent OME from the Medical Research Council (MRC) Otitis Media Study Group to answer some specific questions thrown up by the discussions and to address the issue of the effectiveness of subsequent interventions for OME. The authors decided at an early stage not to review the literature on the effectiveness of

Background and main questions

treatment of OME fully or in a concentrated way, for two main reasons. First, it is very large and heterogeneous, with much of it of poor quality, giving some information but not of the type that can be easily extracted from aggregating the results of the studies of best quality. Secondly, as noted above, hearing is neither the only relevant nor necessarily the ultimate outcome of such treatment. Nevertheless, since there is a high continuing risk of two recent trials particularly relevant to screened caseloads continuing to be misinterpreted in relation to other evidence (arising from the generally poor understanding of the importance of the characteristics of populations selected and the economic pressures

in differing health systems), the interpretation and implications of these particular studies are discussed. Finally, Chapter 6 outlines the strengths and weaknesses of the study, summarises the findings from each strand, draws together the evidence on screening at school entry in the UK from all three strands into a series of conclusions and makes recommendations. The conclusions are used to examine the justification for the SES as a route to identifying children with permanent and temporary hearing impairment in the light of the National Screening Committee’s criteria for screening programmes (Appendix 1).

Health Technology Assessment 2007; Vol. 11: No. 32

Chapter 2 National survey of current screening practice Introduction

●

Scientific background A national survey of paediatricians responsible for the SES in the UK6 confirmed clinical impressions that services had evolved such that there was considerable variation between services in terms of programme organisation, pass/fail criteria, case management and screen performance, despite recommendations to retain and standardise the SES.19 At the time of that survey of 96 services, four services had discontinued their SES programme as a result of local audit.20 In programmes where the SES was not carried out by dedicated screeners, competing programmes, such as immunisation, took priority over the SES, affecting its performance. There was also an awareness that information may have been incomplete as there were no available data from services that may have been provided by non-medical service leads. In recent years consideration has been given to the impact on individual children of unrecognised mild, unilateral or temporary hearing impairment21,22 and the possible need to identify and manage these children. There is also growing debate about the likely impact of newborn hearing screening on the yield of new cases from the SES and about cases that will not be detected by the NHSP.12 Evidence is emerging that services will undergo further revision on an ad hoc basis as a result of the changing pattern in the yield from the SES following the introduction of newborn hearing screening and local variation in the epidemiology of hearing impairment (see Chapter 3).

Methods Ethics and NHS Research and Development Approval The study met the criteria for a multicentre study with no local investigators. Application for full ethical approval for the UK was submitted to the Central Manchester Local Research Ethics Committee. Local research and development (R&D) approval was applied for in all primary care trusts (PCTs) in England, NHS acute trusts which employed an SES lead clinician, the primary care arm of each health board (HB) in Scotland, the NHS trusts in Wales and local health and social care groups (LHSCGs) in Northern Ireland. Some R&D departments for the PCTs are grouped into consortia with administrative responsibility for a number of PCTs, varying from two to 15. Applications for approval were made to 124 departments.

Identification of service leads for SES provision Service delivery of the SES varies across the UK in terms of the organisations responsible for coordinating the programme and employing staff who undertake it. It was therefore necessary to use several lines of enquiry to identify and recruit the service leads. ●

This chapter provides an up-to-date account of current practice and performance of the SES in the UK.

Aims

●

The aims of this study were to describe and analyse in detail current practice of the SES throughout the UK, in order to:

●

quantify variability in screening practice nationally evaluate current screen performance as reported by service leads in terms of screen coverage, referral rates and yield

record the views of SES leads regarding the value of the screen together with their ideas for improvements or alternatives.

●

Letters were sent to all members of the British Association of Community Doctors in Audiology (BACDA) asking them to contact the research team if they were responsible for the SES in their area or, if they were not, to return the name of the responsible person if they knew it. The Directory of Community Nursing 2004/200523 was used to identify school nursing departments. Advertisements were placed in the BACDA newsletter and British Society of Audiology newsletter. Oral and poster presentations were made to the 28th Annual Children’s Hearing Screening Conference. ‘Cold calls’ were made to NHS trusts.

National survey of current screening practice

Development of a postal questionnaire/survey instrument A postal questionnaire (Appendix 2) was developed to establish: ●

●

● ● ●

● ●

●

the target population of children who are routinely entered in the SES programme locations and conditions under which the screen is performed test methods used pass/fail criteria who carries out screening tests and equipment used for screening data management systems used coverage, referral rates and yield of the screen views of the SES leads regarding the screen.

The questionnaire was reviewed by the project’s advisory committee and piloted by seven audiology professionals closely related to the running of the SES in their area. These professionals were identified either via BACDA or through contacts known to members of the research group. Changes were made following the pilot and the questionnaire was finalised.

Data collection Questionnaires were posted to all identified service leads between September and November 2005 with a covering letter giving further details about the study and a reply-paid envelope. If the service lead failed to return the completed survey within 4 weeks a reminder letter and an additional copy of the questionnaire were posted to them. Those leads failing to respond within an additional 2 weeks were contacted by telephone. A final telephone reminder was made after a further 2 weeks and if, at this stage, no reply was received non-response was assumed. Data were entered into an Access database by one researcher (KB) and 10% of questionnaires were entered independently by another (HF) to check for errors in data entry. Data were converted to Excel for analysis and possible completion errors checked by identifying outlier values.

Results Ethics and NHS research and development approval 6

The Central Manchester Local Research Ethics Committee granted final approval for the study in May 2005.

In England, Scotland and Wales 124 R&D departments were approached for approval, covering a total of 304 NHS trusts. Owing to difficulties in identifying the relevant departments, R&D applications could be made to only two out of 15 LHSCGs within Northern Ireland. Six R&D departments covering seven NHS trusts in England did not give approval for the following reasons: ●

●

One required a separate consent sheet with the questionnaire and information letter. Two required full Criminal Records Bureau and locally administered occupational health checks for the principal investigator to secure an honorary contract. Three approvals were still pending at the time of writing.

Questionnaires were not posted to any staff employed by these seven trusts.

Response In the UK, 244 services responsible for the SES were identified. This does not match the number of primary care organisations because it was common for services to cover a geographical area encompassing more than one primary care organisation; that is, the PCT/LSHCGs/Welsh and Scottish HB boundaries did not match the SES service boundaries. Questionnaires were sent to 229 service leads and 195 (85.2%) responded (Table 1). The numbers of PCTs that were covered by a returned questionnaire within each of the 28 strategic health authorities (SHAs) in England are shown in Table 2. In only two of the 28 SHAs the survey failed to achieve 70% coverage of PCTs. Overall, 86.5% of PCTs in England are represented in the survey. Within Scotland questionnaires were returned from ten SES services covering ten out of 15 (66.7%) HBs. In Wales eight SES services returned a questionnaire, covering 16 out of 22 (72.7%) local health boards. In Northern Ireland, one SES service covering one out of 15 LHSCGs returned a questionnaire.

Questionnaire findings The descriptive results for each question in the questionnaire are detailed. The responses from each of the countries have been grouped together to represent the response for the UK as a whole.

Health Technology Assessment 2007; Vol. 11: No. 32

TABLE 1 Number of SES services identified in each country, number sent a questionnaire and number responding Services No. identified

England Scotland Wales Northern Ireland Total

208 15 11 10 244

No. sent a questionnaire (contacted)

Response n

% of identified

% of contacted

201 15 11 2 229

176 10 8 1 195

84.6 66.7 72.7 10.0 79.9

87.6 66.7 72.7 50.0 85.2

TABLE 2 Geographical coverage by PCTs in England PCTs in each SHA SHA Avon, Gloucestershire & Wiltshire Bedford and Hertfordshire Birmingham and the Black Country Cheshire & Merseyside County Durham & Tees Valley Coventry, Warwickshire, Herefordshire & Worcestershire Cumbria & Lancashire Dorset & Somerset Essex Greater Manchester Hampshire and Isle of Wight Kent and Medway Leicestershire, Northamptonshire & Rutland London North Central London North East London North West London South East London South West Norfolk, Suffolk and Cambridgeshire North and East Yorkshire and Northern Lincolnshire Northumberland, Tyne and Wear Shropshire and Staffordshire South West Peninsula South Yorkshire Surrey and Sussex Thames Valley Trent West Yorkshire Total

In all cases ‘n’ refers to the numbers of service leads indicating that response. Unless otherwise indicated, percentages are of the total number (n) of services responding to that question. Missing data are tabulated separately as a percentage of 195 responses. When relevant, the missing values include the ten services who reported that they no longer run an SES (see next subsection). © Queen’s Printer and Controller of HMSO 2007. All rights reserved.

No. within the SHA

No. (%) covered by a returned questionnaire

12 11 12 15 10 8 13 9 13 14 10 9 9 5 7 8 6 5 17 10 6 10 11 9 15 15 19 15 303

12 (100) 6 (54.5) 9 (75) 13 (86.7) 10 (100) 8 (100) 12 (92.3) 8 (88.9) 12 (92.3) 13 (92.9) 9 (90) 8 (88.9) 8 (88.9) 5 (100) 5 (71.4) 8 (100) 5 (83.3) 4 (80) 14 (82.3) 6 (60) 6 (100) 9 (90) 10 (90.9) 8 (88.9) 12 (80) 14 (93.3) 14 (73.7) 14 (93.3) 262 (86.5)

Population entered into the SES programme Twenty-four services (12.2%) no longer run a universal school entry hearing screen; 11 run no screen and 13 implement a targeted screen. Ten services gave reasons for not running a screen at all, including resource limitations (five) and low yield (six). Only two services running a targeted screen gave reasons, both resource limitations. All

National survey of current screening practice

TABLE 3 The extent to which children are routinely entered into the school entry hearing screen (question 1) Some

None

Total responses

Missing n (% of 195 responses)

170 (87.6)

13 (6.7)

11 (5.7)

194 (100)

1 ( 0.5)

Children in private schools

37 (20.4)

52 (28.7)

92 (50.8)

181 (100)

14 (7.2)

Children who are home educated

10 (5.7)

39 (22.4)

125 (71.8)

174 (100)

21 (10.7)

Children in special schools with known physical or sensory disability

85 (47.2)

44 (24.4)

51 (28.3)

180 (100)

15 (7.7)

Children in special schools with known mental disability (excluding those with hearing loss)

79 (44.4)

46 (25.8)

53 (29.8)

178 (100)

17 (8.7)

Children known to have hearing loss

37 (21.3)

60 (34.5)

77 (44.3)

174 (100)

21 (10.8)

All

Children in state schools

Other

Data are shown as n (%).

but two of the remaining 172 services screen all children in state schools. Table 3 indicates the extent to which the SES is offered to children in different educational environments. Only 20.4% (37/181) of services screen all children in private schools. An additional 28.7% (52/181) screen some such children usually when requested to do so. Services that do not routinely screen children in private schools commented that they did perform screening when the school requested it (n = 31), but others said that resources prevented them giving full coverage (n = 10). Several said that private schools have their own arrangements for screening. The proportion of children in the UK attending private schools is up to 7%.24 One service that does not run a universal SES at all does carry out screening in two private schools on a fee-for-service basis. This was reported to be because the private schools insisted on having a screen and the service therefore provided it to make sure that it was conducted properly.

It was found that 55.5% (97/174) of services screen all (21.3%, 37/174) or some (34.5%, 60/174) children who are already known to have a hearing loss. (Note that the term ‘hearing loss’ was used throughout the questionnaire and has therefore been used in this chapter when reporting results.) One reason given for screening children known to be hearing impaired was that doing so was beneficial so as not to exclude children in mainstream education from a whole class activity. Eighteen services also stated that they would not be aware of the child’s hearing status prior to screening unless the child wore a hearing aid.

Most comments supporting not screening children with a known hearing loss referred to the fact that such children all had full audiology cover. Only two services specifically stated that screening a child with known hearing loss would have no value. Children who are educated at home are not screened by 71.8% (125/174) of services. Many services (22/63 commenting) were unaware of home-educated children, but a further 28 claimed to screen such children if they were requested to do so. One service commented that they were “not responsible for children educated at home through parental choice”. The most common reason given for not screening children attending special schools was that such children all receive full audiological testing routinely, or are looked after by specialist services. Additional comments recognised that some children not in state schools would be missed by the service provided, but that responsive services were available. Some services were actively addressing this issue. Table 4 details the arrangements made to screen children who did not attend the first scheduled screen. Children miss the screening opportunity offered in schools for a variety of reasons, including illness, periods of time spent out of the country, travelling families, lack of parental consent and transfer into the school after the screen. The majority of

Health Technology Assessment 2007; Vol. 11: No. 32

TABLE 4 The arrangements in place to screen children for whom consent is obtained but who did not attend the screen for any reason (e.g. through school absence, had a cold) (question 2)

Revisit to the school Appointment arranged at school health clinic Appointment arranged at audiology clinic No arrangement made Othera

All of the time

Most of the time

Some of the time

Rarely

Never

Total responses

Missing n (% of 195 responses)

135 (76.7) 8 (6.0)

26 (14.8) 4 (3.0)

13 (7.4) 34 (25.6)

1 (0.6) 25 (18.8)

1 (0.6) 62 (46.6)

176 (100) 133 (100)

19 (9.7) 62 (31.8)

6 (4.3)

5 (3.6)

28 (20.3)

46 (33.3)

53 (38.4)

138 (100)

57 (29.2)

2 (1.7) 1

14 (12.1) 2

100 (86.2) –

116 (100) 5

79 (40.5) –

– 2

– –

Data are shown as n (%). a Retest in school next year (2), next term (1); direct referral to doctor if concern (1); referral to school nurse audiology clinic (1).

TABLE 5 The school year in which the school entry hearing screen is routinely performed (question 3) n (% of 181 responsesb) Preschool Reception/primary 1a Year 1/primary 2a Year 2/primary 3a Other

1 (0.6) 160 (88.4) 48 (26.5) 6 (3.3) 19 (10.5)

Primary 1, 2 and 3 refer to the system in Scotland and are equivalent to the year numbers in the English system. b Forty-three responses indicated multiple (two or three) answers.

services (91.5%, 161/176) make arrangements to screen non-attendees during a revisit to the school and 13.8% (16/116) of services say they rarely or sometimes make no arrangement to screen. Respondents indicated that they would screen children in later school years if necessary and that the procedures in place for screening such children usually involved recall to the community clinic. The majority of screening (88.4%, 160/181) is performed in the first year of primary school (Table 5). Screening occurring at other times included screening a child in any school year if they were new to the school and had no evidence of undergoing a previous hearing screen; ‘responsive screening’ at any time if concerns were raised about a child’s hearing; and annual screening of special cases such as children with visual impairment or Down’s syndrome. © Queen’s Printer and Controller of HMSO 2007. All rights reserved.

Commenting on the timing of the screen, respondents referred to the conflict that needs to be considered between screening early (in the reception year) to ensure early identification of any problems versus screening later (in year 1) to maximise the child’s maturity and ability to perform the tests and thereby reduce the referral rate.

Screening procedure An important aspect of any screening programme is the information provided to, and consent received from, those covered by the screen. Table 6 lists the documentation available: 123 services were able to provide documentation. Four services used an opt-out system for consent and several others used a global consent for the wider school health check to include consent for hearing screening. Although all respondents reported screening within the school most or some of the time (Table 7) it is notable that comments made highlighted that the conditions under which the screen was frequently performed are very variable (Table 8) and can be problematic, and that suitable conditions are sometimes difficult to identify. Seven respondents said that they would refuse to screen in unsuitable conditions. No services routinely use a sound-treated booth or room (one did use a sound-treated van), but most services operate the screen in ‘quiet’ areas of the school.

Test methods Services were fairly equally divided in whether they always implemented the SES as a stand-alone screen or incorporated it into a wider health check

National survey of current screening practice

TABLE 6 The extent to which written documentation concerning the SES is available (question 4) Yes

Total responses

Missing n (% of 195 responses)

Parent/guardian agreement for the screen

151 (85.3)

26 (14.7)

177 (100)

18 (9.2)

Information provided to the parent/guardian prior to screening

124 (71.3)

50 (28.7)

174 (100)

21 (10.8)

Information provided to the parent/guardian prior to referral

142 (83.5)

28 (16.5)

170 (100)

25 (12.8)

Test protocol

137 (85.1)

24 (14.9)

161 (100)

34 (17.4)

Retest protocol

129 (80.1)

32 (19.9)

161 (100)

34 (17.4)

Referral protocol

133 (84.2)

25 (15.8)

158 (100)

37 (19.0)

Data are shown as n (%). TABLE 7 The location of the first test within the school entry hearing screen (question 5)

School Community clinic Home GP clinic Other

All of the time

Most of the time

Some of the time

Rarely

Never

Total responses

Missing n (% of 195 responses)

156 (86.2) – – – –

25 (13.8) 4 (3.1) – – –

– 19 (15.0) 3 (2.4) 2 (1.6) 2

– 55 (43.3) 39 (31.5) 9 (7.4) 1

– 49 (38.6) 82 (66.1) 111 (91.0) 1

181 (100) 127 (100) 124 (100) 122 (100) 4

14 (7.2) 68 (34.9) 71 (36.4) 73 (37.4)

Data are shown as n (%). TABLE 8 The conditions under which the school entry hearing screen is performed (question 6)

Soundproof booth Sound-treated room Quiet office Noisy office Quiet classroom/area Noisy classroom/area Othera

All of the time

Most of the time

Some of the time

Rarely

Never

Total responses

Missing n (% of 195 responses)

– – 18 (10.8) – 16 (9.6) – 2

– – 87 (52.4) 4 (2.8) 48 (28.9) 6 (4.1) 3

– 3 (2.1) 54 (32.5) 49 (34.0) 65 (39.2) 31 (21.2) 17

1 (0.7) 14 (9.9) 4 (2.4) 36 (25.0) 19 (11.4) 27 (18.5) 2

140 (99.3) 125 (88.0) 3 (1.8) 55 (38.2) 18 (10.8) 82 (56.2) –

141 (100) 142 (100) 166 (100) 144 (100) 166 (100) 146 (100) 24

54 (27.7) 55 (28.2) 29 (14.9) 51 (26.2) 29 (14.9) 49 (25.1) 171 (87.7)

Data are shown as n (%). a Twenty-four services mentioned 14 different areas of the school: medical room (15), staff room (10), library (4), corridor (3), stationery cupboard/store room/broom cupboard (4), main hall (2), sound-treated van, kitchen, hall, toilet, entrance area, head’s office (1 each).

(Table 9), but if those who answered ‘most of the time’ are included, 72.8% (115/158) incorporate screening as part of a wider health check, compared with 52.7% (70/133) who screen for hearing loss on a separate occasion.

The estimates of children that could feasibly be screened in 1 day were very variable (Table 10) and were said to depend on many practical factors, including:

●

child: – child’s understanding and cooperation – ease of testing – attendance rates administration: – mistake-free administration – ability to manage workload staff: – numbers – skills

Health Technology Assessment 2007; Vol. 11: No. 32

TABLE 9 The extent to which children at school entry are screened for hearing loss only or for hearing loss as part of a wider health check (question 9) All of the time

Most of the time

Some of the time

Rarely

Never

Total responses

Missing n (% of 195 responses)

Screen for hearing loss only

65 (48.9)

5 (3.8)

19 (14.3)

24 (18.0)

20 (15.0)

133 (100)

62 (31.8)

Screen for hearing loss as part of a wider health check

95 (60.1)

20 (12.7)

5 (3.2)

33 (20.9)

158 (100)

37 (19.0)

Data are shown as n (%).

TABLE 10 Estimates of the numbers of children (minimum, average and maximum) that could be screened under normal circumstances during the course of a one-day visit to a school (question 10) Number of children

No. of responses

Lowest responsea

Highest response

Median response

94 92 93

1 2 5

45 60 110

10 33 40

1 2 3

20 55 90

5 14 20

(a) When screening for hearing loss only Minimum Average Maximum

(b) When screening for hearing loss as part of a wider health check Minimum 96 Average 99 Maximum 100 a

●

One service reported only testing one child per day.

school: – size and location (several small rural schools entailing greater time spent on travelling) – support provided by schools – level of disturbance – other activities for children – experience of school nurses available time: – length of session – travelling time – number of schools visited.

Not all services screen children for a whole day. Some run the screening in the morning and do the administration and paperwork in school in the afternoon; others attend schools only for half a day at a time. Where services stated the time taken to screen a child, estimates varied from nine per hour to 20 minutes per child. The majority of services (71.7%, 124/173) implement a two-test screen, with only 16.8% (29/173) referring after a single test (Table 11). © Queen’s Printer and Controller of HMSO 2007. All rights reserved.

TABLE 11 The number of tests routinely performed within the screening programme before onward referral (question 7) No. of tests before referral 1 2 3 4 Total Missing responses

n (%) 29 (16.8) 124 (71.7) 18 (10.4) 2 (1.2) 173 (100) 22 (11.3)

Pure tone (PT) sweep testing is used by 97.1% (170/175) of services as the first test. Thirty-one services (18.1%) add in pure tone audiometry (PTA) and/or tympanometry and/or otoscopy at the second test (Table 12). When a two-test screen is carried out, the time between tests varies from doing both tests on the same day up to an interval between tests of more than 12 weeks (Table 13). The majority of referrals are made to ‘audiology’, but details of whether this was a second tier

National survey of current screening practice

TABLE 12 The test methods used within the screening programme (question 7) Test no.

1 2 3

PTA

PT sweep

Tympanometry

Otoscopy

Total responses

Missing n (% of 195 responses)

4 (2.3) 29 (20.3) 16 (72.7)

170 (97.1) 112 (78.3) 5 (22.7)

1 (0.6) 1 (0.7) 1 (4.5)

1 (0.6) 1 (0.7) –

175 (100) 143 (100) 22 (100)

22 (11.3) 52 (26.7) 173 (88.7)

Data are shown as n (%).

TABLE 13 The time between tests for services implementing more than one test (question 7) Between tests

1 and 2 2 and 3

Same day

2 to 12 weeks

59 (44.7) 4 (33.3)

6 (4.5)

Total responses

Missing n (% of 195 responses)

132 (100) 12 (100)

63 (32.3) 183 (93.8)

Data are shown as n (%).

TABLE 14 Service/professional to which onward referral is made on failure of the screen (question 7) Referral to Audiology/hearing clinic GP Children’s hearing assessment clinic Community paediatrician/medical officer ENT Community/hearing/school nurse clinic Total responses Missing responses

n (%) 127 (75.1) 15 (8.8) 7 (4.1) 6 (3.6) 3 (1.8) 11 (6.5) 169 (100) 26 (13.3)

ENT, ear, nose and throat.

community clinic or a tertiary audiology clinic were not given. Fifteen services (8.8%) refer the child to their GP (Table 14).

Pass/fail criteria The test levels at which services decide a child should be retested or referred varied widely with many combinations of levels and frequencies. The details for the first test are shown in Table 15. Onehundred and forty of the 195 services consistently screened at 20 or 25 dB for 1, 2 and 4 kHz, with a variety of other levels and frequencies added to that base.

One-hundred and thirty-five services gave levels at which the child would be said not to have passed the test at test 2. Of these, only 30 were different from the levels at test 1. Seven added a

frequency, 12 increased the level, one decreased the level, nine tested to threshold, and one included fails at tympanometry and otoscopy (unspecified). Twenty-one services gave levels at which the child would be said not to have passed the test at test 3. Two added a fail at tympanometry, six tested to threshold, one decreased the level, five increased the level and one added observation of behaviour.

Screen personnel and equipment Staff from different professional backgrounds are involved in carrying out the SES in different parts of the country (Table 16). Of the respondents, the majority of screeners are school nurses (66.3% of services) or their assistants (18.5% of services). Many services emphasised the importance of training for staff undertaking the SES. A general theme emerged of staff less qualified in audiology undertaking the initial screen(s) followed by referral when necessary to audiologically qualified staff. Resource issues were cited as the reason for using ‘cheaper’ staff, including one instance of ‘mumsy’ ladies who know the schools and children. In contrast, the benefit of employing highly qualified staff, which meant fewer unnecessary referrals, was also mentioned. The majority of services (94.3%) use screening audiometers (Table 17).

Health Technology Assessment 2007; Vol. 11: No. 32

TABLE 15 Pass/fail criteria (levels and frequencies) (question 8) Screening level and frequencies

No. of responses (%)

20 dB at 1, 2 and 4 kHz No other frequencies + 20 dB at 500 Hz only + 25 dB at 500 Hz and/or 250 Hz + 30 dB at 500 Hz only + 20 dB at 500 Hz and/or at 8 kHz and/or at 6 kHz and/or at 250 Hz + 30 dB at 250 and 500 Hz and 8 kHz All other frequencies (not specified) at 20 dB

51 (29.3) 6 16 13 7 4 2 3

25 dB at 1, 2 and 4 kHz No other frequencies + 25 dB at 500 Hz only + 30 dB at 500 Hz and/or 250 Hz and/or 6 kHz + 25 dB at 500 Hz and 8 kHz and/or 250 Hz and/or 6 kHz All other frequencies (not specified) at 25 dB

89 (51.1) 10 37 29 9 4

30dB at 1, 2 and 4 kHz No other frequencies + 30 dB at 500 Hz and 8 kHz and/or 250 Hz + 35 dB at 500 Hz only All other frequencies (not specified) at 30 dB

15 (8.6) 1 8 2 4

Varying between 20 and 30 dB at 1, 2 and 4 kHz No other frequencies + 25–35 dB at 500 Hz only Other combinations Total response Missing or unclassifiable

19 (10.9) 4 10 5 174 (100) 21 (10.8)

TABLE 16 Staff who perform the SES (question 11) n (% of 178 responsesa) School nurse School nurse assistant Nursery nurses Health care assistants/support workers/school health assistants Audiometrician Audiologist/assistant audiologist Technicians Screeners School doctor a

118 (66.3) 33 (18.5) 31 (17.4) 29 (16.3) 28 (15.7) 18 (10.1) 14 (7.9) 11 (6.2) 5 (2.8)

Eighty-eight services mentioned more than one grade of staff.

TABLE 17 Types of equipment and numbers used within the SES (question 12)

a. Screening audiometer b. Diagnostic audiometer c. Screening tympanometer d. Diagnostic tympanometer Othera

Total responding

No. owned

164 (94.3) 30 (29.1) 23 (21.9) 8 (8.33) 7

174 103 105 96

1–48 1–24 1–6

Auroscope (2), otoacoustic emissions test for special schools (1), Quick tymp audiometer/tympanometer (1), sound level meter (2), otoscope (1). Combinations: screening audiometer and screening tympanometer (a + c ) = 12; screening and diagnostic audiometer (a + b ) = 17; screening and diagnostic audiometer and screening tympanometer (a + b + c) = 8.

National survey of current screening practice

TABLE 18 Number of services with a recently performed audit of the SES, data management systems in place, and easily obtainable reports (questions 13, 14 and 15)

Recent audit Data management system Data reports easily obtainable

Total responses

Missing responses

16 (9.5) 112 (69.6) 52 (46.4)

168 (100) 161 (100) 112 (100)

27 (13.8) 34 (17.4) 83 (42.6)

Data are shown as n (%).

TABLE 19 Coverage and referral rates for the SES (question 16) No. of service leads providing reliable data

Mean

Median

Range n 352–10,291

Children eligible for the screen

2704

2229

–

Children undergoing the screen

2512

2176

95.2

234–9483

56.3–100.0

Children referred for further assessment after failing the screen

246

168

7.9

23–1977

1.91–23.4

Several services commented that all equipment, is calibrated annually and that each member of staff had their own equipment, but others referred to equipment that was very old and the inability to replace equipment owing to resource limitations. The use of otoscopes only by staff qualified to use them and the need for explicit parental consent for an invasive procedure were mentioned as reasons for not using them.

Children

–

Fifty-five respondents (28.2% of 195) provided data on children eligible for the screen from which coverage figures could be calculated. The percentage of eligible children who were screened (coverage) ranged from 56.3 to 100%, with a median of 95.2% (mean 91.1%). Nearly threequarters of services (74.5%) achieved more than 90% coverage.

Audit data Services were asked whether an audit of the SES had taken place in the last 2 years, whether they used any sort of data management system and, if so, whether reports were easily obtainable (Table 18).

Forty-six respondents provided data from which referral rates overall could be calculated. The percentage of screened children who were referred for further assessment ranged from 1.91 to 23.4% with a median of 7.9% (mean 7.7%). Eleven of the 57 services (19.3%) gave values of zero for referrals.

Less than 10% of services (16/168) have performed any audit of their service in the last 2 years. Approximately 70% (112/161) of services use some sort of data management system for the SES, but only half of those can easily obtain data reports from it.

The pattern of pass/fail criteria for these 46 services did not differ from the pattern for the services for which data are presented in Table 15. Data were unavailable on the number of children referred at different stages of the screening programme.

Coverage, referral rates and yield Table 18 indicated that few services were able to provide accurate data on coverage and referral rates from the SES. Table 19 indicates the data considered by respondents to be reliable. Data that were estimated or guessed by services have not been included.

Comments made by respondents reflected the poor data management of the SES in general. In particular, several services commented that data were available on numbers eligible, screened and referred (Table 19), but not on outcomes, with fewer than 20 respondents able to give numbers (Table 20). Many emphasised that although (some)

Health Technology Assessment 2007; Vol. 11: No. 32

TABLE 20 Yield (numbers of children) from the SES for different definitions of hearing loss (question 16) Children identified after failing the screen with …

No. of service leads providing reliable data

No. of children: range

18 11 15 7

0–56 0–23 0–305 0–11

… sensorineural hearing loss … permanent conductive hearing loss … temporary conductive hearing loss … other types of hearing loss

TABLE 21 Percentage of children confirmed to have one of three different definitions of hearing loss after referral from the SES as a percentage of those screened and as a percentage of those referred (question 16) Yield

No. of service leads providing reliable data

Median

Range

(a) Sensorineural hearing loss Percentage of those referred (excluding 0%) Percentage of those screened (excluding 0%)

18 15 17 14

1.44 1.71 0.11 0.12

0–12.16 0.62–12.16 0–0.59 0.05–0.59

(b) Permanent conductive hearing loss Percentage of those referred (excluding 0%) Percentage of those screened (excluding 0%)

11 5 10 4

0 3.42 0 0.09

0–17.56 1.24–17.56 0–0.44 0.07–0.44

(c) Temporary conductive hearing loss Percentage of those referred (excluding 0%) Percentage of those screened (excluding 0%)

14 13 14 13

35.88 35.88 2.48 2.77

0–100.0 7.74–100.0 0–7.56 0.72–7.56

data were collected it was not easy or possible to retrieve them in any meaningful form. From the data in Tables 19 and 20, figures for the yield from the SES may be calculated. Table 21 presents the yield as the percentage of children referred and as the percentage of children screened who were subsequently identified as having sensorineural hearing loss, permanent conductive hearing loss or temporary conductive hearing loss. For example, 18 services provided data on the number of children identified with sensorineural hearing loss and the number of children referred. A median yield of 1.44% of those referred can be calculated for the data from these 18 services, with a range of 0 to 12.16%. The rows in italics refer to the data excluding services where no children were identified.

Respondents’ views on the value and continued need for the SES Services were asked whether they had any plans for development or change of the SES. For those © Queen’s Printer and Controller of HMSO 2007. All rights reserved.

services who responded, 28.6% (50/175) are planning to change their current practice in some way, several in response to the implementation of the NHSP (Table 22). Services were also asked to rate the overall usefulness of the SES as it was currently operated. They were asked to indicate one of ten boxes on a range from ‘not useful at all’ to ‘very useful’ (question 20) (Table 23). Thus, 69.3% rated it as 8 or higher, more than one-third (33.5%) rating it as 10. Only 12 services of the 176 responding (6.8%) rated it as 4 or less. Positive comments about the service were made by 184 services. The most common was that the screen effectively identifies children with hearing impairment; in general (38), impairment of late onset or acquired since the newborn screen (30), OME (20), unilateral (15) or unsuspected losses (seven). Other suggested benefits of the screen were that it could exclude hearing loss as a cause

National survey of current screening practice

TABLE 22 Proposed developments in the SES (question 19) Development plans

Changes to the staff running the screen Generally reviewing the screen Considering changes or discontinuation in the light of the introduction of NHSP Considering discontinuation Considering targeting the screen Further development of IT systems Changing the protocol with regard to screening levels Provision of modern equipment (2) or training (2) Improving relationship with second tier audiology services Waiting for these results

15 13 8 4 5 3 2 4 2 2

TABLE 23 Rates of the overall usefulness of the school entry hearing screen as it is currently operated (question 20) 1

Not useful at all No. of responses %

Very useful 1 0.6

5 2.8

3 1.7

10 5.7

9 5.1

23 13.1

35 19.9

28 15.9

59 33.5

Total n = 176.

of a child’s difficulties (24) and that it raised awareness among teachers and parents and allowed surveillance (13). In contrast, only 54 negative comments were made about the screen service. These were concerned with poor test conditions (12), high referral rates and/or low yield (ten), the provision of a less than adequate service owing to lack of resources (seven), poor information on outcomes (six), high rates of non-attendance at follow-up (five), and uncertainties following the introduction of newborn screening (two). Respondents were asked specifically for their suggestions for the future of the SES, either locally or nationally (question 21). Twenty respondents said that the screen must continue and only two specifically said that it should stop. The future of the screen in relation to newborn screening was again highlighted (17), with suggestions for introduction of a targeted SES. Other suggestions were for better standards/guidelines (seven), increased coverage of private schools (six), improved test conditions, and better information technology (IT) support and data collection.

Finally, respondents were invited to add any further comments. Many of these reiterated, for emphasis, comments made earlier in the questionnaire concerning inadequate conditions, IT support, training, etc.

Summary No national protocol exists for the SES programme and there are wide variations in its implementation throughout the UK in terms of the population covered, the physical location and conditions under which the screen is implemented, the test methodology, the criteria used to determine which children to refer, the personnel and the equipment involved, and the ability to collect and then retrieve data. Much of the variation appears to be due to limitations of resources to implement the screen in the light of competing activities for the range of staff employed to run it. Data that could be used to assess outcome/ performance and thereby to determine which methods, if any, were effective in achieving the aims of the screen are not routinely available. This lack of data, combined with the wide variation, meant that the authors were unable to investigate whether significant clusters of services had similar aims, tests and operational characteristics. Despite the difficulties and the lack of robust evidence, most service leads think the screen is useful and do not want to stop using it, even though they recognise that its worth may become even less with the advent of universal newborn hearing screening. Guidelines concerning the value of a selective screen and the population for whom it would be appropriate to target it would be welcomed by many service leads.

Health Technology Assessment 2007; Vol. 11: No. 32

Chapter 3 Possible effects of newborn hearing screening on the school entry hearing screen: evidence from a series of studies in Waltham Forest Introduction Primary preventive measures have affected the underlying epidemiology of permanent childhood hearing impairment (PCHI). There has been a reduction in the incidence of hearing impairment attributable to rhesus incompatibility, and in that remaining as a legacy of congenital rubella, but more recent immunisations for meningococcal and Haemophilus influenzae type b (Hib) disease have also had a demonstrable impact which should have consequences for the occurrence of hearing impairment (see www.hpa.org.uk: Vaccine Preventable Diseases; data generated by the Centre for Infections). Meningitis, measles and mumps have all been implicated as causes of unilateral acquired hearing impairment25 for which in the past the SES has been an important route to identification. Although there are current issues concerning measles, mumps and rubella (MMR) immunisation uptake, the fall in both measles and mumps notifications that followed the introduction of vaccine in 1988 was dramatic, and predictably such preventive measures have consequences for the occurrence of hearing impairment. Improvements in secondary prevention also have implications for the SES. Since the SES became a valued (although rarely evaluated) screen in the 1960s, audiology services and programmes of early detection have developed substantially. Potentially the most significant factor has been the implementation of neonatal screening. The NHSP has been rolled out across England, but the implications for the SES will not be fully realised until the first cohorts reach school age. However, the Whipps Cross University Hospital universal newborn hearing screen (UNHS) has been implemented as an audited service since 1992 predating the country-wide implementation of newborn hearing screening by more than a decade. Although the Whipps Cross UNHS and the NHSP differ in terms of protocol details [transient evoked otoacoustic emission test (TEOAE) followed if failed by a second TEOAE © Queen’s Printer and Controller of HMSO 2007. All rights reserved.

test, with screen referral if either test failed for the former; TEOAE followed if failed by AABR, with screen referral if AABR failed for the latter], longitudinal data from the Whipps Cross screen provide a valuable and quasi-controlled indication of the likely implications for later screens such as the SES. The Whipps Cross UNHS was implemented for the newborn population in the East London district of Waltham Forest. A single audiology service with long-term stability has been responsible for the screen follow-up programme and for the audiological provision for a population of around half a million in Waltham Forest and in the neighbouring district of Redbridge. Whereas the universal screen aimed at screening all newborns in Waltham Forest, a more limited targeted neonatal hearing screen (TNHS), aimed at newborns at risk of hearing impairment, was undertaken in Redbridge. Prospective follow-up of the cohorts has been undertaken by the audiology service and evaluation of process, surrogate outcomes and actual outcome reviewed.26–30 The available information has also been used to evaluate the current worth of the local SES. The necessity for a local evidence base to be available to inform local policy has been emphasised,5 but there are concerns about the applicability of studies with a small population base to inform wider policy. However, the Waltham Forest cohorts will be unique within the UK until the much larger long-term follow-up evaluations from the NHSP are available, and the use of the local cohorts to inform wider policy has therefore been maximised by using longitudinal population studies and by comparing the results with national studies or with data drawn from larger populations.

Changes in the programmes of early screening and identification Approaches to screening for hearing impairment in preschool and primary age children in Waltham Forest are detailed in Figure 1. Changes were based

Possible effects of newborn hearing screening on the school entry hearing screen

Reactive referrals from concerns and from surveillance Sweep test in junior school (8–9 years) SES in first year of primary school (5–6 years) Intermediate screen with toy test or ‘go game’ (3 years) Universal IDT replaced by targeted Neonatal screen with TNHS replaced by UNHS 1977–1981

1982–1986

1987–1991

1992–1996

1997–2001

5-year cohorts FIGURE 1 Components in the Waltham Forest programmes of secondary prevention for 5-year cohorts born over the 25 years from 1977 to 2002

on epidemiological data and screening results from the 1970s to 2002, when local implementations became centrally directed.26–28,31–35 Throughout the 25 years reactive referrals were received from primary health care, from paediatric and ENT services, and also directly from parents. The IDT was undertaken as a universal infant hearing screen at 8 months of age, until 1996. UNHS was introduced after a short period of targeted neonatal screening and the universal IDT was replaced by a targeted screen in 1996. Changes in Redbridge mirrored those in Waltham Forest, but the TNHS was retained from 1990 and the IDT remained a universal screen throughout the period.

The SES has always been considered an important backstop universal screen. It was typical of that used in many other districts and was a sixfrequency sweep from 250 to 8000 Hz at 25 dB HL undertaken in school by school nurses. Screen positives were retested whenever possible in school, and failure at the second test prompted referral to the local clinic where threshold audiometry was undertaken. Children with OME were referred onwards to either their GP or ENT services after a period of conservative management in second tier or school nurse hearing clinics. Children with PCHI of any degree,

or any diagnostic uncertainties, were referred to the audiology services, and children with PCHI reactively referred to the ENT or paediatric service were redirected to audiology. Screen results were initially reported to the health authority Directorate of Information, and from the 1990s were computer recorded on a Regional Interactive Child Health System (RICHS). Data retrieval was routinely undertaken through these sources and through notifications to the educational service for hearing impairment.

Evaluating the SES The investigation consisted of two complementary evaluations.

Evaluation A The changing worth of the SES in terms of the yield of children with a significant PCHI picked up by the screen was measured longitudinally. PCHIs are notified to educational and audiology services and their aggregated details periodically analysed. The number of PCHIs ascertained from cohorts that have received the SES, the identification methods and audiological data were available for comparison of three cohorts with different detection programmes:

Health Technology Assessment 2007; Vol. 11: No. 32

●

an historical 10-year Waltham Forest cohort born from January 1977 to 1987 when no neonatal hearing screen (NNHS) was in place; the size of this NNHS cohort was 31,538 (cohort 1, NNHS) a 10-year Redbridge cohort born from January 1990 to 2000 when a TNHS was in place; the size of this TNHS cohort was 32,890 (cohort 2, TNHS) an 8-year Waltham Forest cohort born from January 1992 to 2000 when a UNHS was in place; the size of this UNHS cohort was 29,132 (cohort 3, UNHS).

The three cohorts thus gave a total cohort of 93,560. Although the primary analyses are concerned with comparisons between cohorts, useful information (e.g. overall prevalence rates) can be derived from some combined analyses (combined cohort).

Evaluation B The SES has also been argued to have an additional ‘useful’ yield of children with a minimal PCHI or with temporary fluctuating OME. Because these children are not routinely and invariably notified to the educational or audiology services an additional population-based crosssectional survey was undertaken by examining the school health records of a 6-year Waltham Forest cohort (January 1993 to 1999) of 19,296 children (cohort 4) who were eligible to have received the SES in the local education authority schools, and who had previously been enrolled into the UNHS. The RICHS was interrogated for the study. It enabled local comparative evaluation of process and results with historical returns from a 3-year SES undertaken on 9301 children from 1986 to 1989 (cohort 5) and with a 5-year Waltham Forest cohort of 15,536 children who were born up to December 1982 (cohort 6) and reported after they had all received the SES;32 neither of these cohorts had had a newborn hearing screen.

Generalising the study Generalisation requires contextualisation of the results within the local demographic. The level of deprivation36 and Asian ethnic background13 are both factors that have been found to increase the odds ratio for PCHI within a community, and inward flow of children into the district from abroad after the newborn or infant screens, and before school entry, are characteristics that are not uniform nationwide. Nevertheless, they are pertinent to the wider interpretation of the © Queen’s Printer and Controller of HMSO 2007. All rights reserved.

current data. The combined population of Redbridge and Waltham Forest in the mid-1990s was 449,500. The live birth rate in Waltham Forest was 3500 per annum, with that in Redbridge being slightly less. Although the districts are immediate neighbours, the populations have some differences. The Department of the Environment Index of Local Conditions using data from the 1991 census ranked Waltham Forest as 20th on the index of the most deprived boroughs, while Redbridge was ranked 120th out of 366 English boroughs. In both there were ethnic minority communities (31% in Waltham Forest and 28% in Redbridge). In Redbridge the largest ethnic community was Indian in origin, and Waltham Forest had the largest Pakistani community, the eighth largest black Caribbean community and the ninth largest Bangladeshi community in London. During the 1990s there was also an increasing number of refugees to both boroughs, principally Turkish, Kurdish and Somalian. The majority grouping in both was Somali. The longitudinal cohort evaluation required assessment of changes in the Waltham Forest population. Data from the 1981 census confirmed that the population in the 1980s was slightly lower than it was a decade later (214,500 in the mid-1980s), with a slightly lower birth rate of around 3000 per year. However, the overall level of deprivation measured by the 1981 census was largely unchanged when compared with later measures from the Index of Local Conditions. Local data on the epidemiology of hearing impairment were compared with those derived from three studies with a national or wider population base: ● ●

●

the prevalence of PCHI in the UK12 the MRC Institute of Hearing Research epidemiology of PCHI in a cohort of 366,480 from the Trent region13 evaluation of the NHSP in England.9

The local population-based cross-sectional survey of the SES process and result was compared with the national BACDA study of 109,505 school hearing screening tests undertaken by 43 services for the school year 2000/01 (the 1995/96 birth cohort).6

Changes in the SES Although SES protocols and procedures remained unchanged throughout the longitudinal cohort comparison, the school nursing services had been

Possible effects of newborn hearing screening on the school entry hearing screen

increasingly used to provide other preventive services and since 2000 this has affected the performance of the SES in Waltham Forest. During the 1980s and up until the end of the 1990s, 90% or over of schoolchildren in the first year of primary school received a SES. Enrolment to the SES for the majority of the cohorts used in the longitudinal comparisons therefore remained high. However, in the cohorts screened since 2000 the proportion receiving the SES has gradually reduced and in 2005 was below 50%. The failure rate was 7.4%, with a mean age of referral for assessment in the school clinic of 5.5 years, and this had remained stable since the 1980s. The non-attendance (DNA) rate for follow-up had also remained stable at 20%. Comparison with the BACDA survey6 confirmed that these results were typical of those achieved elsewhere in the UK. Did the gradually falling SES enrolment after 2000 influence the yield? The non-screening referrals to the audiology service were examined and the age distribution and referral rate from cohort 1, NNHS, were compared with the latest year. In fact, despite all the changes in earlier screens and surveillance, the age distribution and referral rate of reactive vigilant referrals remained remarkably stable. The modal age of referral remained at between 3 and 4 years, with a mean age of referral of 4.81 years in 1986 and 4.65 years in 2005. The reduced coverage of the SES appears therefore not to have resulted in increased numbers identified through reactive referral, and there has been no influx of schoolchildren with PCHI who have been reactively referred. Longitudinal comparison of the epidemiology of hearing impairment was also made to ensure that there was no fall in the number of identified cases in recent years. Comparison was also made with the epidemiological study of PCHI in Trent.13 The prevalence of PCHI at school age also remained longitudinally stable.

Results Evaluation A

Prevalence From the combined cohort of 93,560 in their first year of primary school, 349 children with a unilateral or bilateral PCHI of mild degree or greater (>20 dB HL averaged over 0.5, 1, 2 and 4 kHz) were ascertained. Severity was classified as mild (hearing level 40 dB HL

Bilateral PCHI FIGURE 2 The prevalence of bilateral PCHI and 95% CIs for the combined cohorts (n = 93,560)

cohorts in the prevalence of cases (congenital and acquired) of unilateral hearing impairment (Figure 3). Note that for unilateral hearing impairment it is more reasonable, in terms of the extent of disability, to group moderate impairment with mild rather than severe. Cases of severe or profound unilateral hearing impairment (children colloquially considered to have a ‘dead ear’) fell from 0.95 in 1000 in cohort 1, NNHS, to 0.27 in 1000 in cohort 3, UNHS (␹2 13.338, df 2, p = 0.001), with those with a milder unilateral PCHI remaining stable across the longitudinal cohorts (␹2 0.609, df 2, p = 0.737). The aetiologies were investigated, but causation was usually elusive. Only 18.5% had onset definitely temporally related to illness and the remainder were considered to be congenital, although usually this was because of absence of firm evidence that the unilateral impairment was related to an illness, rather than clear evidence that it was present at birth. In the majority (65%) there was no known cause and it was in this category that there was a highly significant fall in prevalence. The prevalence decreased by a half from 0.86 in 1000 (95% CI 0.53 to 1.18) in cohort 1, NNHS, to 0.41 in 1000 (95% CI 0.18 to 0.64) in cohort 3, UNHS. Once again, comparative prevalence rates were not available from the Trent or national prevalence studies and therefore generalisation is less robust. However, the UK Child Development © Queen’s Printer and Controller of HMSO 2007. All rights reserved.

Studies reported 0.8 in 1000 children aged 7 years to have such an impairment,37 and other historical surveys have reported profound unilateral hearing impairment in 1 in 1000 schoolchildren (many studies cited by Bess and colleagues 198625). These were of the same order as the prevalence rate reported in the cohort 3, NNHS. The prevalence rate of all degrees of unilateral congenital hearing impairment reported from the recent NHSP evaluation was 0.64 in 1000 (95% CI 0.37 to 0.91), of whom less than half had a severe or profound unilateral PCHI.9 Cohort 3, UNHS, used in the current study reported a unilateral congenital prevalence of 0.65 in 1000 (95% CI 0.36 to 0.95), with 0.24 in 1000 (95% CI 0.06 to 0.42) having a severe or profound unilateral PCHI. It is therefore probable that the significant fall in prevalence of profound unilateral hearing impairment can be generalised and it is likely that this is due to measures of primary prevention.

Age of confirmation of hearing impairment Cohort comparisons of the median and interquartile ages when the children were confirmed with a congenital PCHI are detailed in Table 24. The comparison confirmed that there had been a considerable secular reduction in the age when hearing impairment was confirmed when cohort 2, TNHS, and cohort 3, UHNS, that

Possible effects of newborn hearing screening on the school entry hearing screen

1.4 1.29 1.2

Prevalence per 1000

1.0 0.8

0.95 0.78

0.76

0.69 0.61

0.61

0.6 0.51 0.4

0.51

0.46

0.4 0.26

0.2

0.25

0.23

0.18

0.27 0.08

0 Cohort 1, NNHS 1977–1987

Cohort 2, TNHS 1990–2000

Cohort 3, UNHS 1992–2000

Cohort 1, NNHS 1977–1987

Mild or moderate unilateral hearing loss

Cohort 2, TNHS 1990–2000

Cohort 3, UNHS 1992–2000

Severe or profound unilateral hearing loss

FIGURE 3 The prevalence and 95% CIs for unilateral PCHI in the three cohorts

received either a targeted or universal newborn screen were compared with cohort 1, NNHS, when newborn screening was unavailable. In cohort 1, NNHS, the median age when hearing impairment was confirmed in those with severe or profound hearing impairment was at just turned 1 year of age. However, there was a much greater delay in the confirmation of those with a mild or moderate PCHI. This occurred on average after their fourth birthday and in the months running up to primary school entry. Once universal neonatal screening had been established there was a highly significant reduction to 10 weeks of age in confirming the presence of a severe or profound hearing impairment, with those with a moderate PCHI also now being confirmed on average well within the first half of infancy. Those with a mild bilateral PCHI were now being confirmed at around 3 years of age. The same pattern of improvements was also made in the age of confirmation of those with unilateral PCHI, with a highly significant overall reduction when cohort 1, NNHS, was compared with cohort 2, UNHS. Historically, unilateral PCHI had been confirmed when the children had reached their fifth birthday, but once again this was now reduced to the first half of infancy. These were highly significant

improvements in the average age at which all degrees of hearing impairment were identified in cohort 3, UNHS. Such improvements in the median age would be expected following the implementation of effective neonatal screens, but a statistic giving a better indication of late identifications is required to reflect the effectiveness of the system as a whole and the contribution from later screens such as the SES. The 75th centile is thus a more appropriate index for measuring improvements. The interquartile range (IQR) is included in Table 24. Historically, the upper quartile for the age of confirmation was within the first year of primary school for all degrees of PCHI, other than for severe and profound hearing impairment, where it was at 2.5 years of age. Had the remarkable improvements in the median age of confirmation following the implementation of the UNHS also been reflected in a lowering of the 75th centile? No child with a severe or profound bilateral PCHI remained with an unconfirmed hearing impairment after 3 years of age and the 75th centile had been reduced by over 1 year to just under 18 months. However, lesser improvements were seen in the other degrees of PCHI and the

Health Technology Assessment 2007; Vol. 11: No. 32

TABLE 24 Median ages of confirmation of hearing impairment and IQR for those who had remained resident or moved in with a previously unidentified PCHI in the three cohorts (n = 266) Degree of hearing impairment

Bilateral (PTA average BHE 500–4000 Hz)

Median age (IQR) (weeks) 1977–1987 Cohort 1 NNHS (n = 101)

1990–2000 Cohort 2 TNHS (n = 84)

1992–2000 Cohort 3 UNHS (n = 81)

Mild (20–39 dB HL)

218 (166–307)

187 (49–267)

150 (13–257)

Moderate (40–69 dB HL)

218 (130–252)

119 (23–198)

18 (8–247)

53 (47–131)

44 (20–157)

10 (8–76)

265 (194–286)

56 (15–298)

18 (11–243)

Severe/profound (艌70 dB HL) Unilateral (PTA average WHE 500–4000 Hz)

All unilateral (>20 dB HL)

TABLE 25 Median and IQR ages of referral and delays to confirmation for those with a congenital PCHI who had remained resident or moved in with a previously unidentified PCHI in cohort 3, UNHS Degree of hearing impairment

Mild bilateral (n = 28) Moderate bilateral (n = 19) Severe/profound bilateral (n = 16) All unilateral (n = 18)

Median (IQR) (weeks) Age of referral

Age of confirmation

Delay from referral to confirmation

115 (4–236) 11 (5–233) 6 (5–64) 9 (1–237)

150 (13–257) 18 (8–247) 10 (8–76) 18 (11–243)

13 (5–24) 6 (2–11) 3 (2–5) 6 (4–11)

75th centile remained within the weeks leading up to the fifth birthday. Around one-quarter of those children with any degree of PCHI other than a severe or profound hearing impairment therefore still remained to have that hearing impairment confirmed at around school entry, even though the average age of confirmation had been so drastically cut by the introduction of the neonatal screen. When the cumulative distributions of the age of confirmation were examined by severity, 22% of those with a moderate bilateral PCHI, 26% of those with a mild bilateral PCHI and 18% of those with a unilateral hearing impairment in the first year of primary school still remained to have their hearing impairment confirmed after 5 years of age. Although the introduction of neonatal screening had reduced the average age of confirmation, it appears to have done little to reduce the number of ‘stragglers’ in the overall system of detection.

Ages of referral The cohort evaluations used the age when the congenital hearing impairments were confirmed as the most robust and stable indicator available © Queen’s Printer and Controller of HMSO 2007. All rights reserved.

for the longitudinal comparison. However, confirmation delays may have reflected problems in the assessment of children who had actually received a timely referral. The age at referral was therefore examined separately for the cohort that had received the UNHS (cohort 3, UNHS, born 1992–2000) (Table 25). The delay from referral to confirmation was also measured. Median delays from referral to confirmation were less than 1 month for those with a severe or profound hearing impairment. They were slightly longer for those with a lesser degree or a unilateral hearing impairment, but even in those with a mild bilateral impairment the median delay between referral and confirmation was 3 months. The delays in confirmation therefore reflected delays in reactive referral or in referral from the screening programmes. When the cumulative distributions of the age of referral were examined by severity, 16% of those with a moderate bilateral PCHI, 18% of those with a mild bilateral PCHI and 17% of those with a unilateral hearing impairment who were in their first year of primary school still required identification and referral for hearing assessment.

Possible effects of newborn hearing screening on the school entry hearing screen

Unilateral

Mild bilateral

Moderate bilateral

Severe + profound bilateral

1.8

Yield per 1000 cohort

1.6 1.4 1.2 1.0 0.8 0.6 0.4 0.2

Neonatal screen

Parental or professional concerns 0–1 year

IDT

Intermediate screen at 3 years

Parental or professional concerns 1–5 years

Cohort 3, UNHS

Cohort 2, TNHS

Cohort 1, NNHS

Cohort 3, UNHS

Cohort 2, TNHS

Cohort 1, NNHS

Cohort 3, UNHS

Cohort 2, TNHS

Cohort 1, NNHS

Cohort 3, UNHS

Cohort 2, TNHS

Cohort 1, NNHS

Cohort 3, UNHS

Cohort 2, TNHS

Cohort 1, NNHS

Cohort 3, UNHS

Cohort 2, TNHS

Cohort 1, NNHS

SES

FIGURE 4 The yield per 1000 from the three cohorts by the method of identification

Routes to identification The comparative yields per 1000 cohort for the different identification methods in the three cohorts are detailed in Figure 4 and Table 26. There were significant longitudinal changes in the yields from all the identification methods, other than in the yield from parental or professional concerns from 1 to 5 years of age. (The slightly higher overall yield from cohort 1, NNHS, is accounted for by more unilateral cases, probably due to the incidence of mumps at this time.)

a period of 5 years. However, it gave a low yield that consisted entirely of those with a mild or unilateral hearing impairment. The largest yield from cohort 1, NNHS, was from assessments undertaken because of parental or professional concern from 1 to 5 years, and this included 45% of those with a congenital bilateral moderate or worse hearing impairment and 62.5% of those with an acquired or late-onset hearing impairment of this degree. The second largest yield for these cohorts came from the SES (Table 26).

Electrophysiological testing was only introduced for reactive neonatal referrals towards the end of the 10-year period of cohort 1 and early identification was based around the IDT screen at 8 months. The sensitivity of the IDT screen for those with a severe or profound congenital hearing impairment who remained in the district was 77%, but the sensitivity for lesser degrees of bilateral hearing impairment was only 25%. Because this low sensitivity of the IDT screen was widely experienced, the Advisory Committee on Services to Hearing Impaired People (ACSHIP) report (1981) recommended the implementation of an intermediate universal screen around 3 years of age, and this was undertaken in the district for

The implementation of a selective neonatal screen using the auditory brainstem response (ABR) testing of neonates with risk factors based on the American Joint Committee register (1982) gave an overall yield of 1.00 in 1000 neonates with PCHI, with 0.51 in 1000 having a moderate or worse bilateral PCHI. The sensitivity of the district TNHS programme for identifying moderate or worse bilateral congenital PCHI at 46.9% was typical of many other such programmes.5 However, the earlier identification offered by the TNHS was partly offset by a reduced yield from both parental and professional vigilance and from the IDT screen. By combining all three infant detection methods, a yield of 0.76 in 1000 with a

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TABLE 26 Comparison of the yields from the identification methods in the three cohorts Identification method

Yield of all PCHIs per 1000 in cohort (95% CI)

Pearson ␹2 (p)

Cohort 1 No NHS

Cohort 2 Targeted NHS

Cohort 3 Universal NHS

1.003 (0.71–1.41)

1.58 (1.18–2.11)

4.03 (0.045)

Concerns 0–1 year

0.44 (0.26–0.75)

0.12 (0.05–0.31)

0.14 (0.05–0.35)

8.84 (0.01)

IDT screen

0.89 (0.61–1.28)

0.61 (0.39–0.94)

0.10 (0.04–0.30)

17.49 (20 dB at 2 kHz and ENT >25 dB at 4 kHz examination

56%

52%

Hammond Parental et al., 199763 questionnaire

Finally, question 13 addresses the issue of whether any interpretable or intermediate test results were reported. The fact that most studies failed to report these could mean either that there were no uninterpretable test results or that they simply were not recorded, which in turn could be hiding any practical problems encountered in actually applying the tests.

Findings Test accuracy A wide variety of hearing tests (and protocols) was evaluated. Although the reference test varied, most studies used pure tone audiometric testing. Presented below are the sensitivity and specificity values, grouped and tabulated according to the screening test compared where possible to PTA with a hearing impairment cut-off ranging from 15 to 30 dB at various frequencies. Where studies reported multiple comparisons, the sensitivity and specificity values are reported for each comparison separately (Tables 30–37). The two-by-two tables for sensitivity and specificity, where available, are presented in Appendix 10. Parental questionnaires Three studies examined the accuracy of parental questionnaire against PTA (Table 30). Both sensitivity (34–71%) and specificity (52–95%) range widely.

Impedance audiometry/tympanometry Nine studies reported 11 different comparisons of the accuracy of impedance audiometry/ tympanometry compared with PTA, otoscopy or a combined test reference standard (Table 31). Against otoscopy the sensitivity (50–90%) and specificity (65–97%) of tympanometry was moderate to good. However, compared with

Definition of Sensitivity Specificity hearing impairment

>20 dB at 0.5, 1, 2 and 4 kHz

PTA, the test accuracy of tympanometry appeared to be more variable (sensitivity of 40–90% and specificity of 57–85%) and dependent on the tympanometry fail criteria used. FitzZaland and Zink55 reported a good level of tympanometry accuracy (sensitivity 40–93% and specificity 91–100%) against a reference of multiple tests. In part, these findings reflect the differing aims of the tests: tympanometry and otoscopy assess pathology (presence of middle ear effusion) and, unlike PTA, are not tests of hearing sensitivity. Spoken word tests Five studies reported four comparisons of spoken word tests, VASC or SVEP (a test using speech signals), compared with PTA. Sensitivity (51–100%) and specificity (93–96.8%) were moderate to good. Otoscopy Two studies compared otoscopy with PTA. Both sensitivity (23–89%) and specificity values (60–93%) were highly variable. Audiometry Five studies reported comparisons of the pure tone sweep test with PTA. The sensitivity (86–100%) and specificity (65–100%) values were generally high. Indeed, the study by Orlando and Frank73 showed that these high values were consistent across 6-month age groupings between 4 and 6 years. TEOAE Sabo and colleagues54 reported the sensitivity (63%) and specificity (91%) of TEOAE compared with PTA in a smaller study with just 66 children, only 61 of whom completed. They reported

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TABLE 31 Test accuracy of impedance audiometry Source

Test evaluated

Definition of Reference hearing impairment standard

Definition of Sensitivity Specificity hearing impairment

Pang-Ching Tympanometry et al., 199552

Modified ASHA (1990) criteria

Pure tone Score 艌3 sweep test, tympanometry, pneumatic otoscopy, acoustic reflectometry

73%

85%

Olusanya, 200151

Tympanometry

Second test non-type A tympanogram

PTA and pure tone sweep test

>20 dB at 0.5, 1, 2 and 4 kHz

50%

83%

Lyons et al., 200453

Tympanometry

Non-type A tympanogram

Pure tone sweep test

>20 dB at 0.5, 1, 2 and 4 kHz

85%

91%

71%

65%

40–90%a

62–83%a

McCurdy Tympanometry et al., 197671 and stapedius reflex

Type B or C PTA tympanogram or Type A tympanogram and no stapedius reflex

Clark (7) criteria

Nienhuys, Tympanometry et al., 199461

Normal +100 to PTA –99 daPa and 0.3–6 ml

>25 dB at 0.5–4 kHz

Rousch et al., Tympanometry 199259

Traditional ASHA (1990) criteria

Otoscopy

Medical attention required

27%

99%

Rousch Tympanometry et al., 199259

Modified ASHA (1990) criteria

Otoscopy

Medical attention required

64%

97%

FitzZaland and Zink, 198455

1= Type B or C with pressure 艋150 mmH2O 2= Type B or C with pressure 艋175 mmH2O 3= Type B or C with pressure 艋200 mmH2O 4 = Type B only

Combination of Various according history, pure to test tone and air and bone conducted thresholds, tympanometry, acoustic reflex and speech tests

1 = 93% 2 = 93% 3 = 91% 4 = 40%

1 = 91% 2 = 95% 3 = 99% 4 = 100%

Negative pressure of 艌–200 mm OR inability to show compliance at 20 dB at 0.5, 1, 2 and 4 kHz

87%

96%

Maragno and Teatini, 198366

Audiological assessment including PTA

100%

94%

Correctly identifying less than 50% of the test words

PTA

>35 dB HL

83.3%

96.8%

SVEP test

Prescott Voice test et al., 199974

SVEP, Sweep Visual Evoked Potential.

TABLE 33 Test accuracy of otoscopy Source

Test evaluated

Definition of Reference hearing impairment standard

Definition of Sensitivity Specificity hearing impairment

Olusanya, 200151

Otoscopy

Pure tone sweep test and PTA

>20 dB at 0.5, 1, 2, 4 kHz

Wax discharge or lack of tympanic membrane activity

PTA

Clark (7) criteria 0.5–4 kHz

Nienhuys Otoscopy et al., 199461 a

56%

62.4%

23–89%a

60–93%a

Dependent on fail criteria used.

sensitivity (91%) and specificity (99.67%). The study by Square and colleagues60 compared bone conduction tests combined with PTA to impedance screening and found poor sensitivity (26%) and specificity (6.6%). Finally, Pang-Ching and colleagues52 found that reflectometry had poor sensitivity (23%) and specificity (56%) compared with otoscopy results.

Influence of testing environment Only five of the included studies reported using a soundproofed environment in which to carry out the test.52,57,61,66,73 All other studies either reported a non-soundproofed environment or failed to report where the test was conducted. There appeared to be no consistent differences in the sensitivities and specificities of those studies

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TABLE 34 Test accuracy of sweep audiometry Source

Test evaluated

Definition of hearing impairment

Reference standard

Definition of hearing impairment

Sensitivity

Specificity

Sabo et al., 200054

Pure tone sweep test

>25 dB at 0.5 kHz and >20 dB at 1, 2 and 4 kHz

PTA

87%

80%

>25 dB at 0.5, 1, 2 and 4 kHz

PTA

>30 dB

4–4.5 years: 90% 4.5–5 years: 82% 5–5.5 years: 100% 5.5–6 years; 88%

4–4.5 years: 71% 4.5–5 years: 65% 5–5.5 years: 90% 5.6–6 years: 84%

Orlando and Pure tone >25 dB at 0.5, 1, 2 and 4 kHz Frank, 198773 sweep test (audiometer)

PTA

>30 dB

4–4.5 years: 91% 4.5–5 years: 91% 5–5.5 years: 100% 5.5–6 years: 100%

4–4.5 years: 98% 4.5–5 years: 98% 5–5.5 years: 98% 5.5–6 years: 97%

FitzZaland and Zink, 198455

>25 dB at 0.5 and 4 kHz and >20 dB 1 and 2 kHz

Combination Various, of history, dependent pure tone on test and air and bone conducted thresholds, tympanometry, acoustic reflex and speech tests

93%

99%

No response at 20 dB in either ear at any frequency

Examination NR including PTA, tympanometry and ear examination

86%

70.2%

Orlando and Pure tone Frank, 198773 sweep test (audioscope)

Pure tone sweep test

Holtby et al., Pure tone sweep test 199770

TABLE 35 Test accuracy of TEOAE Source

Test evaluated

Definition of Reference hearing impairment standard

Definition of Sensitivity Specificity hearing impairment

Sabo et al., 200054

TEAOE

Response at three PTA frequencies of ⭓3 above the noise floor with a minimum 70% reproducibility at each frequency and a 90% or greater stability

63%

91%

Various

67–100%

80–98%

Nozza et al., TEAOE 199772

PTA

that reported using a controlled test environment and those that did not.

poor to moderate methodological quality (i.e. QUADAS score 20 dB at 0.5, 1, 2 and 4 kHz

97%

86%

Lyons et al., 200453

Protocol 2 DPOAE and tympanometry

DPOAE SNR 艌5 dB Pure tone at 1.9 kHz and sweep test tympanometry results normal

>20 dB at 0.5, 1, 2 and 4 kHz

97%

83%

Lyons et al., 200453

Protocol 3 DPOAE and tympanometry

DPOAE SNR 艌11 dB Pure tone at 1.9 kHz and sweep test tympanometry results normal

>20 dB at 0.5, 1, 2 and 4 kHz

98%

74%

Lyons et al., 200453

Protocol 4 DPOAE and tympanometry

DPOAE SNR 艌5 dB at 1.9 kHz and SNR 艌11 dB at 3.8 kHz and tympanometry results normal

>20 dB at 0.5, 1, 2 and 4 kHz

96%

95%

Pure tone sweep test

SNR, signal to noise ratio.

TABLE 37 Test accuracy of other tests

Source

Test evaluated

Definition of Reference hearing impairment standard

Definition of Sensitivity Specificity hearing impairment

FitzZaland and Zink, 198455

Audiometric Rhinne test

Reporting having heard the tone more loudly at the mastoid location

Audiological assessment

Various, dependent on test

91%

99.67%

Square et al., Bone No response at conduction and + 10 dB and –10 dB 198560 PTA OR no response at = 10 dB

Impedance screening (tympanometry and reflex)

Negative peak pressure at >–150 mm/H2O air pressure or having no pressure peak at all

26%

6.6%

Pang-Ching Acoustic et al., 199552 reflectometry

Tympanometry Score 艌3 and pneumatic otoscopy

23%

56%

Scores of 6–9

was PTA was plotted in the ROC space (Figure 7). Different types of test are given different symbol shading. The diameter of the symbol reflects the sample size of the study, with larger studies having a larger symbol. ‘Good’ tests with both high sensitivity and specificity (e.g. tympanometry and pure tone sweep test) tend to occupy the top left of the plot. In contrast, poorer tests with lower sensitivity and specificity (i.e. parental questionnaires) tend to occupy the bottom right of the plot. It is important to recognise there are at least two caveats in interpreting this figure. First, either or both of two different conditions (middle ear pathology, hearing impairment) are being sought by different studies. Secondly, both the

referral criteria for the screening test and the criterion definition of hearing impairment for the reference test varied across studies. Screen performance The uptake of the screening test was reported by 13 studies (Table 38) and across a variety of screening tests. Regardless of the test applied the uptake rates appeared to be high, ranging from 87.5 to 100%. These high values reflect the fact that the studies are experimental test accuracy assessments rather than ‘real world’ and community-based screening evaluations. None of the studies looked at reported their yield of true cases.

Health Technology Assessment 2007; Vol. 11: No. 32

Orlando73 PTST FitzZaland55 PTST meter FitzZaland55 TY 1.0 Maragno66 SVEP Prescott74 VT 0.8

Orlando73 PTST scope Ritchie57 VASC1

Ritchie57 VASC2 Holtby70 PTST Holtby70 Ty + St Ref

Sabo54 PTST

Nienhuys61 Ty

Sabo54 TEAOE

McCurdy71 Ty + St Ref

Sensitivity

0.6 Hammond63 PQ Olusanya51 Ty

Olusanya51 OTOSC Nenhuys61 OTOSC

0.4

0.2 Olusanya51 PQ 0 0

0.2

0.4

0.6

0.8

1.0

1 – Specificity Parental questionnaires

TEAOE

Impedance

Sweep audiometry

Otoscopy

Spoken word tests

FIGURE 7 Summary ROC (should be interpreted with the results in Tables 30–37)

TABLE 38 Uptake for each study Source Ritchie and Merklein, 197257 FitzZaland and Zink, 198455 Holtby et al., 199770 Lyons et al., 200453 Nienhuys et al., 199461 Olusanya, 200151 Pang-Ching et al., 199552 Prescott et al., 199974 Rodriguez and Melguizo-Yepez, 199456 Rousch et al., 199259 Rousch and Tait, 198562 Sabo et al., 200054 Square et al., 198560

Uptake (%) 100 100 91 100 71 88 100 94 87.5 100 100 99 100

Screen effectiveness It was not possible to assess the potential effectiveness of interventions for children identified by the SES since only the study by Feldman and colleagues46 reported outcomes related to screening test effectiveness. This retrospective cohort study compared two groups of 730 children from different geographical areas in Ontario, Canada. One group received hearing screening (VASC by a public health nurse) before school entry while the other group did not. Hearing impairment was assessed (PTA at 0.5–4 kHz) in both groups at 6–12 months after the hearing screening. As the study found no statistically significant difference in the prevalence of hearing impairment in the groups after 6–12 © Queen’s Printer and Controller of HMSO 2007. All rights reserved.

Systematic review of the effectiveness of school entry hearing screening

months (unscreened group 16.8% versus screened group 14.1%) the authors concluded that preschool hearing screening was ineffective in the sense of leading to interventions which resolved the (presumably temporary) hearing impairments.

●

However, there are some problems with the design of this study that limit the strength of its conclusions. First, it is likely to have been underpowered to detect the small improvement in prevalence of hearing deficit seen in the screened group. Secondly, the observational nature of the study made it open to a number of potential biases. Although the authors attempted to match the children by selecting two geographical areas with similar socio-economic class, there remains considerable potential for differences in the baseline characteristics of the children, so-called selection bias. The baseline characteristics of the children in the two groups were not reported. The assessors conducting the hearing test at 6–12 months may not have been blinded to the screening status of child. In addition to a lack of effectiveness of the hearing screen, there are other potential explanations for the similar level of prevalence of hearing impairment in the two groups (e.g. non-compliance with treatment; ineffective treatment or wrong timescale). Adverse effects None of the included studies reported any adverse effects of screening.

Summary of findings ●

●

There is only level III evidence for the effectiveness of preschool hearing screening, from a single, poor-quality, observational comparative study. Furthermore, this single study was inconclusive in whether preschool screening was more effective than no screening in detecting hearing impairment. No studies were identified that have assessed the long-term impact of preschool hearing screening on outcomes including educational, language and social outcomes, or on the

●

effectiveness of interventions for children identified with hearing impairment via the SES. Several studies with an unacceptable variability in their quality have assessed the accuracy of different hearing screening tests in preschool children. Given the unacceptable variability in methodological quality and reporting of these studies, lack of clarity in the cases of hearing impairment detected (e.g. transient versus permanent hearing impairment), variation in reference test and threshold level for hearing deficit, and range of control over the settings in which these tests were applied, it is difficult to interpret and compare their results. Accepting these caveats and selecting the subset of studies using PTA as the reference test, the findings suggest that: – Studies comparing various screen protocols of pure tone sweep audiometry report high sensitivity and specificity for full PTA and therefore appear to be suitable tests for screening. – Spoken word tests are reported to be a viable option because of their potential acceptable levels of specificity and sensitivity. – Depending on referral criteria, TEOAEs have potentially high specificity, but somewhat lower sensitivity. – Tympanometry and acoustic reflectometry have variable sensitivity and specificity. – Parental questionnaire and otoscopy have poor sensitivity and specificity. Therefore, these tests are likely to be less suitable for screening. – There is insufficient evidence to comment on the accuracy of combinations of tests. A small number of studies indicated a generally high uptake in this age group. However, given the experimental design of the studies, and that they were assessing test accuracy rather than programme accuracy, these findings cannot necessarily be generalised to the uptake of the screen in real-world community screening settings. No studies were found that assessed the potential adverse effects or yield of hearing screening for preschool children.

Health Technology Assessment 2007; Vol. 11: No. 32

Chapter 5 Cost-effectiveness and cost-effectiveness acceptability of the school entry hearing screen Methods Aims and objectives The overall aim of this part of the study was to estimate the cost-effectiveness and cost acceptability of alternative strategies for SES. To achieve this aim the principal objectives were:

accuracy of individual types of test. The key alternatives compared to SES-C were: ●

●

● ●

●

● ●

to conduct a systematic review of the economics literature to estimate the health-related quality of life and utilities associated with the SES programme to estimate the relative costs of the SES to relate the costs and health-related quality of life and utilities and compare alternative models of SES.

Overall approach The analysis assessed the cost-effectiveness and cost acceptability of alternative models of SES. It was designed to investigate the extent to which the differences in the cost-effectiveness strategies for hearing screening result in differences in costs, resource use, health status, and hearing-related disability and quality of life. The perspectives of the NHS and education services, patients and family were used to approximate a societal perspective. To address the research questions a decisionanalytic model was developed to synthesise clinical and economic data from a number of sources. The model was used to estimate the relative costeffectiveness of alternative SES programmes and no SES. As recommended by guidelines for economic evaluation,77–79 the alternatives for comparison were chosen to reflect the range of SES programmes reported in the survey of UK current practice (Chapter 2). A composite SES programme (SES-C) was defined that included a combination of the categories or types of SES tests reported in the survey of current practice. To define the composite, the probability of each type of test being used was estimated. Using the data from the survey of current practice, SES-C was defined as pure tone sweep audiometry (99%) and tympanometry (1%). This was used to weight the probability and cost data relating to the costs and © Queen’s Printer and Controller of HMSO 2007. All rights reserved.

●

● ●

universal SES, using pure tone sweep audiometry only (SES-PTS) universal SES, using parental questionnaire only (SES-PQ) universal SES, using tympanometry only (SES-T) universal SES, using spoken word tests only (SES-SW) no SES targeted SES.

The decision model includes events relevant to the effectiveness, subsequent diagnosis and treatment/management, patient outcomes, resource use and costs of the screening packages. The outcomes assessed with the model were cost per quality-adjusted life-year (QALY) gained, and cost per year gained with no, minimal or mild hearing impairment (YNHI). The cost per QALY gained was the primary outcome measure. QALYs weight life-years by the utility or value attached to health states and improvements in health and are recommended in cases where the health outcome of interest is change in morbidity rather than simply survival.77–79 If the data on utility required to estimate QALYs are derived from inappropriate instruments or low-quality evaluations, then the estimates of QALYs may be inaccurate, which would bias the results of the analysis. An alternative measure is years with no disability due to hearing impairment. However, there were insufficient data to extrapolate from years with different levels of hearing impairment to estimate years with different levels of disability. Therefore, the cost per year with no to mild hearing impairment (YNHI) was included as an alternative measure, to assess whether the results of the economic model would differ substantially according to the method used to evaluate and value hearing levels. The outcome of cost per year with no, minimal or mild hearing impairment is a potentially less sensitive measure of health associated with hearing impairment. It assumes that minimal and mild hearing impairment have no impact on the overall utility or value of a year

Cost-effectiveness and cost-effectiveness acceptability of the school entry hearing screen

with these levels of hearing impairment compared with years of life with no hearing impairment. In addition, any benefit accruing to years of life with more severe hearing impairment are not included in the estimate of outcome; in other words, the value of years of life with moderate to severe hearing impairment is set to zero. If different methods of SES affect the distribution of children between different levels of hearing impairment, and each level of hearing is associated with a different utility or value, then use of the YNHI will results in misestimation of the benefit of alternative SES programmes. The cost per true positive case of hearing impairment detected is an alternative outcome measure that is less sensitive than the cost per YNHI. It was decided to include this in the sensitivity analysis only if the conclusions of the economic model did not differ substantially between cost per QALY gained and cost per YNHI gained. The time-horizon used for the primary analysis was from the day of screening up to 1 year. Secondary analyses explored longer time-horizons of 6 years (from the day of screening to secondary school age) and 11 years (from the day of screening to 16 years of age). As outlined in Chapter 1, children in the UK enter school at around 5 years of age and are usually screened in their first (reception) year at school. Differences between education authorities mean that they enter school any time from their fourth birthday up to their fifth birthday. So, the most appropriate population for the analysis is children 4–6 years of age; that is, 4 years and 1 day to 5 years and 364 days (i.e. the time at which school entry screening could take place in the UK). The selected age range also reflects an assumption that identification and management of previously undetected hearing impairment relatively early in a child’s school life are beneficial. Data for the model were derived from the survey of current SES practice reported in Chapter 2, the systematic review of test accuracy and effectiveness reported in Chapter 4, and two additional reviews of the economics literature and national databases and statistical sources for the UK.

Reviews of economic literature and databases

A systematic review of economic evaluations of screening for hearing impairment in children aged 4–6 years (the age range of interest for this economic evaluation) was conducted. The objectives of the review were:

●

to assess the costs, effectiveness and costeffectiveness of school entry screening to identify decision models reported in the literature to identify economic data for the model used in this study.

The review used a focused systematic search of studies and databases that report resource use, quality of life data, costs or patient outcomes associated with screening for hearing impairment. A second review of literature that reported resource use, costs or outcomes of management interventions for hearing impairment was also conducted. The objective of this review was to identify economic data that could be used to populate the economic model. The search strategies for both reviews were implemented in the following electronic databases: ● ● ●

● ●

●

MEDLINE (1966 to 2005 week 3) EMBASE (1980 to 2005 week 31) Cumulative Index to Nursing & Allied Health Literature (CINAHL) (1982 to August 2005 week 5) Econlit (1969–2002 and 2003–2005) Cochrane Library (Wiley) NHS Economic Evaluation Database (NHS EED) (2005 Issue 2) Office of Health Economics Database (OHE HEED) (July 2005 issue).

The searches were limited to electronic databases. The detailed search strategy for the review of economic evaluations of screening is reproduced in Appendix 11. Appendix 12 details the search strategy for the management of hearing impairment review. The search strategies were developed by the project team and an information specialist with extensive experience in literature searching. The search strategy was modified and optimised for each electronic database. The economic terms used on MEDLINE, CINAHL and EMBASE were adapted from the York Centre for Reviews and Dissemination (CRD)78 filter. The clinical terms for the screening search strategy were based on filters developed at the University of Birmingham for the review of effectiveness of SES (Chapter 4). The NHS National Electronic Library for Health (http://libraries.nelh.nhs.uk), the PRODIGY guideline website (www.prodigy.nhs.uk) and the Scottish Intercollegiate Guideline Network website (www.sign.ac.uk) were used to identify relevant interventions and search terms for the management of hearing impairment review. Only

Health Technology Assessment 2007; Vol. 11: No. 32

interventions with a clinical benefit recommended in these guidelines were included in the search strategy for management of hearing impairment.

screening programmes and subsequent management interventions. 6. Resource use and cost were reported separately.

The syntax of the search strategies was mapped accordingly, to translate directly the thesaurus of MEDLINE, EMBASE and CINAHL. The search strategy imposed no language, date or other similar limitations.

The full paper was included in the review of the cost-effectiveness of screening only if it met criteria 1, 2 and 3 and at least one of 4, 5 and 6. To identify decision models and data for the economic model, studies were included if they met criterion 1 and one or more of criteria 2–6.

A screening form for inclusion/exclusion was used to screen titles and abstracts and exclude any studies that did not report resource use, utility values or costs related to SES or management of hearing impairment. Articles were only rejected on initial screen if the reviewer could determine from the title and abstract that the article did not meet the prespecified inclusion/exclusion criteria. If a title/abstract could not have been rejected with certainty, the full text of the article was obtained for further evaluation to assess whether they met the inclusion/exclusion criteria. One reviewer (GV) then screened all of the retrieved papers. The following inclusion and exclusion criteria were applied for the review of economic evaluations of screening studies (Appendix 13): 1. The studies were based on primary data or used data from systematic literature reviews, reported detailed data on costs and outcomes for extraction and use in the economic model, were conducted in a range of settings (e.g. education services, primary/secondary/tertiary healthcare, other local community services, or the family home) and were generalisable to the UK setting. 2. The paper reported data relevant to the population of interest (children between 4 and 6 years of age). 3. The evaluations compared at least two of the following interventions: PTA, tympanometry, acoustic reflex, otoadmittance tests, ABR, medical examinations (which entail a hearing screening), distraction tests, behavioural tests, speech perception tests, questionnaires, otoacoustic emissions and no screen. 4. The paper reported at least one of the following outcomes: year with no or mild/moderate disability due to hearing impairment, year with moderate or severe disability due to hearing impairment, QALYs gained, utility measure and health status measure. 5. The paper reported at least one of the following types of economic data: resource use, costs or utilities associated with hearing © Queen’s Printer and Controller of HMSO 2007. All rights reserved.

For the review of management strategies for hearing impairment the following inclusion and exclusion criteria were applied (Appendix 14): 1. The studies were based on primary data or used data from systematic literature reviews, reported detailed data on costs and outcomes for extraction and use in the economic model, were conducted in a range of settings (e.g. education services, primary/secondary/tertiary healthcare, other local community services, or the family home, and were generalisable to the UK setting. 2. The paper reported data relevant to children with identified hearing impairment aged from birth to 12 years, undergoing any of the following interventions: hearing aids, autoinflation, middle ear ventilation, myringotomy/grommets, adenoidectomy, speech and language therapy, hearing tactics (family, community, school), referral to specialists or cochlear implantation (only for comparative purposes). 3. Studies assessed one of the following outcomes: year with no or mild/moderate disability due to hearing impairment, year with moderate or severe disability due to hearing impairment, QALYs gained, utility or health status. 4. Studies reported resource use, costs or utilities associated with subsequent management interventions. 5. Studies reported resource use and cost separately. To identify data on the costs or outcomes of management strategies for hearing impairment to populate the economic model, studies were included if they met criterion 1 and one or more of criteria 2–5. A second reviewer (LD) independently screened any papers where the first reviewer (GV) was unclear of inclusion. Any uncertainties in the reviewers’ assessment of the studies were resolved by discussion and, when necessary, in consultation with the rest of the project team. Reviewers were

Cost-effectiveness and cost-effectiveness acceptability of the school entry hearing screen

not masked to the source and authors of the studies. Data were extracted to populate the economic model using a data abstraction form. The form was based on the criteria to assess abstracts for the NHS EED database (Appendix 15). Data were extracted from included studies on: participants – study population; study design including type of intervention and perspective; screening procedure including comparator interventions and setting of the screen; resource use including choice and combination of screening tools; costs (direct and indirect where reported) and outcomes, including information of synthesis of costs and benefits, data on quality of life, dates to which data relate, discounting rates and side-effects; statistical/ sensitivity analyses; and study findings. All economics papers included in the analysis were quality assessed. The purpose of this assessment was to examine whether the methodology was appropriate and the results were valid and generalisable to other settings (with focus on the NHS). Quality assessment was based on the critical appraisal criteria cited in the CRD Report Number 6.78 Quality assessment questions were included in the data extraction form (see Appendix 15) and the papers were scanned accordingly.

Economic model The decision-analytic model was developed and validated by discussion with experts in the provision of hearing and school entry screening

services. TreeAge Pro 2005 software was used. A static decision tree model was used. The timehorizon of the model is finite and limited for both the primary and secondary analyses. A static decision-tree model works well in analysing chance events with limited recurrence or change over time (such as the probability that a person has hearing impairment or not). This is the case for most events associated with SES. The main exception is the occurrence of transient hearing impairment due to OME. OME can resolve and recur more than once in a 12-month time-frame. However, the consequences of an initial episode of OME detected by the SES (including subsequent recurrences) are not likely to last for more than 12 months. In addition, OME is not likely to result in long-term hearing impairment and only minimal or mild hearing impairment within an episode of OME. Therefore, it was decided to estimate the mean number of recurrences of transient hearing impairment and use these to model the impact on costs and outcomes of transient hearing impairment (and associated recurrences) at the end of the 1-year time-horizon (6- and 11-year time-frames for the secondary analyses). The first split in a decision-tree model is a ‘decision node’ (sometimes called choice node) and is represented by a square box. Decision nodes reflect a choice to be made between alternatives. Later splits occur at ‘chance nodes’, which are represented by circles. Chance nodes occur when there is a number of subsequent events that could happen. Each event is assigned a

No SES SES-PTS SES-T

2nd test within screen

SES-SW School entry screen (SES)

Fails 1st screening test

No 2nd test

Passes 1st screening test

Referred for diagnostic evaluation

Child screened

SES-PQ SES-C

Identified, diagnostic evaluation Has hearing impairment

1 Child not screened

Not identified No hearing impairment 2

Targeted SES

Child identified at risk, eligible for SES Child not identified at risk, not screened

FIGURE 8 Decision tree, choice of screening method

Child screened Child not screened

Health Technology Assessment 2007; Vol. 11: No. 32

Referred for diagnostic evaluation Fails 2nd screening test

3rd test within screen No 3rd test

Child tested

True –ves Passes 2nd screening test

Identified, diagnostic evaluation False –ves

2nd test within screen

Not identified Identified, diagnostic evaluation Has hearing impairment Child not tested

Not identified No hearing impairment

FIGURE 9 Decision subtree for third test within a screen

Diagnostic evaluation Referred for diagnostic evaluation

Has hearing impairment No hearing impairment

Transient hearing impairment Permanent hearing impairment

Non-surgical intervention Surgical intervention

Has hearing impairment No diagnostic evaluation No hearing impairment

FIGURE 10 Decision subtree for children referred for diagnostic evaluation

Minimal (0), a lower ICER is preferred to a higher one. For negative ICERs there is no clear decision rule. A negative ICER may occur when one intervention is both more costly and less effective than another and is not cost-effective, or when an intervention is less costly and more effective than another and is clearly cost-effective. No predefined target ceiling ratio (i.e. the maximum a decision-maker is willing to pay for a unit of effect) for cost-effectiveness was chosen. This was because there is no evidence on what a single target ceiling ratio should be. A range of ceiling ratios was used, from decisionmakers being willing to pay £0 to gain 1 QALY to

Cost-effectiveness and cost-effectiveness acceptability of the school entry hearing screen

Potentially relevant publications identified for retrieval 169

Papers excluded on the basis of abstract and title 113

Full papers obtained for more detailed evaluation 56

Total numbers of papers for data extraction 2

FIGURE 13 Flow diagram of included studies for the cost-effectiveness of screening

decision-makers being willing to pay £30,000 to gain 1 QALY. The ICER was the primary outcome measure used to compare each alternative with the composite universal SES programme (SES-C). PSA was used to generate mean expected costs and outcomes and statistical measures of expected variance around the likely estimate of each variable in the model and its distribution. Each variable was assigned a base-case or average value and a distribution of possible values. The probabilistic analysis sums the results of multiple analyses (iterations). Each iteration samples values for the variables at random from the specified distributions. The sampling method used was Monte Carlo, expected value. The simulation software was TreeAge Pro 2005 plus Healthcare module.

Cost-effectiveness acceptability curves (CEACs) were plotted and used as a method of summarising the uncertainty around the generated cost-effectiveness ratios. CEACs plot the probability that an intervention is cost-effective against the value of a ceiling ratio (i.e. the maximum a decision-maker is willing to pay for a unit of effect).

The CEAC estimates the probability that SES-C is cost-effective. This is done by first bootstrapping the estimates of cost per QALY (ICER) from the PSA. The proportion of bootstrapped estimates where the cost per QALY is lower than the ceiling ratio is calculated out of the total number of bootstrapped estimates of the ICER. This is repeated for each of the ceiling ratios (in this case the ceiling ratios were £0 per QALY gained to £30,000 per QALY gained, in increments of £1000). The probability that SES-C is cost-effective is then estimated as the proportion of bootstrapped estimates of the ICER that are lower than each ceiling ratio. These estimates are plotted graphically, against each of the ceiling ratios, to derive a CEAC. Net benefit statistics were estimated by revaluing the bootstrapped estimates of QALYs, using the ceiling ratios or willingness to pay (WTP) to gain one unit of outcome used for the CEAC analysis (i.e. £0 per QALY gained to £30,000 per QALY gained, in increments of £1000). For each WTP threshold, the net benefit (NB) is estimated as NB = E * WTP – C

Health Technology Assessment 2007; Vol. 11: No. 32

where E is the incremental QALY gained by an intervention, WTP is willingness to pay to gain 1 QALY, and C is the incremental cost of the intervention. The CEAC summarises the information at each value of WTP to gain a QALY. The net benefit statistic gives an estimate of the monetary value of a QALY or other measure of effectiveness. Sensitivity analysis was used to explore the impact of structural uncertainty by estimating costs, effects, ICERs and CEACs for each of the alternative outcomes estimated in the analysis, for the impact of universal newborn hearing screen on the numbers of children with unidentified hearing impairment at school entry, and for temporary versus permanent hearing impairment.

Results Systematic review of the costeffectiveness of screening and of management strategies The search strategy for the cost-effectiveness of screening identified 169 articles. Of these, 113 were excluded. The full texts of 56 articles were obtained and reviewed. Data from two papers were extracted for inclusion in the economic model (Figure 13). Neither paper reported a full economic evaluation (Appendix 16).

school TEOAE programme were marginally greater than for the school pure tone programme. The cost per hearing-impaired child with sensorineural or mixed impairment of at least a moderate degree was substantially greater than the cost per hearing-impaired child identified with any hearing-impairment, across all programmes. The ultimate yield of hearing-impaired cases was not affected by the age at screening. The difference between the costs of children with hearing impairment for all programmes is related to the low yield of cases diagnosed with sensorineural/mixed hearing impairment and the higher yield of subjects with conductive, and possibly transient pathologies. The costs for the infant programme were substantially less than most reports in the literature of TEOAE-based universal neonatal screening programmes. With regard to the first school programme, the use of TEOAE resulted in a marginally higher cost per child and cost per child with hearing impairment. Higher total programme costs were incurred in the first school programme as opposed to the second one owing, in part, to difference in the cost, maintenance and efficiency of the screening equipment. No indirect costs were included, but this omission is unlikely to affect the model’s results. Costs and quantities were reported separately; however, no sensitivity analysis or any other statistical analysis was performed to evaluate the uncertainty around costs of quantities. The study did not discount costs owing to the short time-frame of the analysis. The price year was not reported.

One paper reported a cost study88 to compare the costs associated with a TEOAE infant screening programme, a TEOAE school entry programme and a pure tone screening school-entry programme. There was no justification for the selection of the comparator programmes; however, pure tone screening is used in routine practice in the UK and so makes the comparison relevant to this review. The perspective was not clearly stated, making it difficult to judge whether all relevant costs had been assessed. The data for the screening programmes were obtained from observational studies in Brisbane. A total of 1305 infants entered the infant screening programme. The age of this population was 2 months. The school screening programme sample for TEOAE was 940 children, with a mean age of 6.2 years, recruited from 22 primary schools throughout Brisbane. No selection criteria were reported. The same schoolchildren were tested with PTA.

The second paper reported an effectiveness study of hearing screening89 that included some cost information. The aims were to evaluate impedance measurements against PTA as a screening method for the detection of middle ear changes associated with hearing impairment in infant school children. Justification of the choice of PTA as the comparator programme was that it represents usual practice. The study used an NHS perspective. Indirect costs were not included in the analysis. The cost data were collected on the sample of children observed in the study. The study did not discount costs owing to the short time-frame of the analysis. No sensitivity analysis or any other statistical analysis was used to evaluate the uncertainty around costs of quantities. The price year of the resources was 1998. The study did not formally relate costs to outcomes.

The results indicated that the costs for TEOAE infant screening were higher than either school screening programme. Moreover, costs for the

The electronic and bibliographic searches of papers reporting resource use, costs or outcomes of management interventions identified 960

Cost-effectiveness and cost-effectiveness acceptability of the school entry hearing screen

TABLE 39 Probability of events related to screening and diagnosis Item/event

Mean

Range

Child is screened Fails first screening test Referred for diagnostic evaluation following first screen test Child attends diagnostic evaluation Hearing impairment (true positives) No hearing impairment (true negatives) Referred for second test within screen Misses second test within screen Fails second screening test Referred for diagnostic evaluation following second screen test Third test within screen Referred for diagnostic evaluation following third screen test

0.92 0.08a 0.37b 0.80c 0.53a 0.99a 0.42b 0.08b 0.50a 0.50b 0.10b 0.85b

0.56–1.00 0.05–0.26a 0.27–0.47b 0.60–1.00c 0.23–0.83a 0.98–1.00a 0.32–0.52b 0.00–0.44b 0.22–0.78a 0.17–0.83b 0.00–0.20b 0.70–1.00b

Systematic review of effectiveness, Chapter 4 of this report.54,55 Survey of current practice, Chapter 2 of this report. c Waltham Forest study, Chapter 3 of this report. b

potentially relevant titles and abstracts (Appendix 11). Of these, 824 were excluded on the basis of title and abstract and 136 full-text papers/abstracts were collected for more detailed evaluation. Of these 136 papers, no studies could be used to populate the model. Thirty-four were excluded because they did not report detailed data on costs and outcomes for extraction and use in the economic model, 33 studies were not generalisable to the UK setting, 19 papers evaluated the wrong intervention, 16 looked at a different population cohort, 12 papers assessed a different outcome measure, seven analyses did not report resource use and costs separately, five studies reported utilities not associated with management interventions, three were not based on primary data collection or systematic reviews, and seven were excluded for other reasons.

Data used as inputs to the economic model The probability of using PTA or pure tone sweep audiometry in SES programmes in the UK was estimated as 0.99 (survey of current practice, Chapter 2) for the first and second tests within a screen. The remaining services used tympanometry. The probability of using PTA or pure tone sweep audiometry for a third test within a screen was reduced to 0.95 and the probability of tympanometry increased to 0.05 (survey of current practice, Chapter 2).

Table 39 gives the probabilities of events related to screening for and diagnosis of hearing impairment. These data were derived from the survey of current practice, the Waltham Forest

study and the systematic review of effectiveness reported in Chapters 2, 3 and 4, respectively. Table 40 shows the probability of having hearing impairment and the distribution of hearing impairment between transient and permanent and by severity of hearing impairment. To estimate the probability of hearing impairment it was assumed that only children with bilateral hearing impairment, lasting for more than 1 month, were likely to have hearing impairment.90 The probability for transient hearing impairment or OME lasting for more than a month was calculated from published literature.91 The prevalence of permanent hearing impairment was estimated from published studies.12,17,30 Table 41 presents the probability of interventions for hearing impairment by different levels and types of hearing impairment. Children with transient hearing impairment will have either surgical or non-surgical intervention. Children with minimal, mild or moderate permanent unilateral hearing impairment will have either no intervention (or watchful waiting) or hearing aids. Children with severe or profound permanent hearing impairment will have either hearing aids or surgery (i.e. cochlear implant). Table 42 shows the staff and equipment used for screening and the unit costs estimated for the analysis. The acquisition costs of equipment were estimated as: screening audiometer = £950 plus VAT, screening tympanometer = £2147 plus VAT (£1995–2300) and otoscope = £80. The estimate of throughput (number of children screened) for

Health Technology Assessment 2007; Vol. 11: No. 32

TABLE 40 Probability and distribution of hearing impairment in the general population of children Item/event Prevalence of hearing impairment Transient hearing impairment Unilateral transient hearing impairment, given transient hearing impairment Permanent hearing impairment Permanent unilateral hearing impairment, given permanent hearing impairment Minimal unilateral hearing impairment (71 dB) Permanent bilateral hearing impairment, given permanent hearing impairment Minimal permanent bilateral hearing impairment (105 dB)

0.85a 0.677b 0.616b 0.497b 0.353b

0.75–1.00a 0.652–0.702b 0.469–0.634b 0.469–0.525b 0.327–0.379b

a b

Expert opinion and Dolan et al. (1995).85 Barton et al. (2004).86

additional costs for children with moderate hearing impairment were estimated to be £0 (range £0–7280) for social care and £6747 (range £0–8460) for education. The additional costs for children with severe hearing impairment were estimated to be £7280 (range £0–7280) for social care and £6747 (range £0–8460) for education. The additional costs for children with profound hearing impairment were estimated to be £7280 (range £7280–31,500) for social care and £18460 (range £6747–18,460) for education. Table 45 shows the estimated utilities for different levels of hearing impairment. The utility for no hearing impairment was estimated as 0.99, which is the population norm for the general population under 25 years of age.85 The utility values for minimal and mild hearing impairment were estimated from expert opinion. The utility values for moderate to profound hearing impairment were estimated from one study.86 This study used the Health Utilities Index, a generic validated © Queen’s Printer and Controller of HMSO 2007. All rights reserved.

measure, and associated utility weights to evaluate health status.

Results of the economic model One-year time-horizon, alternative measures of effect Tables 46–48 and Figure 14 show the results of the primary analysis for the 1-year time-horizon. Table 46 indicates that universal SES using current practice (SES-C) costs a total of £10 per child, which is more than no SES (less than £1 per child) and is associated with higher QALYs (0.983) than no SES (0.979). The ICER for SES-C is calculated as the net cost of SES-C (£10 minus £0.22) divided by the net QALYs of SES-C (0.983 minus 0.979), which is £2445 per QALY gained. This is within the threshold WTP to gain 1 QALY suggested by NICE guidelines (£30,000 per QALY gained).96 The cost-effectiveness acceptability analysis suggests that SES-C was more cost-effective than no SES in more than 50% of simulations, if decision-makers are willing

Cost-effectiveness and cost-effectiveness acceptability of the school entry hearing screen

TABLE 46 Expected cost and QALYs of screening, 1 year Expected cost (£) Expected QALY SES-C No SES Mean SD 2.5% percentile 97.5% percentile

10 6 6 27

0.22 0.19 0.01 0.74

SES-C No SES 0.983 0.005 0.973 0.992

0.979 0.007 0.964 0.991

TABLE 47 Probability that SES-C is cost-effective, 1 year, QALYs WTP to gain 1 QALY (£)

Probability SES-C is cost-effective

1 2250 5250 30,000

0.00 0.05 0.70 0.90

to pay £2250 or more to gain 1 QALY (Table 47 and Figure 14). Given the data used, this indicates that there is a high probability that SES-C is likely to be more cost-effective than no SES. The data in Table 48 show the results of the net benefit analysis. The net benefit statistics were estimated by revaluing the bootstrapped estimates of QALYs, using different ceiling ratios or WTP to gain one QALY. For each WTP threshold, the net benefit is estimated as the incremental QALY gained by SES-C multiplied by the WTP value, minus the incremental cost of SES-C. If decisionmakers are prepared to pay less than £2250 to gain 1 QALY, then there is no net benefit associated with SES-C and no SES would be more cost-effective. If decision-makers are prepared to pay more than £2250 to gain 1 QALY, then SES-C is associated with an estimated net benefit of up to £112 per child (at a WTP threshold of

TABLE 48 Net benefit of SES-C, 1 year, QALYs WTP (£)

Net benefit of SES-C compared with no SES (£)

1 2250 5250 30,000

Mean

2.5% percentile

97.5% percentile

–10 0 12 112

6 12 22 110

–27 –32 –40 –118

–5 19 53 341

1.0

Proportion cost-effective

0.9 0.8 0.7 0.6 0.5 Universal SES

0.4 0.3 0.2 0.1 0 0

16 WTP (£,000)

FIGURE 14 CEAC of SES-C, 1 year, QALYs

Health Technology Assessment 2007; Vol. 11: No. 32

TABLE 49 Expected cost and YNHI of screening, 1 year

£30,000 per QALY). However, the range of estimates does cross zero (i.e. the estimate for the 2.5% percentile is negative). This indicates that there may be a high level of uncertainty in the data.

relevant measure of effectiveness. The costeffectiveness acceptability analysis and net benefit analysis suggest that SES-C is not likely to be cost-effective compared with no SES. However, this analysis gives equal weight to children with no hearing impairment, minimal hearing impairment and mild hearing impairment. If minimal and mild hearing impairment adversely affect health status and health-related utility, then using YNHI as a measure of effect will underestimate the benefit of SES-C. The cost per YNHI was included as an alternative measure to assess whether the results of the economic model would differ substantially according to the method used to evaluate and value hearing levels.

Tables 49 and 50 and Figure 15 show the results of the analysis for the 1-year time-horizon, using year with no to mild hearing impairment as the measure of effect (YNHI). These data indicate that universal SES using current practice costs more, and is associated with lower YNHIs than no SES. This suggests that no SES is more cost-effective than SES-C if years gained with no to mild hearing impairment is considered the most

The cost per true-positive case of hearing impairment detected is an alternative outcome measure that is less sensitive than the cost per YNHI. Use of this measure would give similar results to using the YNHI as an indicator of the impact of SES-C. That is, SES-C would not be cost-effective compared with no SES if the cost per true positive case were used as the outcome measure of interest.

Expected cost (£) Expected YNHI SES-C No SES Mean SD 2.5% percentile 97.5% percentile

10 6 6 27

0.22 0.19 0.01 0.74

SES-C No SES 0.999 0.001 0.997 1.000

1.000 0.000 1.000 1.000

TABLE 50 Net benefit of SES-C, 1 year, YNHI WTP (£)

Net benefit of SES-C compared with no SES (£)

1 30,000

Mean

2.5% percentile

97.5% percentile

–10 –28

5 26

–26 –113

–5 –10

1.0

Proportion cost-effective

0.9 0.8 0.7 0.6 0.5 0.4

Universal SES

0.3 0.2 0.1 0 0

Cost-effectiveness and cost-effectiveness acceptability of the school entry hearing screen

Six- and 11-year time-horizons, QALYs Tables 51 and 52 and Figure 16 show the results of the primary analysis for the 6-year time-horizon. The cost-effectiveness acceptability analysis suggests that SES-C was more cost-effective than no SES in more than 99% of simulations, if decision-makers are willing to pay £2000 or more to gain 1 QALY. Given the data used, this indicates that there is a high probability that SESC is likely to be more cost-effective than no SES. Tables 53 and 54 and Figure 17 show the results of the primary analysis for the 11-year time-horizon. The cost-effectiveness acceptability analysis

TABLE 51 Expected cost and QALYs of screening, 6 years Expected cost (£) Expected QALY SES-C No SES Mean SD 2.5% percentile 97.5% percentile

25 10 11 49

SES-C No SES

2 2 0.36 6

5.37 0.04 5.28 5.44

5.27 0.07 5.13 5.39

suggests that SES-C was more cost-effective than no SES in more than 99% of simulations, if decision-makers are willing to pay £2000 or more to gain 1 QALY. Given the data used, this indicates that there is a high probability that SES-C is likely to be more cost-effective than no SES.

Comparison of SES using less accurate screen tests, 1-year time-horizon, QALYs Tables 55 and 56 and Figure 18 show the results when less accurate screening tests (SES-T, SES-PQ and SES-SW) are compared with no SES for a 1 year time-horizon. The CEACs for SES-PQ and SES-SW suggest that they are less cost-effective than no screening. The CEAC for SES-T suggests that it is more cost-effective than no SES in 50–70% per cent of simulations, if decision-makers are willing to pay £5000 or more to gain 1 QALY. Table 57 shows the net benefit of SES-C compared with less accurate screening tests (the cost and QALY information is given in Tables 46 and 57). Figure 19 shows the CEAC when less accurate screening tests (SES-T, SES-PQ and SES-SW) are compared with SES-C for a 1-year time-horizon.

TABLE 52 Net benefit of SES-C, 6 years, QALYs WTP (£)

Net benefit of SES-C compared with no SES (£)

1 30,000

Mean

2.5% percentile

97.5% percentile

–23 2853

25 1376

–46 721

–9 6034

1.0

Proportion cost-effective

0.9 0.8 0.7 0.6 0.5 0.4

Universal SES

0.3 0.2 0.1 0 0

16 WTP (£,000)

FIGURE 16 CEAC of SES-C, 6 years, QALYs

Health Technology Assessment 2007; Vol. 11: No. 32

Comparison of SES-C with low-accuracy targeted SES, 1-year time-horizon, QALYs Tables 59 and 60 and Figure 21 show the results when targeted SES is compared with SES-C for a 1-year time-horizon. The targeted SES assumes that only children identified as being at risk of hearing impairment are screened (10%). For this analysis, the probability that children are accurately identified was set equal to the probability that parental questionnaires are an accurate screen, which was relatively low. The costeffectiveness acceptability analysis suggests that in this case SES-C was more cost-effective than targeted SES in around 75–90% of simulations, if decision-makers are willing to pay £5000 or more to gain 1 QALY. Given the data used, this indicates that there is a high probability that SES-C is likely to be more cost-effective than targeted SES, if the accuracy of identifying children at risk is low.

The CEACs for SES-T, SES-PQ and SES-SW suggest that they are less cost-effective than SES-C.

Comparison of SES-C with no SES using more accurate screen tests, 1-year time-horizon, QALYs SES using pure tone sweep only (SES-PTS) was more accurate than SES-C. The expected costs of SES-PTS were similar to those of SES-C (£10.39 SES-PTS and £9.90 SES-C) and the expected QALYs were the same at 0.983. Table 58 shows the net benefit of SES-PTS compared with SES-C. The CEAC in Figure 20 suggests that SES-C is costeffective in 60% of simulations compared with SES-PTS.

TABLE 53 Expected cost and QALYs of screening, 11 years Expected cost (£) Expected QALY SES-C No SES Mean SD 2.5% percentile 97.5% percentile

30 13 12 64

SES-C No SES

3 2 0.45 9

9.07 0.07 8.93 9.19

Comparison of SES-C with high-accuracy targeted SES, 1-year time-horizon, QALYs Table 61 and Figure 22 show the results when SESC is compared with targeted SES for a 1-year timehorizon. The targeted SES assumes that only children identified as being at risk of hearing

8.91 0.11 8.68 9.10

TABLE 54 Net benefit of SES-C, 11 years, QALYs WTP (£)

Net benefit of SES-C compared with no SES (£)

1 30,000

Mean

2.5% percentile

97.5% percentile

–27 4867

13 2327

–59 1270

–10 10,242

1.0

Proportion cost-effective

0.9 0.8 0.7 0.6 0.5 0.4

Universal SES

0.3 0.2 0.1 0 0

Cost-effectiveness and cost-effectiveness acceptability of the school entry hearing screen

TABLE 55 Expected cost and QALYs of less effective screening, 1 year Expected cost (£)

Expected QALY

SES-T

SES-PQ

SES-SW

SES-T

SES-PQ

SES-SW

10 2 6 15

23 12 8 44

30 24 9 84

0.975 0.019 0.912 0.991

0.977 0.006 0.964 0.989

0.964 0.022 0.907 0.989

Mean SD 2.5% percentile 97.5% percentile

TABLE 56 Net benefit of less effective screening versus no screening, 1 year, QALYs WTP (£)

Net benefit of screening compared with no SES (£) Mean

2.5% percentile

97.5% percentile

–10 –121

2 568

–15 –1989

–6 286

SES-PQ 1 30,000

–23 –81

12 145

–44 –355

–8 207

SES-SW 1 30,000

–30 –464

24 645

–83 –2033

–9 236

SES-T 1 30,000

1.0

SES-T

0.9

SES-SW

Proportion cost-effective

0.8

SES-PQ

0.7 0.6 0.5 0.4 0.3 0.2 0.1 0 0

20 WTP (£,000)

FIGURE 18 CEAC of less accurate SES programmes versus no SES, 1 year, QALYs

Health Technology Assessment 2007; Vol. 11: No. 32

TABLE 57 Net benefit of less effective screening versus SES-C, 1 year, QALYs WTP (£)

Net benefit of less accurate screening compared with SES-C (£) Mean

2.5% percentile

97.5% percentile

SES-T 1 30,000

0 –244

6 566

–6 –2132

16 131

SES-PQ 1 30,000

–13 –193

13 133

–35 –449

13 64

SES-SW 1 30,000

–20 –576

24 649

–74 –2203

12 100

1.0

SES-T

0.9

SES-SW

Proportion cost-effective

0.8

SES-PQ

0.7 0.6 0.5 0.4 0.3 0.2 0.1 0 0

WTP (£,000)

FIGURE 19 CEAC of SES-C versus less accurate tests, 1 year, QALYs

TABLE 58 Net benefit of more accurate screening versus SES-C, 1 year, QALYs WTP (£)

SES-PTS 1 30,000

Net benefit of more accurate screening compared with SES-C (£) Mean

2.5% percentile

97.5% percentile

0 –4

8 85

–19 –220

17 194

1.0 0.9 0.8 0.7

SES-PTS

Proportion cost-effective

Cost-effectiveness and cost-effectiveness acceptability of the school entry hearing screen

0.6 0.5 0.4 0.3 0.2 0.1

1.0 0.9 0.8 0.7

Targeted SES

0.6 0.5 0.4 0.3 0.2 0.1

0 0

WTP (£,000)

FIGURE 20 CEAC of SES-PTS versus SES-C, 1 year, QALYs

FIGURE 21 CEAC of low accuracy targeted SES versus SES-C, 1 year, QALYs

TABLE 59 Expected cost and QALYs of targeted screening versus universal SES-C, 1 year Expected cost (£)

Mean SD 2.5% percentile 97.5% percentile

Expected QALY

Low-accuracy targeted screening

High-accuracy targeted screening

3 3 1 12

1 1 0 2

Low-accuracy High-accuracy targeted screening targeted screening 0.980 0.006 0.967 0.992

0.988 0.004 0.981 0.996

TABLE 60 Net benefit of low accuracy targeted screening versus SES-C, 1 year, QALYs WTP (£)

Net benefit of less accurate targeted screening compared with SES-C (£) Mean

2.5% percentile

97.5% percentile

Low-accuracy targeted screening 1 6 30,000 –74

4 78

–3 –251

20 76

TABLE 61 Net benefit of high accuracy targeted screening versus SES-C, 1 year, QALYs WTP (£)

Net benefit of more accurate targeted screening compared with SES-C (£) Mean

High-accuracy targeted screening 1 9 30,000 173

5 77

2.5% percentile 5 65

97.5% percentile 25 364

Health Technology Assessment 2007; Vol. 11: No. 32

Comparison of SES-C with no SES, low prevalence of hearing impairment in target population, 1-year time-horizon, QALYs Tables 62 and 63 and Figure 23 show the results when the prevalence of unidentified permanent hearing impairment is assumed to be lower, as would be the case if a proportion of cases of hearing impairment were identified via the NHSP. SES-C is compared with no SES for a 1-year timehorizon. In this analysis, the prevalence of unidentified permanent hearing impairment is reduced from 3.5 in 1000 to 0.34 in 1000. This was chosen to reflect the potential impact of the introduction of the NHSP (Waltham Forest study, Chapter 3). In addition, the probability that someone with hearing impairment has any permanent hearing impairment (including minimal and mild hearing impairment) is reduced from 0.04 to 0.01. The cost-effectiveness acceptability analysis suggests that in this case SES-C was still more cost-effective than no SES in over 50% of simulations, if decision-makers are willing to pay £5000 or more to gain 1 QALY. Given the data used, this indicates that there is a high probability that SES-C is likely to be more cost-effective than no SES, when the prevalence of unidentified permanent hearing impairment is reduced to 0.34 in 1000 and the proportion of people with hearing impairment who have any permanent hearing impairment is reduced to 1%.

Proportion cost-effective

1.0 0.9 0.8 0.7

Targeted SES

0.6 0.5 0.4 0.3 0.2 0.1 0 0

WTP (£,000)

FIGURE 22 CEAC of SES-C versus high accuracy targeted SES, 1 year, QALYs

impairment are screened (10%). For this analysis, the probability that children are accurately identified was set equal to 90%. The costeffectiveness acceptability analysis suggests that in this case targeted SES was more cost-effective than SES-C in around 80–90% of simulations, if decision-makers are willing to pay £1 or more to gain 1 QALY. Given the data used, this indicates that there is a high probability that SES-C is likely to be less cost-effective than targeted SES, if the process to identify at-risk children currently is at approximately 90%.

Tables 64 and 65 and Figure 24 show the results if the NHSP or other previous screening programmes mean that the prevalence of any

TABLE 62 Expected cost and QALYs of SES-C versus no screening, low prevalence of hearing impairment, 1 year Expected cost (£)

Mean SD 2.5% percentile 97.5% percentile

Expected QALY

SES-C

No screening

SES-C

No screening

9 5 5 22

0.11 0.09 0.007 0.33

0.985 0.005 0.976 0.933

0.983 0.005 0.973 0.993

TABLE 63 Net benefit of SES-C versus no screening, low prevalence of hearing impairment, 1 year, QALYs WTP (£)

Low prevalence of hearing impairment 1 30,000

Net benefit of more accurate targeted screening compared with SES-C (£) Mean

2.5% percentile

97.5% percentile

–9 47

5 78

–22 –154

–5 176

Cost-effectiveness and cost-effectiveness acceptability of the school entry hearing screen

1.0

Proportion cost-effective

0.9 0.8 0.7 0.6 0.5 Universal SES

0.4 0.3 0.2 0.1 0 0

WTP (£,000)

FIGURE 23 CEAC of SES-C versus no SES, lower prevalence of moderate or worse permanent hearing impairment, 1 year, QALYs

Comparison of SES-C with no SES, 1-year timehorizon, true cases of hearing impairment detected Tables 66 and 67 and Figures 25 and 26 present the results of comparing SES-C with no screening, using the limited outcome measure of number of true cases of hearing impairment detected by screening. When true cases of any hearing impairment are used as the effect measure, the data support the results of the primary analysis, that SES-C is likely to be costeffective. However, if the appropriate effect

hearing impairment still to be found is halved, and the proportion of people with hearing impairment who have permanent hearing impairment is reduced to 1%. In this case, SES-C is still cost-effective compared with no SES, but the probability that it is cost-effective is reduced to 50% in 60% of simulations. The amount that decision-makers would need to be willing to pay to gain 1 QALY also increases from £2000 to over £6000. However, this is still less than the value implied by previous healthcare decisions (£30,000 per QALY gained96).

TABLE 64 Expected cost and QALYs of SES-C versus no screening, prevalence of hearing impairment halved, 1 year Expected cost (£)

Mean SD 2.5% percentile 97.5% percentile

Expected QALY

SES-C

No screening

SES-C

No screening

9 5 5 23

0.08 0.07 0.05 0.27

0.986 0.005 0.977 0.994

0.985 0.005 0.975 0.994

TABLE 65 Net benefit of SES-C versus no screening, prevalence of hearing impairment halved, 1 year, QALYs WTP (£)

Low prevalence of hearing impairment 1 30,000

Net benefit of more accurate targeted screening compared with SES-C (£) Mean

2.5% percentile

97.5% percentile

–8 16

4 73

–23 –179

–5 138

Health Technology Assessment 2007; Vol. 11: No. 32

1.0

Proportion cost-effective

0.9 0.8 0.7 0.6 0.5 Universal SES

0.4 0.3 0.2 0.1 0 0

WTP (£,000)

FIGURE 24 CEAC of SES-C versus no SES, prevalence of still-to-be-found hearing impairment is halved, 1 year, QALYs TABLE 66 Expected cost and true cases of any hearing impairment detected of SES-C versus no screening, 1 year Expected true cases of any hearing impairment

Mean SD 2.5% percentile 97.5% percentile

Expected true cases of permanent hearing impairment

SES-C

No screening

SES-C

No screening

0.018 0.018 0.007 0.07

0.001 0.000 0.000 0.002

0.00038 0.00053 0.00003 0.00216

0.00002 0.00002 0.00000 0.00009

TABLE 67 Net benefit of SES-C versus no screening, true cases of any hearing impairment detected, 1 year, QALYs WTP (£)

Net benefit of more accurate targeted screening compared with SES-C (£) Mean

True cases of any hearing impairment 1 –9 30,000 502 True cases of permanent hearing impairment 1 –9 30,000 2

2.5% percentile

97.5% percentile

5 524

–24 195

–5 2058

5 12

24 –7

5 39

measure is thought to be true cases of permanent hearing impairment detected by screening, then SES-C is less likely to be cost-effective, with less than 50% of simulations showing SES-C as costeffective. Table 68 shows the total cost and number of true cases of any hearing impairment detected for a 1-year cohort of children entering school and eligible for screening in England. This is based © Queen’s Printer and Controller of HMSO 2007. All rights reserved.

on the population of 1.129 million children aged between 4 and 6 years in 2005 (http://www.statistics.gov.uk/statbase/Expodata/ Spreadsheets/D9390.xls). The data indicate that the universal screening programme represented by SES-C is likely to detect an additional 1497 true cases of any hearing impairment in 1 year compared with no screening, including an additional 32 cases of permanent hearing impairment.

Cost-effectiveness and cost-effectiveness acceptability of the school entry hearing screen

1.0

0.9 0.8 0.7

Universal SES

Proportion cost-effective

1.0

0.6 0.5 0.4 0.3 0.2 0.1

0.9 0.8 0.7

Universal SES

0.6 0.5 0.4 0.3 0.2 0.1

0 0

WTP (£,000)

FIGURE 25 CEAC of SES-C versus no SES, 1 year, true cases of any hearing impairment detected

FIGURE 26 CEAC of SES-C versus no SES, 1 year, true cases of permanent hearing impairment detected

Summary

economic model was the data included in the systematic review reported in Chapter 4. This was supplemented by data on the prevalence and distribution of hearing impairment, the probability of a child being screened, diagnosed and treated, the costs of screening, diagnosis and treatment, and the outcomes of screening from published literature, the survey of current practice, the observational study conducted in Waltham Forest, national statistics and databases, local accounts and expert opinion.

The literature was systematically searched to identify published economic evaluations that assessed the cost-effectiveness of SES. No full economic evaluations were found. Two partial economic evaluations were found. Overall, the quality of these papers was judged to be low according to the quality assessment criteria used. In particular, there were insufficient data available to judge the validity and robustness of the economic and clinical data used in the analyses, or the relevance of the data and results to the UK setting. Cost studies were also reviewed to extract any relevant resource use and unit cost data for the economic model. However, as with the economic evaluations, the quality and applicability of the data (to the UK setting) from these studies were limited. A decision-analytic model was developed to assess the costs, effectiveness and net benefit of SES compared with no SES and SES using alternative tests within the screen. The primary source of data about the accuracy of the screening tests for the

For the 1-year time-horizon used in the primary analysis, SES-C was associated with higher costs and slightly higher QALYs compared with no SES and other SES programmes. The range of expected costs, QALYs and net benefits was broad, with the 2.5th to 97.5th percentiles of differences in expected costs and outcomes crossing zero. CEACs and measures of net benefit provide a means of assessing the robustness of differences in expected costs and outcomes, when combined into ICERs. These analyses allow for the fact that there may be a relationship between resource use and costs and outcomes, so that, for example, higher

TABLE 68 Total costs and true cases detected for a 1-year cohort of children eligible for screening Total costs (£)

SES-C No screening SES-C minus no screening

880,854 19,379 861,475

Total true cases detected Any hearing impairment

Permanent hearing impairment

1,585 88 1,497

33 2 32

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resource use and therefore costs may be associated with improved outcomes. Overall, the primary analysis indicated that SES-C was cost-effective compared with all the other SES and no SES programmes. A number of secondary analyses was used to explore subgroups of the data and test assumptions used in the model. However, the use of subsets of the data, with relatively few studies to

combine and small sample sizes, means that these analyses can be exploratory only. The secondary analyses supported the result that SES-C is costeffective compared with no SES and alternative SES programmes. The most cost-effective method of implementing SES is using SES-PTS. This is more cost-effective than no SES, SES-C and less accurate tests used in SES.

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Chapter 6 Summary and conclusions Introduction: strengths and weaknesses of the study This study is the first comprehensive attempt in the UK to address issues surrounding screening for hearing impairment at school entry (around age 4–6 years). Previous studies have reported surveys of practice, audits of screen performance and test accuracy for specific conditions such as OME. This study aims to bring three strands of work together: a survey of practice across the UK (Chapter 2), a systematic review of the accuracy of alternative tests and the effectiveness of interventions (Chapter 4), and modelling costs and cost-effectiveness (Chapter 5). In addition, the authors were able to access some primary data from the team in Waltham Forest (PW) that address changes that are likely to occur owing to the introduction of newborn hearing screening (Chapter 3).

Survey of current practice For the survey of current practice a postal questionnaire was developed for service leads for the SES across the UK. Robust results from postal survey methodology rely on both identifying the appropriate population and achieving a sufficiently high response rate to be able to generalise. Identification of the population to which the questionnaire was to be sent raised several procedural difficulties. The aim was to survey the total population, that is, all staff responsible, as service leads, for the SES programme in the UK. The service is not provided in the same way throughout the UK, probably for historical reasons, and hence identifying the leads involved a series of different approaches (listed in Chapter 2), including advertisements in professional newsletters and cold-calling NHS trusts. This last method highlighted the difficulty, encountered often, of identifying anyone within the trust who knew about the screen and/or knew who had responsibility for it. This raises issues of managerial responsibility for the screen, and consequent local and national accountability for screen performance. After considerable effort and time a contact was secured for every trust in England, every board in Wales and Scotland and © Queen’s Printer and Controller of HMSO 2007. All rights reserved.

for every LSHCG in Northern Ireland. The 244 service leads identified for the SES, many of whom covered more than one PCT, comprise a much higher number of SES leads than in previous surveys,6 and are likely to represent almost total coverage, although it is possible that a very few services run entirely by educational services would be missed. Research governance approval was sought from 124 R&D directorates covering 304 PCTs in England, Scotland and Wales, but in Northern Ireland it was only possible to approach two R&D offices from 15 LSHCGs. R&D procedural difficulties meant that 229 out of 244 services could be sent a questionnaire. There was an extremely high return rate of questionnaires for this sort of study of just over 85%. Bearing in mind the data and comments from respondents, the high return rate seemed to reflect the willingness of people to take the time to tell the researchers about their service and their views, in the knowledge that the information would be used to contribute to the development of a service which they felt was important but in need of some guidance. The response rate may also be due to the fact that respondents were not asked to allocate time to report data that were not easily available. This means that some of the information provided lacks depth and that the data on yield and screen performance are based on a relatively small proportion of all services; even the data from these services may lack accuracy. Nevertheless, the survey of current practice is the most complete carried out in the UK, with wide geographical coverage and a high response rate.

Waltham Forest observational studies The authors were fortunate to be able to access good-quality data from the Waltham Forest services. In UK terms this service is unique since it offers robust data from three sizeable cohorts, all of which had the SES, but following a universal newborn screen for one, a targeted newborn screen for the second and traditional infant screens for the third. This is a rare set of comparative cohorts, and although they do not conjointly comprise a single longitudinal study, they allow some valuable comparisons concerning the possible future impact of the now national

Summary and conclusions

implementation of the NHSP on the SES, as well as some other secular data. However, there are features of the Waltham Forest populations (e.g. ethnicity, mobility) that undermine highly detailed or quantitative generalisation from this local evidence base to the national cohort covered (since March 2006) by the NHSP, once the latter has reached school entry age (in around 2010). Nevertheless, the epidemiological characteristics of those children with a permanent hearing impairment in the Waltham Forest cohorts was not significantly different from that reported from larger and national studies undertaken in the UK, and it is probable that the population of hearingimpaired children reported is reasonably typical of that present elsewhere in the UK. The newborn screen protocol used in the Waltham Forest cohorts differed from that implemented in the NHSP; nevertheless, the yield of moderate or worse permanent bilateral hearing impairment obtained from the NHSP (1.0 in 1000, 95% CI 0.78 to 1.22)9 was extremely similar to the yield of 1.03 in 1000 (95% CI 0.66 to 1.40) achieved in the Waltham Forest studies. The yield of children with a permanent unilateral hearing impairment in the Waltham Forest studies was 0.55 (95% CI 0.28 to 0.82) and this was similar to the 0.64 in 1000 (95% CI 0.37 to 0.91) reported from the NHSP. These similarities suggest that generalisation is justified. The Waltham Forest studies were ascertainment studies, that is, they depended on collecting data from all known cases of children with permanent hearing impairment in the three cohorts. Thus, cases not yet identified, for whatever reason, would not be included, and the strength of the data is crucially dependent on both the quality of the service at identifying cases and the robustness of the ascertainment procedures. Furthermore, the ability of services accurately to identify childhood hearing impairment reduces with lower levels of impairment, so in any study of this type, despite the greater number of milder cases there remains some uncertainty about their ascertainment rate.

Systematic review of accuracy and effectiveness of screening

The principal strength of the systematic review was its comprehensiveness. Compared with three previous similar reviews in this area more studies were identified, in part because these previous reviews focused more narrowly, for example on preschool screening for OME or on a particular screening test (whisper voice test).

The main limitation of the review was a lack of good-quality evidence on the effectiveness of SES on long-term outcomes, including educational, language and social outcomes. The authors acknowledge the challenge of demonstrating effectiveness at two stages, both case finding and intervention. In addition, although several studies have assessed the accuracy of screening tests, their quality and the quality of reporting were unacceptably variable. Two particular problems were the inconsistent reference standard applied and variability in the criterion definition for hearing impairment that was used. Although full diagnostic PTA was used as the reference test in many studies, this was not the case in all. A reference standard test is a key element of any study of diagnostic accuracy. A high or low level of accuracy derived from a comparison of a given test against a range of reference standards, none of which is stated to be a gold standard, is difficult to interpret. Furthermore, studies often failed to specify clearly the criterion threshold that defined hearing impairment, and different studies applied different hearing thresholds for their case definition. Without a consistent case definition it is difficult not only to interpret the accuracy of results of a given study, but also to apply these results to real-world clinical practice.

Cost-effectiveness Overall, the evidence base to support the economic model was weak. As noted in Chapter 4, the robustness of the available evidence about the accuracy of screening tests was undermined by the variable quality of the studies investigating test accuracy. In addition, no evidence about the shortor long-term effectiveness (impact on disability and quality of life) or cost-effectiveness of SES was found. The data used to estimate other probabilities, costs and outcomes were synthesised from a variety of sources, including surveys of clinical practice and expert opinion. These may affect the robustness of the conclusions. Although the variables that used data from surveys or expert opinion were assigned wide distributions wherever possible, this also increases the uncertainty in the model parameters and reduces the likelihood or probability that an intervention is cost-effective. The economic model was static in nature and based on a short time-frame of 1 year for the primary analysis. The time-horizon was extended to 11 years in secondary analyses. The static structure of the model was based on the assumption that the values of the variables included would not change significantly over time. There is no evidence to suggest that this is an

Health Technology Assessment 2007; Vol. 11: No. 32

unreasonable assumption for the 1-year timeframe considered in the primary analysis. However, if hearing impairment identified by the school screen is progressive, or the impact on quality of life and health status of hearing impairment identified by the SES changes significantly over time, then the results of the economic model may not apply. High and low estimates of utility and long-term costs were included in the primary and secondary analyses. The results of the cost acceptability and net benefit analyses reflected the high uncertainty about the input parameters, but broadly supported the main conclusions of the economic analyses. The structure of the model was developed from the reviews of clinical and economic evidence and discussion with experts in audiology. The primary and secondary analyses used QALYs as the outcome measure to estimate ICERs, net benefit and CEACs. QALYs take into account differences in potential life expectancy and the impact of adverse events on overall health-related quality of life; they tend to be weighted by mortality and produce only small differences among adverse events that have a short or relatively low impact on quality of life. This may bias the analysis if one or more interventions are associated with high rates of adverse events that individually have a relatively low impact on health, but cumulatively could have a significant impact on health and health-related quality of life. Overall, the estimates of expected QALYs differentiated between different SES programmes and levels of hearing impairment. These factors would suggest that the QALY is a reasonable measure for the economic analysis (and is consistent with the approach used for reports to NICE).

Summary of findings Survey of current practice The SES is usually performed in the first year of primary education, in school, and usually (72%) with prior written information to parents and guardians. The survey indicated that there is wide variation in the implementation of the screen throughout the UK. This variation applies to the population covered, with 51% of respondent services not screening children entering private education and 72% not screening home-educated children; the physical location and conditions under which the screen is implemented, with little © Queen’s Printer and Controller of HMSO 2007. All rights reserved.

evidence of commitment by schools to offer suitable locations, and little commitment by management to provide training and replacement equipment; test methodology, with different numbers and types of tests and retests (17% referring after the first test, 72% after a second, 10% after a third); the time of repeat tests (same day to 12 weeks); the criteria determining which children to refer, which varied from 20 dB HL across up to seven frequencies to 30 dB HL at only three frequencies (the dB scale is a logarithmic ratio scale, with 0 dB HL being the average normal hearing threshold and the difference between 20 and 30 dB HL representing a ten-fold increase in power and more than a doubling of subjective loudness); the personnel and the equipment involved; and the ability to collect and then retrieve data. There is little or non-existent robust audit at local (and therefore national) level, absent or inadequate data management systems, and a lack of explicit procedures for quality assurance. The one area of consistency concerned the pure tone sweep test, which was used by 97% of responding services as the first test in the screen. The fact of existing protocol variation between services could provide for an evaluation of what might be the most appropriate, successful and efficient implementation of the screening programme. However, good data on yield and screen performance are necessary for such an evaluation; although nearly 70% of services claimed to have data management systems in place, only 50% of those said that they could easily obtain data reports. Coverage and referral data were available for approximately one-third of services (n = 55), but fewer than 20 services could provide any robust data on the numbers of children identified as hearing impaired. The uptake data from those that could provide them indicated a median uptake rate for those offered the screen of over 90%. At least half the responding services screen those children already known to some part of the service (but often not to the SES service) to have a hearing impairment, again indicative of poor information sharing. Despite these marked difficulties and the lack of robust audit there was a very high response rate to the survey questionnaire, indicating a high level of interest; there were clear indications from comments offered that the majority of service leads regard the screen as useful and would prefer it to continue, even though it was recognised that the value of the screen may reduce with the advent of universal newborn hearing screening. A small

Summary and conclusions

proportion of respondents (12.2%) have abandoned the screen, and a few noted that they were awaiting national guidance on its future. A significant number of respondents stated that they would welcome such national guidance. Other service leads would welcome guidelines on the value of a selective (targeted) screen and on the population for whom it would be appropriate. Most concerns about the continued relevance of the screen as a universal screen focused on the impact of the introduction of universal newborn hearing screening, the inadequate resources (time, personnel and facilities) available to implement the screen, and the inadequacy of systems and technological support for data management and retrieval. Support for the continuing value of the screen focused on its ability to identify children who would otherwise not be identified, either because they had been missed by previous screens or surveillance for whatever reason, or because they had entered the system having had no previous screens.

Waltham Forest observational studies The evidence from the cohort comparisons in Waltham Forest reported here for the first time in a single source suggests strongly that there is a material effect of the introduction of newborn hearing screening on the SES, in addition to other secular changes that have occurred in recent years. Of the latter, the most important are probably the immunisation programmes that seem to have been accompanied by a significant reduction in the proportion of children with severe and profound unilateral hearing impairment. With regard to the possible changing pattern of routes to identification of permanent childhood hearing impairment, before the introduction of newborn screening the yield from the IDT, intermediate screens and parental/professional concern throughout infancy and up to school entry was around 73% of the yield of all PCHI cases resident and currently known to the service, while the final screen, the SES, accounted for the remainder at a rate of 1.11 in 1000, of which 0.63 in 1000 were unilateral impairments. The evidence suggests that since the introduction of universal newborn screening, over 90% of the cases known to services now have been identified via newborn screening and parental/professional concern in infancy and up to school entry, with only 0.34 in 1000 identified by the SES (of which only 0.07 in 1000 were unilateral).

Thus, in Waltham Forest, newborn screening has reduced the yield of the SES for permanent hearing impairment. However, postnewborn

routes to identification remain important, in large part because of late-onset and acquired cases, those who had ‘moved in’, and those with a congenital impairment that had not been picked up by the newborn screening programme. The prevalence of mild and greater bilateral and unilateral hearing impairment at school age was 3.47 in 1000, similar to findings from other studies. Parental and professional concern remained a steady source of identification (1.31 in 1000) postnatally, but still, at school entry 16% of moderate and greater bilateral, 18% of mild bilateral and 17% of unilateral permanent hearing impairments remained to be identified. The evidence for long-term effects of moderate or greater congenital bilateral hearing impairment is well documented,5 and there are known and demonstrably beneficial interventions based around the early provision of hearing aids. Longstanding beliefs in the necessity of intervention have made it impractical to conduct controlled trials on benefits of intervention for moderate hearing impairment which is first identified at school entry, and the present research has furthermore failed to identify statistically controlled studies with age of identification as a major factor that enable some conclusions to be drawn in the way that it is possible in the earlier years. Nevertheless, it is reasonable to assume that the effects, if untreated, would be marked, especially for significant but unidentified impairment at the transition to formal schooling. These arguments suggest that some sort of systematic approach to identification of moderate or greater permanent hearing impairment at, or approaching school entry age is required. The evidence on the effects of mild bilateral and unilateral hearing impairment on long-term outcomes is largely absent, and the same argument is therefore difficult to make on the basis of available evidence. However, from what is known of mild hearing impairment and the acoustics of classrooms, it would be reasonable to extend the argument to include the need to find and manage these not previously known mild and unilateral hearing impairments as well.

Systematic review of accuracy and effectiveness of screening There was only level III evidence for the effectiveness of preschool hearing screening, from a single, poor-quality, observational comparative study. Furthermore, this single study was inconclusive in whether preschool screening was more effective than no screening in detecting hearing impairment. No studies were identified that have assessed the long-term impact of

Health Technology Assessment 2007; Vol. 11: No. 32

preschool hearing screening on educational, language and social outcomes. Several studies have assessed the accuracy of different hearing screening tests in preschool children. Given the unacceptable variability in methodological quality and reporting of these studies, the lack of clarity over the cases of hearing impairment detected (e.g. transient versus permanent hearing impairment), the variation in reference test and threshold level for hearing deficit, and the range of settings in which these tests were applied, it is difficult to interpret and compare their results. Nevertheless, accepting these caveats and selecting the subset of studies using PTA as the reference test, the findings suggest that pure tone sweep audiometry has high sensitivity and specificity for full PTA and therefore appears to be a suitable test for screening. Other possible tests, about which more and better evidence is required, are spoken word tests and TEOAEs. For OME, tympanometry and reflectometry have variable reported sensitivity and specificity as screening tests (although note that tympanometry is a wellestablished and valuable diagnostic test which would be expected to be part of the follow-up diagnostic test battery), and parental report is found to have poor sensitivity and specificity. There is insufficient evidence to comment on the accuracy of combinations of tests. A small number of studies indicated a generally high uptake in this age group. However, given the experimental design of the studies and the fact that they were assessing test accuracy rather than programme effectiveness, these findings cannot be generalised to the uptake of the screen in realworld community screening settings. The two published studies with evidence of uptake of screening at school entry in real-world settings suggest uptake in excess of 90%, reflecting the ‘captive’ nature of the population to be screened.6,97

Cost-effectiveness There are no good-quality published studies that assess the cost-effectiveness of SES, and no full economic evaluations. The two partial economic evaluations that were found were of poor quality and uncertain relevance. A decision-analytic model was developed to assess the costs, effectiveness and net benefit of SES when compared with no SES and SES using alternative screening tests. The primary source of data about the accuracy of the screening tests for © Queen’s Printer and Controller of HMSO 2007. All rights reserved.

the economic model was the data included in the systematic review reported in Chapter 4. This was supplemented by data on the prevalence and distribution of hearing impairment, the probability of a child being screened, diagnosed and treated, the costs of screening, diagnosis and treatment, and the outcomes of screening from published literature, the survey of current practice (Chapter 2), the Waltham Forest observational study (Chapter 3), national statistics and databases, local accounts and expert opinion. For the 1-year time-horizon used in the primary analysis, SES-C was associated with higher costs and slightly higher QALYs compared with no SES and other SES alternatives. The ICER for SES-C is around £2500 per QALY gained. The range of expected costs, QALYs and net benefits was broad, with the 2.5th to 97.5th percentiles of differences in expected costs and outcomes crossing zero, indicating a high level of uncertainty in the conclusions. CEACs and measures of net benefit provide a means of assessing the robustness of differences in expected costs and outcomes, when combined into ICERs. These analyses allow for the fact there may be a relationship between resource use and costs and outcomes, so that, for example, higher resource use and therefore costs may be associated with improved outcomes. Overall, the primary analysis indicated that SES-C was costeffective compared with all the other SES programmes evaluated and with no SES. The costs of individual SES tests (rather than screening programmes) were estimated to be approximately £8 per screening test (see Table 42, Chapter 5). It is the costs of the screening tests that dominate the total expected costs of screening. The costs of the screening tests may have been overestimated if the duration of the tests reported in the survey of current practice also included the time needed to conduct wider health checks. The survey of current practice (Chapter 2) indicated that up to 60% of programmes included the SES in wider health checks all the time; if this is the case, then the cost of each screening test may be lower and the costeffectiveness of SES higher than estimated here. A number of secondary analyses was used to explore subgroups of the data and test assumptions used in the model. However, the use of subsets of the data, with relatively few studies to combine and small sample sizes, means that these analyses can be exploratory only. The secondary analyses supported the result that SES-C is costeffective compared with no SES and alternative SES models. Furthermore, the analyses using the 6- and 11-year time-horizons supported this

Summary and conclusions

conclusion, with SES-C being more cost-effective in over 99% of simulations. In the economic model, SES-C is a weighted composite reflecting the pure tone sweep test (99%) with tympanometry (1%). When SES-C was compared with the SES as it is mostly practised at present in the UK (i.e. pure tone sweep only), the latter was more cost-effective in 80% of simulations. The estimates of the costs and QALYs of SES-C were based primarily on the pure tone sweep as the screening test. The studies included in the systematic review (Chapter 4) indicate that this test has relatively high sensitivity and specificity compared with alternative tests; however, the accuracy of the pure tone sweep was assessed in trials settings, rather than in the varied and less than ideal settings encountered in routine practice. This may mean that the accuracy and therefore the cost-effectiveness of SES-C are overestimated, compared with no SES. However, the analysis comparing SES using alternative tests with lower accuracy indicated that SES might still be more cost-effective than no SES. When SES-C was compared with targeted screening at school entry, if the targeting accurately detected 90% of children with a hearing impairment then targeted screening was more cost-effective than universal screening. However, if the identification of children at risk of hearing impairment for targeted screening was associated with low sensitivity and specificity, then universal screening was more cost-effective than targeted screening. The economic analyses used a relatively low prevalence of moderate or worse previously unidentified permanent hearing impairment. Reducing this further to model the potential impact of newborn hearing screening reduced the proportion of simulations when SES was costeffective to around 60%. Decision-makers also needed to be willing to pay over £6000 to gain 1 QALY for SES to be cost-effective in more than 50% of cases. Overall, because of the lack of primary data and the necessarily wide limits for variables in the modelling, these results must be considered indicative and exploratory only.

The OME issue and some further analyses 80

Hearing impairment of a mild degree is also associated with transient episodes of OME, which

is much more prevalent in children than is permanent hearing impairment.15 Some people have argued the case for a screen at school entry to identify previously unknown cases of children with OME that is of a severity and/or persistence sufficient to require treatment. There has been extensive although generally poor-quality research on the treatments for OME in children, much of which fails to address the question of which subtypes of ‘OME child’ benefit from treatments. A recent meta-analysis98 accessing individual patient data from several trials confirmed the accepted conclusion that, for well-defined cases, ventilation tubes (grommets) do improve hearing for so long as they are in place. However, for the most persistent or recurrent cases (i.e. those for whom the certainty of selection for surgery is greatest) the condition tends to return, leading to the need for reinsertion(s) of grommets. Age, within the range of about 3–8 years, does not seem to be a characteristic of major importance for results, provided that children meet a criterion for persistence and severity. The most comprehensive and sophisticated evidence on candidature for intervention in OME is emerging from the UK TARGET randomised trial,99 of which the aspects of particular relevance here are mostly yet to be published. The trial does not contradict the above simple statements, but documents more fully the breadth and duration of benefits from adjuvant adenoidectomy and the criteria for selection of a subgroup within which the combined treatment is highly effective (Haggard M, University of Cambridge: personal communication, 2006). Professor Haggard informed the review team via several presentations given at international meetings and extensive annotated analyses which show that the largely null results on young mild cases emerging from screening did not apply to older (>3.75 years) and better selected cases, but that consistent if modest benefits are shown in the TARGET data. Thus, in relation to the Wilson–Jungner principles for screening, at one level an effective and available intervention does exist for children of school entry age. There is still no convincing and favourable evidence for types of treatment other than these surgical operations. The implementation of an overall screening and treatment programme is less satisfactory than the above statement suggests. The evidence on this point comes from children of younger age than school entry, but there is no good reason for it not

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to apply. Two trials of ventilation tubes in OME have been published in recent years, making essentially the same point.100,101 They are summarised here because, although restricted, they are of high internal validity and are particularly relevant to the issue of the caseloads that screening tends to find: the marginal rather than the extreme. The Rovers trial was done on children referred from the implementation of the 8-month IDT screen in The Netherlands, although by the time they had been through confirmation of fluid in the ears, they were around 2 years of age. As well as an ear status measure, there was a language test and a quality of life scale. The Paradise trial recruited slightly older children in the USA to whom it was possible to give a wider range of assessments of valued outcomes including performance tests. These children had been referred by paediatricians who exercise a highly surveillant semi-specialised form of childcare in the medically insured part of the US population. Both of these trials found that the placement of ventilation tubes did give the known short-term benefits to ear status or hearing, but did not improve wider valued measures of outcome. These null results make useful political points for the two countries concerned, both having high intervention rates, about overtreatment in routine practice in the past, due to selection of cases that are too mild and/or insufficiently persistent to benefit. A widespread misinterpretation of these trials, imagining that they suggest that ventilation tubes ‘do not work’, has led to unnecessary avoidance of their real message. In many conditions it is hard to show a knock-on from either disease or treatment into valued outcomes, in relation to other powerful sources of influence on those same outcomes. The indirect knock-on from fluctuating hearing impairment and physical health problems in OME, and its treatment, into language and other developmental outcomes make that challenge particularly hard. The knockon benefits would be expected to be rather slight. The evidence suggests that it is particularly slight in very young caseloads emerging from screening or surveillance, where the rate of spontaneous remission in untreated controls is high, and particularly so where the case entry criteria are mild. For the preceding reasons, TARGET recruited children from the NHS who had already undergone gate-keeping. This typically includes some initial caution by the GP over the need for referral, several months (in most districts) of © Queen’s Printer and Controller of HMSO 2007. All rights reserved.

waiting to be seen in secondary care, and being subject to a further 3-month formal watchful waiting period to establish persistence or recurrence. These were older children (3.5–7 years) and only randomised on meeting a severity criterion of hearing thresholds of 20 dB HL or greater in the better ear a second time after 3 months of watchful waiting. TARGET did produce some statistically significant and clinically material benefits to physical health and development over the 2 years following treatment as well as to hearing, but these benefits were rather modest taken as a whole. Given awareness of this difference in caseload, the results from TARGET and the other two trials are not inconsistent. Further analyses of the hearing level data from TARGET established that children with more severe hearing levels do indeed receive more benefit to hearing, as expected. The finding is encouraging for tests of hearing being relevant for screens for OME. The issues then for screening in OME as a fluctuating condition are: (1) how few sequential stages will suffice after some initial universal screen to define a small caseload that approximates the severity and persistence of that in TARGET, and (2) whether for the cost of such successive testing, the incremental yield over what would have been referred reactively at this age is worth that cost. There is no published evidence that addresses the foregoing two questions. Both a national study6 and the historical cohort from Waltham Forest (Chapter 3) have shown that just under 3% of those screened were referred from the SES with OME. This represents a large proportion of all those referred by the SES, with data from the survey of current practice suggesting a median for positive predictive value for temporary conductive hearing impairment of 36%. In the Waltham Forest cohort, new cases (i.e. not previously known to services) amounted to 1.4% and of these half (0.7%) needed ENT referral; details thereafter were not available, and interpretation of the value of making these referrals is therefore difficult. However, some light can be thrown on the contribution of the present SES system by a further analysis of cases seen in the TARGET recruitment stages. SES screen referrals to ENT services for possible surgery tend to go through community paediatricians who specialise in audiology (e.g. members of BACDA), rather than through GPs. This distinction is not hard and fast: not all community referrals will have originated with screens, although many will, and nearly all ex-screen referrals will arrive from this source. The

Summary and conclusions

TARGET RCT had already shown on its database of over 3000 cases that referrals from community paediatricians had higher positive predictive value than those direct from GPs.99 Because the lead-in stages to the randomised trial had recruited at a dozen ENT departments throughout the UK, the trial chief investigator (Haggard) and colleagues were asked to probe whether these analyses could be extended to say anything about the yield from SES.

●

A set of analyses was run on over 4000 referrals and a document annotating the results was supplied to the present team, of which the following is a summary. Although the TARGET study had not asked about screening at the level of either individual case or contributing district (clinic), it had a useful degree of indirect leverage via (1) the age distribution being centred on 5 years, the age of maximal relevance to SES and (2) a distinction between GP and community referrals. The question that this permits to be answered is one of yield: whether, taking GPreferred cases as a control set, the number or severity of community-referred children in this database increases from around 5 years of age, when contrasting the preceding 2 years of crosssectional age with the following 2 years. This involves an interaction with age. The main-effect advantage for community mentioned above was again found to be pervasive and reflects the availability in community services of audiometry, some specialist expertise and a tendency to retest to establish persistence. Analyses of this type broken down by age and source were conducted on the severity (a marker of positive predictive value and specificity) and numbers of cases (yield). Effectiveness of screening would predict the highest values to occur in the ex-community over5-year-olds. However, in neither numbers nor severity was there an interaction between source (GP/community) and age band (before/after modal SES age). Thus, the combination of number and severity of cases coming through the community specifically after SES age is not large enough to show up in the ENT caseload. It is therefore probably not large enough either to represent a distinct societal benefit.

●

The evidence from the observational studies in Waltham Forest is that: ●

●

Conclusions The evidence from the national survey of current practice is that: ●

the SES is in place in most areas of England, Wales and Scotland; data from Northern

Ireland were too few to draw any conclusions that might generalise there; just over 10% of respondents had abandoned the screen, while others were awaiting guidance in the light of the national implementation of newborn hearing screening coverage of the SES is variable, but is often over 90% for children in state schools; coverage is poor for private schools and home-educated children referral rates are variable, with a median of about 8% the test used for the screen is in all cases the pure tone sweep test; however, there is a wide variety of implementations of this, with differing frequencies, pass criteria and retest protocols; written examples of protocols were often poor and ambiguous there is no national approach to data collection, audit and quality assurance, and there are variable approaches at local level; a small proportion of services was able to provide audit data on coverage referral rates and yields, but these were often of doubtful quality, especially with respect to yield the screen is performed in less than ideal test conditions; this probably increases the referral rates and decreases accuracy resources for replacement equipment, calibration and screener training are said to be limited and impacting on the quality of the screen in many areas.

●

the prevalence of permanent childhood hearing impairment continues to increase through infancy owing to acquired, late-onset and progressive hearing impairment, in line with published evidence; of the 3.47 in 1000 children with a permanent hearing impairment at school screen age, 1.89 in 1000 required identification after the newborn screen; a high proportion of these appeared to have identifiable risk factors the introduction of newborn hearing screening is likely to reduce significantly the yield of a universal SES for permanent bilateral and unilateral hearing impairments; the yield of the SES in Waltham Forest for such impairments has fallen from about 1.11 in 1000 before newborn screening to about 0.34 in 1000 for cohorts who have had newborn screening, of which only 0.07 in 1000 are unilateral impairments surveillance procedures, comprising (at least) reactive services to parental and professional

Health Technology Assessment 2007; Vol. 11: No. 32

●

concern, are an important route to identification of cases throughout infancy and the preschool years in the Waltham Forest cohort studies, small but material numbers of children with permanent hearing impairment remained to be found at or before school entry; just under 20% of permanent moderate or greater bilateral, mild bilateral and unilateral impairments, known to services as 6-year-olds or older, remained to be identified around the time of school entry; some of these were late onset or acquired, some ‘moved in’ and some congenital cases not identified by the newborn screen.

The evidence from the systematic review of the accuracy of alternative tests for the SES and of the effectiveness of interventions is that: ●

●

there were no good-quality published comparative trials identified of alternative screens or tests for SES there was one poor quality study which compares screening to no screening, but the results are inconclusive studies concerned with the relative accuracy (in terms of sensitivity and specificity) of alternative screening tests are difficult to compare and often flawed by differing referral criteria and differing case definitions; nevertheless, using full PTA as the reference test, the pure tone sweep test appears to have high sensitivity and high specificity for minimal, mild and greater hearing impairments, better than alternative tests for which evidence was identified (otoacoustic emissions, tympanometry, reflectometry, parental questionnaire); some evidence suggests that spoken word tests can have high sensitivity and specificity; no goodquality evidence was identified addressing the accuracy of combinations of tests there is insufficient evidence to draw any conclusions about possible harm of the screen as currently performed there were no published studies identified which examined the possible effects of SES and subsequent interventions on longer term language, educational or social outcomes.

●

The evidence suggests that a national screening programme for permanent hearing impairment at school entry meets all but three of the criteria for a screening programme; namely, knowledge of the distribution of test values in the population with agreed cut-offs, RCTs showing that the screen reduces morbidity, and a national protocol with quality assurance and audit (Appendix 1). With regard specifically to the issue of the value or effectiveness of the screen finding cases of transient hearing impairment associated with middle ear disorder, the evidence comes from the survey of current practice, a recently published survey,6 the Waltham Forest cohort studies and a recent meta-analysis, alongside data from a large and well-controlled UK study as yet largely unpublished (Haggard M, Cambridge University: personal communication, 2006). Collectively, this material suggests that: ●

●

The evidence from the cost-effectiveness and cost modelling study is that: ● ●

●

no good-quality published cost-effectiveness studies or economic evaluations of SES were identified lack of primary data and the necessarily wide limits for variables in the modelling mean that

any conclusions must be considered indicative and exploratory only using decision-analytic modelling, and taking into account all types of hearing impairment, a universal SES based largely or completely on pure tone sweep tests was associated with higher costs and slightly higher QALYs compared with no screen and other screen alternatives; the ICER for such a screen is around £2500 per QALY gained; the range of expected costs, QALYs and net benefits was broad, indicating a considerable degree of uncertainty targeted screening can be more cost-effective than universal school entry screening; this depends on there being identifiable risk factors.

surgical intervention (ventilation tubes) for children with OME improves hearing levels as expected, and has modest but measurable effects on longer term outcomes (physical, developmental) for more severe and persistent cases, but not for milder and marginal cases about 3% of those screened are referred with OME, representing perhaps 40% of referrals; some of these cases are already known to services, and some will not require treatment; about one-quarter may require further otological management including surgery; these estimates will have wide confidence intervals there is no evidence that the SES is a better source of referrals of more severe and persistent cases of OME than reactive GP referrals, although data with a more rigorous sampling frame and prospective analysis are required to confirm that there is evidence for no effect

Summary and conclusions

●

at least six of the accepted criteria for a screening programme for OME are currently not met (Appendix 1).

Conclusions: closing comment

In 1987, Stewart-Brown and Haslum3 published the results of a survey of national practice (in England and Wales) with regard to screening for hearing impairment in childhood. In it, they commented that “the number of times that children were screened at school varied considerably … the hearing level at which children were referred after sweep audiometry varied among districts … only 73 [out of 165] could report the referral rate … very few districts were collecting the sort of data that would allow them to make even the most rudimentary assessment of their screening programmes, far less any evaluation of cost consequences or benefits … secretory otitis media fulfils few of the criteria that should be met before a screening programme can be considered likely to be either effective or ethical”. While a considerable number of lead clinicians for the SES have tried to implement an improved programme in the face of resource constraints and competing priorities, the overall similarity between the present team’s findings in 2005 and Stewart-Brown and Haslum’s in 19843 is striking. They proceeded to comment on the need for well-controlled studies to underpin policy changes; two decades on, the lack of a good-quality evidence base to drive change in this area remains a serious problem.

Implications for practice

There is some evidence that significant numbers of children with permanent hearing impairment remain to be identified at school entry. There is evidence that the pure tone sweep test if properly implemented as a screen can have reasonable levels of sensitivity and specificity as a route to identification of these children. Although there is little evidence on the effectiveness of the SES, there is also little evidence to judge whether it is ineffective. The implications of the conclusions summarised in the section ‘Conclusions’ (p. 82) are that services already implementing the SES (the overwhelming majority in the UK) should continue to do so, pending later evidence-based policy decisions (see in the next section), but that they should make every effort to implement a clear test and screen protocol, and that they should audit the screen performance for cases of PCHI not already known to services. However, the case for using the SES as a route for finding

children with OME suitable for intervention is weak (see Appendix 1).

Recommendations for future research It is evident that in most areas of the UK there are service leads who value the SES, and who head up services that deliver a screen in the main based on the pure tone sweep test to most children in public education. However, test and screen protocols vary between districts, there is little evidence of explicit quality assurance procedures, and data management systems are generally poor or nonexistent, so that data on screen performance, let alone longer term outcomes, are largely absent. The public health context in which the SES is delivered is changing significantly, and by about 2010 almost all babies born in the UK will have undergone newborn hearing screening. Not all PCHI will be identified by newborn screening: late-onset and acquired impairments, children moving in who have not had a newborn screen and cases not picked up by the newborn screen (largely mild and minimal hearing impairments) will remain to be identified. The justifiable means by which such cases could be identified include parental and professional concern, formal surveillance of some kind at defined ages, targeted school entry screening and universal school entry hearing screening. The evidence required to make policy decisions between such alternatives does not exist. The following recommendations for future research and audit are made with the overall aim of being able to make evidence-based policy decisions in or around 2012, when all school entry cohorts will have had newborn hearing screening. A priority need is for the establishment of a single, agreed national protocol for those services delivering the SES to make future studies and future audit of screen performance more directly comparable. On the basis of the evidence, such a protocol should be based on detection of pure tones. It is known that the greater the level of permanent hearing impairment, the poorer the quality of life is likely to be, while the lower the pass threshold for the screening test, the poorer the test and screen specificity. Furthermore, PCHI tends to be worse in the higher frequencies, and the testing of low and mid-frequencies in the conditions under which the SES has to be performed is particularly subject to noise interference. Thus, there are persuasive arguments for a single, high-frequency (4 kHz) pure tone

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detection test set at a level likely to be most effective and efficient (25 dB HL). Although the case for screening for OME at school entry is weak (see Appendix 1), there may be persistent or severe cases who remain to be identified at school entry and who would benefit from surgical intervention; a high-frequency 25 dB HL criterion for referral would, as an incidental benefit, increase the positive predictive value for such cases. Alongside an agreed national protocol must go systems for data monitoring and quality assurance so that robust data on screen accuracy and effectiveness can be collected. Such systems are in place for newborn hearing screening, and R&D is required to develop a single data system as part of a national plan around screening for childhood hearing impairment and paediatric audiology services. There is a need to establish with greater certainty the prevalence of permanent mild and minimal hearing impairment at school entry that could be identified by a suitable quality-assured screen protocol, and to confirm the prevalence and severity distribution of congenital unilateral hearing impairment. Comparative trials are needed to compare the effectiveness, efficacy and efficiency of alternative approaches to the identification of permanent hearing impairment postnewborn screen.

Specifically, a comparison of reactive services, a formal surveillance procedure between fourth and fifth birthdays,7 targeted screening between fourth and fifth birthdays, and universal screening at school entry age would establish the necessary evidence for policy decisions. Targeted screening would be based on children who had not received or completed newborn screening, children attending child development centres, children coming from families with a history of permanent childhood deafness, and children who have suffered bacterial meningitis or a childhood viral illness leading to doubt about their hearing. The lack of prospective controlled studies on the effectiveness of hearing screening and subsequent interventions in terms of later outcomes for children with permanent mild, minimal and unilateral hearing impairment identified at school entry represents a major gap in the evidence base. However, it is not clear that such studies would take priority over better data on alternative protocols, uptake, yield and diagnostic accuracy; furthermore, there are real problems in identifying appropriate outcome measures that have sufficient sensitivity. The distribution of detection thresholds for pure tones in the population at school entry, and how different cut-off criteria would relate to measures of hearing disability (Wilson and Jungner criterion no. 538), are not known. Research is needed to establish these.

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Acknowledgements e thank Professor Mark Haggard (Multicentre Otitis Media Study Group, the University of Cambridge), who has acted as an expert advisor for the project and has provided invaluable guidance and input on all aspects of the study. We also thank the members of our advisory committee for their contribution: Ms Susan Hamrouge (Speech and Language Therapist and Educational Psychologist), Mr Andrew Ford (parent of a deaf child) and Mr Kevin Gibbin (ENT Consultant Surgeon). Sincere thanks to all the SES service leads who completed and returned a questionnaire. We thank Anne Shafe and Sarah Armstrong (Medical Statisticians, Trent Research and Development Support Unit) for statistical advice in Chapter 3, Dr Gill Painter (Consultant Community Paediatrician – Audiology, Central Manchester PCT) for help and advice in piloting the questionnaire and her knowledge of the school health system, Ms Sue Bayliss (Department of Epidemiology and Public Health, the University of Birmingham) for designing the literature searches used in the systematic review, Dr Martin Connock (Department of Epidemiology and Public Health, the University of Birmingham) for the preparation of the summary ROC curve for the systematic review, and Mrs Tracey Keeble and Mrs Julie Robinson for the excellent administrative and secretarial support they have provided.

Contribution of authors John Bamford (Professor, Specialist in Paediatric Audiology and Deaf Education) led the research

team. Heather Fortnum (Associate Professor, Epidemiologist) led the survey and associated analyses of current practice and coordinated the editing of the final report. Kirsty Bristow (Research Assistant, Epidemiologist) conducted the survey and analyses of current practice, and coordinated effort across the team. Jenny Smith (Research Assistant, Epidemiologist) conducted the reviews of accuracy and effectiveness of screening tests, and coordinated the editing of the draft report. Georgios Vamvakas (Research Assistant, Health Economist) conducted the health economics reviews, modelling and analyses. Linda Davies (Reader, Heath Economist) led the health economics reviews, modelling and analyses. Rod Taylor (Reader, Health Statistician) led the reviews of accuracy and effectiveness of screening tests. Pete Watkin (Consultant Community Paediatrician, Physician in Paediatric Audiology) led the data collection and analyses of the Waltham Forest studies. Sarita Fonseca (Consultant Community Paediatrician, Physician in Paediatric Audiology) advised on the survey of current practice and provided a direct link with the previous recent study of current practice. Adrian Davis (Professor, Director of NHSP, Epidemiologist) advised on all aspects of the research and provided a direct link with the newborn hearing screening programme. Sally Hind (Senior MRC Scientist, Developmental Psychologist) advised on the survey of current practice. All authors contributed to writing and editing.

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References 1.

Ewing A. The sweep frequency method of making screening tests of the hearing of school children. BMJ 1955;4904:41–2.

Court SDM. Fit for the Future. The Report of the Committee on Child Health Services. London: HMSO; 1976.

14.

Bess FH, Dodd-Murphy J, Parker RA. Children with minimal sensorineural hearing loss: prevalence, education performance and functional status. Ear Hear 1998;19:339–54.

15.

Haggard MP, Hughes E. Screening children’s hearing. London: HMSO; 1991.

16.

Zielhuis GA, Straatman H, Rach GH, van den Broek P. Analysis and presentation of data on the natural course of otitis media with effusion in children. Int J Epidemiol 1990;19:1037–44.

Stewart-Brown S, Haslum MN. Screening for hearing loss in childhood: a study of national practice. BMJ 1987;6584:1386–8.

Haggard M. Research and development of effective services for hearing-impaired people. London: Nuffield Provincial Hospitals Trust; 1993.

17.

Davis A, Bamford J, Wilson I, Ramkalawan T, Forshaw M, Wright S. A critical review of the role of neonatal hearing screening in the detection of congenital hearing impairment. Health Technol Assess 1997;1(10).

Midgley EJ, Dewey C, Pryce K, Maw AR. The frequency of otitis media with effusion in British pre-school children: a guide for treatment. Clin Otolaryngol 2000;25:485–91.

18.

Butler CC, van der Linden MK, MacMillan HL, van der Wouden JC. Should children be screened to undergo early treatment for otitis media with effusion? A systematic review of randomized trials. Child Care Health Dev 2003;29:425–32.

19.

Polnay L. Health needs of school age children: report of a joint working party. London: British Paediatric Association; 1995.

20.

Henderson RE, Newton N. Is a universal school entry hearing screen worthwhile? Ambulatory Child Health 2000;6:247–52.

Fonseca S, Forsyth H, Neary W. School hearing programme in the UK: practice and performance. Arch Dis Child 2005;90:154–64.

Hall D, editor. Health for all children. 4th ed. Oxford: Oxford University Press; 2003.

McCormick B. Screening for hearing impairment in young children. London: Croom Helm; 1988.

Bamford J, Uus K, Davis A. Screening for hearing loss in childhood: issues, evidence and current approaches in the UK. J Med Screen 2005; 3:119–24.

21.

Tharpe AM, Bess FH. Minimal, progressive and fluctuating hearing loss in children: characteristics, identification and management. Pediatr Clin N Am 1999;46:65–78.

10.

Bamford J, Ankjell H, Crockett R, Marteau T, McCracken W, Parker D, et al. Evaluation of the newborn hearing screening programme (NHSP) in England: Report of the evaluation of the first phase of implementation of the NHSP. 2006; National Library for Health.

22.

Smith WR, Pither RE. Long term follow-up of children with mild hearing impairment pre and post intervention. J BATOD 1993;17(4):99–103.

23.

Directory of Community Nursing 2004/2005. Chichester: Professional, Managerial and Healthcare Publications; 2004.

24.

Independent Schools Council. URL: www.isc.co.uk. Accessed May 2006.

25.

Bess FH, Klee T, Cuthbertson JL. Identification assessment and management of children with unilateral sensorineural hearing loss. Ear Hear 1986;7:43–5.

26.

Watkin PM. Neonatal otoacoustic emission screening and the identification of deafness. Arch Dis Child Fetal Neonatal Ed 1996;74:F16–25.

27.

Watkin PM. Outcomes of neonatal screening for hearing loss by otoacoustic emission. Arch Dis Child Fetal Neonatal Ed 1996;75:F158–68.

11.

Yoshinga-Itano C, Sedley A, Coulter DK, Mehl AL. Language of early- and later-identified children with hearing loss. Pediatrics 1998;102:1161–71.

12.

Fortnum HM, Summerfield AQ, Marshall DH, Davis AC, Bamford JM. Prevalence of permanent childhood hearing impairment in the United Kingdom and implications for universal neonatal hearing screening: questionnaire based ascertainment study. BMJ 2001;323:536–40.

13.

Fortnum HM. Epidemiology of permanent childhood hearing impairment: implications for neonatal hearing screening. J Audiol Med 2003;1:155-64.

References

28.

Watkin PM, Baldwin M. Confirmation of deafness in infancy. Arch Dis Child 1999;81:380–9.

29.

Kennedy C, McCann D, Campbell MJ, Law C, Mullee M, Petrou S, et al. Language ability after early detection of permanent childhood hearing impairment. N Engl J Med 2006; 353:2131–41.

30.

31.

Watkin PM, McCann D, Law C, Petrou S, Stevenson J, Worsford S, et al. Language ability in children with permanent hearing impairment: the influence of early management and family participation. Pediatrics 2007; in press. Watkin PM, Baldwin M, Laoide S. Parental suspicion and identification of hearing impairment. Arch Dis Child 1990;65:846–50.

43.

Barlow J, Stewart-Brown S, Fletcher J. Systematic review of the school entry medical examination. [review]. Arch Dis Child 1998;78:301–11.

44.

New Zealand Health Technology Assessment. Screening programmes for the detection of otitis media with effusion and conductive hearing loss in pre-school and new entrant school children: a critical appraisal of the literature [structured abstract]. NZ Health Technol Assess 1998;3(61).

45.

Zielhuis GA, Rach GH, van der Broek P. Screening for otitis media with effusion in preschool children. Lancet 1989;i:311–14.

46.

Feldman W, Sackett B, Milner RA, Gilbert S. Effects of preschool screening for vision and hearing on prevalence of vision and hearing problems 6–12 months later. Lancet 1980;ii:1014–16.

47.

Pirozzo S, Papinczak T, Glasziou P. Whispered voice test for screening for hearing impairment in adults and children: systematic review. BMJ 2003;327:967.

32.

Watkin PM. The age of identification of childhood deafness – improvements since the 1970s. Public Health 1991;105:303–12.

33.

Watkin PM, Baldwin M, McEnery G. Neonatal at risk screening and the identification of deafness. Arch Dis Child 1991;66:1130–5.

34.

Watkin PM. Controlling the quality of universal neonatal hearing screens. Public Health 1999;113:171–6.

48.

35.

Watkin P, Hasan J, Baldwin M, Ahmed M. Neonatal hearing screening: have we taken the right road? Results from a 10-year targeted screen longitudinally followed up in a single district. Audiol Med 2005;3:175–84.

Groen JJ. Pure tone audiometry and whispered voice test. Conformities and differences in test results. ORL J Otorhinolaryngol Relat Spec 1973; 35:65–70.

49.

Dempster JH, Mackenzie K. Clinical role of freefield voice tests in children. Clin Otolaryngol 1992;17:54–6.

50.

Prescott CA, Omoding SS, Fermor J, Ogilvy D. An evaluation of the ‘voice test’ as a method for assessing hearing in children with particular reference to the situation in developing countries. Int J Pediatr Otorhinolaryngol 1999;51:165–70.

51.

Olusanya B. Early detection of hearing impairment in a developing country: what options? Audiology 2001;40:141–7.

52.

Pang-Ching G, Robb M, Heath R, Takumi M. Middle ear disorders and hearing loss in native Hawaiian preschoolers. Language 1995;26:33–8.

53.

Lyons A, Kei J, Driscoll C. Distortion product otoacoustic emissions in children at school entry: a comparison with pure-tone screening and tympanometry results. J Am Acad Audiol 2004;15:702–15.

54.

Sabo MP, Winston R, Macias JD. Comparison of pure tone and transient otoacoustic emissions screening in a grade school population. Am J Otolaryngol 2000;21:88–91.

36.

Sutton G, Rowe S. Risk factors for childhood deafness in the Oxford region. Br J Audiol 1997;31:39–54.

37.

Peckham CS, Stark O, Dudgeon JA, Martin JA, Hawkins G. Congenital cytomegalovirus infection: a cause of sensorineural hearing loss. Arch Dis Child 1987;62:1233–7.

38.

UK National Screening Committee. Criteria for appraising the viability effectiveness and appropriateness of a screening programme. URL: http://www.nsc.nhs.uk/ pdfs/criteria.pdf. Accessed May 2006.

39.

Sackett DL, Haynes RB, Tugwell P, Guyatt GH. Clinical epidemiology: a basic science for clinical medicine. Philadelphia, PA: Lippincott Williams & Wilkins; 1991.

40.

Whiting P, Rutjies AW, Dinnes J, Reitsma J, Bossuyt PM, Kleijnen J. Development and validation of methods for assessing the quality of diagnostic accuracy studies. Health Technol Assess 2004;8(25).

41.

Oxman AD. User’s guide to the medical literature. VI How to use an overview. JAMA 1994;272:1367–71.

55.

FitzZaland RE, Zink GD. A comparative study of hearing screening procedures. Ear Hear 1984; 5:205–10.

42.

Whiting P. The development of QUADAS: a tool for the quality assessment of studies of diagnostic accuracy included in systematic reviews. BMC Med Res Methodol 2003;3:25.

56.

Rodriguez MA, Melguizo-Yepez M. [Screening for hearing disorders in school children of Medellin, Colombia, 1993]. [Spanish]. Salud Publica Mex 1994;36:670–2.

Health Technology Assessment 2007; Vol. 11: No. 32

71.

Hamill B. Comparing two methods of preschool and kindergarten hearing screening. J Sch Health 1988;58:95–7.

McCurdy JA, Goldstein JL, Gorski D. Auditory screening of preschool-children with impedance audiometry – comparison with pure-tone audiometry – detecting otologic disease prior to onset of hearing-loss. Clin Pediatr 1976;15:436–41.

72.

Roush J, Drake A, Sexton JE. Identification of middle ear dysfunction in young children: a comparison of tympanometric screening procedures. Ear Hear 1992;13:63–9.

Nozza RJ, Sabo DL, Mandel EM. A role for otoacoustic emissions in screening for hearing impairment and middle ear disorders in schoolage children. Ear Hear 1997;18:227–39.

73.

Square R, et al. An alternative to impedance screening: unoccluded frontal bone conduction screening. Journal of Childhood Communication Disorders 1985;8:123–30.

Orlando MS, Frank T. Audiometer and AudioScope hearing screening compared with threshold test in young children. J Pediatr 1987;110:261–3.

74.

75.

Skurr BA, Jones DL. Testing the hearing level of schoolchildren: comparison of several currently used screening techniques. Med J Aust 1981; 2:248–9.

76.

Holtby I, Forster DP, Kumar U. Pure tone audiometry and impedance screening of school entrant children by nurses: evaluation in a practical setting. J Epidemiol Community Health 1997;51:711–15.

57.

Ritchie BC, Merklein RA. An evaluation of the efficiency of the Verbal Auditory Screening for Children (VASC). J Speech Hear Res 1972;15:280–6.

58.

59.

60.

61.

Nienhuys TG, Boswell JB, McConnel FB. Middle ear measures as predictors of hearing loss in Australian aboriginal schoolchildren. Int J Pediatr Otorhinolaryngol 1994;30:15–27.

62.

Roush J, Tait CA. Pure-tone and acoustic immittance screening of preschool-aged children: an examination of referral criteria. Ear Hear 1985;6:245–50.

63.

Hammond PD, Gold MS, Wigg NR, Volkmer RE. Preschool hearing screening: evaluation of a parental questionnaire. J Paediatr Child Health 1997;33:528–30.

64.

Schell NB. Report on Nassau County Pilot study on preschool screening program for hearing. New York State J Med 1970;70:55.

77.

National Institute for Clinical Excellence. Guide to the methods of technology appraisal No. 515. London: National Institute for Clinical Excellence; 2004.

65.

Hind SE, Atkins RL, Haggard MP, Brady D, Grinham G. Alternatives in screening at school entry: comparison of the Childhood Middle Ear Disease and Hearing Questionnaire (CMEDHQ) and the pure tone sweep test. Br J Audiol 1999;33:403–14.

78.

Vanoli A, Drummond M. Improving access to costeffectiveness information for health care decision making: the NHS Economic Evaluation Database. CRD Report No. 6. 2nd ed. York: Centre for Review and Dissemination; 2001.

79.

66.

Maragno C, Teatini GP. [Measurement of the efficacy of the Rimondini–Rossi test for audiometric screening] [Italian]. Acta Otorhinolaryngol Ital 1983;3:483–90.

Gold MR, Siegel JE, Russell LB, Weinstein MC. Cost effectiveness in health and medicine. New York: Oxford University Press; 1996.

80.

Ahmed AU, Curley JWA, Newton VE, Mukherjee D. Hearing aids versus ventilation tubes in persistent otitis media with effusion: a survey of clinical practice. J Laryngol Otol 2001;115:274–9.

81.

Mills RP, McKerrow WS. Management of childhood otitis media with effusion by Scottish otolaryngologists. J R Coll Surg Edinb 2000;45:321–5.

82.

Niskar AS, Kieszak SM, Holmes A, Esteban E, Rubin C, Brody DJ. Prevalence of hearing loss among children 6 to 19 years of age: the third national health and nutrition examination survey. JAMA 1998;279:1071–5.

83.

Curtis L, Netten A. Unit costs of health and social care. Canterbury: Personal Social Services Research Unit; 2005.

84.

UK Cochlear Implant Study Group. UK Cochlear Implant Study Group Criteria for candidacy for

67.

British Society of Audiology. Descriptors for pure tone audiograms. Br J Audiol 1988;22:123.

68.

Abou Haidar L, Blond MH, Chautemps D, Ployet MJ, Lescanne E. [‘Audio 4’: a simple and quick speech audiometry test for moderate hearing loss screening in four-year-old children] [French]. Arch Pediatr 2005;12:264–72.

69.

Gomes M, Lichtig I. Evaluation of the use of a questionnaire by non-specialists to detect hearing loss in preschool Brazilian children. Int J Rehabil Res 2005;28:171–4.

70.

Holtby I, Forster DP, Kumar U. Pure tone audiometry and impedance screening of school entrant children by nurses: evaluation in a practical setting. J Epidemiol Community Health 1997;51:711–15.

References

unilateral cochlear implantation in postlingually deafened adults. II: Cost effectiveness analysis. Ear Hear 2004;25:336–60. 85.

Dolan P, Gudex C, Kind P, William A. A social tariff for EuroQuol: results from a UK general population survey. Centre for Health Economics Discussion Paper 138. 1995; York: University of York.

86.

Barton GR, Bankart J, Davis AC, Summerfield QA. Comparing utility scores before and after hearing aid provision. Appl Health Econ Health Policy 2004;3:103–5.

87.

Drummond M, O’Brien B, Stoddart GL, Torrance GW. Methods for economic evaluation of health care programmes. Oxford: Oxford University Press; 1997.

88.

Driscoll C, Kei J, McPherson B. Transient evoked otoacoustic emissions in 6-year-old school children: a normative study. Scand Audiol 2000;29:103–10.

89.

Holtby I, Forster DP. Evaluation of pure tone audiometry and impedance screening in infant schoolchildren. J Epidemiol Community Health 1992;46:21–5.

90.

91.

Fria TJ. Hearing acuity of children with otitis media with effusion. Arch Otolaryngol 1985;111:10–16.

92.

NHS. Agenda for change: NHS terms and conditions of service handbook. January 2005. URL: www.nhsemployers.org/pay-conditions/payconditions-707.cfm. Accessed April 2006.

93.

Butler CC, van-der-Linden MK, MacMillan H, van der Wouden JC. Screening children in the first four years of life to undergo early treatment for otitis media with effusion. Cochrane Database of Systematic Reviews (Issue 2). Chichester: John Wiley & Sons; 2003.

Department for Education and Skills. Statistics of education. 2005. URL: http//:www.dfes.gov.uk. Accessed April 2006.

94.

Department for Education and Skills. Children in need in England: results of a survey of activity and expenditure as reported by local authority social services' children and families teams for a survey week in February 2005. National Statistics 2005;(vweb 01-2005). http://www.dfes.gov.uk/ rsgateway/DB/VOL/v000647/vweb02-2006.pdf. Accessed May 2006.

95.

Department of Health. NHS Reference Costs 2003 and National Tariff 2004. URL: http://www.dh.gov.uk/PolicyAndGuidance/ OrganisationPolicy/FinanceAndPlanning/ NHSReferenceCosts/fs/en. Accessed April 2006.

96.

Rawlins MD, Culyer AJ. National Institute for Clinical Excellence and its value judgments. BMJ 2006;329:224–7.

97.

Rosen J, Johnson C, Wilkinson H. School-entry hearing screening: an audit of referrals in a three year period. Aust NZ J Audiol 2004;26: 142–7.

98.

Rovers MM, Black N, Browning GG, Maw R, Zielhuis GA, Haggard MP. Grommets in otitis media with effusion: an individual patient data meta-analysis. Arch Dis Child 2005;90:480–5

99.

MRC Multi-centre Otitis Media Study Group. Risk factors for persistence of bilateral otitis media with effusion. Clin Otolaryngol 2001;26:147–56

100. Rovers MM, Straatman H, Ingels K, van der Wilt GJ, van den Broek P, Zielhuis GA. The effect of ventilation tubes on language development in infants with otitis media with effusion: a randomised trial. Pediatrics 2000; 106:E42. 101. Paradise JL, Dollaghan CA, Campbell TF, Feldman HM, Bernard BS, Colborn DK, et al. Otitis media and tympanostomy tube insertion during the first three years of life: developmental outcomes at the age of four years. Pediatrics 2003;112:265–77.

Health Technology Assessment 2007; Vol. 11: No. 32

Appendix 1 Screening at school entry for childhood hearing impairment: an appraisal against National Screening Committee criteria Screening for permanent childhood hearing impairment (June 2006) Criteria

Supporting evidence

The condition 1. The condition should be an important public health problem

Bilateral PCHI can have a devastating impact on communication skills (Conrad, 1979), educational attainment (Wood et al., 1986), and quality of life (Gregory, 1995; Cheng et al., 2000), with a high cost to society (Mohr et al., 2000). Unilateral hearing impairment would be expected to affect auditory perception in various predictable ways (e.g. poor localisation of sound sources, difficulty in noisy or reverberant environments such as schools), and there is some evidence of detrimental effects on academic progress (e.g. Bess et al., 1998) Authors’ summary opinion: satisfied

2. (i)

The epidemiology of the condition should be known (ii) The natural history of the condition should be understood (iii) There should be a recognised latent period or early symptomatic stage

No national register of hearing-impaired children exists for the UK, and accurate estimates of the prevalence of PCHI and of its profile across all ages and all degrees of impairment are unavailable PCHI of a moderate degree or greater (i.e. detection thresholds >40 dB HL averaged across 0.5, 1, 2 and 4 kHz) is present at birth at a rate of about 1.6 per 1000 live births, of which approximately 1.0 in 1000 are bilateral and 0.6 in 1000 are unilateral impairments (Davis et al., 1997; Bamford et al., 2006). In terms of incidence, this means that in the UK about 800 children per year will be born with permanent bilateral hearing impairment of a moderate or greater degree, and about 500 per year will be born with unilateral hearing impairment (i.e. hearing within normal limits in one ear, hearing impairment of moderate or greater degree in the other ear) There is good evidence that the prevalence of permanent bilateral moderate or greater hearing impairment increases through the first decade of childhood (Fortnum et al., 2001; Fortnum, 2003). It is possible that the prevalence of bilateral moderate or greater impairment reaches 2 in 1000 by the age of about 9 years. If permanent mild hearing impairments are included, evidence from retrospective ascertainment studies in this review suggests that by school entry the prevalence is around 3.5 in 1000. There are four situations where children with permanent hearing impairment may not be identified by a screening test within a few days of birth: ●

●

Some children will have no impairment at birth, but will acquire the impairment later in their life as a result of some traumatic event such as infection (usually bacterial meningitis, Fortnum, 1992), head injury (Zimmerman et al., 1993), ototoxic therapy (Casano et al., 1999) or chemotherapy (Littman et al., 1998; Berg et al., 1999). Some children may have an impairment at birth, but of a severity insufficient to be detected by the newborn screening procedures. As the child grows this mild impairment may represent a significant disabling condition in itself, or the impairment may progress to a greater severity (Hayes and Dreith, 2000). The causes of progressive impairments include hereditary hearing loss and syndromal associations such as Alport, Waardenburg type II and Alström (Gorlin et al., 1995; Zwirner and Wilichowski, 2001), infectious diseases (Williamson et al., 1992), anatomical malformations (Zalzal et al., 1995; Shetty et al., 1997), perinatal events and treatments (Fujiwaka et al., 1995; Lasky et al., 1998) and ototoxic drugs (Pasic and Dobie, 1991; Borradori et al., 1997). continued

Appendix 1

Criteria

Supporting evidence ●

●

Some children develop genuine late-onset impairment that develops with no obvious causative factor and hence is not truly acquired (Parker, 1999). A fourth group of children contributing to those who may not be identified at birth are those who should undergo the screen but who do not. Reasons for this at the time of screening include early discharge and/or parental refusal, but children who migrate into an area or country implementing a neonatal screening programme from an area/country which does not, also fall into this group.

Children in all four of these categories comprise those who require identification postneonatal screen and who will need some form of follow-up to be established The evidence on permanent unilateral hearing loss is more limited. Although it appears from NHSP data that the prevalence at birth is about 0.6 in 1000, it is not known whether there are significant numbers of later onset cases, whether some of the losses are progressive, and whether there is a tendency for congenital or postnatal unilateral hearing loss to progress to bilateral loss PCHI of whatever cause does not improve. It may remain stable or worsen (progressive) There is no latent period or early symptomatic stage in PCHI Authors’ summary opinion: satisfied 3. All cost-effective primary prevention interventions should have been implemented as far as practicable

Primary prevention includes immunisation for conditions that are known to cause permanent hearing impairment, both prenatally and postnatally (e.g. rubella, mumps, meningitis); reduction in the use of, and monitoring of levels of, ototoxic antibiotics such as gentamycin in the neonatal period; and genetic counselling for people with affected children or at higher risk of having an affected child Authors’ summary opinion: satisfied

The test 4. There should be a simple, safe, precise and validated screening test

The procedures for the SES vary in their implementation, but all are relatively simple. There is no known danger to the child or to the screener in performing the test. Limited quality evidence suggests that the test has high sensitivity and specificity for full PTA (Orlando and Frank, 1987; FitzZaland and Zink, 1984; Holtby et al.. 1997; Sabo et al., 2000) There has until recently been a widespread if implicit consensus that “the pure tone sweep test has value educationally and as a safety net to catch any deficiencies of the earlier screening system in the overall public health provision” (Haggard, 1993), a position broadly endorsed by Hall (2003) The review of current practice indicated that all but 12.2% of respondents operate a universal school entry screen and use the pure tone sweep test; however, the protocols used are unacceptably variable Authors’ summary opinion: satisfied

5. The distribution of test values in the population should be known and a suitable cut-off level defined and agreed

There are no published data on population values for pure tone sweep audiometry. A cutoff level has not been defined and agreed and varies across the national provision Population data on pure tone audiometric levels are not available for children of school entry age; adult norms are used. This may have marginal effects on case identification of mild and minimal hearing impairments, but not on moderate and greater impairments Authors’ summary opinion: not satisfied

6. The test should be acceptable to the population

The pure tone sweep test and PTA are well-established tests and appear to be acceptable to the population (children) and their parents, although no data have been published that address this issue Authors’ summary opinion: satisfied

continued

Health Technology Assessment 2007; Vol. 11: No. 32

Criteria

Supporting evidence

7. If the test is for mutations the criteria used to select subset of mutations to be covered by screening should be clearly set out

Although hearing impairment may be caused by inherited or novel mutations, this screening test is not designed to identify them. Further diagnostic evaluations may include selective mutation screening/testing

8. There should be an agreed policy on the further diagnostic investigation of individuals with a positive test and on the choices available to those individuals

In 1976 the Court Report recommended that hearing screens be carried out at least twice in school (Court, 1976). However, there was no nationally agreed protocol for the screen, and implementation thus varied in small but possibly important details across services

Authors’ summary opinion: not relevant

Guidelines on diagnostic investigation and subsequent treatment choices have been developed by the British Society of Audiology in collaboration with the National Deaf Children’s Society (NDCS, 2006). For moderate and greater bilateral hearing impairments in school-age children, diagnostic procedures are well established and reliable when performed by trained paediatric audiologists. Intervention options include amplification (hearing aids), communication advice, educational support and social care support. For mild and unilateral hearing impairment, diagnostic procedures are more challenging, especially if transient middle ear conditions are also present, and require good-quality paediatric audiology services and paediatric audiologists, of which there is a national shortage. Intervention options are similar to those with more severe impairments, but evidence on the cost-effectiveness of these options is largely missing Authors’ summary opinion: partially satisfied

The treatment 9. There should be an effective treatment or intervention for patients identified through early detection

It has long been believed that earlier identification of hearing impairment must lead to better outcomes, and there is now reliable evidence that this is so in the domains of communication, educational achievement and quality of life (Davis et al., 1997; YoshinagoItano et al., 2000; Moeller, 2000). Few people now disagree with the statement that identification of congenital impairments in the first few months of life and consequent habilitation is desirable. For late-onset, progressive and otherwise not previously known impairments identified at school entry age, evidence on the effectiveness of the interventions (provision of hearing aids and regular follow-up with appropriate rehabilitative support particularly in education) is absent. It is reasonable to assume that the intervention will be effective for moderate and greater bilateral impairments; more evidence is required about intervention for mild, minimal and unilateral impairments. However, since these could be at risk for worsening impairment, identification and monitoring is arguably desirable as a minimum Authors’ summary opinion: satisfied

10. There should be agreed evidence-based policies covering which individuals should be offered treatment and the appropriate treatment to be offered

There is still debate over the lower level of hearing impairment for which provision of hearing aids is beneficial. More evidence is required with respect to interventions for mild, minimal and unilateral impairments identified at school age. The evidence for the type and extent of intervention for children with moderate and greater bilateral impairments is relatively clear

11. Clinical management of the condition and patient outcomes should be optimised by all healthcare providers prior to participation in a screening programme

There is evidence from the NHSP (www.nhsp.info) that the quality of paediatric audiology services in the UK is unacceptably variable. This is likely to be a resource and training issue and is receiving attention

Authors’ summary opinion: partially satisfied

continued

Appendix 1

Criteria

Supporting evidence

12. There should be evidence from high-quality RCTs that the screening programme is effective in reducing mortality and morbidity

There is no evidence from high-quality RCTs that the screening programme is effective in reducing morbidity

13. There should be evidence that the complete screening programme (test, diagnostic procedures, treatment/intervention) is clinically, socially and ethically acceptable to health professionals and the public

Screening and intervention for childhood hearing impairment are clinically, socially and ethically acceptable to all health professionals concerned with the issue and to most of the public. The majority of health professionals who took part in the survey of current practice were strongly in favour of the SES. There is an important minority of the Deaf community who subscribe to a social/cultural model of deafness and who do not support some of the ‘corrective’ interventions for severe/profound hearing impairment; however, this does not apply to the mild/moderate levels detected by the SES

14. The benefit from the screening programme should outweigh the physical and psychological harm (caused by the test, diagnostic procedure and treatment)

The extent of the beneficial effects of early identification for children with permanent hearing impairment on developmental outcomes in general and communication in particular has been demonstrated for young preschool infants (e.g. Yoshinaga-Itano et al., 1998); there is no evidence of either benefit or harm associated with the SES

15. The opportunity cost of the screening programme (including testing, diagnosis and treatment) should be economically balanced in relation to expenditure on medical care as a whole

There is no good-quality published evidence of the costs and effectiveness of the screen. The cost-effectiveness modelling carried out suggested that each screen costs £8, and that a universal school entry screen based largely or completely on pure tone sweep tests was associated with higher costs and slightly higher QALYs compared with no screen and other screen alternatives; the ICER for such a screen is around £2500 per QALY gained; the range of expected costs, QALYs and net benefits was broad, indicating a considerable degree of uncertainty

Authors’ summary opinion: not satisfied

Authors’ summary opinion: satisfied

Authors’ summary opinion: partially satisfied

Authors’ summary opinion: partially satisfied 16. There should be a plan for monitoring and managing the screening programme and an agreed set of quality assurance standards

Several reviews (Stewart-Brown and Haslum, 1987; Haggard and Hughes, 1991; Davis et al., 1997; Fonseca et al., 2005) have recommended monitoring and management strategies for the SES; the evidence from the survey of current practice suggests that none has been widely implemented, and that little has changed since the review of Stewart-Brown and Haslum. There is no national protocol or quality assurance plan Authors’ summary opinion: not satisfied

17. Adequate staffing facilities for testing, diagnosis, treatment and programme management should be available prior to the commencement of the screening programme

18. All other options for managing the condition should have been considered (e.g. improving treatment, providing other services)

Suggestions for universal screens for hearing impairment at different ages have been made and implemented. The most important is the universal newborn hearing screen, fully implemented in England as the NHSP with effect from March 2006

Authors’ summary opinion: partially satisfied

Interventions have remained stable in recent years Authors’ summary opinion: satisfied

Health Technology Assessment 2007; Vol. 11: No. 32

Bamford J, Ankjell H, Crockett R, Marteau T, McCracken W, Parker D, et al. Evaluation of the newborn hearing screening programme (NHSP) in England. 2006 URL: http://libraries.nelh.nhs.uk/screening/ Berg AL, Spitzer JB, Garvin JH Jr. Ototoxic impact of cisplatin in pediatric oncology patients. Laryngoscope 1999;109:1806–14. Bess FH, Dodd-Murphy J, Parker RA. Children with minimal sensorineural hearing loss: prevalence, education performance and functional status. Ear Hear 1998,19:339–54. Borradori C, Fawer C-L, Buclin T, Calame A. Risk factors for sensorineural hearing loss in preterm infants. Biol Neonate 1997;71:1–10. British Society of Audiology. Descriptors for pure-tone audiograms. Br J Audiol 1988;22:123. Casano RA, Johnson DF, Bykhovskaya Y, Torricelli F, Bigozzi M, Fischel-Ghodsian N. Inherited susceptibility to aminoglycoside ototoxicity: genetic heterogeneity and clinical implications. Am J Otol 1999;20:151–6. Cheng AK, Rubin HR, Powe NR, Mellon NK, Francis HW, Niparko JK. Cost–utility analysis of the cochlear implant in children. JAMA 2000;284:850–6. Conrad R. The deaf schoolchild: language and cognitive function. London: Harper and Row; 1979. Court, SDM, editor. Fit for the Future. The Report of the Committee on Child Health Services. London: HMSO; 1976. Davis A, Bamford J, Wilson I, Ramkalawan T, Forshaw M, Wright S. A critical review of the role of neonatal hearing screening in the detection of congenital hearing impairment. Health Technol Assess 1997;1(10). FitzZaland RE, Zink GD. A comparative study of hearing screening procedures. Ear Hear 1984;5:205–10. Fonseca S, Forsyth H, Neary W. School hearing programme in the UK: practice and performance. Arch Dis Child 2005,90:154–6. Fortnum HM. Hearing impairment after bacterial meningitis: a review. Arch Dis Child 1992;67:1128–33. Fortnum HM. Epidemiology of permanent childhood hearing impairment: implications for neonatal hearing screening J Audiol Med 2003,1:155–64. Fortnum HM, Summerfield AQ, Marshall DH, Davis AC, Bamford JM. Prevalence of permanent childhood hearing impairment in the UK and implications for universal neonatal hearing screening: questionnaire based ascertainment study. BMJ, 2001;323:536–40.

Gregory S. Deaf children and their families. Cambridge: Cambridge University Press; 1995. Haggard M, Research in the development of effective services for hearing-impaired people. London: Nuffield Provincial Hospitals Trust; 1993. Haggard MP, Hughes E. Screening children’s hearing. London: HMSO; 1991. Hall D, editor. Health for all children. 4th ed. Oxford: Oxford University Press; 2003. Hayes D, Dreith S. Catastrophic progressive hearing loss in childhood. J Am Acad Audiol 2000;11:300–8. Holtby I, Forster DP, Kumar U. Pure tone audiometry and impedance screening of school entrant children by nurses: evaluation in a practical setting. J Epidemiol Community Health 1997;51:711–15. Lasky RE, Wiorek L, Becker TR. Hearing loss in survivors of neonatal extracorporeal membrane oxygenation (ECMO) therapy and high-frequency oscillatory (HFO) therapy. J Am Acad Audiol 1998;9:47–58. Littman TA, Magruder A, Strother DR. Monitoring and predicting ototoxic damage using distortion-product otoacoustic emissions: pediatric case study. J Am Acad Audiol 1998;9:257–62. Moeller MP. Early intervention and language development in children who are deaf and hard of hearing. Pediatrics 2000;106:E43. Mohr PE, Feldman JJ, Dunbar JL, McConkey-Robbins A, Niparko JK, Rittenhouse RK, et al. The societal costs of severe to profound hearing loss in the United States. Int J Technol Assess 2000;16:1120–35. NDCS. Quality standards in paediatric audiology, 2006, Vol. 3. URL: http://www.ndcs.org.uk/information/ professional_focus/professional_publications/health_prof essionals/quality_3.html Orlando MS, Frank T. Audiometer and AudioScope hearing screening compared with threshold test in young children. J Pediatr 1987;110:261–3. Parker G. Children with significant hearing loss who pass neonatal hearing screening [MSc thesis]. Manchester: University of Manchester; 1999. Pasic TR, Dobie RA. Cis-platinum ototoxicity in children. Laryngoscope 1991;101:985–91. Sabo MP, Winston R, Macias JD. Comparison of pure tone and transient otoacoustic emissions screening in a grade school population. Am J Otol 2000;21:88–91.

Fujikawa S, Yang L, Waffern F, Lerner M. Persistent pulmonary hypertension of the newborn (PPHN) treated with inhaled nitric oxide: preliminary hearing outcomes. J Am Acad Audiol 1995;8:263–8.

Shetty PG, Shroff MM, Kirtane MV, Karmarker SS. Cerebrospinal fluid otorhinorrhea in patients with defects through the lamina cribosa of the internal auditory canal. Am J Neuroradiol 1997;18:478–81.

Gorlin RJ, Toriello HV, Cohen MM Jr. Hereditary hearing loss and its syndromes. New York: Oxford University Press; 1995.

Stewart-Brown S, Haslum MN, Screening for hearing loss in childhood: a study of national practice. BMJ 1987;294:1386–8.

Appendix 1

Williamson WD, Demmler GJ, Percy AK, Catlin FI. Progressive hearing loss in infants with asymptomatic congenital cytomegalovirus infection. Pediatrics 1992;90: 862–6. Wood D, Wood H, Griffiths A, Howarth I. Teaching and talking with deaf children. Chichester: John Wiley and Sons; 1986. Yoshinago-Itano C, Coulter D, Thomson V. The Colorado newborn hearing screening project: effects on speech and language development for children with hearing loss. J Perinatol 2000;20:S132–7. Yoshinaga-Itano C, Sedey AL, Coulter DK. Language of

early- and later-identified children with hearing loss. Pediatrics 1998;102:1161–71. Zalzal GH, Tomaski SM, Vezina LG, Bjornsti P, Grundfast KM. Enlarged vestibular aqueduct and sensorineural hearing loss in childhood. Arch Otolaryngol Head Neck Surg 1995;121:2–28. Zimmerman WD, Ganzel TM, Windmill IM, Nazar GB, Phillips M. Peripheral hearing loss following head trauma in children. Laryngoscope 1993;103:87–91. Zwirner P, Wilichowski E. Progressive sensorineural hearing loss in children with mitochondrial encephalomyopathies. Laryngoscope 2001;111:515–21.

Screening for temporary childhood hearing impairment (June 2006) Criteria

Supporting evidence

The condition 1. The condition should be an important public health problem

Temporary hearing impairment associated with middle ear fluid (OME, sometimes known as glue ear) is a common condition in childhood, particularly up to about 8 years of age (Casselbrant and Mandel, 2003). The hearing loss may be unilateral or more commonly bilateral, minimal, mild or occasionally moderate in degree; evidence for long-term effects is sparse Authors’ summary opinion: partially satisfied

2. (i)

The epidemiology of the condition should be known (ii) The natural history of the condition should be understood (iii) There should be a recognised latent period or early symptomatic stage

The period prevalence (0–8 years) for OME is around 80% (Casselbrant and Mandel, 2003), while the point prevalence may be as high as 20% at 2 and 4 years of age (Zielhuis et al., 1990). Risk factors include socio-economic group, passive smoking, bottle feeding, upper respiratory tract infections, craniofacial anomalies and time on the neonatal intensive care unit at birth (Casselbrant and Mandel, 2003) The natural history of the condition is only partly understood. Spontaneous remission is common, with no long-term effects. About 5% of cases exhibit severity of the hearing impairment and persistence/recurrence of the condition sufficient to cause concern Hearing impairment is a major symptom, caused by middle ear fluid impeding the passage of acoustic energy from outer to inner ear. The degree of impairment varies with the presence and viscosity of the fluid Authors’ summary opinion: satisfied

3. All cost-effective primary prevention interventions should have been implemented as far as practicable

Public health initiatives for children and families with young infants are likely to have an important effect on the condition Authors’ summary opinion: satisfied

The test 4. There should be a simple, safe, precise and validated screening test

The procedures for the SES vary in their implementation, but all are relatively simple. There is no known danger to the child or to the screener in performing the test. Limited quality evidence suggests that the test has high sensitivity and specificity for full PTA (Orlando and Frank, 1987; FitzZaland and Zink;1984, Holtby et al., 1997; Sabo et al., 2000) There has until recently been a widespread if implicit consensus that “the pure tone sweep test has value educationally and as a safety net to catch any deficiencies of the earlier screening system in the overall public health provision” (Haggard, 1993), a position broadly endorsed by Hall (2003)

continued

Health Technology Assessment 2007; Vol. 11: No. 32

Criteria

Supporting evidence The review of current practice indicated that all but 12.2% of respondents operate a universal school entry screen and use the pure tone sweep test; however, the protocols used are unacceptably variable Authors’ summary opinion: satisfied

5. The distribution of test values in the population should be known and a suitable cut-off level defined and agreed

There are no published data on population values for pure tone sweep audiometry. A cutoff level has not been defined and agreed and varies across the national provision. Evidence on a suitable cut-off level for severity of hearing impairment associated with OME is emerging from the TARGET trial data (Haggard M, University of Cambridge: personal communication, 2006), but the requirement of persistence as a marker of potential to benefit requires repeat tests (screen or follow-up) Population data on pure tone audiometric levels are not available for children of school entry age; adult norms are used. This may affect case identification of mild and minimal hearing impairments Authors’ summary opinion: not satisfied

6. The test should be acceptable to the population

7. If the test is for mutations the criteria used to select subset of mutations to be covered by screening should be clearly set out

Authors’ summary opinion: not relevant

8. There should be an agreed policy on the further diagnostic investigation of individuals with a positive test and on the choices available to those individuals

Further diagnostic investigation to confirm hearing impairment and middle ear fluid involves air and bone conduction PTA and acoustic impedance measures, with otoscopy and ENT examination; these are standard procedures. Authors’ summary opinion: satisfied

The treatment 9. There should be an effective treatment or intervention for patients identified through early detection

The main treatment option is surgery to drain the fluid and insert ventilation tubes (grommets), possibly with adjuvant adenoidectomy. This restores hearing to normal, but there is no evidence for longer term benefits in marginal cases (Rovers et al., 2005). In severe and persistent cases there is emerging evidence for modest benefit from treatment on physical and developmental measures (Haggard M: personal communication). It is reasonable to argue that reactive services will know of these cases before school entry, but evidence is lacking. Education-based interventions may also be effective in reducing temporary disability Authors’ summary opinion: partially satisfied

10. There should be agreed evidence-based policies covering which individuals should be offered treatment and the appropriate treatment to be offered

In severe and persistent cases there is emerging but as yet unpublished evidence for modest longer term benefit from surgical intervention, as well as the immediate expected benefit of hearing restored to near normal (Haggard M: personal communication). More work is required on case definition and markers of likely benefit from surgery Authors’ summary opinion: partially satisfied

continued

Appendix 1

Criteria

Supporting evidence

11. Clinical management of the condition and patient outcomes should be optimised by all healthcare providers prior to participation in a screening programme

There is considerable practice variability between services in the UK; management and outcomes therefore vary in ways not related to evidence

12. There should be evidence from high-quality RCTs that the screening programme is effective in reducing mortality and morbidity

There is no evidence from high-quality RCTs that the screening programme is effective in reducing morbidity (Simpson et al., 2003)

There is no evidence that screening and intervention for childhood hearing impairment associated with OME are clinically, socially and ethically unacceptable to health professionals concerned with the issue and to the public. Universal SES is undertaken by all but 12.2% of service-lead respondents in the current nationwide survey of current practice; surgical intervention for OME is relatively straightforward and risk free, and is very common; parental refusal is thought to be rare. Robust evidence for these statements is sparse, however

14. The benefit from the screening programme should outweigh the physical and psychological harm (caused by the test, diagnostic procedure and treatment)

The extent of the beneficial effects of early identification for children with transient hearing impairment on developmental outcomes in general and communication in particular has not been demonstrated; there is no evidence of either benefit or harm associated with the SES and treatment for OME, other than evidence of some postsurgical changes noted to the tympanic membrane in some cases who have repeat ventilation tubes (Rosenfeld, 2003)

15. The opportunity cost of the screening programme (including testing, diagnosis and treatment) should be economically balanced in relation to expenditure on medical care as a whole

There is no good-quality published evidence of the costs and effectiveness of the screen. The cost-effectiveness modelling carried out suggested that each screen costs £8, and that a universal school entry screen based largely or completely on pure tone sweep tests was associated with higher costs and slightly higher QALYs compared with no screen and other screen alternatives; the range of expected costs, QALYs and net benefits was broad, indicating a considerable degree of uncertainty

16. There should be a plan for monitoring and managing the screening programme and an agreed set of quality assurance standards

Authors’ summary opinion: not satisfied

Authors’ summary opinion: partially satisfied

Authors’ summary opinion: not satisfied

Authors’ summary opinion: satisfied

Authors’ summary opinion: not satisfied. 17. Adequate staffing facilities for testing, diagnosis, treatment and programme management should be available prior to the commencement of the screening programme

100

Resources to perform the screen vary across different implementations of the programme nationally. Many responses to the survey in the current report highlight deficiencies in staff numbers and experience, facilities and equipment. Resources in paediatric otology departments are variable, with variable linkage with good-quality paediatric audiology; waiting times for surgery for OME in children are variable Authors’ summary opinion: partially satisfied continued

Health Technology Assessment 2007; Vol. 11: No. 32

Criteria

Supporting evidence

18. All other options for managing the condition should have been considered (e.g. improving treatment, providing other services)

The most common route for referral of children with this condition is via GPs. It is not clear from the published evidence that improvement of reactive services and surveillance programmes in the preschool period (Hall and Elliman, 2003) would not result in the identification of the children who would benefit from surgery (i.e. those with severe and persistent symptoms) Authors’ summary opinion: not satisfied

Casselbrant ML, Mandel E. Epidemiology. In Rosenfeld RR, Bluestone CD, editors. Evidence-based otitis media. Hamilton and London: BC Decker; 2003. pp. 147–62. Davis A, Bamford J, Wilson I, Ramkalawan T, Forshaw M, Wright S. A critical review of the role of neonatal hearing screening in the detection of congenital hearing impairment. Health Technol Assess 1997;1(10). FitzZaland RE, Zink GD. A comparative study of hearing screening procedures. Ear Hear 1984;5:205–10. Fonseca S, Forsyth H, Neary W. School hearing programme in the UK: practice and performance. Arch Dis Child 2005;90:154–64. Haggard M. Research and development of effective services for hearing-impaired people. London: Nuffield Provincial Hospitals Trust; 1993. Haggard MP, Hughes E. Screening children’s hearing. London: HMSO; 1991. Hall D, editor. Health for all children. 4th ed. Oxford: Oxford University Press; 2003. Holtby I, Forster DP, Kumar U. Pure tone audiometry and impedance screening of school entrant children by nurses: evaluation in a practical setting. J Epidemiol Community Health 1997;51:711–15. Orlando MS, Frank T. Audiometer and AudioScope hearing screening compared with threshold test in young children. J Pediatr 1987;110:261–3.

Rosenfeld RR. Tympanostomy tube care and consequences. In Rosenfeld RR, Bluestone CD, editors. Evidence-based otitis media. Hamilton and London: BC Decker; 2003. pp. 460–81. Rovers MM, Black N, Browning GG, Maw R, Zielhuis GA, Haggard MP. Grommets in otitis media with effusion: an individual patient data meta-analysis. Arch Dis Child 2005;90:480–5. Sabo MP, Winston R, Macias JD. Comparison of pure tone and transient otoacoustic emissions screening in a grade school population. Am J Otol 2000;21: 88–91. Simpson SA, Thomas CL, van der Linden MK, Macmillan H, van der Wouden JC, Butler C. Identification of children in the first four years of life for early treatment for otitis media with effusion. Cochrane Database Syst Rev 2003;(2):CD004163. (Updated: Cochrane Database Syst Rev 2007;(1): CD004163.) Stewart-Brown S, Haslum MN. Screening for hearing loss in childhood: a study of national practice. BMJ 1987;6584:1386–8. Zielhuis GA, Rach GH, van den Broek P. The natural course of otitis media with effusion in preschool children. Eur Arch Otorhinol 1990;247:215–21.

Health Technology Assessment 2007; Vol. 11: No. 32

Appendix 2 Questionnaire used in the survey of national practice

Health Technology Assessment Programme Sponsored Project Use of this logo does not constitute endorsement

Current practice, accuracy, effectiveness and cost-effectiveness of the School Entry hearing Screen (SES) A research project commissioned by the NHS R&D Health Technology Assessment Programme

District / Area / PCT ..................................................................................... Service Lead Name ....................................................................................... Name of person completing the questionnaire (if different) ............................................................................

Appendix 2

Who should fill in this questionnaire? This questionnaire should be filled in by the person who is considered the clinical service lead for the school entry hearing screen in your area. If this is not you then please pass the questionnaire on to the most appropriate person for completion.

Confidentiality Your answers will be stored on a computer at the University of Manchester and will meet the conditions of the Data Protection Act. Your answers will be anonymised before they are inputted into the computer. All responses will be kept confidential and they will be seen only by members of the research team.

Questions If you have any questions or would like to receive a summary report of our findings then please do not hesitate to contact: Kirsty Bristow Human Communication and Deafness School of Psychological Sciences Humanities Building (Devas) University of Manchester Oxford Road Manchester M13 9PL Telephone: 0161 275 8575 e-mail:

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[emailprotected]

Health Technology Assessment 2007; Vol. 11: No. 32

BEFORE YOU START Does your area have a written protocol for the School Entry Hearing Screen AND can a copy of this protocol be sent to us? 䡺 NO If you have answered NO please go to page 3 and answer as many questions as possible. If you are not sure how to answer a question then please give the best answer you can and write additional comments if you want to. 䡺 YES, a hard copy of the written protocol has been included with the completed questionnaire 䡺 YES, a hard copy of the written protocol has been sent separately 䡺 YES, an electronic version of the written protocol has been emailed to Kirsty Bristow ([emailprotected])

If you have answered YES to the above question please read the following statement – Some of the questions within this questionnaire may already be answered within your written School Entry Hearing Screen protocol. So…. If the answer is adequately covered by information already given in the protocol just write ‘protocol’. However, this will not always be the case and some questions may need more detail or ask for your opinion. Therefore we ask that you read all sections of the questionnaire as carefully as possible.

Please feel free to add comments on any question in the spaces provided at the end of each section or on additional pages. All comments will be read, so please write as many as you wish

Appendix 2

WHO DO YOU TEST? These questions are designed to find out the kinds of children that are routinely tested by the School Entry Hearing Screen in your area. 1 Please indicate which children are routinely entered into the school entry hearing screen in your area. Please tick one box for each category.

All

Some

None

Children in state schools Children in private schools Children who are home educated Children in special schools with known physical or sensory disability (excluding hearing loss) Children in special schools with known mental disability (excluding children who also have hearing loss ) Children known to have hearing loss Other (please specify in the space below) ……………………………...................................................

If you have answered some or none to any of the above categories it would be very helpful if you could give further details in the space below .......................................................................................................................................................................... .......................................................................................................................................................................... .......................................................................................................................................................................... .......................................................................................................................................................................... ..........................................................................................................................................................................

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2 What arrangements (if any) are in place within your area to screen children, for whom you have consent, who did not attend the screen for any reason (e.g. through school absence, had a cold)? Please tick one box for each category All of the time

Most of the time

Some of the time

Rarely

Never

Revisit to the school Appointment arranged at school health clinic Appointment arranged at Audiology clinic No arrangement made Other (please specify in the space below) ……………………………………………….

3 In which school year is the school entry hearing screen routinely performed in your area? Please tick as many answers as apply. Preschool Reception/Primary 1 Year 1/Primary 2 Year 2/Primary 3 Other (please specify in the space below) .......................................................................................................................................................................... Any Comments? .......................................................................................................................................................................... .......................................................................................................................................................................... ..........................................................................................................................................................................

Appendix 2

HOW DO YOU PERFORM THE SCHOOL ENTRY HEARING SCREEN? For the purposes of this study we need to know what audiological tests are used in the School Entry Hearing Screen, under what conditions these tests are performed and in what locations. By answering all the questions in this section you will enable us to understand these factors for your area

Before completing this section please be aware that for the purposes of this questionnaire we are applying the following definitions – Screen – the entire remit of tests that a child undergoes before either passing or being referred for further hearing assessment Test – the individual assessments, which when taken together, form a screen For example, the diagram below details one screen that consists of either one or two tests – Screen Example – Test One

Pass

Fail

Test two (three weeks later)

Pass screen

Pass

Fail

Pass screen

Referred for further assessment

4 Does your area have any written documentation for the following? (please tick one box for each category) Documentation? Yes Parent/guardian agreement for the screen Information provided to the parent/guardian prior to screening Information provided to the parent/guardian prior to referral Test protocol Re-test protocol Referral protocol If you answered yes to any of the above categories, could a copy of the document(s) be sent to us? Yes, they have been included with this completed questionnaire Yes, they have been sent separately Copy unavailable

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5 Where is the first test within the school entry hearing screen typically carried out in your area? (please tick one box for each location)

All of the time

Most of the time

Some of the time

Rarely

Never

School Community clinic Home GP clinic Other (please specify in the space below) ………………………………………………

6 Under what conditions is the school entry hearing screen in your area performed? (please tick one box for each category) All of the time

Most of the time

Some of the time

Rarely

Never

Soundproof booth Sound treated room Quiet office Noisy office Quiet classroom/area Noisy classroom/area Other (please specify in the space below) …………………………………........................

Appendix 2

7 Which tests are used as part of the school entry hearing screen in your area? How are these tests combined into a whole screen protocol? Please describe via words or flow diagram. To indicate the required level of detail an example is shown in the box below. The information we require is shown in italics in the diagram and detailed in the following bullet pointed list – ● ● ● ●

the tests used at each stage of the screen how much time passes between each test how many times the child is tested before referral takes place which service the child is referred on to

Test 1 Sweep pure tone audiometry

Your text or diagram –

110

Pass

Fail

No further action

Test 2 Sweep pure tone audiometry Test 2 takes place two weeks after test 1

Pass

Fail

No further action

Refer to Audiology

Health Technology Assessment 2007; Vol. 11: No. 32

8 Please indicate at what level a child will be said to have not passed each test you use in the screen. Frequency screened and level at which a child will not have passed Test 1 Test 2 Test 3

9 When screening children at school entry do you ……. (please tick one box for each category) All of the time

Most of the time

Some of the time

Rarely

Never

… screen for hearing loss only … screen for hearing loss as part of a wider health check

10 Please estimate the minimum, average and maximum numbers of children that could be screened under normal circumstances during the course of a one day visit to a school in your area.

When screening for hearing loss only

When screening for hearing loss as part of a wider health check

Minimum number Average number Maximum number

Any Comments? .......................................................................................................................................................................... .......................................................................................................................................................................... .......................................................................................................................................................................... ..........................................................................................................................................................................

Appendix 2

WHO PERFORMS THE TESTING? These questions are designed to find out which staff are involved in the implementation of the School Entry Hearing Screen in your area. 11 Please indicate which staff perform the school entry hearing screen tests in your area. Please tick all that apply to your area. School nurse Audiometrician Audiologist Health Visitor School Doctor Other (please specify in the space below) ..........................................................................................................................................................................

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WHAT EQUIPMENT DO YOU USE? 12 Please indicate the types of equipment used within the school entry hearing screen as run in your area. Please then say, as accurately as possible, the number of each you have. Use? No

If yes, how many are in use? Yes

Screening audiometer Diagnostic audiometer Screening tympanometer Diagnostic tympanometer Other (please specify) ……………………………………………

Appendix 2

AUDIT DATA In order for us to make evidence-based recommendations for the future of the School Entry Hearing Screen to the NHS we need to know the referral rates and yield of the screen. Your answers to these questions are therefore extremely important. However, we do NOT expect you to undertake an exhaustive and time consuming note review process. Please answer the questions below with details of audit data ONLY if it can be easily obtained. 13 Has an audit of the School Entry Hearing Screen in your area been performed in the last two years AND can a copy of this audit be made available to us? No .................................................................................................................

please go to question 14

Yes, a hard copy of the audit has been included with this questionnaire ................................................................................................

please go to question 18

Yes, a hard copy of the audit has been sent separately ...............................

please go to question 18

Yes, an electronic version of the audit has been emailed to Kirsty Bristow ([emailprotected]) ...................................

please go to question 18

14 Please indicate if the school entry hearing screen in your area employs a data management system. No .................................................................................................................

please go to question 17

Yes, an IT system Yes, paper system Yes, other If other, please specify below ..........................................................................................................................................................................

15 Can you easily get data reports from this data management system? No ................................................................................................................. Yes

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please go to question 17

Health Technology Assessment 2007; Vol. 11: No. 32

16 Please indicate the following for the most recent academic year possible – (e.g. 2003–2004, 2002–2003 etc. Please indicate below which year) For the academic year ………………. Number How many children were eligible for the school entry hearing screen in your area? How many children were screened with the school entry hearing screen in your area? How many children were referred for further audiological assessment following failure of the school entry hearing screen in your area? How many children with sensorineural hearing loss were identified by the school entry hearing screen in your area? How many children with permanent conductive hearing loss were identified by the school entry hearing screen in your area? How many children with temporary conductive hearing loss were identified by the school entry hearing screen in your area? How many children with other types of hearing loss were identified by the school entry hearing screen in your area? (please specify types of hearing loss) ……………………………………………………………………………………..

17 Do you have any other documentation that you feel may be of use to us? No Yes If yes, could a copy this documentation be made available to us? Yes, a hard copy has been included with this completed questionnaire Yes, a hard copy has been sent separately Yes, an electronic version has been emailed to Kirsty Bristow ([emailprotected]) No

Appendix 2

18 Is information regarding the costing of the School Entry Hearing Screen within the last two years routinely available in your district? No Yes If yes, please provide the contact details of a member of staff that could provide us with this costing data in the space below Name

.....................................................................................................................................................

Position

.....................................................................................................................................................

Address

..................................................................................................................................................... .....................................................................................................................................................

E-mail

.....................................................................................................................................................

Telephone

.....................................................................................................................................................

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YOUR VIEWS The questions so far may not have addressed all the issues you wish to raise. This section allows you to say what you think about the usefulness and future of the School Entry Hearing Screen. 19 Are there any plans for development or change of the school entry hearing screen in your area? No Yes If yes, please give details below .......................................................................................................................................................................... .......................................................................................................................................................................... .......................................................................................................................................................................... .......................................................................................................................................................................... 20 Please cross one of the boxes below to indicate how useful overall you think the School Entry Hearing Screen is in your area as it is currently operated. Not useful at all

Very useful

Please add further comments to explain your answer .......................................................................................................................................................................... .......................................................................................................................................................................... .......................................................................................................................................................................... .......................................................................................................................................................................... .......................................................................................................................................................................... 21 Please add below any suggestions for the future of the School Entry Hearing Screen either in your area or nationally .......................................................................................................................................................................... .......................................................................................................................................................................... ..........................................................................................................................................................................

Appendix 2

Any further comments? .......................................................................................................................................................................... .......................................................................................................................................................................... .......................................................................................................................................................................... .......................................................................................................................................................................... ..........................................................................................................................................................................

Thank you for taking the time to complete this questionnaire We may need to contact you again to discuss some matters raised in your questionnaire. We will endeavour to do so only when strictly necessary and will keep any correspondence with you to an absolute minimum. Do we have your permission to contact you if required? No Yes If yes could you please give your preferred form of contact below– Telephone Number .......................................................................................................................... OR E-mail

Address

.......................................................................................................................... .......................................................................................................................... ..........................................................................................................................

OR Post

Address

.......................................................................................................................... .......................................................................................................................... ..........................................................................................................................

Please return this completed questionnaire, and any other supporting documents you wish to include, in the pre-paid envelope supplied to– Kirsty Bristow Human Communication and Deafness School of Psychological Sciences Humanities Building (Devas) University of Manchester Oxford Road Manchester M13 9PL

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Appendix 3 Prevalence rates for permanent childhood hearing impairment for three cohorts in Waltham Forest and Redbridge Districts and for one cohort in Trent Region of the UK TABLE 69 Prevalence of PCHI by the end of the first year in primary school in cohort 1, NNHS: 31,538 children born from January 1977 to 1987 (n = 131) Degree of hearing impairment

All PCHI n Prevalence/1000 (95% CI)

Congenital PCHI n Prevalence/1000 (95% CI)

Acquired PCHI n Prevalence/1000 (95% CI)

Bilateral (PTA average 500–4000 Hz) in BHE

Mild (20–39 dB HL)

37 1.17 (0.8 to 1.55)

32 1.01 (0.66 to 1.37)

5 0.16 (0.02 to 0.3)

Moderate (40–69 dB HL)

24 0.76 (0.46 to 1.07)

18 0.57 (0.31 to 0.83)

6 0.19 (0.04 to 0.34)

Severe (70–94 dB HL)

12 0.38 (0.17 to 0.6)

10 0.32 (0.12 to 0.51)

2 0.06 (0 to 0.15)

Profound (艌95 dB HL)

12 0.38 (0.16 to 0.6)

12 0.38 (0.17 to 0.6)

0 0.00 (0.00 to 0.00)

All (艌40 dB HL)

48 1.52 (1.09 to 1.95)

40 1.27 (0.88 to 1.66)

8 0.25 (0.08 to 0.43)

All bilateral

85 2.69 (2.12 to 3.27)

72 2.28 (1.76 to 2.81)

13 0.41 (0.19 to 0.64)

Mild/moderate (20–69 dB HL)

16 0.51 (0.26 to 0.76)

15 0.48 (0.23 to 0.72)

1 0.03 (0 to 0.09)

Severe/profound (艌70 dB HL)

30 0.95 (0. 61 to 1.29)

23 0.73 (0.43 to 1.03)

7 0.22 (0.06 to 0.39)

All unilateral

46 1.46 (1.04 to 1.88)

38 1.21 (0.82 to 1.59)

8 0.25 (0.08 to 0.43)

131 4.15 (3.44 to 4.86)

110 3.49 (2.84 to 4.14)

21 0.67 (0.38 to 0.95)

Unilateral (PTA average 500–4000 Hz) in WHE

Total all degrees

Appendix 3

TABLE 70 Prevalence of PCHI by the end of the first year in primary school in cohort 2, TNHS: 32,980 children born from January 1990 to 2000 (n = 117) Degree of hearing impairment

All PCHI n Prevalence/1000 (95% CI)

Congenital PCHI n Prevalence/1000 (95% CI)

Acquired PCHI n Prevalence/1000 (95% CI)

Bilateral (PTA average 500–4000 Hz) in BHE

Mild (20–39 dB HL)

40 1.22 (0.84 to 1.59)

29 0.88 (0.56 to 1.2)

11 0.33 (0.14 to 0.53)

Moderate (40–69 dB HL)

24 0.73 (0.44 to 1.02)

19 0.58 (0.32 to 0.84)

5 0.15 (0.02 to 0.29)

Severe (70–94 dB HL)

14 0.43 (0.2 to 0.65)

12 0.36 (0.16 to 0.57)

2 0.06 (0 to 0.15)

Profound (艌95 dB HL)

11 0.33 (0.14 to 0.53)

0 0.0 (0.00 to 0.00)

All (艌40 dB HL)

49 1.49 (1.07 to 1.91)

42 1.28 (0.89 to 1.66)

7 0.21 (0.06 to 0.37)

All bilateral

89 2.71 (2.14 to 3.27)

71 2.16 (1.66 to 2.66)

18 0.55 (0.29 to 0.8)

Mild/moderate (20–69 dB HL)

13 0.39 (0.18 to 0.61)

10 0.30 (0.12 to 0.49)

3 0.09 (0 to 0.19)

Severe/profound (艌70 dB HL)

15 0.46 (0.23 to 0.69)

12 0.36 (0.16 to 0.57)

3 0.09 (0 to 0.19)

All unilateral

28 0.85 (0.54 to 1.17)

22 0.67 (0.39 to 0.95)

6 0.18 (0.04 to 0.33)

117 3.56 (2.91 to 4.2)

93 2.83 (2.25 to 3.4)

24 0.73 (0.44 to 1.02)

Unilateral (PTA average 500–4000 Hz) in WHE

Total all degrees

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TABLE 71 Prevalence of PCHI by the end of the first year in primary school in cohort 3, UNHS: 29,132 children born from January 1992 to 2000 (n = 101) Degree of hearing impairment

All PCHI n Prevalence/1000 (95% CI)

Congenital PCHI n Prevalence/1000 (95% CI)

Acquired PCHI n Prevalence/1000 (95% CI)

Bilateral (PTA average 500–4000 Hz) in BHE

Mild (20–39 dB HL)

36 1.24 (0.83 to 1.64)

28 0.96 (0.61 to 1.32)

8 0.27 (0.08 to 0.46)

Moderate (40–69 dB HL)

23 0.79 (0.47 to 1.11)

19 0.65 (0.36 to 0.95)

4 0.14 (0 to 0.27)

Severe (70–94 dB HL)

9 0.31 (0.11 to 0.51)

0 0.00 (0.00 to 0.00)

Profound (艌95 dB HL)

10 0.34 (0.13 to 0.56)

8 0.27 (0.08 to 0.46)

2 0.07 (0 to 0.16)

All (艌40 dBHL)

42 1.44 (1.01 to 1.88)

36 1.24 (0.83 to 1.64)

6 0.21 (0.04 to 0.37)

All bilateral

78 2.68 (2.08 to 3.27)

64 2.20 (1.63 to 2.7)

14 0.48 (0.25 to 0.78)

Mild/moderate (20–69 dB HL)

15 0.51 (0.25 to 0.78)

12 0.41 (0.18 to 0.64)

3 0.10 (0 to 0.22)

Severe/profound (艌70 dB HL)

8 0.27 (0.08 to 0.46)

7 0.24 (0.06 to 0.42)

1 0.03 (0 to 0.1)

All unilateral

23 0.79 (0.47 to 1.11)

19 0.65 (0.36 to 0.95)

4 0.14 (0 to 0.27)

101 3.47 (2.79 to 4.14)

83 2.85 (2.21 to 3.42)

18 0.62 (0.36 to 0.95)

Unilateral (PTA average 500–4000 Hz) in WHE

Total all degrees

Appendix 3

TABLE 72 Prevalence of PCHI for the three cohorts combined and for the Trent cohort, and an analysis of the difference in the prevalence rates between the three cohorts and between the combined cohort and the Trent cohort Degree of hearing impairment

Bilateral (PTA average 500–4000 Hz) in BHE

Unilateral (PTA average 500–4000 Hz) in WHE

Combined cohort Prevalence/1000 (95% CI)

Comparison of prevalence in three cohorts Pearson ␹2 (p-value)

Trent cohort Prevalence/1000 (95% CI)

Mild (20–39 dB HL)

1.21 (0.99 to 1.43)

0.052 (0.974)

Not available

Moderate (40–69 dB HL)

0.76 (0.58 to 0.94)

0.073 (0.964)

0.68 (0.61 to 0.78)

0.583 (0.445)

Severe (70–94 dB HL)

0.37 (0.25 to 0.5)

0.568 (0.753)

0.28 (0.23 to 0.34)

2.151 (0.142)

Profound (艌95 dB HL)

0.35 (0.23 to 0.47)

0.108 (0.948)

0.31 (0.26 to 0.37)

0.463 (0.496)

All (艌40 dB HL)

1.49 (1.24 to 1.73)

0.060 (0.967)

1.27 (1.16 to 1.39)

2.532 (0.112)

All bilateral

2.69 (2.36 to 3.03)

0.005 (0.998)

Not available

Mild/moderate (20–69 dB HL)

0.47 (0.33 to 0.61)

0.609 (0.737)

Not available

Severe/profound (艌70 dB HL)

0.57 (0.41 to 0.72)

13.338 (0.001)***

Not available

All unilateral

1.04 (0.83 to 1.24)

8.229 (0.016)*

Not available

*** p < 0.001, * p < 0.05.

122

Comparison of prevalence in Trent and combined cohorts Pearson ␹2 (p-value)

Health Technology Assessment 2007; Vol. 11: No. 32

Appendix 4 Search strategies used in the systematic review Ovid MEDLINE(R) 1966 to April week 4 2005 1 2 3 4 5 6 7 8 9 10 11 12 13 14 15 16 17 18 19 20 21 22 23 24 25 26 27 28 29 30 31 32 33 34 35 36 37 38 39 40 41 42 43 44 45

hearing loss$.mp. or exp Hearing Loss/ (hearing adj (disorder$ or difficult$ or problem$ or impair$)).mp. exp Hearing Disorders/ or/1-3 exp Child, Preschool/ or school entry.mp. exp Child Development/ early detect$.mp. infant school$.mp. exp Schools, Nursery/ or exp Nurseries/ or exp Child Day Care Centers/ or kindergarten$.mp. nursery school$.mp. or/5-10 screen$.mp. or exp Mass Screening/ 4 and 12 13 and 11 (school entry adj3 (screen$ or exam$)).mp. (medical exam$ adj2 school$).mp. or/15-16 14 or 17 randomized controlled trial.pt. controlled clinical trial.pt. randomized controlled trials.sh. random allocation.sh. double blind method.sh. single-blind method.sh. or/19-24 (animals not human).sh. 25 not 26 clinical trial.pt. exp clinical trials/ (clin$ adj25 trial$).ti,ab. ((singl$ or doubl$ or trebl$ or tripl$) adj25 (blind$ or mask$)).ti,ab. placebos.sh. placebo$.ti,ab. random$.ti,ab. research design.sh. or/28-35 36 not 26 37 not 27 comparative study.sh. exp evaluation studies/ follow up studies.sh. prospective studies.sh. (control$ or prospectiv$ or volunteer$).ti,ab. or/39-43 44 not 26

46 45 not (27 or 38) 47 27 or 38 or 46 48 18 and 47

Ovid MEDLINE(R) In-Process & Other Non-Indexed Citations 6 June 2005 1 2 3 4 5 6 7 8 9 10 11 12 13 14 15 16 17 18 19 20 21 22 23 24 25

hearing loss$.mp. or exp Hearing Loss/ (hearing adj (disorder$ or difficult$ or problem$ or impair$)).mp. exp Child, Preschool/ or school entry.mp. early detect$.mp. infant school$.mp. exp Schools, Nursery/ or exp Nurseries/ or exp Child Day Care Centers/ or kindergarten$.mp. nursery school$.mp. screen$.mp. or exp Mass Screening/ (school entry adj3 (screen$ or exam$)).mp. (medical exam$ adj2 school$).mp. randomized controlled trial.pt. controlled clinical trial.pt. clinical trial.pt. (clin$ adj25 trial$).ti,ab. ((singl$ or doubl$ or trebl$ or tripl$) adj25 (blind$ or mask$)).ti,ab. placebo$.ti,ab. random$.ti,ab. (control$ or prospectiv$ or volunteer$).ti,ab. or/1-2 or/3-7 or/8-10 or/11-18 19 and 20 21 and 23 22 and 24

EMBASE 1980 to 2005 week 19 1 2

hearing loss$.mp. or exp Hearing Loss/ (hearing adj (disorder$ or difficult$ or problem$ or impair$)).mp. 3 exp Hearing Disorder/ 4 or/1-3 5 school entry.mp. 6 (pre adj school).mp. 7 (nursery adj school$).mp. 8 exp nursery school/ 9 kindergarten$.mp. or exp kindergarten/ 10 exp Day Care/ 11 infant school$.mp.

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Appendix 4

12 13 14 15 16 17 18 19 20 21 22 23 24 25 26 27 28 29 30 31 32 33 34 35 36 37 38 39

early detect$.mp. exp Child Development/ or/5-13 screen$.mp. exp mass screening/ or exp screening/ or exp auditory screening/ or exp screening test/ or/15-16 (school entry adj3 (screen$ or exam$)).mp. (medical exam$ adj2 school$).mp. or/18-19 4 and 17 21 and 14 22 or 20 randomized controlled trial/ exp clinical trial/ exp controlled study/ double blind procedure/ randomization/ placebo/ single blind procedure/ (control$ adj (trial$ or stud$ or evaluation$ or experiment$)).mp. ((singl$ or doubl$ or trebl$ or tripl$) adj5 (blind$ or mask$)).mp. (placebo$ or matched communities or matched schools or matched populations).mp. (comparison group$ or control group$).mp. (clinical trial$ or random$).mp. (quasiexperimental or quasi experimental or pseudo experimental).mp. matched pairs.mp. or/24-37 23 and 38

CINAHL – Cumulative Index to Nursing & Allied Health Literature 1982 to May week 1 2005 1 2

124

3 4 5 6 7 8 9 10 11 12 13 14 15 16 17 18 19

hearing loss$.mp. or exp Hearing Loss/ (hearing adj (disorder$ or difficult$ or problem$ or impair$)).mp. exp Hearing Disorder/ or/1-3 school entry.mp. (pre adj school).mp. (nursery adj school$).mp. exp nursery school/ kindergarten$.mp. or exp kindergarten/ exp day care/ infant school$.mp. early detect$.mp. exp child development/ or/5-13 (school entry adj3 (screen$ or exam$)).mp. (medical exam$ adj2 school$).mp. or/15-16 exp health screening/ or screen$.mp. exp hearing screening/

20 21 22 23 24

or/18-19 4 and 18 21 or 19 22 and 14 23 or 17

Cochrane Library (Wiley) 2005 Issue 2 #1 #2 #3 #4 #5 #6 #7 #8 #9 #10 #11 #12 #13 #14 #15 #16 #17 #18 #19 #20 #21 #22 #23 #24 #25 #26 #27

exp Hearing Loss/ Hearing next loss hearing next disorder* hearing next difficult* hearing next problem* hearing next impair* exp Hearing disorders/ #1 or #2 or #3 or #4 or #5 or #6 or #7 screening exp Mass screening/ #9 or #10 school next entr* early detect* infant next school* nursery next school* kindergarten* exp Schools, Nursery/ exp Child day care centers/ exp Child, Pre School exp Child Development #12 or #13 or #14 or #15 or #16 or #17 or #18 or #19 or #20 #8 and #11 #21 and #22 school next entr* medical near/3 exam* near/3 school* #24 or #25 #23 or #26

ERIC (Cambridge Scientific Abstracts) Searched 7 June 2005 #1 #2 #3 #4

school entry or preschool screen* or test* hearing loss or hearing impair* #1 and #2 and #3

Science Citation Index (Web of Knowledge) Searched 7 June 2005 #1 #2 #3 #4

school entry or preschool screen* or test* hearing loss or hearing impair* #1 and #2 and #3

PsycINFO 1985 to May week 1 2005 1 2

(hearing adj (disorder$ or impair$ or problem$ or difficul$)).mp. exp Hearing Disorders/ or hearing loss.mp.

Health Technology Assessment 2007; Vol. 11: No. 32

3 4 5 6 7 8 9 10 11

12 13 14 15 16 17 18 19 20

or/1-2 exp Screening Tests/ or screen$.mp. 1 and 4 Exp CHILD DAY CARE/ (School Adj Entry).Mp (Pre Adj School).Mp. Nurser$.Mp. Exp Kindergarten Students/ Or Exp KINDERGARTENS/ Or Kindergarten$.Mp. Exp Preschool Students/ Or Exp Preschool Education/ Or Exp Nursery School Students/ Or Infant School$.Mp. Early Detect$.Mp. (School Entry Adj3 (Screen$ Or Exam$)).Mp. Child$.Mp. Or/6-14 5 And 15 (School Entry Adj3 (Screen$ Or Exam$)).Mp. (Medical Exam$ Adj2 School$).Mp. Or/17-18 16 Or 19

Search strategies: accuracy of diagnostic tests Ovid MEDLINE(R) 1966 to January week 4 2006 1 2 3 4 5 6 7 8 9 10 11 12 13 14 15 16 17 18 19 20 21 22 23 24

audiometry.mp. or audiometry/ or exp audiometry, pure-tone/ exp otoacoustic emissions, spontaneous/ or otoacoustic emission$.mp. exp acoustic impedance tests/ or acoustic impedance.mp. exp hearing tests/is, mt [instrumentation, methods] hearing test$.mp. sweep audio.mp. sweep test$.mp. (hearing adj2 questionnaire$).mp. cmedhq.mp. conventional audiometry.mp. conditioned play audiometry.mp. cpa.mp. exp audiometry, evoked response/ audiologic$ assessment$.mp. acoustic intermittance.tw. tympanometry.mp. otoscopy.mp. or exp otoscopy/ or exp diagnostic techniques, otological/ otological exam$.mp. acoustic reflex test$.mp. teoae.mp. dpoae.mp. (impedance adj screening).mp. (impedance adj method$).mp. fixed frequency audio.mp.

25 26 27 28 29 30 31 32 33 34 35 36 37 38 39 40 41 42 43 44 45 46 47 48 49 50 51 52 53 54 55 56 57 58

(speech adj2 noise).mp. reflectometry.mp. acoustic impedance.mp. or/1-27 exp hearing loss, sensorineural/pc, di exp hearing disorders/di, pc exp otitis media/pc, di exp hearing loss, high-frequency/pc, di exp hearing loss/di, pc hearing impairment$.mp. exp hearing loss, conductive/di, pc (hearing adj3 screen$).mp. exp "sensitivity and specificity"/ exp predictive value of tests/ (diagnos$ adj2 accura$).mp. or/37-39 exp child, preschool/ or school entry.mp. exp child development/ early detect$.mp. infant school$.mp. exp schools, nursery/ or exp nurseries/ or exp child day care centers/ or kindergarten$.mp. nursery school$.mp. or/41-46 screen$.mp. or exp mass screening/ (school entry adj3 (screen$ or exam$)).mp. (medical exam$ adj2 school$).mp. or/49-50 28 and 40 47 or 51 or/29-36 52 and 53 40 and 54 53 and 56 55 or 57

EMBASE 1980 to 2006 week 11 1

exp pure tone audiometry/ or exp audiometry/ or audiometry.mp. 2 otoacoustic emission$.mp. or exp spontaneous otoacoustic emission/ or exp otoacoustic emission/ 3 acoustic impedance.mp. or exp acoustic impedance/ 4 hearing test$.mp. 5 exp hearing test/ 6 sweep audio.mp. 7 sweep test$.mp. 8 (hearing adj2 questionnaire$).mp. 9 cmedhq.mp. 10 ((conventional or conditioned play) adj audiometry).mp. 11 cpa.mp. 12 exp evoked response audiometry/ 13 (audiologic$ adj assessment$).mp. 14 (acoustic adj intermittance).mp.

125

Appendix 4

15 16 17 18 19 20 21 22 23 24 25 26 27 28 29 30 31 32 33 34 35 36 37 38 39 40 41 42 43 44 45 46 47 48 49 50 51 52 53 54 55 56 57 58 59 60

tympanometry.mp. or exp tympanometry/ otoscopy.mp. or exp otoscopy/ (otological adj2 technique$).mp. (otological adj2 (exam$ or technique$)).mp. teoae.mp. dpoae.mp. or exp distortion product otoacoustic emission/ (impedance adj (screen$ or method$)).mp. fixed frequency audio.mp. (speech adj2 noise).mp. reflectometry.mp. or exp reflectometry/ or/1-24 hearing loss/pc, di [prevention, diagnosis] hearing disorder/pc, di [prevention, diagnosis] otitis media/pc, di [prevention, diagnosis] hearing impair$.mp. hearing impairment/pc, di [prevention, diagnosis] (hearing adj3 screen$).mp. exp "sensitivity and specificity"/ (predictive adj2 test$).mp. (diagnos$ adj2 accura$).mp. or/32-34 or/26-31 25 and 35 school entry.mp. pre-school.mp. child/ exp child development/ early detect$.mp. infant school$.mp. nursery school$.mp. or exp nursery school/ exp child care/ or child day care.mp. kindergarten$.mp. or exp kindergarten/ or/38-46 screen$.mp. exp mass screening/ (school entry adj3 (screen$ or exam$)).mp. (medical exam$ adj2 school$).mp. or/48-49 or/50-51 25 and 35 and 47 35 and 36 and 47 54 or 55 or/52-53 47 or 57 35 and 36 and 58 56 or 59

CINAHL 1982 to March week 3 2006 1

126

audiometry.mp. or exp audiometry, evoked response/ or exp audiometry/ or exp audiometry, pure-tone/ otoacoustic emission$.mp. or exp otoacoustic emissions, spontaneous/

3 4 5 6 7 8 9 10 11 12 13 14 15 16 17 18 19 20 21 22 23 24 25 26 27 28 29 31 32 33 34 35 36 37 38 39 40 41 42 43 44 45 46 47 48 49 51 52 53 54 55 56 57

exp acoustic impedance tests/ or acoustic impedance.mp. hearing test$.mp. or exp hearing tests/ sweep test$.mp. sweep audio.mp. (hearing adj2 questionnaire$).mp. cmedhq.mp. (conventional adj2 audiometry).mp. (conditioned adj2 audiometry).mp. cpa.mp. evoked response.mp. or exp evoked potentials/ (audiologic$ adj assessment$).mp. (acoustic adj intermittance).mp. tympanometry.mp. otoscopy.mp. (otological adj2 technique$).mp. (otological adj2 exam$).mp. teoae.mp. dpoae.mp. or exp otoacoustic emissions, evoked/ (impedance adj (screen$ or method$)).mp. fixed frequency audio.mp. (speech adj2 noise).mp. reflectometry.mp. or/1-24 hearing disorders/di, pc hearing impair$.mp. exp hearing screening/ (hear$ adj2 screen$).mp. exp "sensitivity and specificity"/ exp "predictive value of tests"/ (predictive adj2 test$).mp. (diagnos$ adj2 accura$).mp. or/31-34 school entry.mp. exp child, preschool/ or pre-school.mp. exp child development/ early detect$.mp. infant school$.mp. or exp infant development/ nursery school$.mp. or exp schools, nursery/ child day care.mp. or exp child day care/ kindergarten$.mp. or/36-43 screen$.mp. exp hearing screening/ exp school admissions/ (school entry adj2 (screen$ or exam$)).mp. (medical exam$ adj2 school$).mp. or/47-49 25 and 35 and 44 30 and 35 and 44 52 or 53 50 or 51 or 44 30 and 35 and 55 54 or 56

Health Technology Assessment 2007; Vol. 11: No. 32

PSYCINFO 1967 to March week 4 2006 1 2 3 4 5 6 7 8 9 10 11 12 13 14 15 16 17 18 19 20 21 22 23 24 25 26 27 28 29 30 31

exp bone conduction audiometry/ or exp audiometry/ or audiometry.mp. otoacoustic emission$.mp. acoustic impedance.mp. hearing test$.mp. sweep audio.mp. sweep test$.mp. (hearing adj2 questionnaire$).mp. cmedhq.tw. cpa.mp. evoked response audiometry.mp. audiologic$ assessment$.mp. acoustic intermittance.tw. tympanometry.mp. otoscopy.mp. (otological adj2 diagnos$).mp. otological exam$.mp. acoustic reflex test$.mp. teoae.mp. dpoae.mp. (impedance adj screening).mp. (impedance adj method$).mp. fixed frequency audio.mp. (speech adj2 noise).mp. reflectometry.mp. acoustic impedance.mp. or/1-25 (sensitivity adj2 specificity).mp. (predictive value adj2 test$).mp. (diagnos$ adj2 accurac$).mp. or/27-29 ((hearing loss$ or hearing disorder$ or hearing impair$ or otitis media) adj3 (diagnos$ or screen$)).mp.

32 33 34 35

36 37 38 39 40 41

30 or 31 26 and 32 child$.mp. or exp child day care/ exp early childhood development/ or exp preschool education/ or exp preschool students/ or pre-school.mp. or exp nursery schools/ kindergarten$.mp. or exp kindergartens/ nursery school$.mp. exp elementary school students/ or infant school$.mp. exp early intervention/ or early detect$.mp. or/34-39 33 and 40

ERIC (CSA) 1966 to present Search date: 28 March 2006 (hearing or otitis) and (diagnos* or screen* or test*) and (school*or nurser* or infant*) and (accur* or predictive or sensitiv*)

Science Citation Index (Web Of Knowledge) 1970 to present Search date: 28 March 2006 (Hearing or otitis or deaf*) and (screen* or test* or diagnos*) and (accura* or predictive or sensitive) and (pre-school or infant* or nurser* or kindergarten*)

Health Technology Assessment 2007; Vol. 11: No. 32

Appendix 5 Quality criteria for systematic reviews CRITICAL APPRAISAL SKILLS PROGRAMME Making sense of evidence about clinical effectiveness FIN

E IS

ACT

CASP

APPR

10 questions to help you make sense of a review

These questions consider the following: Are the results of the review valid? (SECTION A) What are the results? (SECTION B) Will the results help locally? (SECTION C) A number of italicised prompts are given after each question. These are designed to remind you why the question is important. There will not be time in the small groups to answer them all in detail!

These materials were developed by the Critical Appraisal Skills Programme (CASP) and we thank them for permission to use the materials.

Appendix 5

A/ Are the results of the review valid? Screening Questions 1. Did the review address a clearly focused question?

Yes 䡺

Can’t tell 䡺

No 䡺

Yes 䡺

Can’t tell 䡺

No 䡺

Yes 䡺

Can’t tell 䡺

No 䡺

Yes 䡺

Can’t tell 䡺

No 䡺

Yes 䡺

Can’t tell 䡺

No 䡺

HINT: An issue can be ‘focused’ in terms of ● ● ●

the population studied the intervention given the outcome considered

2. Did the authors look for the appropriate sort of papers? HINT: The ‘best sort of studies’ would – address the review’s question – have an appropriate study design (usually RCTs for papers evaluating interventions)

Is it worth continuing?

Detailed Questions 3. Do you think the important, relevant studies were included? HINT: Look for – – – – –

which bibliographic databases were used follow-up from reference lists personal contact with experts search for unpublished as well as published studies search for non-English language studies

4. Did the review’s authors do enough to assess the quality of the included studies? HINT: The authors need to consider the rigour of the studies they have identified. Lack of rigour may affect the studies’ results (“All that glisters is not gold” Shakespeare, the Merchant of Venice, Act II) 5. If the results of the review have been combined, was it reasonable to do so? HINT: Consider whether – – – –

130

the the the the

results were similar from study to study results of all the included studies are clearly displayed results of the different studies are similar reasons for any variations in results are discussed

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B/ What are the results? 6. What are the overall results of the reviews? HINT: Consider – if you are clear about the review’s ‘bottom line’ results: – what these are (numerically if appropriate) – how were the results expressed (NNT, odds ratio etc.) 7. How precise are the results? HINT: Look at the confidence intervals, if given

C/ Will the results help locally? 8. Can the results be applied to the local population?

Yes 䡺

Can’t tell 䡺

No 䡺

Yes 䡺

Can’t tell 䡺

No 䡺

HINT: Consider whether – the patients covered by the review could be sufficiently different to your population to cause concern – your local setting is likely to differ much from that of the review 9. Were all important outcomes considered?

10. Are the benefits worth the harms and costs? Even if this is not addressed by the review, what do you think?

Health Technology Assessment 2007; Vol. 11: No. 32

Appendix 6 Quality criteria for diagnostic test studies Quality assessment form for studies looking at diagnostic accuracy (Taken from the QUADAS checklist) Title:

First Author: Date:

Question 1

Yes

Was the spectrum of patients representative of the patients who will receive the test in practice?

No Unclear Comments

Question 2

Yes

Were the selection criteria clearly described?

No Unclear Comments

Question 3

Yes

Is the reference standard likely to correctly classify the target condition?

No Unclear Comments

Appendix 6

Question 4

Yes

Is the time period between reference standard and index test short enough to be reasonable?

No Unclear Comments

Question 5

Yes

Did the whole sample or a random selection of the sample receive verification using a reference standard?

No Unclear Comments

Question 6

Yes

Did patients receive the same regardless of the index test result?

No Unclear Comments

Question 7

Yes

Was the reference standard independent of the index test? (i.e. the index test did not form part of the reference standard)

No Unclear Comments

Question 8

Yes

Was the execution of the index test described in sufficient detail to permit replication of the test?

No Unclear Comments

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Question 9

Yes

Was the execution of the reference standard described in sufficient detail to permit its replication?

No Unclear Comments

Question 10

Yes

Were the index test results interpreted without knowledge of the results of the reference standard?

No Unclear Comments

Question 11

Yes

Were the reference standard results interpreted without knowledge of the results of the index test?

No Unclear Comments

Question 12

Yes

Were the same clinical data available when test results were interpreted as would be available when the test is used in practice?

No Unclear Comments

Question 13

Yes

Were uninterpretable/intermediate test results reported?

No Unclear Comments

Appendix 6

Question 14

Yes

Were withdrawals from the study explained?

No Unclear Comments

136

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Appendix 7 Excluded studies and reasons for exclusions Aguero AL, Borria JJ, de Mola M, Asnaghi P, Cansler A, Edelstein S, et al. The audiometric evaluation of Buenos Aires schoolchildren. Boletin de la Oficina Sanitaria Panamericana 1995;119:292–8. [Non-comparative study.] Al Allaf AMY, Ali A, Muneer M. Deafness in children and the need for cochlear implants. [Arabic]. Journal of the Bahrain Medical Society 2003;15:219–22. [Noncomparative study.] Arslan E, Turrini M, Lupi G, Genovese E, Orzan E. Hearing threshold assessment with auditory brainstem response (ABR) and ElectroCochleoGraphy (ECochG) in uncooperative children. Scand Audiol Suppl 1997;46:32–7. [Looking at uncooperative children, general anaesthetic was applied.] Augustsson I, Nilson C, Engstrand I. The preventive value of audiometric screening of preschool and young school-children. Int J Pediatr Otorhinolaryngol 1990;20:51–62. [Non-comparative study.] Babb MJ, Hilsinger RL Jr, Korol HW, Wilcox RD. Modern acoustic reflectometry: accuracy in diagnosing otitis media with effusion. Ear Nose Throat J 2004;83:622–4. [Not looking at screening.] Bamford J, Davis A, Boyle J, Law J, Chapman S, Brown SS, et al. Preschool hearing, speech, language, and vision screening. Qual Health Care 1998;7:240–7. [Audit.] Baumann U, Schorn K. Early detection of pediatric hearing loss. Visual and automated procedures compared. HNO 2001;49:118–25. [Children too young.] Beppu R, Hattori T, Yanagita N. Comparison of TEOAE with Play audiometry for screening hearing problems in children. Auris Nasus Larynx 1997;24:367–71. [Children too young.] Brunner M, Pfeiffer B, Heinrich C, Proschel U. Evaluation of the new Heidelberg preschool screening for auditory perception and language processing. Folia Phoniatr Logop 2005;57:48-58. [Not looking at screening.] Cadman D, Walter SD, Chambers LW, Ferguson R, Szatmari P, Johnson N, et al. Predicting problems in school performance from preschool health, developmental and behavioural assessments. CMAJ 1988;139:31–6. [Not looking at hearing.] Cadman D, Chambers LW, Walter SD, Feldman W, Smith K, Ferguson R. The usefulness of the Denver Developmental Screening Test to predict kindergarten problems in a general community population. Am J Public Health 1984;74:1093–7. [Not looking at hearing.] © Queen’s Printer and Controller of HMSO 2007. All rights reserved.

Combs JT. Predictive value of the angle of acoustic reflectometry. Pediatr Infect Dis J 1991;10:214–16. [Not looking at screening/hearing.] Combs JT. Single vs. double acoustic reflectometry tracings. Pediatr Infect Dis J 1989;8:616–20. [Not looking at screening/hearing.] Dancer J, Burl NT, Waters S. Effects of unilateral hearing loss on teacher responses to the SIFTER. Screening Instrument for Targeting Educational Risk. Am Ann Deaf 1995;140:291–4. [Some of the subjects had previously diagnosed hearing problems.] Dempster JH, Mackenzie K. Clinical role of free-field voice tests in children. Clin Otolaryngol Allied Sci 1992;17:54–6. [Looking at children who had previously been referred to a hearing clinic.] Dempster JH, Mackenzie K. Tympanometry in the detection of hearing impairments associated with otitis media with effusion. Clin Otolaryngol Allied Sci 1991;16:157–9. [Children had already been referred.] Douniadakis DE, Nikolopoulos TP, Tsakanikos MD, Vassiliadis SV, Apostolopoulos NJ. Evaluation of acoustic reflectometry in detecting otitis media in children. Br J Audiol 1993;27:409–14. [Children had already been referred for hearing problems.] Driscoll C, Kei J, McPherson B. Hearing screening for children in community settings using transient evoked otoacoustic emissions. Asia Pac J Speech Lang Hear 2003;8:179–84. [Screening is not comparative.] Driscoll C, Kei J, McPherson B. Transient evoked otoacoustic emissions in 6-year-old school children: a normative study. Scand Audiol Suppl 2000;29:103–10. [Looking at disabled children.] Elliott M, Jones JC, Jones R, Pritchard VG, Robinson BE. An inter-district audit of the school entry medical examination in Cheshire. Public Health 1994;108:203–10. [Not a study, just an audit of medical examinations.] Emmer MB, Silman S. The prediction of hearing loss in persons with cerebral palsy using contralateral acoustic reflex threshold for broad-band noise. Am J Audiol 2003;12:91–5. [Children studied had cerebral palsy.] Finitzo T, Friel-Patti S, Chinn K, Brown O. Tympanometry and otoscopy prior to myringotomy: issues in diagnosis of otitis media. Int J Pediatr Otorhinolaryngol 1992;24:101–10. [Children had recurring otitis media.] Flanary VA, Flanary CJ, Colombo J, Kloss D. Mass hearing screening in kindergarten students. Int J Pediatr Otorhinolaryngol 1999;50:93–8. [Non-comparative study.]

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Appendix 7

Garrubba V, Grandori F, Lamoretti M, Nicolai P, Zanetti D, Antonelli AR. Electric response audiometry in infants and preschool children. Long-term control of the results. Acta Otolaryngol Suppl 1991;482:36–43. [Children all under 4.] Gershel J, Kruger B, Giraudi-Perry D, Chobot J, Rosenberg M, Shapiro IM, et al. Accuracy of the Welch Allyn AudioScope and traditional hearing screening for children with known hearing loss. J Pediatr 1985;106:15–20. [Some children had previously diagnosed hearing problems.] Gray S, Yamauchi T. Preschool screening of speech, language and hearing – model program of early identification and intervention. Clin Res 1976;24:A175. [Non-comparative study.] Guo Y, Yao D. The application of otoacoustic emissions in paediatric hearing screening. Chung-Kuo i Hsueh Ko Hsueh Yuan Hsueh Pao Acta Academiae Medicinae Sinicae 1996;18:284–7 [Used high-risk infants only.] Haggard MP, Wood EJ, Carroll S. Speech, admittance and tone tests in school screening. Reconciling economics with pathology and disability perspectives. Br J Audiol 1984;18:133–53. [Outcomes not relevant.] Heath RW, et al. Hearing dysfunction in Hawaiian preschoolers: its relation to educational achievement and family characteristics. Internal report, January 1987. [Paper could not be retrieved.] Herer GR, Glattke TJ, Rafitis IA, Cummiskey C. Detection of hearing loss in young children and adults using otoacoustic emissions. Folia Phoniatr Logop 1996;48:117–21 [Non-comparative study.] Holmes AE, Jones-Muir KC, Kember FJ. Acoustic reflectometry versus tympanometry in pediatric middle ear screenings. Lang Speech Hear Serv Sch 1989;20:41–9. [Some children had previously diagnosed hearing problems.] Holtby I, Forster DP. Evaluation of pure tone audiometry and impedance screening in infant schoolchildren. J Epidemiol Commun Health 1992;46:21–5. [Children too old.] Jones J, Batchelor L, Gordon N, West M. The preschool medical: an evaluation of this examination and its role in child health surveillance. Child Care Health Dev 1989;15:425–34. [Non-comparative study.] Kaleida PH, Stool SE. Assessment of otoscopists’ accuracy regarding middle-ear effusion. Otoscopic validation. Am J Dis Child 1992;146:433–5. [Physician validation.] Kanasaku M, Suzuki S, Notoya M, et al. The screening level of pure tone audiometry for young children. Audiol Jpn 1977;20:702–8. [Non-comparative study.]

138

Karzon RG. Validity and reliability of tympanometric measures for pediatric patients. J Speech Hear Res 1991;34:386–90. [Children had already been referred for hearing problems.]

Kazanas SG, Maw AR. Tympanometry, stapedius reflex and hearing impairment in children with otitis media with effusion. Acta Otolaryngol 1994;114:410–14 [Children already had diagnosed hearing problems.] Koike KJ, Wetmore SJ. Interactive effects of the middle ear pathology and the associated hearing loss on transient-evoked otoacoustic emission measures. Otolaryngol Head Neck Surg 1999;121:238–44. [Children were deaf and blind and already had suspected ear problems.] Krueger WWO, Ferguson L. A comparison of screening methods in school-aged children. Otolaryngol Head Neck Surg 2002;127:516–19. [Children too old.] Lee DH, Yeo SW. Clinical diagnostic accuracy of otitis media with effusion in children, and significance of myringotomy: diagnostic or therapeutic? J Korean Med Sci 2004;19:739–43 [Children already had OME.] Lous J. Secretory otitis-media in schoolchildren – is screening for secretory otitis-media advisable? Dan Med Bull 1995;42:71–99. [Audit.] McKenzie E, Magian V, Stokes R. A study of the recommended pass/fail criteria for impedance audiometry in a school screening program. J Otolaryngol 1982;11:40–5. [Comparative results not valid for this review.] Mackie K, Dermody P. Use of a Monosyllabic Adaptive Speech Test (MAST) with young children. Research Note. J Speech Hear Res 1986;29:275–81. [Included children with learning difficulties.] Magnusson M, Rasmussen F, Sundelin C. Early identification of children with communication disabilities – evaluation of a screening programme in a Swedish county. Acta Paediatr 1996;85:1319–26. [Children too young.] Maki-Torkko E, Sorri M, Jarvelin MR. Conditions for paediatric hearing screening: a survey in 28 Finnish child welfare clinics. Public Health 1998;112:47–51 [A study looking at nurses and practitioners, not children.] Marriage J, King J, Briggs J, Lutman ME. The reliability of the SCAN test: results from a primary school population in the UK. Br J Audiol 2001;35:199–208. [Children too old.] Matkin ND. Analysis of a recorded test for the measurement of hearing in children. December 1969. [Paper could not be retrieved.] Matusiak M, Wierzbicka M, Szyfter W. [Prevalence of conductive hypoacusis in children aged 5–9 years old from rural area in Poland – prospective screening of healthy subjects] [Polish]. Otolaryngol Polska 2002;56:459–66. [Non-comparative study.] Maw AR, Tiwari RS. Children with glue ear: how do they present? Clin Otolaryngol Allied Sci 1988;13:171–7. [Wrong outcomes.] de Melker RA. Evaluation of the diagnostic value of pneumatic otoscopy in primary care using the results of

Health Technology Assessment 2007; Vol. 11: No. 32

tympanometry as a reference standard. Br J General Pract 1993;43:22–4. [Children included were deaf.] Merer DM, Gravel JS. Screening infants and young children for hearing loss: examination of the CAST procedure. J Am Acad Audiol 1997;8:233–42. [Some children had previously diagnosed hearing loss.] National Institutes of Health. Consensus: early identification of hearing impairment in infants and young children. Summary. Am J Otol 1994;1:130–1. [Not a study.] Ng J, Yun HL. Otoacoustic emissions (OAE) in paediatric hearing screening – the Singapore experience. J Singapore Paediatr Soc 1992;34:1–5. [Children included described as being ‘at risk’.] Okalidou A, Kampanaros M. Teacher perceptions of communication impairment at screening stage in preschool children living in Patras, Greece. Int J Lang Commun Disord 2001;36:489–502. [Looking at prevalence of communication problems, cross-sectional study that only gives one figure for the prevalence of hearing impairment in their study population.] Piskorski P, Keefe DH, Simmons JL, Gorga MP. Prediction of conductive hearing loss based on acoustic ear-canal response using a multivariate clinical decision theory. J Acoust Soc Am 1999;105:1749–64. [Not looking at screening.] Proschel U, Eysholdt U. Evoked otoacoustic emissions in children in relation to middle ear impedance. Folia Phoniatr 1993;45:288–94. [The children already had conductive hearing loss in at least one ear.] Richardson MP, Williamson TJ, Lenton SW, Tarlow MJ, Rudd PT. Otoacoustic emissions as a screening test for hearing impairment in children. Arch Dis Child 1995;72:294–7. [No breakdown of age.] Rothman R, Owens T, Simel DL. Does this child have acute otitis media? [review]. JAMA 2003;290:1633–40. [Not looking at screening.] Sagalovich BM, Shimanskaia EI. Age-related characteristics of dynamic indicators of acoustic impedance of the middle ear in children. Vestn Otorinolaringol 1992;(3):9–13. [Non-comparative.] Scaldwell WA. Prevalence of otis media in Cree and Ojibway school children in six Ontario communities. Journal of American Indian Education 1985;25:1–5. [Not screening.] Schuster M, Kummer P, Hoppe U, Eysholdt U, Weber A, Rosanowski F. Guidelines and their practical application in congenital hearing loss. Gesundheitswesen 2003;65:566–71. [Children already had diagnosed hearing problems.]

Silman S, Silverman CA, Arick DS. Pure-tone assessment and screening of children with middle-ear effusion. J Am Acad Audiol 1994;5:173–82. [Children already had OME.] Stewart MG, Ohlms LA, Friedman EM, Sulek M, Duncan NO III, Fernandez AD, et al. Is parental perception an accurate predictor of childhood hearing loss? A prospective study. Otolaryngol Head Neck Surg 1999;120:340–4. [Children already had OME.] Sturner RA, Green JA, Funk SG. Cognitive-development related to performance on preschool hearing screeningtests. J Dev Behav Pediatr 1983;4:94–8. [Not a comparison of hearing tests.] Swedish Council on Technology Assessment in Health Care. Universal newborn hearing screening – early assessment briefs 2004. [Wrong population.] Takata GS, Chan LS, Morphew T, Mangione-Smith R, Morton SC, Shekelle P. Evidence assessment of the accuracy of methods of diagnosing middle ear effusion in children with otitis media with effusion. Pediatrics 2003;112:1379–87. [Just looking at OME diagnosis.] Taylor CL, Brooks RP. Screening for hearing loss and middle-ear disorders in children using TEOAEs. Am J Audiol 2000;9:50–5. [Children had been referred.] Westerlund M, Sundelin C. Screening for developmental language disability in 3-year-old children. Experiences from a field study in a Swedish municipality. Child Care Health Dev 2000;26:91–110. [Children too young.] Wood EJ, Lutman ME, Fernandes MA. Validation of a screening oto-admittance instrument. Br J Audiol 1982;16:273–5. [Wrong population.] Yasuhara A, Hori A. A comparison of the threedimensional auditory brainstem response and the conventional auditory brainstem response in children. Brain Dev 2002;24:750–7. [Not looking at hearing.] Yockel NJ. A comparison of audiometry and audiometry with tympanometry to determine middle ear status in school-age children. J Sch Nurs 2002;18:287–92. [Noncomparative study.] Zakzouk SM. Epidemiology and etiology of hearing impairment among infants and children in a developing country. Part I. J Otolaryngol 1997;26:335–44. [Noncomparative study.] Zielhuis GA, Gerritsen AAM, Gorissen WHM, Dekker LJ, Rovers MM, van der Wilt GJ, et al. Hearing deficits at school age; the predictive value of otitis media in infants. Int J Pediatr Otorhinolaryngol 1998;44:227–34 [Not looking at screening.]

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Appendix 8 Summary of quality of systematic reviews TABLE 73 Quality of systematic reviews Barlow et al., 199843

NZHTA, 199844

Pirozzo et al., 200347

Yes Yes Yes Yes NA Can’t tell Yes Yes

Yes Can’t tell Can’t tell Yes NA Can’t tell Yes Yes

Was the aim stated clearly? Were the appropriate sort of papers sought? Are the important relevant studies included? Was the quality of the studies appropriately assessed? If the results are combined was it appropriate to do so? Can the results be applied to the local population? Were all important outcomes considered? Are the benefits worth the harms and costs?

Health Technology Assessment 2007; Vol. 11: No. 32

Appendix 9 Quality of primary studies

144

1. 2. 3. 4. 5. 6.

Yes Yes Yes Yes Yes Yes 18

Yes Yes Yes Yes Unclear Yes 12

Yes Yes Yes Yes Yes Yes Yes Yes Yes Yes Yes Yes Yes Yes Yes Yes Yes Yes

Q. 5

Yes Yes Yes Yes Yes Yes 20

Yes Yes Yes Yes Yes Yes Yes Yes Yes Yes Yes Yes Yes Yes Yes Yes Yes Yes

Q. 6

Yes Unclear Yes Unclear Yes Yes 19

Yes Yes Yes Yes Yes Yes Yes Yes Yes Unclear Yes Yes Yes Yes Yes Yes Yes Unclear

Q. 7 Yes Yes Yes No Yes Yes Yes Unclear Yes Unclear Yes Unclear Yes No Yes Yes Yes Unclear

Q. 9 Unclear Unclear No Unclear Yes Unclear Unclear Yes Unclear Unclear Unclear Unclear Yes Unclear Yes Unclear Unclear Unclear

Q. 10

Yes Yes Unclear Yes Yes Unclear Yes Yes Unclear Unclear Unclear Unclear No No Unclear Yes Yes Unclear 16 16 4

Yes Yes Unclear Yes Yes Yes Yes Yes Yes Unclear Yes Yes Yes Yes Yes Yes Yes Unclear

Q. 8

Yes Unclear Unclear Unclear Unclear Unclear 5

Unclear Unclear No Unclear Yes Unclear Unclear Yes Unclear Unclear Unclear Unclear Yes Unclear Yes Unclear Unclear Unclear

Q. 11

Was the spectrum of patients representative of the patients who will receive the test in practice? Were the selection criteria clearly described? Is the reference standard likely to correctly classify the target condition? (PTA has been taken as the most suitable reference standard.) Is the time period between reference standard and index test short enough to be reasonable? Did the whole sample or a random selection of the sample receive verification using a reference standard? Did patients receive the same regardless of the index test result?

Questions

Unclear Yes Unclear Unclear Unclear Unclear 17

Yes Unclear Unclear Unclear Yes No 11

Q. 4

Yes Yes Yes Yes Yes Yes 20

Q. 3

Yes Yes Yes No Yes Yes No Yes Unclear Yes Yes Yes No Yes Yes Yes Yes Yes Yes Yes Yes Yes Yes Yes No Yes Yes Unclear Yes Yes Yes Yes Yes Yes Yes Yes Yes Unclear Yes No Yes Yes Yes Yes Yes Yes Yes Unclear Unclear Yes Yes Unclear Unclear Yes

Q. 2

Yes Yes Yes Yes Yes Yes Yes Yes Yes Yes Yes Yes Yes Yes Yes Yes Yes Yes

Q. 1

Non-English studies. Total number of ‘yes’ responses to each criteria. c Total number of ‘yes’ scores for each study.

Ritchie and Merklein, 197257 FitzZaland and Zink, 198455 Gomes and Lichtig, 200569 Abou Haidar et al., 200568 Hamill, 198858 Hammond et al., 199763 Hind et al., 199965 Holtby et al., 199770 Lyons et al., 200453 Maragno and Teatini, 198366a McCurdy et al.,197671 Nienhuys et al., 199461 Nozza et al., 199772 Olusanya, 200151 Orlando and Frank, 198773 Pang-Ching et al., 199552 Prescott et al., 199950 Rodriguez and Melguizo-Yepez, 199456a Rousch et al., 199259 Rousch and Tait, 198562 Sabo et al., 200054 Schell, 197064a Skurr and Jones, 198175 Square et al., 198560 Totalsb

TABLE 74 Quality of primary studies

Unclear Unclear Unclear Unclear Unclear Unclear 0

Unclear Unclear Unclear Unclear Unclear Unclear Unclear Unclear Unclear Unclear Unclear Unclear Unclear Unclear Unclear Unclear Unclear Unclear

Q. 12

Unclear Unclear Unclear Unclear Unclear Unclear 0

Unclear Unclear Unclear Unclear Unclear Unclear Unclear Unclear Unclear Unclear Unclear Unclear Unclear Unclear Unclear Unclear Unclear Unclear

Q.13

Yes Yes Yes Unclear No Yes 16

Yes Yes Yes No Yes No Yes Yes Yes Unclear No Yes Yes Yes Yes Yes Unclear Unclear

Q.14

continued

10 8 8 NA 5 8

10 9 7 8 11 9 10 11 9 NA 9 9 11 8 12 9 8 NA

Scorec

Appendix 9

7. 8. 9. 10. 11. 12. 13. 14.

Was the reference standard independent of the index test? (i.e. the index test did not form part of the reference standard) Was the execution of the index test described in sufficient detail to permit replication of the test? Was the execution of the reference standard described in sufficient detail to permit its replication? Were the index test results interpreted without knowledge of the results of the reference standard? Were the reference standard results interpreted without knowledge of the results of the index test? Were the same clinical data available when test results were interpreted as would be available when the test is used in practice? Were uninterpretable/intermediate test results reported? Were any withdrawals from the study explained?

TABLE 74 Quality of primary studies (cont’d)

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145

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Appendix 10 Two by two tables for sensitivity and specificity where available Ritchie and Merklein57 TABLE 75 VASC screen (protocol 1) versus PTA Failed intervention

Passed intervention

5 21

116 20

Passed control Failed control

Sensitivity 51% Specificity 96%

TABLE 76 VASC screen (protocol 2) versus PTA Failed intervention

Passed intervention

8 24

113 17

Passed control Failed control

Sensitivity 59% Specificity 93%

FitzZaland and Zink55 TABLE 77 Pure tone sweep versus combination of tests

Passed control Failed control

Failed intervention

Passed intervention

39 128

3334 9

Sensitivity 93% Specificity 99%

TABLE 78 Rhinne audiometric test versus combination of tests

Passed control Failed control

Failed intervention

Passed intervention

11 124

3362 13

Sensitivity 91% Specificity 99.67%

TABLE 79 Tympanometry (negative pressure 艌–150 mm) versus combination of tests

Passed control Failed control

Failed intervention

Passed intervention

298 127

3057 10

Sensitivity 93% Specificity 91%

Appendix 10

TABLE 80 Tympanometry (negative pressure 艌–175 mm) versus combination of tests Failed intervention

Passed intervention

183 127

3190 10

Passed control Failed control

Sensitivity 91% Specificity 99%

TABLE 81 Tympanometry (negative pressure 艌–200 mm) versus combination of tests Failed intervention

Passed intervention

73 125

3300 12

Failed intervention

Passed intervention

0 55

3373 82

Passed control Failed control

Sensitivity 91% Specificity 99%

TABLE 82 Tympanometry type B only

Passed control Failed control

Sensitivity 40% Specificity 100%

Hammond and colleagues63 TABLE 83 Questionnaire versus combination of tests Failed intervention

Passed intervention

259 10

227 8

Passed control Failed control

Sensitivity 56% Specificity 52%

Hamill58 TABLE 84 VASC versus pure tone sweep test Failed intervention

Passed intervention

23 39

508 6

Passed control Failed control

Sensitivity 87% Specificity 96%

Lyons and colleagues53 TABLE 85 Tympanometry versus pure tone sweep test

Passed control Failed control

148

Failed intervention

Passed intervention

171 94

1725 16

Sensitivity 85% Specificity 91%

Health Technology Assessment 2007; Vol. 11: No. 32

TABLE 86 DPOAE (protocol 1) versus pure tone sweep test Failed intervention

Passed intervention

241 273

1484 8

Passed control Failed control

Sensitivity 97% Specificity 86%

TABLE 87 DPOAE (protocol 2) versus pure tone sweep test Failed intervention

Passed intervention

289 273

1436 8

Passed control Failed control

Sensitivity 97% Specificity 83%

TABLE 88 DPOAE (protocol 3) versus pure tone sweep test Failed intervention

Passed intervention

440 277

1285 4

Passed control Failed control

Sensitivity 98% Specificity 74%

TABLE 89 DPOAE (protocol 4) versus pure tone sweep test Failed intervention

Passed intervention

92 269

1633 12

Passed control Failed control

Sensitivity 96% Specificity 95%

Maragno and Teatini66 TABLE 90 SVEP test versus hearing assessment

Passed control Failed control

Failed intervention

Passed intervention

0 31

48 2

Sensitivity 100% Specificity 94%

McCurdy and colleagues71 TABLE 91 Tympanometry plus stapedius reflex versus PTA

Passed control Failed control

Failed intervention

Passed intervention

24 57

45 23

Sensitivity 71% Specificity 65%

Appendix 10

Olusanya51 TABLE 92 Questionnaire versus pure tone audiometry Failed intervention

Passed intervention

15 23

291 45

Failed intervention

Passed intervention

50 25

253 25

Failed intervention

Passed intervention

116 28

193 22

Failed intervention

Passed intervention

106 33

429 5

Failed intervention

Passed intervention

48 24

487 14

Passed control Failed control

Sensitivity 34% Specificity 95%

TABLE 93 Tympanometry versus PTA

Passed control Failed control

Sensitivity 50% Specificity 83%

TABLE 94 Otoscopy versus PTA

Passed control Failed control

Sensitivity 56% Specificity 62.4%

Sabo and colleagues54 TABLE 95 Pure tone sweep test versus PTA

Passed control Failed control

Sensitivity 87% Specificity 80%

TABLE 96 TEOAE versus PTA

Passed control Failed control

Sensitivity 63% Specificity 91%

Square and colleagues60 TABLE 97 Bone conduction versus impedance audiometry

Passed control Failed control

150

Failed intervention

Passed intervention

56 14

4 39

Sensitivity 26% Specificity 6.6%

Health Technology Assessment 2007; Vol. 11: No. 32

Appendix 11 Economic search strategies MEDLINE 1966 to August week 3 2005 Search date: 2 August 2005 Number of records: 74 Hearing loss$.mp or exp Hearing loss/ or (hearing adj (disorder$ or difficult$ or problem$ or impair$)).mp or Hearing Disorders/ AND exp child, preschool/ or school entry.mp or exp Child Development/ or early detect$.mp or infant school$.mp or exp Schools, Nursery/ or kindergarten$.mp or exp Child Day Care Centers/ or exp Nurseries/ or nursery school$.mp AND screen$.mp or exp Mass Screening/ AND (school entry adj3 (screen$ or exam$)).mp or (medical exam$ adj2 school$).mp AND economics.mp or exp ECONOMICS, NURSING/ or exp ECONOMICS, MEDICAL/ or exp ECONOMICS/ or exp ECONOMICS, HOSPITAL/ or exp ECONOMICS, PHARMACEUTICAL/ or (econom$ or cost or costs or costly or costing or costed or price or prices or pricing or pharmacoeconomic$).tw or (expenditure$ not energy).tw or (value adj1 money).tw or budget$.tw or cost-effectiveness.mp or cost utili$.mp or cost benefit.mp or exp Cost-Benefit Analysis/ or cost minimi$.mp or exp Health Care Costs/ or economic evaluation$.mp or exp ‘Costs and Cost Analysis’/ or financ$.mp or exp Resource Allocation/ or health resource allocation.mp or Health Resources/ or health resource utilization.mp or preference?.ab,ti,kw or qaly?.ab,ti,kw or quality adjusted.ab,ti,kw or (utility or utilities).ab,ti,kw

EMBASE 1980 to 2005 week 31 Search date: 2 August 2005 Number of records: 38 Hearing loss$.mp or exp Hearing loss/ or (hearing adj (disorder$ or difficult$ or problem$ or impair$)).mp or Hearing Disorders/ AND school entry.mp or (pre adj school).mp or (nursery adj school$).mp or exp Nursery School/ or kindergarten$.mp or exp KINDERGARTEN/ or exp Day Care/ or infant school$.mp early © Queen’s Printer and Controller of HMSO 2007. All rights reserved.

detect$.mp or exp Child Development/ AND screen$.mp or exp MASS SCREENING/ or exp SCREENING/ or exp AUDITORY SCREENING/ or exp SCREENING TEST/ AND (school entry adj3 (screen$ or exam$)).mp or (medical exam$ adj2 school$).mp AND ‘Cost Benefit Analysis’/ or ‘Cost-effectiveness Analysis’/ or ‘Cost Minimization Analysis’/ or ‘Cost Utility Analysis’/ or Economic Evaluation/ or (cost or costs or costed or costly or costing).tw or (economic$ or pharmacoeconomic$ or price$ or pricing).tw or (technology adj assessment$).tw

CINAHL 1982 to August 2005 week 5 Search date: 2 August 2005 Number of records: 32 Hearing loss$.mp or exp Hearing loss/ or (hearing adj (disorder$ or difficult$ or problem$ or impair$)).mp or Hearing Disorders/ AND school entry.mp or (pre adj school).mp or (nursery adj school$).mp or exp Schools, Nursery/ or kindergarten$.mp or exp Day Care/ or infant school$.mp or early detect$.mp or exp Child Development/ AND (school entry adj3 (screen$ or exam$)).mp or (medical exam$ adj2 school$).mp AND exp Health Screening/ or screen$.mp or exp Hearing Screening/ AND (cost or costs or costed or costly or costing).tw or (economic$ or pharmacoeconomic$ or price$ or pricing).tw or (technology adj assessment$).tw or cost benefit analysis.mp or exp ‘Cost Benefit Analysis’/ or costeffectiveness.mp or cost minimization.mp or exp Health Care Costs/ or cost utility.mp or economic evaluation$.mp or exp ‘Economic Aspects of Illness’/ or exp ECONOMICS/ or exp ECONOMICS, PHARMACEUTICAL/ or health resource allocation.mp or exp Health Resource Allocation/ or health resource utilization.mp or exp Health Resource Utilization/

Cochrane Library (Wiley) NHS EED 2005 Issue 2 Search date: 28 July 2005 Number of records: 12

151

Appendix 11

Exp hearing loss/ or hearing next loss or hearing next disorder* or hearing next difficult* or hearing next problem* or hearing next impair* or exp hearing disorders/ AND screening or exp mass screening/ AND school next entr* or early detect* or infant next school* or nursery next school* or kindergarten* or exp schools, nursery/ or exp Child Day Care Centers/ or exp Child, Preschool/ or exp Child Development/ OR school next entr* or medical near/3 exam* near/3 school*

OHE HEED July 2005 issue Search date: 26 July 2005 Number of records: 18

ECONLIT

Hearing AND screen* AND child*

1969–2002 and 2003–2005

Total references (after de-duplication): 164

Search date: 28 July 2005 Number of records: 11

152

Hearing loss* or hearing disorder* or hearing difficult* or hearing problem* or hearing impair* or deafness or hypoacusis or hypacusis or hard of hearing of hard-of-hearing

Health Technology Assessment 2007; Vol. 11: No. 32

Appendix 12 Subsequent management intervention search strategies MEDLINE

EMBASE

1966 to November week 3 2005

1980 to 2006 week 3

Search date: 17 November 2005 Number of records: 397

Search date: 16 January 2006 Number of records: 190

Hearing loss$.mp or exp Hearing loss/ or (hearing adj (disorder$ or difficult$ or problem$ or impair$)).mp or Hearing Disorders/ or deaf$.mp or otitis media with effusion.mp or exp Otitis Media with Effusion/ or OME.mp or glue ear.mp or (hard adj1 hearing).mp AND economics.mp or exp ECONOMICS, NURSING/ or exp ECONOMICS, MEDICAL/ or exp ECONOMICS/ or exp ECONOMICS, HOSPITAL/ or exp ECONOMICS, PHARMACEUTICAL/ or (econom$ or cost or costs or costly or costing or costed or price or prices or pricing or pharmacoeconomic$).tw or (expenditure$ not energy).tw or (value adj1 money).tw or budget$.tw or cost-effectiveness.mp or cost utili$.mp or cost benefit.mp or exp Cost-Benefit Analysis/ or cost minimi$.mp or exp Health Care Costs/ or economic evaluation$.mp or exp ‘Costs and Cost Analysis’/ or financ$.mp or exp Resource Allocation/ or health resource allocation.mp or Health Resources/ or health resource utilization.mp or preference?.ab,ti,kw or qaly?.ab,ti,kw or quality adjusted.ab,ti,kw or (utility or utilities).ab,ti,kw AND hearing aid$.mp or exp Hearing Aids/ or cochlear implant$.mp or exp Cochlear Implants/ or exp ‘Rehabilitation of Hearing Impaired’/ or hearing tactic$.mp or autoinflation.mp or exp Middle Ear Ventilation/ or grommet$.mp or tympanostomy.mp or myringotomy.mp or adenoidectomy.mp or exp ADENOIDECTOMY/ or exp Language Therapy/ or exp Speech Therapy/ or speech language therapy.mp or ear nose throat.mp or exp EDUCATION/ or education$.mp or exp EDUCATION, SPECIAL/ or exp Teaching/ or teach$.mp or exp Comprehensive Health Care/ or audiolog$.mp or otolaryngology$.mp or pediatric$.mp or clinician$.mp or exp NURSE CLINICIANS/ or exp Nursing Staff/ AND limit to (‘newborn infant (birth to 1 month)’ or ‘infant (1 to 23 months)’ or ‘preschool child (2 to 5 years)’ or ‘child (6 to 12 years)’)

‘hearing loss’/exp or ‘hearing loss’ or ‘hearing disorder’/exp or ‘hearing disorder’ or ‘hearing difficulty’/exp or ‘hearing difficulty’ or ‘hearing problem’ or ‘hearing impairment’/exp or ‘hearing impairment’ or deaf* or ‘otitis media with effusion’/exp or ‘otitis media with effusion’ or ome or ‘glue ear’/exp or ‘glue ear’ or ‘hard *1 hearing’ AND ‘cost benefit analysis’/exp or ‘cost benefit analysis’ or ‘cost-effectiveness analysis’/exp or ‘cost-effectiveness analysis’ or ‘cost minimization analysis’/exp or ‘cost minimization analysis’ or ‘economic evaluation’/exp or ‘economic evaluation’ or (‘cost’/exp or ‘cost’) or costs or costed or costly or costing or economic* or pharmacoeconomic* or price or pricing or ‘technology *3 assessment’ or ‘health resources’/exp or ‘health resources’ or ‘quality of life’/exp or ‘quality of life’ AND ‘hearing aid’/exp or ‘hearing aid’ or ‘cochlear prosthesis’/exp or ‘cochlear prosthesis’ or ‘auditory rehabilitation’/exp or ‘auditory rehabilation’ or autoinflation or ‘middle ear ventilation’/exp or ‘middle ear ventilation’ or ‘tympanostomy tube’/exp or ‘tympanostomy tube’ or grommet* or ‘myringotomy’/exp or ‘myringotomy’ or adenoidectomy’/exp or ‘adenoidectomy’ or ‘speech therapy’/exp or ‘speech therapy’ or ‘otorhinolaryngology’/exp or ‘otorhinolaryngology’ or ‘ear nose throat surgery’/exp or ‘ear nose throat surgery’ or ‘education’/exp or ‘education’ or ‘teaching’/exp or ‘teaching’ or ‘audiology’/exp or ‘audiology’ or pediatric* or nurs* AND [embase]/lim AND ([newborn]/lim or [infant]/lim or [preschool]/lim or [school]/lim)

CINAHL 1982 to December 2005 week 1 Search date: 1 December 2005 Number of records: 263 Hearing loss$.mp or exp Hearing loss/ or (hearing adj (disorder$ or difficult$ or problem$ or

153

Appendix 12

impair$)).mp or Hearing Disorders/ or deaf$.mp or exp Deafness/ or otitis media with effusion.mp or exp Otitis Media with Effusion/ or OME.mp or glue ear.mp or (hard adj1 hearing).mp AND (cost or costs or costed or costly or costing).tw or (economic$ or pharmacoeconomic$ or price$ or pricing).tw or (technology adj assessment$).tw or cost benefit analysis.mp or exp ‘Cost Benefit Analysis’/ or cost-effectiveness.mp or cost minimization.mp or exp Health Care Costs/ or cost utility.mp or economic evaluation$.mp or exp ‘Economic Aspects of Illness’/ or health resource allocation.sh,mp or health resource utilization.sh,mp or preference?.mp or exp Life Expectancy/ or exp ‘Outcomes (Health Care)’/ or exp Quality of Life’/ or qaly?.mp or exp Health Status/ or quality adjusted.mp or (utility or utilities).mp AND hearing aid$.mp or exp Hearing Aids/ or cochlear implant$.mp or exp Cochlear Implant/ or Communication Skills/ or exp ‘Rehabilitation of Hearing Impaired’/ or hearing tactic$.mp or Conversation/ or Counseling/ or autoinflation.mp or exp Middle Ear Ventilation/ or grommet$.mp or tympanostomy.mp or myringotomy.mp or adenoidectomy.mp or exp ADENOIDECTOMY/ or exp Education, Speech-Language Pathology/ or exp ‘Rehabilitation, Speech and Language’/ or speech language therapy.mp or exp Speech Therapy/ or exp ‘Education, Continuing (Credit)’/ or exp Surgery, Otorhinolagyngologic/ or exp Specialties, Nursing/ or ear nose throat.mp or exp DEAF EDUCATION/ or exp PATIENT EDUCATION/ or exp PARENTING EDUCATION/ or education$.mp or exp EDUCATION, SPECIAL/ or exp ‘OUTCOMES OF EDUCATION’/ or exp EDUCATION, CONTINUING/ or exp EDUCATION, AUDIOLOGY/ or exp EDUCATION, SPEECHLANGUAGE PATHOLOGY/ or exp EDUCATION/ or teach$.mp or audiolog$.mp or otolaryngolog$.mp or exp PEDIATRICS/ or pediatric$.mp or clinician$.mp AND limit to (newborn infant or infant or preschool child or child )

Cochrane Library (Wiley internet version) NHS EED 2006 Issue 1 Search date: 31 January 2006 Number of records: 69 Hearing next (loss or disorder* or difficulty or problem or impairment) or deaf or otitis next media or OME or glue next ear or hard near/2 hearing AND hearing next aid* or cochlear next implant* or rehabilitation near/2 hearing or hearing next tactics or autoinflation or middle next ear next ventilation or grommet* or tympanostomy or myringotomy or adenoidectomy or language next therapy or speech next therapy or ear next nose or education or teaching or comprehensive next health or audiology or otolaryngology or paediatrician* or clinician* AND restrict to NHS EED database

ECONLIT 1969–2002 and 2003–2005/12 Search date: 11 January 2006 Number of records: 39 hearing or deaf* or otitis AND manage* or treat* or intervene* or otolaryngology* or audiolog* or speech or rehabilit* or nurs* or aid* or implant* or grommet* or autoinflation or tympanostomy or myringotomy or adenoid* or language

OHE HEED January 2006 issue Search date: 10 January 2006 Number of records: 189 hearing or deaf* or otitis AND manage* or treat* or intervene* or otolaryngology* or audiolog* or speech or rehabilit* or nurs* or aid* or implant* or grommet* or autoinflation or tympanostomy or myringotomy or adenoid* or language Total references (after de-duplication): 960

154

Health Technology Assessment 2007; Vol. 11: No. 32

Appendix 13 Inclusion/exclusion economic data form Trial author and date:

Ref. no: Yes

1. Based on primary data collection or systematic review? 2. Children 4–6 years old, undergoing any of the following interventions? Pure tone audiometry Questionnaires Reflectometry Otoadmittance tests Speech audiometry Otoacoustic emissions Medical examinations (which entail a hearing screen) 3. Including at least two of the following interventions? Pure tone audiometry Tympanometry Acoustic reflex Otoadmittance tests Auditory brainstem response Medical examinations (which entail a hearing screen) Speech perception tests Distraction test Behavioural test Questionnaires Otoacoustic emissions No screen 4. Assessing any of the following outcomes? Year with no or mild/moderate disability due to hearing loss Year with moderate or severe disability due to hearing loss Quality-adjusted life-year gained Utility measure Health status measure 5. Resource use and costs and utilities associated with screening programmes and subsequent management? 6. Report resource use and cost separately? 7. Report sufficient detail to extract costs and outcome data relevant to each alternative comparison of screening programmes?

Health Technology Assessment 2007; Vol. 11: No. 32

Appendix 14 Inclusion/exclusion subsequent interventions data form Trial author and date:

Ref. no: Yes

1. Based on primary data collection or systematic review? 2. Children from birth to 12 years of age, undergoing any of the following interventions? Hearing aids Autoinflation Middle ear ventilation Myringotomy Adenoidectomy Speech and language therapy Hearing tactics (family, community, school) Referral to specialists Cochlear implantation 3. Assessing any of the following outcomes? Year with no or mild/moderate disability due to hearing loss Year with moderate or severe disability due to hearing loss Quality-adjusted life-year gained Utility measure Health status measure 4. Resource use and costs and utilities associated with subsequent management interventions? 5. Report resource use and cost separately?

Health Technology Assessment 2007; Vol. 11: No. 32

Appendix 15 Economic data extraction form Economic Data Extraction Form General Information Paper Reference No. Author/Year:

Reviewer ID:

Date:

Title: Sub Title: Journal: Source of funding: Notes/Comments:

Study Characteristics Health Technology: Comparator: Type of Intervention

Economic Study Type

Perspective

Primary Prevention Secondary Prevention Screening Diagnosis Treatment Rehabilitation Palliative Care Other (Please Specify) Not Reported Hypothesis/Study Question:

Cost-effectiveness Analysis Cost-utility Analysis Cost-benefit Analysis Cost-consequence Analysis Cost-Study Not Reported

NHS Societal Hospital Not Stated Other (Please Specify) Setting:

Study Population:

Dates to which Data Relate

Modelling

Effectiveness Evidence Resource Use

Was a model used? Yes No If yes state purpose and type:

Price Year

159

Appendix 15

Source of Data Source of Effectiveness Data Single Study Synthesis of Prev. Pub.

Source of Cost Data Actual Source Literature Source

Link between Effectiveness and Costs

Effectiveness data from a single study Study Sample:

Study design:

Power calculation

RCT

Duration of follow-up:

Number subjects in intervention group

Non-RCT with concurrent controls

Loss to follow-up:

Number subjects in control group

Cohort study

Any blinding for assessment of outcomes:

Recruitment rate

Historical controls

Analysis of clinical study:

Number excluded from study

Before and after study

Treatment completers

Method of sample selection:

Case series

Intention to treat

Other (specify)

Effectiveness results:

Not reported Number of centres Effectiveness data from a synthesis of previous studies (model) Study inclusion criteria:

160

Study designs included:

Number of primary studies included:

Health Technology Assessment 2007; Vol. 11: No. 32

Study exclusion criteria reported:

RCT

Method of combination of primary studies:

Sources searched reported:

Non-RCT with concurrent controls

Meta-analysis

Criteria used to judge validity: Cohort study

Narrative method

Concealment of randomisation

Historical controls

Other (specify)

Blind assessment

Before and after study

Results of the review:

Low drop out rates

Case series

Other (specify)

Not reported

Appendix 15

Economic Evaluation Measure of Benefits used in the Economic Analysis

No Measure of Benefit (CCA or CMA) Direct costs: Health service

Direct costs: Patient

Estimation of Direct Costs Based On: A Guess Actual Data Derived using Modelling Other Not Reported Estimation of Patient Direct Costs Based On: A Guess Actual Data Derived using Modelling Other Not Reported

Source of Direct Cost Data:

Discounting Undertaken?

Price Year: Yes No Currency:

162

Discount Rate

Health Technology Assessment 2007; Vol. 11: No. 32

Economic Evaluation (continued) Indirect Costs

Estimation of Indirect Costs Based On:

A Guess Actual Data Derived using Modelling Other Not Reported Source of Indirect Cost Data

Discounting Undertaken?

Price Year: Yes No Currency:

Discount Rate:

Conversion Rates Used:

Statistical/Sensitivity Analyses Statistical Tests Carried Out?

Types of test used in Analysis of Costs:

Yes No

Appendix 15

Type of Sensitivity Analysis:

Areas of Uncertainty Tested:

One-way Analysis Two-way Analysis Multi-way Analysis Threshold Analysis Analysis of Extremes Probabilistic Analysis Other Not Reported Not Carried Out

Results Clinical Outcome/Benefit:

Duration of Benefits: Cost results:

Side Effects Considered?

Cost of Adverse Events Considered? Y N How were the estimates of Costs and Results of Synthesis of Costs and Benefits: Benefits Combined? Cost/Life Saved

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Cost/Life Gained Cost/QALY

Not Relevant

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Net Benefit Incremental Net Benefit Other Not Combined Author’s Conclusions:

Reviewer’s Conclusions:

Overall assessment of study quality:

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Appendix 16 Description of included papers

168

Comparators

Authors: Holtby and Forster, 199289 Setting: Infant or primary schools, UK Perspective: NHS Funding: Not stated

Impedance measurements against PTA

Authors: Driscoll, et al., NA 200088 Setting: Audiological clinic, Australia Perspective: Not stated Funding: University of Queensland

Study characteristics

TABLE 98 Description of included papers

Study: Effectiveness Health technology: Impedance measurements Type of intervention: Screening Population: 6-year-old schoolchildren Sources: Single study for the effectiveness data. The source for the cost data was not reported

Study: Study Health technology: TEOAE and PTA Type of intervention: Screening Population: Infants with a mean age of 2 months (range 1.5–2.5) and children with a mean age of 6.2 years (range 5.2–7.9) Sources: Actual study, synthesis of data from previous publications for effectiveness

Economic study type and population

where S = salary of screener, R = rate of tests, C = cost of equipment, M = cost of annual maintenance, L = expected life of equipment in years, and N = number of children screened per instrument per year

Date: 1988 Effectiveness: Sensitivity, specificity, repeatability, predictive value, screening rate Resource use: Nurses, audiometers Price year: NR Direct costs: Salary, cost of equipment, cost of annual maintenance Indirect costs: NR Currency: UK sterling Link between effectiveness and costs: Cost per child screened per instrument = S/R + (C + (M*L)/(N*L))

The total annual cost (TC) of the programmes was determined using the following equation: TC = CPC × N. The cost per hearing impaired child (CPHIa) was calculated using the equation: CPHIa = TC/#HL (where #HL is the number of children diagnosed with a unilateral or bilateral, sensorineural or mixed hearing impairment of at least a moderate degree (3FA 艌41)). The cost per child with sensorineural/mixed/conductive hearing impairment was calculated based on the number of children diagnosed with any hearing impairment

where S = salary of screener, R = rate of tests/hour, C = cost of initial equipment purchase, M = cost of annual maintenance, L = expected life of equipment in years, and N = number of cases per year

Date: 2000 Effectiveness: Rate of tests per hour Resource use: Audiologists, equipment Price year: Not clear Direct costs: Hourly wage of a full-time audiologist, cost of equipment, cost of annual equipment maintenance, cost per child screened Indirect costs: NR Currency: Australian dollars Link between effectiveness and costs: Cost per child = S/R + (C + (M*L)/(N*L))

Data

Appendix 16

Health Technology Assessment 2007; Vol. 11: No. 32

Health Technology Assessment reports published to date Volume 1, 1997 No. 1 Home parenteral nutrition: a systematic review. By Richards DM, Deeks JJ, Sheldon TA, Shaffer JL. No. 2 Diagnosis, management and screening of early localised prostate cancer. A review by Selley S, Donovan J, Faulkner A, Coast J, Gillatt D. No. 3 The diagnosis, management, treatment and costs of prostate cancer in England and Wales. A review by Chamberlain J, Melia J, Moss S, Brown J. No. 4 Screening for fragile X syndrome. A review by Murray J, Cuckle H, Taylor G, Hewison J. No. 5 A review of near patient testing in primary care. By Hobbs FDR, Delaney BC, Fitzmaurice DA, Wilson S, Hyde CJ, Thorpe GH, et al. No. 6 Systematic review of outpatient services for chronic pain control. By McQuay HJ, Moore RA, Eccleston C, Morley S, de C Williams AC. No. 7 Neonatal screening for inborn errors of metabolism: cost, yield and outcome. A review by Pollitt RJ, Green A, McCabe CJ, Booth A, Cooper NJ, Leonard JV, et al. No. 8 Preschool vision screening. A review by Snowdon SK, Stewart-Brown SL. No. 9 Implications of socio-cultural contexts for the ethics of clinical trials. A review by Ashcroft RE, Chadwick DW, Clark SRL, Edwards RHT, Frith L, Hutton JL. No. 10 A critical review of the role of neonatal hearing screening in the detection of congenital hearing impairment. By Davis A, Bamford J, Wilson I, Ramkalawan T, Forshaw M, Wright S.

No. 11 Newborn screening for inborn errors of metabolism: a systematic review. By Seymour CA, Thomason MJ, Chalmers RA, Addison GM, Bain MD, Cockburn F, et al. No. 12 Routine preoperative testing: a systematic review of the evidence. By Munro J, Booth A, Nicholl J. No. 13 Systematic review of the effectiveness of laxatives in the elderly. By Petticrew M, Watt I, Sheldon T. No. 14 When and how to assess fast-changing technologies: a comparative study of medical applications of four generic technologies. A review by Mowatt G, Bower DJ, Brebner JA, Cairns JA, Grant AM, McKee L.

Volume 2, 1998 No. 1 Antenatal screening for Down’s syndrome. A review by Wald NJ, Kennard A, Hackshaw A, McGuire A. No. 2 Screening for ovarian cancer: a systematic review. By Bell R, Petticrew M, Luengo S, Sheldon TA. No. 3 Consensus development methods, and their use in clinical guideline development. A review by Murphy MK, Black NA, Lamping DL, McKee CM, Sanderson CFB, Askham J, et al. No. 4 A cost–utility analysis of interferon beta for multiple sclerosis. By Parkin D, McNamee P, Jacoby A, Miller P, Thomas S, Bates D. No. 5 Effectiveness and efficiency of methods of dialysis therapy for end-stage renal disease: systematic reviews. By MacLeod A, Grant A, Donaldson C, Khan I, Campbell M, Daly C, et al.

No. 6 Effectiveness of hip prostheses in primary total hip replacement: a critical review of evidence and an economic model. By Faulkner A, Kennedy LG, Baxter K, Donovan J, Wilkinson M, Bevan G. No. 7 Antimicrobial prophylaxis in colorectal surgery: a systematic review of randomised controlled trials. By Song F, Glenny AM. No. 8 Bone marrow and peripheral blood stem cell transplantation for malignancy. A review by Johnson PWM, Simnett SJ, Sweetenham JW, Morgan GJ, Stewart LA. No. 9 Screening for speech and language delay: a systematic review of the literature. By Law J, Boyle J, Harris F, Harkness A, Nye C. No. 10 Resource allocation for chronic stable angina: a systematic review of effectiveness, costs and cost-effectiveness of alternative interventions. By Sculpher MJ, Petticrew M, Kelland JL, Elliott RA, Holdright DR, Buxton MJ. No. 11 Detection, adherence and control of hypertension for the prevention of stroke: a systematic review. By Ebrahim S. No. 12 Postoperative analgesia and vomiting, with special reference to day-case surgery: a systematic review. By McQuay HJ, Moore RA. No. 13 Choosing between randomised and nonrandomised studies: a systematic review. By Britton A, McKee M, Black N, McPherson K, Sanderson C, Bain C. No. 14 Evaluating patient-based outcome measures for use in clinical trials. A review by Fitzpatrick R, Davey C, Buxton MJ, Jones DR.

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No. 15 Ethical issues in the design and conduct of randomised controlled trials. A review by Edwards SJL, Lilford RJ, Braunholtz DA, Jackson JC, Hewison J, Thornton J. No. 16 Qualitative research methods in health technology assessment: a review of the literature. By Murphy E, Dingwall R, Greatbatch D, Parker S, Watson P.

No. 6 Assessing the costs of healthcare technologies in clinical trials. A review by Johnston K, Buxton MJ, Jones DR, Fitzpatrick R.

No. 17 The costs and benefits of paramedic skills in pre-hospital trauma care. By Nicholl J, Hughes S, Dixon S, Turner J, Yates D.

No. 7 Cooperatives and their primary care emergency centres: organisation and impact. By Hallam L, Henthorne K.

No. 18 Systematic review of endoscopic ultrasound in gastro-oesophageal cancer. By Harris KM, Kelly S, Berry E, Hutton J, Roderick P, Cullingworth J, et al.

No. 8 Screening for cystic fibrosis. A review by Murray J, Cuckle H, Taylor G, Littlewood J, Hewison J.

No. 19 Systematic reviews of trials and other studies. By Sutton AJ, Abrams KR, Jones DR, Sheldon TA, Song F. No. 20 Primary total hip replacement surgery: a systematic review of outcomes and modelling of cost-effectiveness associated with different prostheses. A review by Fitzpatrick R, Shortall E, Sculpher M, Murray D, Morris R, Lodge M, et al.

Volume 3, 1999 No. 1 Informed decision making: an annotated bibliography and systematic review. By Bekker H, Thornton JG, Airey CM, Connelly JB, Hewison J, Robinson MB, et al. No. 2 Handling uncertainty when performing economic evaluation of healthcare interventions. A review by Briggs AH, Gray AM. No. 3 The role of expectancies in the placebo effect and their use in the delivery of health care: a systematic review. By Crow R, Gage H, Hampson S, Hart J, Kimber A, Thomas H.

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No. 5 Methods for evaluating area-wide and organisation-based interventions in health and health care: a systematic review. By Ukoumunne OC, Gulliford MC, Chinn S, Sterne JAC, Burney PGJ.

No. 4 A randomised controlled trial of different approaches to universal antenatal HIV testing: uptake and acceptability. Annex: Antenatal HIV testing – assessment of a routine voluntary approach. By Simpson WM, Johnstone FD, Boyd FM, Goldberg DJ, Hart GJ, Gormley SM, et al.

No. 9 A review of the use of health status measures in economic evaluation. By Brazier J, Deverill M, Green C, Harper R, Booth A. No. 10 Methods for the analysis of quality-oflife and survival data in health technology assessment. A review by Billingham LJ, Abrams KR, Jones DR. No. 11 Antenatal and neonatal haemoglobinopathy screening in the UK: review and economic analysis. By Zeuner D, Ades AE, Karnon J, Brown J, Dezateux C, Anionwu EN. No. 12 Assessing the quality of reports of randomised trials: implications for the conduct of meta-analyses. A review by Moher D, Cook DJ, Jadad AR, Tugwell P, Moher M, Jones A, et al. No. 13 ‘Early warning systems’ for identifying new healthcare technologies. By Robert G, Stevens A, Gabbay J. No. 14 A systematic review of the role of human papillomavirus testing within a cervical screening programme. By Cuzick J, Sasieni P, Davies P, Adams J, Normand C, Frater A, et al. No. 15 Near patient testing in diabetes clinics: appraising the costs and outcomes. By Grieve R, Beech R, Vincent J, Mazurkiewicz J. No. 16 Positron emission tomography: establishing priorities for health technology assessment. A review by Robert G, Milne R.

No. 17 (Pt 1) The debridement of chronic wounds: a systematic review. By Bradley M, Cullum N, Sheldon T. No. 17 (Pt 2) Systematic reviews of wound care management: (2) Dressings and topical agents used in the healing of chronic wounds. By Bradley M, Cullum N, Nelson EA, Petticrew M, Sheldon T, Torgerson D. No. 18 A systematic literature review of spiral and electron beam computed tomography: with particular reference to clinical applications in hepatic lesions, pulmonary embolus and coronary artery disease. By Berry E, Kelly S, Hutton J, Harris KM, Roderick P, Boyce JC, et al. No. 19 What role for statins? A review and economic model. By Ebrahim S, Davey Smith G, McCabe C, Payne N, Pickin M, Sheldon TA, et al. No. 20 Factors that limit the quality, number and progress of randomised controlled trials. A review by Prescott RJ, Counsell CE, Gillespie WJ, Grant AM, Russell IT, Kiauka S, et al. No. 21 Antimicrobial prophylaxis in total hip replacement: a systematic review. By Glenny AM, Song F. No. 22 Health promoting schools and health promotion in schools: two systematic reviews. By Lister-Sharp D, Chapman S, Stewart-Brown S, Sowden A. No. 23 Economic evaluation of a primary carebased education programme for patients with osteoarthritis of the knee. A review by Lord J, Victor C, Littlejohns P, Ross FM, Axford JS.

Volume 4, 2000 No. 1 The estimation of marginal time preference in a UK-wide sample (TEMPUS) project. A review by Cairns JA, van der Pol MM. No. 2 Geriatric rehabilitation following fractures in older people: a systematic review. By Cameron I, Crotty M, Currie C, Finnegan T, Gillespie L, Gillespie W, et al.

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No. 3 Screening for sickle cell disease and thalassaemia: a systematic review with supplementary research. By Davies SC, Cronin E, Gill M, Greengross P, Hickman M, Normand C.

No. 14 The determinants of screening uptake and interventions for increasing uptake: a systematic review. By Jepson R, Clegg A, Forbes C, Lewis R, Sowden A, Kleijnen J.

No. 24 Outcome measures for adult critical care: a systematic review. By Hayes JA, Black NA, Jenkinson C, Young JD, Rowan KM, Daly K, et al.

No. 4 Community provision of hearing aids and related audiology services. A review by Reeves DJ, Alborz A, Hickson FS, Bamford JM.

No. 15 The effectiveness and cost-effectiveness of prophylactic removal of wisdom teeth. A rapid review by Song F, O’Meara S, Wilson P, Golder S, Kleijnen J.

No. 25 A systematic review to evaluate the effectiveness of interventions to promote the initiation of breastfeeding. By Fairbank L, O’Meara S, Renfrew MJ, Woolridge M, Sowden AJ, Lister-Sharp D.

No. 5 False-negative results in screening programmes: systematic review of impact and implications. By Petticrew MP, Sowden AJ, Lister-Sharp D, Wright K. No. 6 Costs and benefits of community postnatal support workers: a randomised controlled trial. By Morrell CJ, Spiby H, Stewart P, Walters S, Morgan A. No. 7 Implantable contraceptives (subdermal implants and hormonally impregnated intrauterine systems) versus other forms of reversible contraceptives: two systematic reviews to assess relative effectiveness, acceptability, tolerability and cost-effectiveness. By French RS, Cowan FM, Mansour DJA, Morris S, Procter T, Hughes D, et al. No. 8 An introduction to statistical methods for health technology assessment. A review by White SJ, Ashby D, Brown PJ. No. 9 Disease-modifying drugs for multiple sclerosis: a rapid and systematic review. By Clegg A, Bryant J, Milne R. No. 10 Publication and related biases. A review by Song F, Eastwood AJ, Gilbody S, Duley L, Sutton AJ. No. 11 Cost and outcome implications of the organisation of vascular services. By Michaels J, Brazier J, Palfreyman S, Shackley P, Slack R. No. 12 Monitoring blood glucose control in diabetes mellitus: a systematic review. By Coster S, Gulliford MC, Seed PT, Powrie JK, Swaminathan R. No. 13 The effectiveness of domiciliary health visiting: a systematic review of international studies and a selective review of the British literature. By Elkan R, Kendrick D, Hewitt M, Robinson JJA, Tolley K, Blair M, et al.

No. 16 Ultrasound screening in pregnancy: a systematic review of the clinical effectiveness, cost-effectiveness and women’s views. By Bricker L, Garcia J, Henderson J, Mugford M, Neilson J, Roberts T, et al. No. 17 A rapid and systematic review of the effectiveness and cost-effectiveness of the taxanes used in the treatment of advanced breast and ovarian cancer. By Lister-Sharp D, McDonagh MS, Khan KS, Kleijnen J. No. 18 Liquid-based cytology in cervical screening: a rapid and systematic review. By Payne N, Chilcott J, McGoogan E. No. 19 Randomised controlled trial of nondirective counselling, cognitive–behaviour therapy and usual general practitioner care in the management of depression as well as mixed anxiety and depression in primary care. By King M, Sibbald B, Ward E, Bower P, Lloyd M, Gabbay M, et al. No. 20 Routine referral for radiography of patients presenting with low back pain: is patients’ outcome influenced by GPs’ referral for plain radiography? By Kerry S, Hilton S, Patel S, Dundas D, Rink E, Lord J. No. 21 Systematic reviews of wound care management: (3) antimicrobial agents for chronic wounds; (4) diabetic foot ulceration. By O’Meara S, Cullum N, Majid M, Sheldon T. No. 22 Using routine data to complement and enhance the results of randomised controlled trials. By Lewsey JD, Leyland AH, Murray GD, Boddy FA. No. 23 Coronary artery stents in the treatment of ischaemic heart disease: a rapid and systematic review. By Meads C, Cummins C, Jolly K, Stevens A, Burls A, Hyde C.

No. 26 Implantable cardioverter defibrillators: arrhythmias. A rapid and systematic review. By Parkes J, Bryant J, Milne R. No. 27 Treatments for fatigue in multiple sclerosis: a rapid and systematic review. By Brañas P, Jordan R, Fry-Smith A, Burls A, Hyde C. No. 28 Early asthma prophylaxis, natural history, skeletal development and economy (EASE): a pilot randomised controlled trial. By Baxter-Jones ADG, Helms PJ, Russell G, Grant A, Ross S, Cairns JA, et al. No. 29 Screening for hypercholesterolaemia versus case finding for familial hypercholesterolaemia: a systematic review and cost-effectiveness analysis. By Marks D, Wonderling D, Thorogood M, Lambert H, Humphries SE, Neil HAW. No. 30 A rapid and systematic review of the clinical effectiveness and costeffectiveness of glycoprotein IIb/IIIa antagonists in the medical management of unstable angina. By McDonagh MS, Bachmann LM, Golder S, Kleijnen J, ter Riet G. No. 31 A randomised controlled trial of prehospital intravenous fluid replacement therapy in serious trauma. By Turner J, Nicholl J, Webber L, Cox H, Dixon S, Yates D. No. 32 Intrathecal pumps for giving opioids in chronic pain: a systematic review. By Williams JE, Louw G, Towlerton G. No. 33 Combination therapy (interferon alfa and ribavirin) in the treatment of chronic hepatitis C: a rapid and systematic review. By Shepherd J, Waugh N, Hewitson P.

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Health Technology Assessment reports published to date

No. 34 A systematic review of comparisons of effect sizes derived from randomised and non-randomised studies. By MacLehose RR, Reeves BC, Harvey IM, Sheldon TA, Russell IT, Black AMS. No. 35 Intravascular ultrasound-guided interventions in coronary artery disease: a systematic literature review, with decision-analytic modelling, of outcomes and cost-effectiveness. By Berry E, Kelly S, Hutton J, Lindsay HSJ, Blaxill JM, Evans JA, et al. No. 36 A randomised controlled trial to evaluate the effectiveness and costeffectiveness of counselling patients with chronic depression. By Simpson S, Corney R, Fitzgerald P, Beecham J. No. 37 Systematic review of treatments for atopic eczema. By Hoare C, Li Wan Po A, Williams H. No. 38 Bayesian methods in health technology assessment: a review. By Spiegelhalter DJ, Myles JP, Jones DR, Abrams KR. No. 39 The management of dyspepsia: a systematic review. By Delaney B, Moayyedi P, Deeks J, Innes M, Soo S, Barton P, et al. No. 40 A systematic review of treatments for severe psoriasis. By Griffiths CEM, Clark CM, Chalmers RJG, Li Wan Po A, Williams HC.

Volume 5, 2001 No. 1 Clinical and cost-effectiveness of donepezil, rivastigmine and galantamine for Alzheimer’s disease: a rapid and systematic review. By Clegg A, Bryant J, Nicholson T, McIntyre L, De Broe S, Gerard K, et al. No. 2 The clinical effectiveness and costeffectiveness of riluzole for motor neurone disease: a rapid and systematic review. By Stewart A, Sandercock J, Bryan S, Hyde C, Barton PM, Fry-Smith A, et al. No. 3 Equity and the economic evaluation of healthcare. By Sassi F, Archard L, Le Grand J.

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No. 4 Quality-of-life measures in chronic diseases of childhood. By Eiser C, Morse R.

No. 5 Eliciting public preferences for healthcare: a systematic review of techniques. By Ryan M, Scott DA, Reeves C, Bate A, van Teijlingen ER, Russell EM, et al.

No. 15 Home treatment for mental health problems: a systematic review. By Burns T, Knapp M, Catty J, Healey A, Henderson J, Watt H, et al.

No. 6 General health status measures for people with cognitive impairment: learning disability and acquired brain injury. By Riemsma RP, Forbes CA, Glanville JM, Eastwood AJ, Kleijnen J.

No. 16 How to develop cost-conscious guidelines. By Eccles M, Mason J.

No. 7 An assessment of screening strategies for fragile X syndrome in the UK. By Pembrey ME, Barnicoat AJ, Carmichael B, Bobrow M, Turner G. No. 8 Issues in methodological research: perspectives from researchers and commissioners. By Lilford RJ, Richardson A, Stevens A, Fitzpatrick R, Edwards S, Rock F, et al. No. 9 Systematic reviews of wound care management: (5) beds; (6) compression; (7) laser therapy, therapeutic ultrasound, electrotherapy and electromagnetic therapy. By Cullum N, Nelson EA, Flemming K, Sheldon T. No. 10 Effects of educational and psychosocial interventions for adolescents with diabetes mellitus: a systematic review. By Hampson SE, Skinner TC, Hart J, Storey L, Gage H, Foxcroft D, et al. No. 11 Effectiveness of autologous chondrocyte transplantation for hyaline cartilage defects in knees: a rapid and systematic review. By Jobanputra P, Parry D, Fry-Smith A, Burls A. No. 12 Statistical assessment of the learning curves of health technologies. By Ramsay CR, Grant AM, Wallace SA, Garthwaite PH, Monk AF, Russell IT. No. 13 The effectiveness and cost-effectiveness of temozolomide for the treatment of recurrent malignant glioma: a rapid and systematic review. By Dinnes J, Cave C, Huang S, Major K, Milne R. No. 14 A rapid and systematic review of the clinical effectiveness and costeffectiveness of debriding agents in treating surgical wounds healing by secondary intention. By Lewis R, Whiting P, ter Riet G, O’Meara S, Glanville J.

No. 17 The role of specialist nurses in multiple sclerosis: a rapid and systematic review. By De Broe S, Christopher F, Waugh N. No. 18 A rapid and systematic review of the clinical effectiveness and costeffectiveness of orlistat in the management of obesity. By O’Meara S, Riemsma R, Shirran L, Mather L, ter Riet G. No. 19 The clinical effectiveness and costeffectiveness of pioglitazone for type 2 diabetes mellitus: a rapid and systematic review. By Chilcott J, Wight J, Lloyd Jones M, Tappenden P. No. 20 Extended scope of nursing practice: a multicentre randomised controlled trial of appropriately trained nurses and preregistration house officers in preoperative assessment in elective general surgery. By Kinley H, Czoski-Murray C, George S, McCabe C, Primrose J, Reilly C, et al. No. 21 Systematic reviews of the effectiveness of day care for people with severe mental disorders: (1) Acute day hospital versus admission; (2) Vocational rehabilitation; (3) Day hospital versus outpatient care. By Marshall M, Crowther R, AlmarazSerrano A, Creed F, Sledge W, Kluiter H, et al. No. 22 The measurement and monitoring of surgical adverse events. By Bruce J, Russell EM, Mollison J, Krukowski ZH. No. 23 Action research: a systematic review and guidance for assessment. By Waterman H, Tillen D, Dickson R, de Koning K. No. 24 A rapid and systematic review of the clinical effectiveness and costeffectiveness of gemcitabine for the treatment of pancreatic cancer. By Ward S, Morris E, Bansback N, Calvert N, Crellin A, Forman D, et al.

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No. 25 A rapid and systematic review of the evidence for the clinical effectiveness and cost-effectiveness of irinotecan, oxaliplatin and raltitrexed for the treatment of advanced colorectal cancer. By Lloyd Jones M, Hummel S, Bansback N, Orr B, Seymour M. No. 26 Comparison of the effectiveness of inhaler devices in asthma and chronic obstructive airways disease: a systematic review of the literature. By Brocklebank D, Ram F, Wright J, Barry P, Cates C, Davies L, et al. No. 27 The cost-effectiveness of magnetic resonance imaging for investigation of the knee joint. By Bryan S, Weatherburn G, Bungay H, Hatrick C, Salas C, Parry D, et al. No. 28 A rapid and systematic review of the clinical effectiveness and costeffectiveness of topotecan for ovarian cancer. By Forbes C, Shirran L, Bagnall A-M, Duffy S, ter Riet G. No. 29 Superseded by a report published in a later volume. No. 30 The role of radiography in primary care patients with low back pain of at least 6 weeks duration: a randomised (unblinded) controlled trial. By Kendrick D, Fielding K, Bentley E, Miller P, Kerslake R, Pringle M. No. 31 Design and use of questionnaires: a review of best practice applicable to surveys of health service staff and patients. By McColl E, Jacoby A, Thomas L, Soutter J, Bamford C, Steen N, et al. No. 32 A rapid and systematic review of the clinical effectiveness and costeffectiveness of paclitaxel, docetaxel, gemcitabine and vinorelbine in nonsmall-cell lung cancer. By Clegg A, Scott DA, Sidhu M, Hewitson P, Waugh N. No. 33 Subgroup analyses in randomised controlled trials: quantifying the risks of false-positives and false-negatives. By Brookes ST, Whitley E, Peters TJ, Mulheran PA, Egger M, Davey Smith G. No. 34 Depot antipsychotic medication in the treatment of patients with schizophrenia: (1) Meta-review; (2) Patient and nurse attitudes. By David AS, Adams C.

No. 35 A systematic review of controlled trials of the effectiveness and costeffectiveness of brief psychological treatments for depression. By Churchill R, Hunot V, Corney R, Knapp M, McGuire H, Tylee A, et al. No. 36 Cost analysis of child health surveillance. By Sanderson D, Wright D, Acton C, Duree D.

Volume 6, 2002

No. 9 Zanamivir for the treatment of influenza in adults: a systematic review and economic evaluation. By Burls A, Clark W, Stewart T, Preston C, Bryan S, Jefferson T, et al. No. 10 A review of the natural history and epidemiology of multiple sclerosis: implications for resource allocation and health economic models. By Richards RG, Sampson FC, Beard SM, Tappenden P.

No. 1 A study of the methods used to select review criteria for clinical audit. By Hearnshaw H, Harker R, Cheater F, Baker R, Grimshaw G.

No. 11 Screening for gestational diabetes: a systematic review and economic evaluation. By Scott DA, Loveman E, McIntyre L, Waugh N.

No. 2 Fludarabine as second-line therapy for B cell chronic lymphocytic leukaemia: a technology assessment. By Hyde C, Wake B, Bryan S, Barton P, Fry-Smith A, Davenport C, et al.

No. 12 The clinical effectiveness and costeffectiveness of surgery for people with morbid obesity: a systematic review and economic evaluation. By Clegg AJ, Colquitt J, Sidhu MK, Royle P, Loveman E, Walker A.

No. 3 Rituximab as third-line treatment for refractory or recurrent Stage III or IV follicular non-Hodgkin’s lymphoma: a systematic review and economic evaluation. By Wake B, Hyde C, Bryan S, Barton P, Song F, Fry-Smith A, et al. No. 4 A systematic review of discharge arrangements for older people. By Parker SG, Peet SM, McPherson A, Cannaby AM, Baker R, Wilson A, et al. No. 5 The clinical effectiveness and costeffectiveness of inhaler devices used in the routine management of chronic asthma in older children: a systematic review and economic evaluation. By Peters J, Stevenson M, Beverley C, Lim J, Smith S. No. 6 The clinical effectiveness and costeffectiveness of sibutramine in the management of obesity: a technology assessment. By O’Meara S, Riemsma R, Shirran L, Mather L, ter Riet G. No. 7 The cost-effectiveness of magnetic resonance angiography for carotid artery stenosis and peripheral vascular disease: a systematic review. By Berry E, Kelly S, Westwood ME, Davies LM, Gough MJ, Bamford JM, et al. No. 8 Promoting physical activity in South Asian Muslim women through ‘exercise on prescription’. By Carroll B, Ali N, Azam N.

No. 13 The clinical effectiveness of trastuzumab for breast cancer: a systematic review. By Lewis R, Bagnall A-M, Forbes C, Shirran E, Duffy S, Kleijnen J, et al. No. 14 The clinical effectiveness and costeffectiveness of vinorelbine for breast cancer: a systematic review and economic evaluation. By Lewis R, Bagnall A-M, King S, Woolacott N, Forbes C, Shirran L, et al. No. 15 A systematic review of the effectiveness and cost-effectiveness of metal-on-metal hip resurfacing arthroplasty for treatment of hip disease. By Vale L, Wyness L, McCormack K, McKenzie L, Brazzelli M, Stearns SC. No. 16 The clinical effectiveness and costeffectiveness of bupropion and nicotine replacement therapy for smoking cessation: a systematic review and economic evaluation. By Woolacott NF, Jones L, Forbes CA, Mather LC, Sowden AJ, Song FJ, et al. No. 17 A systematic review of effectiveness and economic evaluation of new drug treatments for juvenile idiopathic arthritis: etanercept. By Cummins C, Connock M, Fry-Smith A, Burls A. No. 18 Clinical effectiveness and costeffectiveness of growth hormone in children: a systematic review and economic evaluation. By Bryant J, Cave C, Mihaylova B, Chase D, McIntyre L, Gerard K, et al.

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No. 19 Clinical effectiveness and costeffectiveness of growth hormone in adults in relation to impact on quality of life: a systematic review and economic evaluation. By Bryant J, Loveman E, Chase D, Mihaylova B, Cave C, Gerard K, et al. No. 20 Clinical medication review by a pharmacist of patients on repeat prescriptions in general practice: a randomised controlled trial. By Zermansky AG, Petty DR, Raynor DK, Lowe CJ, Freementle N, Vail A. No. 21 The effectiveness of infliximab and etanercept for the treatment of rheumatoid arthritis: a systematic review and economic evaluation. By Jobanputra P, Barton P, Bryan S, Burls A. No. 22 A systematic review and economic evaluation of computerised cognitive behaviour therapy for depression and anxiety. By Kaltenthaler E, Shackley P, Stevens K, Beverley C, Parry G, Chilcott J. No. 23 A systematic review and economic evaluation of pegylated liposomal doxorubicin hydrochloride for ovarian cancer. By Forbes C, Wilby J, Richardson G, Sculpher M, Mather L, Reimsma R. No. 24 A systematic review of the effectiveness of interventions based on a stages-ofchange approach to promote individual behaviour change. By Riemsma RP, Pattenden J, Bridle C, Sowden AJ, Mather L, Watt IS, et al. No. 25 A systematic review update of the clinical effectiveness and costeffectiveness of glycoprotein IIb/IIIa antagonists. By Robinson M, Ginnelly L, Sculpher M, Jones L, Riemsma R, Palmer S, et al. No. 26 A systematic review of the effectiveness, cost-effectiveness and barriers to implementation of thrombolytic and neuroprotective therapy for acute ischaemic stroke in the NHS. By Sandercock P, Berge E, Dennis M, Forbes J, Hand P, Kwan J, et al.

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No. 27 A randomised controlled crossover trial of nurse practitioner versus doctor-led outpatient care in a bronchiectasis clinic. By Caine N, Sharples LD, Hollingworth W, French J, Keogan M, Exley A, et al.

No. 28 Clinical effectiveness and cost – consequences of selective serotonin reuptake inhibitors in the treatment of sex offenders. By Adi Y, Ashcroft D, Browne K, Beech A, Fry-Smith A, Hyde C. No. 29 Treatment of established osteoporosis: a systematic review and cost–utility analysis. By Kanis JA, Brazier JE, Stevenson M, Calvert NW, Lloyd Jones M. No. 30 Which anaesthetic agents are costeffective in day surgery? Literature review, national survey of practice and randomised controlled trial. By Elliott RA Payne K, Moore JK, Davies LM, Harper NJN, St Leger AS, et al. No. 31 Screening for hepatitis C among injecting drug users and in genitourinary medicine clinics: systematic reviews of effectiveness, modelling study and national survey of current practice. By Stein K, Dalziel K, Walker A, McIntyre L, Jenkins B, Horne J, et al. No. 32 The measurement of satisfaction with healthcare: implications for practice from a systematic review of the literature. By Crow R, Gage H, Hampson S, Hart J, Kimber A, Storey L, et al. No. 33 The effectiveness and cost-effectiveness of imatinib in chronic myeloid leukaemia: a systematic review. By Garside R, Round A, Dalziel K, Stein K, Royle R. No. 34 A comparative study of hypertonic saline, daily and alternate-day rhDNase in children with cystic fibrosis. By Suri R, Wallis C, Bush A, Thompson S, Normand C, Flather M, et al. No. 35 A systematic review of the costs and effectiveness of different models of paediatric home care. By Parker G, Bhakta P, Lovett CA, Paisley S, Olsen R, Turner D, et al.

No. 2 Systematic review of the effectiveness and cost-effectiveness, and economic evaluation, of home versus hospital or satellite unit haemodialysis for people with end-stage renal failure. By Mowatt G, Vale L, Perez J, Wyness L, Fraser C, MacLeod A, et al. No. 3 Systematic review and economic evaluation of the effectiveness of infliximab for the treatment of Crohn’s disease. By Clark W, Raftery J, Barton P, Song F, Fry-Smith A, Burls A. No. 4 A review of the clinical effectiveness and cost-effectiveness of routine anti-D prophylaxis for pregnant women who are rhesus negative. By Chilcott J, Lloyd Jones M, Wight J, Forman K, Wray J, Beverley C, et al. No. 5 Systematic review and evaluation of the use of tumour markers in paediatric oncology: Ewing’s sarcoma and neuroblastoma. By Riley RD, Burchill SA, Abrams KR, Heney D, Lambert PC, Jones DR, et al. No. 6 The cost-effectiveness of screening for Helicobacter pylori to reduce mortality and morbidity from gastric cancer and peptic ulcer disease: a discrete-event simulation model. By Roderick P, Davies R, Raftery J, Crabbe D, Pearce R, Bhandari P, et al. No. 7 The clinical effectiveness and costeffectiveness of routine dental checks: a systematic review and economic evaluation. By Davenport C, Elley K, Salas C, Taylor-Weetman CL, Fry-Smith A, Bryan S, et al. No. 8 A multicentre randomised controlled trial assessing the costs and benefits of using structured information and analysis of women’s preferences in the management of menorrhagia. By Kennedy ADM, Sculpher MJ, Coulter A, Dwyer N, Rees M, Horsley S, et al.

Volume 7, 2003

No. 9 Clinical effectiveness and cost–utility of photodynamic therapy for wet age-related macular degeneration: a systematic review and economic evaluation. By Meads C, Salas C, Roberts T, Moore D, Fry-Smith A, Hyde C.

No. 1 How important are comprehensive literature searches and the assessment of trial quality in systematic reviews? Empirical study. By Egger M, Jüni P, Bartlett C, Holenstein F, Sterne J.

No. 10 Evaluation of molecular tests for prenatal diagnosis of chromosome abnormalities. By Grimshaw GM, Szczepura A, Hultén M, MacDonald F, Nevin NC, Sutton F, et al.

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No. 11 First and second trimester antenatal screening for Down’s syndrome: the results of the Serum, Urine and Ultrasound Screening Study (SURUSS). By Wald NJ, Rodeck C, Hackshaw AK, Walters J, Chitty L, Mackinson AM.

No. 21 Systematic review of the clinical effectiveness and cost-effectiveness of tension-free vaginal tape for treatment of urinary stress incontinence. By Cody J, Wyness L, Wallace S, Glazener C, Kilonzo M, Stearns S, et al.

No. 12 The effectiveness and cost-effectiveness of ultrasound locating devices for central venous access: a systematic review and economic evaluation. By Calvert N, Hind D, McWilliams RG, Thomas SM, Beverley C, Davidson A.

No. 22 The clinical and cost-effectiveness of patient education models for diabetes: a systematic review and economic evaluation. By Loveman E, Cave C, Green C, Royle P, Dunn N, Waugh N.

No. 13 A systematic review of atypical antipsychotics in schizophrenia. By Bagnall A-M, Jones L, Lewis R, Ginnelly L, Glanville J, Torgerson D, et al. No. 14 Prostate Testing for Cancer and Treatment (ProtecT) feasibility study. By Donovan J, Hamdy F, Neal D, Peters T, Oliver S, Brindle L, et al. No. 15 Early thrombolysis for the treatment of acute myocardial infarction: a systematic review and economic evaluation. By Boland A, Dundar Y, Bagust A, Haycox A, Hill R, Mujica Mota R, et al. No. 16 Screening for fragile X syndrome: a literature review and modelling. By Song FJ, Barton P, Sleightholme V, Yao GL, Fry-Smith A. No. 17 Systematic review of endoscopic sinus surgery for nasal polyps. By Dalziel K, Stein K, Round A, Garside R, Royle P. No. 18 Towards efficient guidelines: how to monitor guideline use in primary care. By Hutchinson A, McIntosh A, Cox S, Gilbert C.

No. 23 The role of modelling in prioritising and planning clinical trials. By Chilcott J, Brennan A, Booth A, Karnon J, Tappenden P. No. 24 Cost–benefit evaluation of routine influenza immunisation in people 65–74 years of age. By Allsup S, Gosney M, Haycox A, Regan M. No. 25 The clinical and cost-effectiveness of pulsatile machine perfusion versus cold storage of kidneys for transplantation retrieved from heart-beating and nonheart-beating donors. By Wight J, Chilcott J, Holmes M, Brewer N. No. 26 Can randomised trials rely on existing electronic data? A feasibility study to explore the value of routine data in health technology assessment. By Williams JG, Cheung WY, Cohen DR, Hutchings HA, Longo MF, Russell IT. No. 27 Evaluating non-randomised intervention studies. By Deeks JJ, Dinnes J, D’Amico R, Sowden AJ, Sakarovitch C, Song F, et al.

No. 19 Effectiveness and cost-effectiveness of acute hospital-based spinal cord injuries services: systematic review. By Bagnall A-M, Jones L, Richardson G, Duffy S, Riemsma R.

No. 28 A randomised controlled trial to assess the impact of a package comprising a patient-orientated, evidence-based selfhelp guidebook and patient-centred consultations on disease management and satisfaction in inflammatory bowel disease. By Kennedy A, Nelson E, Reeves D, Richardson G, Roberts C, Robinson A, et al.

No. 20 Prioritisation of health technology assessment. The PATHS model: methods and case studies. By Townsend J, Buxton M, Harper G.

No. 29 The effectiveness of diagnostic tests for the assessment of shoulder pain due to soft tissue disorders: a systematic review. By Dinnes J, Loveman E, McIntyre L, Waugh N.

No. 30 The value of digital imaging in diabetic retinopathy. By Sharp PF, Olson J, Strachan F, Hipwell J, Ludbrook A, O’Donnell M, et al. No. 31 Lowering blood pressure to prevent myocardial infarction and stroke: a new preventive strategy. By Law M, Wald N, Morris J. No. 32 Clinical and cost-effectiveness of capecitabine and tegafur with uracil for the treatment of metastatic colorectal cancer: systematic review and economic evaluation. By Ward S, Kaltenthaler E, Cowan J, Brewer N. No. 33 Clinical and cost-effectiveness of new and emerging technologies for early localised prostate cancer: a systematic review. By Hummel S, Paisley S, Morgan A, Currie E, Brewer N. No. 34 Literature searching for clinical and cost-effectiveness studies used in health technology assessment reports carried out for the National Institute for Clinical Excellence appraisal system. By Royle P, Waugh N. No. 35 Systematic review and economic decision modelling for the prevention and treatment of influenza A and B. By Turner D, Wailoo A, Nicholson K, Cooper N, Sutton A, Abrams K. No. 36 A randomised controlled trial to evaluate the clinical and costeffectiveness of Hickman line insertions in adult cancer patients by nurses. By Boland A, Haycox A, Bagust A, Fitzsimmons L. No. 37 Redesigning postnatal care: a randomised controlled trial of protocol-based midwifery-led care focused on individual women’s physical and psychological health needs. By MacArthur C, Winter HR, Bick DE, Lilford RJ, Lancashire RJ, Knowles H, et al. No. 38 Estimating implied rates of discount in healthcare decision-making. By West RR, McNabb R, Thompson AGH, Sheldon TA, Grimley Evans J.

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No. 39 Systematic review of isolation policies in the hospital management of methicillinresistant Staphylococcus aureus: a review of the literature with epidemiological and economic modelling. By Cooper BS, Stone SP, Kibbler CC, Cookson BD, Roberts JA, Medley GF, et al. No. 40 Treatments for spasticity and pain in multiple sclerosis: a systematic review. By Beard S, Hunn A, Wight J. No. 41 The inclusion of reports of randomised trials published in languages other than English in systematic reviews. By Moher D, Pham B, Lawson ML, Klassen TP. No. 42 The impact of screening on future health-promoting behaviours and health beliefs: a systematic review. By Bankhead CR, Brett J, Bukach C, Webster P, Stewart-Brown S, Munafo M, et al.

Volume 8, 2004 No. 1 What is the best imaging strategy for acute stroke? By Wardlaw JM, Keir SL, Seymour J, Lewis S, Sandercock PAG, Dennis MS, et al. No. 2 Systematic review and modelling of the investigation of acute and chronic chest pain presenting in primary care. By Mant J, McManus RJ, Oakes RAL, Delaney BC, Barton PM, Deeks JJ, et al. No. 3 The effectiveness and cost-effectiveness of microwave and thermal balloon endometrial ablation for heavy menstrual bleeding: a systematic review and economic modelling. By Garside R, Stein K, Wyatt K, Round A, Price A. No. 4 A systematic review of the role of bisphosphonates in metastatic disease. By Ross JR, Saunders Y, Edmonds PM, Patel S, Wonderling D, Normand C, et al. No. 5 Systematic review of the clinical effectiveness and cost-effectiveness of capecitabine (Xeloda® ) for locally advanced and/or metastatic breast cancer. By Jones L, Hawkins N, Westwood M, Wright K, Richardson G, Riemsma R.

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No. 6 Effectiveness and efficiency of guideline dissemination and implementation strategies. By Grimshaw JM, Thomas RE, MacLennan G, Fraser C, Ramsay CR, Vale L, et al.

No. 7 Clinical effectiveness and costs of the Sugarbaker procedure for the treatment of pseudomyxoma peritonei. By Bryant J, Clegg AJ, Sidhu MK, Brodin H, Royle P, Davidson P. No. 8 Psychological treatment for insomnia in the regulation of long-term hypnotic drug use. By Morgan K, Dixon S, Mathers N, Thompson J, Tomeny M. No. 9 Improving the evaluation of therapeutic interventions in multiple sclerosis: development of a patient-based measure of outcome. By Hobart JC, Riazi A, Lamping DL, Fitzpatrick R, Thompson AJ. No. 10 A systematic review and economic evaluation of magnetic resonance cholangiopancreatography compared with diagnostic endoscopic retrograde cholangiopancreatography. By Kaltenthaler E, Bravo Vergel Y, Chilcott J, Thomas S, Blakeborough T, Walters SJ, et al. No. 11 The use of modelling to evaluate new drugs for patients with a chronic condition: the case of antibodies against tumour necrosis factor in rheumatoid arthritis. By Barton P, Jobanputra P, Wilson J, Bryan S, Burls A. No. 12 Clinical effectiveness and costeffectiveness of neonatal screening for inborn errors of metabolism using tandem mass spectrometry: a systematic review. By Pandor A, Eastham J, Beverley C, Chilcott J, Paisley S. No. 13 Clinical effectiveness and costeffectiveness of pioglitazone and rosiglitazone in the treatment of type 2 diabetes: a systematic review and economic evaluation. By Czoski-Murray C, Warren E, Chilcott J, Beverley C, Psyllaki MA, Cowan J. No. 14 Routine examination of the newborn: the EMREN study. Evaluation of an extension of the midwife role including a randomised controlled trial of appropriately trained midwives and paediatric senior house officers. By Townsend J, Wolke D, Hayes J, Davé S, Rogers C, Bloomfield L, et al.

No. 15 Involving consumers in research and development agenda setting for the NHS: developing an evidence-based approach. By Oliver S, Clarke-Jones L, Rees R, Milne R, Buchanan P, Gabbay J, et al. No. 16 A multi-centre randomised controlled trial of minimally invasive direct coronary bypass grafting versus percutaneous transluminal coronary angioplasty with stenting for proximal stenosis of the left anterior descending coronary artery. By Reeves BC, Angelini GD, Bryan AJ, Taylor FC, Cripps T, Spyt TJ, et al. No. 17 Does early magnetic resonance imaging influence management or improve outcome in patients referred to secondary care with low back pain? A pragmatic randomised controlled trial. By Gilbert FJ, Grant AM, Gillan MGC, Vale L, Scott NW, Campbell MK, et al. No. 18 The clinical and cost-effectiveness of anakinra for the treatment of rheumatoid arthritis in adults: a systematic review and economic analysis. By Clark W, Jobanputra P, Barton P, Burls A. No. 19 A rapid and systematic review and economic evaluation of the clinical and cost-effectiveness of newer drugs for treatment of mania associated with bipolar affective disorder. By Bridle C, Palmer S, Bagnall A-M, Darba J, Duffy S, Sculpher M, et al. No. 20 Liquid-based cytology in cervical screening: an updated rapid and systematic review and economic analysis. By Karnon J, Peters J, Platt J, Chilcott J, McGoogan E, Brewer N. No. 21 Systematic review of the long-term effects and economic consequences of treatments for obesity and implications for health improvement. By Avenell A, Broom J, Brown TJ, Poobalan A, Aucott L, Stearns SC, et al. No. 22 Autoantibody testing in children with newly diagnosed type 1 diabetes mellitus. By Dretzke J, Cummins C, Sandercock J, Fry-Smith A, Barrett T, Burls A.

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No. 23 Clinical effectiveness and costeffectiveness of prehospital intravenous fluids in trauma patients. By Dretzke J, Sandercock J, Bayliss S, Burls A. No. 24 Newer hypnotic drugs for the shortterm management of insomnia: a systematic review and economic evaluation. By Dündar Y, Boland A, Strobl J, Dodd S, Haycox A, Bagust A, et al. No. 25 Development and validation of methods for assessing the quality of diagnostic accuracy studies. By Whiting P, Rutjes AWS, Dinnes J, Reitsma JB, Bossuyt PMM, Kleijnen J. No. 26 EVALUATE hysterectomy trial: a multicentre randomised trial comparing abdominal, vaginal and laparoscopic methods of hysterectomy. By Garry R, Fountain J, Brown J, Manca A, Mason S, Sculpher M, et al. No. 27 Methods for expected value of information analysis in complex health economic models: developments on the health economics of interferon-␤ and glatiramer acetate for multiple sclerosis. By Tappenden P, Chilcott JB, Eggington S, Oakley J, McCabe C. No. 28 Effectiveness and cost-effectiveness of imatinib for first-line treatment of chronic myeloid leukaemia in chronic phase: a systematic review and economic analysis. By Dalziel K, Round A, Stein K, Garside R, Price A. No. 29 VenUS I: a randomised controlled trial of two types of bandage for treating venous leg ulcers. By Iglesias C, Nelson EA, Cullum NA, Torgerson DJ on behalf of the VenUS Team. No. 30 Systematic review of the effectiveness and cost-effectiveness, and economic evaluation, of myocardial perfusion scintigraphy for the diagnosis and management of angina and myocardial infarction. By Mowatt G, Vale L, Brazzelli M, Hernandez R, Murray A, Scott N, et al. No. 31 A pilot study on the use of decision theory and value of information analysis as part of the NHS Health Technology Assessment programme. on K, By Claxton K Ginnelly L, Sculpher M, Philips Z, Palmer S.

No. 32 The Social Support and Family Health Study: a randomised controlled trial and economic evaluation of two alternative forms of postnatal support for mothers living in disadvantaged inner-city areas. By Wiggins M, Oakley A, Roberts I, Turner H, Rajan L, Austerberry H, et al. No. 33 Psychosocial aspects of genetic screening of pregnant women and newborns: a systematic review. By Green JM, Hewison J, Bekker HL, Bryant, Cuckle HS. No. 34 Evaluation of abnormal uterine bleeding: comparison of three outpatient procedures within cohorts defined by age and menopausal status. By Critchley HOD, Warner P, Lee AJ, Brechin S, Guise J, Graham B. No. 35 Coronary artery stents: a rapid systematic review and economic evaluation. By Hill R, Bagust A, Bakhai A, Dickson R, Dündar Y, Haycox A, et al. No. 36 Review of guidelines for good practice in decision-analytic modelling in health technology assessment. By Philips Z, Ginnelly L, Sculpher M, Claxton K, Golder S, Riemsma R, et al. No. 37 Rituximab (MabThera®) for aggressive non-Hodgkin’s lymphoma: systematic review and economic evaluation. By Knight C, Hind D, Brewer N, Abbott V. No. 38 Clinical effectiveness and costeffectiveness of clopidogrel and modified-release dipyridamole in the secondary prevention of occlusive vascular events: a systematic review and economic evaluation. By Jones L, Griffin S, Palmer S, Main C, Orton V, Sculpher M, et al. No. 39 Pegylated interferon ␣-2a and -2b in combination with ribavirin in the treatment of chronic hepatitis C: a systematic review and economic evaluation. By Shepherd J, Brodin H, Cave C, Waugh N, Price A, Gabbay J. No. 40 Clopidogrel used in combination with aspirin compared with aspirin alone in the treatment of non-ST-segmentelevation acute coronary syndromes: a systematic review and economic evaluation. By Main C, Palmer S, Griffin S, Jones L, Orton V, Sculpher M, et al.

No. 41 Provision, uptake and cost of cardiac rehabilitation programmes: improving services to under-represented groups. By Beswick AD, Rees K, Griebsch I, Taylor FC, Burke M, West RR, et al. No. 42 Involving South Asian patients in clinical trials. By Hussain-Gambles M, Leese B, Atkin K, Brown J, Mason S, Tovey P. No. 43 Clinical and cost-effectiveness of continuous subcutaneous insulin infusion for diabetes. By Colquitt JL, Green C, Sidhu MK, Hartwell D, Waugh N. No. 44 Identification and assessment of ongoing trials in health technology assessment reviews. By Song FJ, Fry-Smith A, Davenport C, Bayliss S, Adi Y, Wilson JS, et al. No. 45 Systematic review and economic evaluation of a long-acting insulin analogue, insulin glargine By Warren E, Weatherley-Jones E, Chilcott J, Beverley C. No. 46 Supplementation of a home-based exercise programme with a class-based programme for people with osteoarthritis of the knees: a randomised controlled trial and health economic analysis. By McCarthy CJ, Mills PM, Pullen R, Richardson G, Hawkins N, Roberts CR, et al. No. 47 Clinical and cost-effectiveness of oncedaily versus more frequent use of same potency topical corticosteroids for atopic eczema: a systematic review and economic evaluation. By Green C, Colquitt JL, Kirby J, Davidson P, Payne E. No. 48 Acupuncture of chronic headache disorders in primary care: randomised controlled trial and economic analysis. By Vickers AJ, Rees RW, Zollman CE, McCarney R, Smith CM, Ellis N, et al. No. 49 Generalisability in economic evaluation studies in healthcare: a review and case studies. By Sculpher MJ, Pang FS, Manca A, Drummond MF, Golder S, Urdahl H, et al. No. 50 Virtual outreach: a randomised controlled trial and economic evaluation of joint teleconferenced medical consultations. By Wallace P, Barber J, Clayton W, Currell R, Fleming K, Garner P, et al.

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Volume 9, 2005 No. 1 Randomised controlled multiple treatment comparison to provide a cost-effectiveness rationale for the selection of antimicrobial therapy in acne. By Ozolins M, Eady EA, Avery A, Cunliffe WJ, O’Neill C, Simpson NB, et al. No. 2 Do the findings of case series studies vary significantly according to methodological characteristics? By Dalziel K, Round A, Stein K, Garside R, Castelnuovo E, Payne L. No. 3 Improving the referral process for familial breast cancer genetic counselling: findings of three randomised controlled trials of two interventions. By Wilson BJ, Torrance N, Mollison J, Wordsworth S, Gray JR, Haites NE, et al. No. 4 Randomised evaluation of alternative electrosurgical modalities to treat bladder outflow obstruction in men with benign prostatic hyperplasia. By Fowler C, McAllister W, Plail R, Karim O, Yang Q. No. 5 A pragmatic randomised controlled trial of the cost-effectiveness of palliative therapies for patients with inoperable oesophageal cancer. By Shenfine J, McNamee P, Steen N, Bond J, Griffin SM. No. 6 Impact of computer-aided detection prompts on the sensitivity and specificity of screening mammography. By Taylor P, Champness J, GivenWilson R, Johnston K, Potts H. No. 7 Issues in data monitoring and interim analysis of trials. By Grant AM, Altman DG, Babiker AB, Campbell MK, Clemens FJ, Darbyshire JH, et al. No. 8 Lay public’s understanding of equipoise and randomisation in randomised controlled trials. By Robinson EJ, Kerr CEP, Stevens AJ, Lilford RJ, Braunholtz DA, Edwards SJ, et al.

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No. 9 Clinical and cost-effectiveness of electroconvulsive therapy for depressive illness, schizophrenia, catatonia and mania: systematic reviews and economic modelling studies. By Greenhalgh J, Knight C, Hind D, Beverley C, Walters S.

No. 10 Measurement of health-related quality of life for people with dementia: development of a new instrument (DEMQOL) and an evaluation of current methodology. By Smith SC, Lamping DL, Banerjee S, Harwood R, Foley B, Smith P, et al. No. 11 Clinical effectiveness and costeffectiveness of drotrecogin alfa (activated) (Xigris®) for the treatment of severe sepsis in adults: a systematic review and economic evaluation. By Green C, Dinnes J, Takeda A, Shepherd J, Hartwell D, Cave C, et al. No. 12 A methodological review of how heterogeneity has been examined in systematic reviews of diagnostic test accuracy. By Dinnes J, Deeks J, Kirby J, Roderick P. No. 13 Cervical screening programmes: can automation help? Evidence from systematic reviews, an economic analysis and a simulation modelling exercise applied to the UK. By Willis BH, Barton P, Pearmain P, Bryan S, Hyde C. No. 14 Laparoscopic surgery for inguinal hernia repair: systematic review of effectiveness and economic evaluation. By McCormack K, Wake B, Perez J, Fraser C, Cook J, McIntosh E, et al. No. 15 Clinical effectiveness, tolerability and cost-effectiveness of newer drugs for epilepsy in adults: a systematic review and economic evaluation. By Wilby J, Kainth A, Hawkins N, Epstein D, McIntosh H, McDaid C, et al. No. 16 A randomised controlled trial to compare the cost-effectiveness of tricyclic antidepressants, selective serotonin reuptake inhibitors and lofepramine. By Peveler R, Kendrick T, Buxton M, Longworth L, Baldwin D, Moore M, et al. No. 17 Clinical effectiveness and costeffectiveness of immediate angioplasty for acute myocardial infarction: systematic review and economic evaluation. By Hartwell D, Colquitt J, Loveman E, Clegg AJ, Brodin H, Waugh N, et al.

No. 18 A randomised controlled comparison of alternative strategies in stroke care. By Kalra L, Evans A, Perez I, Knapp M, Swift C, Donaldson N. No. 19 The investigation and analysis of critical incidents and adverse events in healthcare. By Woloshynowych M, Rogers S, Taylor-Adams S, Vincent C. No. 20 Potential use of routine databases in health technology assessment. By Raftery J, Roderick P, Stevens A. No. 21 Clinical and cost-effectiveness of newer immunosuppressive regimens in renal transplantation: a systematic review and modelling study. By Woodroffe R, Yao GL, Meads C, Bayliss S, Ready A, Raftery J, et al. No. 22 A systematic review and economic evaluation of alendronate, etidronate, risedronate, raloxifene and teriparatide for the prevention and treatment of postmenopausal osteoporosis. By Stevenson M, Lloyd Jones M, De Nigris E, Brewer N, Davis S, Oakley J. No. 23 A systematic review to examine the impact of psycho-educational interventions on health outcomes and costs in adults and children with difficult asthma. By Smith JR, Mugford M, Holland R, Candy B, Noble MJ, Harrison BDW, et al. No. 24 An evaluation of the costs, effectiveness and quality of renal replacement therapy provision in renal satellite units in England and Wales. By Roderick P, Nicholson T, Armitage A, Mehta R, Mullee M, Gerard K, et al. No. 25 Imatinib for the treatment of patients with unresectable and/or metastatic gastrointestinal stromal tumours: systematic review and economic evaluation. By Wilson J, Connock M, Song F, Yao G, Fry-Smith A, Raftery J, et al. No. 26 Indirect comparisons of competing interventions. By Glenny AM, Altman DG, Song F, Sakarovitch C, Deeks JJ, D’Amico R, et al. No. 27 Cost-effectiveness of alternative strategies for the initial medical management of non-ST elevation acute coronary syndrome: systematic review and decision-analytical modelling. By Robinson M, Palmer S, Sculpher M, Philips Z, Ginnelly L, Bowens A, et al.

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No. 28 Outcomes of electrically stimulated gracilis neosphincter surgery. By Tillin T, Chambers M, Feldman R. No. 29 The effectiveness and cost-effectiveness of pimecrolimus and tacrolimus for atopic eczema: a systematic review and economic evaluation. By Garside R, Stein K, Castelnuovo E, Pitt M, Ashcroft D, Dimmock P, et al. No. 30 Systematic review on urine albumin testing for early detection of diabetic complications. By Newman DJ, Mattock MB, Dawnay ABS, Kerry S, McGuire A, Yaqoob M, et al. No. 31 Randomised controlled trial of the costeffectiveness of water-based therapy for lower limb osteoarthritis. By Cochrane T, Davey RC, Matthes Edwards SM. No. 32 Longer term clinical and economic benefits of offering acupuncture care to patients with chronic low back pain. By Thomas KJ, MacPherson H, Ratcliffe J, Thorpe L, Brazier J, Campbell M, et al. No. 33 Cost-effectiveness and safety of epidural steroids in the management of sciatica. By Price C, Arden N, Coglan L, Rogers P. No. 34 The British Rheumatoid Outcome Study Group (BROSG) randomised controlled trial to compare the effectiveness and cost-effectiveness of aggressive versus symptomatic therapy in established rheumatoid arthritis. By Symmons D, Tricker K, Roberts C, Davies L, Dawes P, Scott DL. No. 35 Conceptual framework and systematic review of the effects of participants’ and professionals’ preferences in randomised controlled trials. By King M, Nazareth I, Lampe F, Bower P, Chandler M, Morou M, et al. No. 36 The clinical and cost-effectiveness of implantable cardioverter defibrillators: a systematic review. By Bryant J, Brodin H, Loveman E, Payne E, Clegg A. No. 37 A trial of problem-solving by community mental health nurses for anxiety, depression and life difficulties among general practice patients. The CPN-GP study. By Kendrick T, Simons L, Mynors-Wallis L, Gray A, Lathlean J, Pickering R, et al.

No. 38 The causes and effects of sociodemographic exclusions from clinical trials. By Bartlett C, Doyal L, Ebrahim S, Davey P, Bachmann M, Egger M, et al. No. 39 Is hydrotherapy cost-effective? A randomised controlled trial of combined hydrotherapy programmes compared with physiotherapy land techniques in children with juvenile idiopathic arthritis. By Epps H, Ginnelly L, Utley M, Southwood T, Gallivan S, Sculpher M, et al. No. 40 A randomised controlled trial and costeffectiveness study of systematic screening (targeted and total population screening) versus routine practice for the detection of atrial fibrillation in people aged 65 and over. The SAFE study. By Hobbs FDR, Fitzmaurice DA, Mant J, Murray E, Jowett S, Bryan S, et al. No. 41 Displaced intracapsular hip fractures in fit, older people: a randomised comparison of reduction and fixation, bipolar hemiarthroplasty and total hip arthroplasty. By Keating JF, Grant A, Masson M, Scott NW, Forbes JF. No. 42 Long-term outcome of cognitive behaviour therapy clinical trials in central Scotland. By Durham RC, Chambers JA, Power KG, Sharp DM, Macdonald RR, Major KA, et al. No. 43 The effectiveness and cost-effectiveness of dual-chamber pacemakers compared with single-chamber pacemakers for bradycardia due to atrioventricular block or sick sinus syndrome: systematic review and economic evaluation. By Castelnuovo E, Stein K, Pitt M, Garside R, Payne E.

No. 46 The effectiveness of the Heidelberg Retina Tomograph and laser diagnostic glaucoma scanning system (GDx) in detecting and monitoring glaucoma. By Kwartz AJ, Henson DB, Harper RA, Spencer AF, McLeod D. No. 47 Clinical and cost-effectiveness of autologous chondrocyte implantation for cartilage defects in knee joints: systematic review and economic evaluation. By Clar C, Cummins E, McIntyre L, Thomas S, Lamb J, Bain L, et al. No. 48 Systematic review of effectiveness of different treatments for childhood retinoblastoma. By McDaid C, Hartley S, Bagnall A-M, Ritchie G, Light K, Riemsma R. No. 49 Towards evidence-based guidelines for the prevention of venous thromboembolism: systematic reviews of mechanical methods, oral anticoagulation, dextran and regional anaesthesia as thromboprophylaxis. By Roderick P, Ferris G, Wilson K, Halls H, Jackson D, Collins R, et al. No. 50 The effectiveness and cost-effectiveness of parent training/education programmes for the treatment of conduct disorder, including oppositional defiant disorder, in children. By Dretzke J, Frew E, Davenport C, Barlow J, Stewart-Brown S, Sandercock J, et al.

Volume 10, 2006 No. 1 The clinical and cost-effectiveness of donepezil, rivastigmine, galantamine and memantine for Alzheimer’s disease. By Loveman E, Green C, Kirby J, Takeda A, Picot J, Payne E, et al.

No. 44 Newborn screening for congenital heart defects: a systematic review and cost-effectiveness analysis. By Knowles R, Griebsch I, Dezateux C, Brown J, Bull C, Wren C.

No. 2 FOOD: a multicentre randomised trial evaluating feeding policies in patients admitted to hospital with a recent stroke. By Dennis M, Lewis S, Cranswick G, Forbes J.

No. 45 The clinical and cost-effectiveness of left ventricular assist devices for end-stage heart failure: a systematic review and economic evaluation. By Clegg AJ, Scott DA, Loveman E, Colquitt J, Hutchinson J, Royle P, et al.

No. 3 The clinical effectiveness and costeffectiveness of computed tomography screening for lung cancer: systematic reviews. By Black C, Bagust A, Boland A, Walker S, McLeod C, De Verteuil R, et al.

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No. 4 A systematic review of the effectiveness and cost-effectiveness of neuroimaging assessments used to visualise the seizure focus in people with refractory epilepsy being considered for surgery. By Whiting P, Gupta R, Burch J, Mujica Mota RE, Wright K, Marson A, et al. No. 5 Comparison of conference abstracts and presentations with full-text articles in the health technology assessments of rapidly evolving technologies. By Dundar Y, Dodd S, Dickson R, Walley T, Haycox A, Williamson PR. No. 6 Systematic review and evaluation of methods of assessing urinary incontinence. By Martin JL, Williams KS, Abrams KR, Turner DA, Sutton AJ, Chapple C, et al. No. 7 The clinical effectiveness and costeffectiveness of newer drugs for children with epilepsy. A systematic review. By Connock M, Frew E, Evans B-W, Bryan S, Cummins C, Fry-Smith A, et al. No. 8 Surveillance of Barrett’s oesophagus: exploring the uncertainty through systematic review, expert workshop and economic modelling. By Garside R, Pitt M, Somerville M, Stein K, Price A, Gilbert N. No. 9 Topotecan, pegylated liposomal doxorubicin hydrochloride and paclitaxel for second-line or subsequent treatment of advanced ovarian cancer: a systematic review and economic evaluation. By Main C, Bojke L, Griffin S, Norman G, Barbieri M, Mather L, et al. No. 10 Evaluation of molecular techniques in prediction and diagnosis of cytomegalovirus disease in immunocompromised patients. By Szczepura A, Westmoreland D, Vinogradova Y, Fox J, Clark M.

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No. 11 Screening for thrombophilia in high-risk situations: systematic review and costeffectiveness analysis. The Thrombosis: Risk and Economic Assessment of Thrombophilia Screening (TREATS) study. By Wu O, Robertson L, Twaddle S, Lowe GDO, Clark P, Greaves M, et al.

No. 12 A series of systematic reviews to inform a decision analysis for sampling and treating infected diabetic foot ulcers. By Nelson EA, O’Meara S, Craig D, Iglesias C, Golder S, Dalton J, et al.

No. 20 A systematic review of the clinical effectiveness and cost-effectiveness of enzyme replacement therapies for Fabry’s disease and mucopolysaccharidosis type 1. By Connock M, Juarez-Garcia A, Frew E, Mans A, Dretzke J, Fry-Smith A, et al.

No. 13 Randomised clinical trial, observational study and assessment of costeffectiveness of the treatment of varicose veins (REACTIV trial). By Michaels JA, Campbell WB, Brazier JE, MacIntyre JB, Palfreyman SJ, Ratcliffe J, et al.

No. 21 Health benefits of antiviral therapy for mild chronic hepatitis C: randomised controlled trial and economic evaluation. By Wright M, Grieve R, Roberts J, Main J, Thomas HC on behalf of the UK Mild Hepatitis C Trial Investigators.

No. 14 The cost-effectiveness of screening for oral cancer in primary care. By Speight PM, Palmer S, Moles DR, Downer MC, Smith DH, Henriksson M et al. No. 15 Measurement of the clinical and costeffectiveness of non-invasive diagnostic testing strategies for deep vein thrombosis. By Goodacre S, Sampson F, Stevenson M, Wailoo A, Sutton A, Thomas S, et al. No. 16 Systematic review of the effectiveness and cost-effectiveness of HealOzone® for the treatment of occlusal pit/fissure caries and root caries. By Brazzelli M, McKenzie L, Fielding S, Fraser C, Clarkson J, Kilonzo M, et al. No. 17 Randomised controlled trials of conventional antipsychotic versus new atypical drugs, and new atypical drugs versus clozapine, in people with schizophrenia responding poorly to, or intolerant of, current drug treatment. By Lewis SW, Davies L, Jones PB, Barnes TRE, Murray RM, Kerwin R, et al.

No. 22 Pressure relieving support surfaces: a randomised evaluation. By Nixon J, Nelson EA, Cranny G, Iglesias CP, Hawkins K, Cullum NA, et al. No. 23 A systematic review and economic model of the effectiveness and costeffectiveness of methylphenidate, dexamfetamine and atomoxetine for the treatment of attention deficit hyperactivity disorder in children and adolescents. By King S, Griffin S, Hodges Z, Weatherly H, Asseburg C, Richardson G, et al. No. 24 The clinical effectiveness and costeffectiveness of enzyme replacement therapy for Gaucher’s disease: a systematic review. By Connock M, Burls A, Frew E, Fry-Smith A, Juarez-Garcia A, McCabe C, et al. No. 25 Effectiveness and cost-effectiveness of salicylic acid and cryotherapy for cutaneous warts. An economic decision model. By Thomas KS, Keogh-Brown MR, Chalmers JR, Fordham RJ, Holland RC, Armstrong SJ, et al.

No. 18 Diagnostic tests and algorithms used in the investigation of haematuria: systematic reviews and economic evaluation. By Rodgers M, Nixon J, Hempel S, Aho T, Kelly J, Neal D, et al.

No. 26 A systematic literature review of the effectiveness of non-pharmacological interventions to prevent wandering in dementia and evaluation of the ethical implications and acceptability of their use. By Robinson L, Hutchings D, Corner L, Beyer F, Dickinson H, Vanoli A, et al.

No. 19 Cognitive behavioural therapy in addition to antispasmodic therapy for irritable bowel syndrome in primary care: randomised controlled trial. By Kennedy TM, Chalder T, McCrone P, Darnley S, Knapp M, Jones RH, et al.

No. 27 A review of the evidence on the effects and costs of implantable cardioverter defibrillator therapy in different patient groups, and modelling of costeffectiveness and cost–utility for these groups in a UK context. By Buxton M, Caine N, Chase D, Connelly D, Grace A, Jackson C, et al.

Health Technology Assessment 2007; Vol. 11: No. 32

No. 28 Adefovir dipivoxil and pegylated interferon alfa-2a for the treatment of chronic hepatitis B: a systematic review and economic evaluation. By Shepherd J, Jones J, Takeda A, Davidson P, Price A.

No. 37 Cognitive behavioural therapy in chronic fatigue syndrome: a randomised controlled trial of an outpatient group programme. By O’Dowd H, Gladwell P, Rogers CA, Hollinghurst S, Gregory A.

No. 46 Etanercept and efalizumab for the treatment of psoriasis: a systematic review. By Woolacott N, Hawkins N, Mason A, Kainth A, Khadjesari Z, Bravo Vergel Y, et al.

No. 29 An evaluation of the clinical and costeffectiveness of pulmonary artery catheters in patient management in intensive care: a systematic review and a randomised controlled trial. By Harvey S, Stevens K, Harrison D, Young D, Brampton W, McCabe C, et al.

No. 38 A comparison of the cost-effectiveness of five strategies for the prevention of nonsteroidal anti-inflammatory drug-induced gastrointestinal toxicity: a systematic review with economic modelling. By Brown TJ, Hooper L, Elliott RA, Payne K, Webb R, Roberts C, et al.

No. 47 Systematic reviews of clinical decision tools for acute abdominal pain. By Liu JLY, Wyatt JC, Deeks JJ, Clamp S, Keen J, Verde P, et al.

No. 30 Accurate, practical and cost-effective assessment of carotid stenosis in the UK. By Wardlaw JM, Chappell FM, Stevenson M, De Nigris E, Thomas S, Gillard J, et al.

No. 39 The effectiveness and cost-effectiveness of computed tomography screening for coronary artery disease: systematic review. By Waugh N, Black C, Walker S, McIntyre L, Cummins E, Hillis G.

No. 31 Etanercept and infliximab for the treatment of psoriatic arthritis: a systematic review and economic evaluation. By Woolacott N, Bravo Vergel Y, Hawkins N, Kainth A, Khadjesari Z, Misso K, et al. No. 32 The cost-effectiveness of testing for hepatitis C in former injecting drug users. By Castelnuovo E, Thompson-Coon J, Pitt M, Cramp M, Siebert U, Price A, et al. No. 33 Computerised cognitive behaviour therapy for depression and anxiety update: a systematic review and economic evaluation. By Kaltenthaler E, Brazier J, De Nigris E, Tumur I, Ferriter M, Beverley C, et al. No. 34 Cost-effectiveness of using prognostic information to select women with breast cancer for adjuvant systemic therapy. By Williams C, Brunskill S, Altman D, Briggs A, Campbell H, Clarke M, et al.

No. 40 What are the clinical outcome and costeffectiveness of endoscopy undertaken by nurses when compared with doctors? A Multi-Institution Nurse Endoscopy Trial (MINuET). By Williams J, Russell I, Durai D, Cheung W-Y, Farrin A, Bloor K, et al. No. 41 The clinical and cost-effectiveness of oxaliplatin and capecitabine for the adjuvant treatment of colon cancer: systematic review and economic evaluation. By Pandor A, Eggington S, Paisley S, Tappenden P, Sutcliffe P. No. 42 A systematic review of the effectiveness of adalimumab, etanercept and infliximab for the treatment of rheumatoid arthritis in adults and an economic evaluation of their costeffectiveness. By Chen Y-F, Jobanputra P, Barton P, Jowett S, Bryan S, Clark W, et al. No. 43 Telemedicine in dermatology: a randomised controlled trial. By Bowns IR, Collins K, Walters SJ, McDonagh AJG.

No. 35 Psychological therapies including dialectical behaviour therapy for borderline personality disorder: a systematic review and preliminary economic evaluation. By Brazier J, Tumur I, Holmes M, Ferriter M, Parry G, Dent-Brown K, et al.

No. 44 Cost-effectiveness of cell salvage and alternative methods of minimising perioperative allogeneic blood transfusion: a systematic review and economic model. By Davies L, Brown TJ, Haynes S, Payne K, Elliott RA, McCollum C.

No. 36 Clinical effectiveness and costeffectiveness of tests for the diagnosis and investigation of urinary tract infection in children: a systematic review and economic model. By Whiting P, Westwood M, Bojke L, Palmer S, Richardson G, Cooper J, et al.

No. 45 Clinical effectiveness and costeffectiveness of laparoscopic surgery for colorectal cancer: systematic reviews and economic evaluation. By Murray A, Lourenco T, de Verteuil R, Hernandez R, Fraser C, McKinley A, et al.

No. 48 Evaluation of the ventricular assist device programme in the UK. By Sharples L, Buxton M, Caine N, Cafferty F, Demiris N, Dyer M, et al. No. 49 A systematic review and economic model of the clinical and costeffectiveness of immunosuppressive therapy for renal transplantation in children. By Yao G, Albon E, Adi Y, Milford D, Bayliss S, Ready A, et al. No. 50 Amniocentesis results: investigation of anxiety. The ARIA trial. By Hewison J, Nixon J, Fountain J, Cocks K, Jones C, Mason G, et al.

Volume 11, 2007 No. 1 Pemetrexed disodium for the treatment of malignant pleural mesothelioma: a systematic review and economic evaluation. By Dundar Y, Bagust A, Dickson R, Dodd S, Green J, Haycox A, et al. No. 2 A systematic review and economic model of the clinical effectiveness and cost-effectiveness of docetaxel in combination with prednisone or prednisolone for the treatment of hormone-refractory metastatic prostate cancer. By Collins R, Fenwick E, Trowman R, Perard R, Norman G, Light K, et al. No. 3 A systematic review of rapid diagnostic tests for the detection of tuberculosis infection. By Dinnes J, Deeks J, Kunst H, Gibson A, Cummins E, Waugh N, et al. No. 4 The clinical effectiveness and costeffectiveness of strontium ranelate for the prevention of osteoporotic fragility fractures in postmenopausal women. By Stevenson M, Davis S, Lloyd-Jones M, Beverley C.

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Health Technology Assessment reports published to date

No. 5 A systematic review of quantitative and qualitative research on the role and effectiveness of written information available to patients about individual medicines. By Raynor DK, Blenkinsopp A, Knapp P, Grime J, Nicolson DJ, Pollock K, et al.

No. 13 A systematic review and economic evaluation of epoetin alfa, epoetin beta and darbepoetin alfa in anaemia associated with cancer, especially that attributable to cancer treatment. By Wilson J, Yao GL, Raftery J, Bohlius J, Brunskill S, Sandercock J, et al.

No. 6 Oral naltrexone as a treatment for relapse prevention in formerly opioid-dependent drug users: a systematic review and economic evaluation. By Adi Y, Juarez-Garcia A, Wang D, Jowett S, Frew E, Day E, et al.

No. 14 A systematic review and economic evaluation of statins for the prevention of coronary events. By Ward S, Lloyd Jones M, Pandor A, Holmes M, Ara R, Ryan A, et al.

No. 22 A systematic review of the routine monitoring of growth in children of primary school age to identify growth-related conditions. By Fayter D, Nixon J, Hartley S, Rithalia A, Butler G, Rudolf M, et al.

No. 15 A systematic review of the effectiveness and cost-effectiveness of different models of community-based respite care for frail older people and their carers. By Mason A, Weatherly H, Spilsbury K, Arksey H, Golder S, Adamson J, et al.

No. 23 Systematic review of the effectiveness of preventing and treating Staphylococcus aureus carriage in reducing peritoneal catheter-related infections. By McCormack K, Rabindranath K, Kilonzo M, Vale L, Fraser C, McIntyre L, et al.

No. 7 Glucocorticoid-induced osteoporosis: a systematic review and cost–utility analysis. By Kanis JA, Stevenson M, McCloskey EV, Davis S, Lloyd-Jones M. No. 8 Epidemiological, social, diagnostic and economic evaluation of population screening for genital chlamydial infection. By Low N, McCarthy A, Macleod J, Salisbury C, Campbell R, Roberts TE, et al. No. 9 Methadone and buprenorphine for the management of opioid dependence: a systematic review and economic evaluation. By Connock M, Juarez-Garcia A, Jowett S, Frew E, Liu Z, Taylor RJ, et al. No. 10 Exercise Evaluation Randomised Trial (EXERT): a randomised trial comparing GP referral for leisure centre-based exercise, community-based walking and advice only. By Isaacs AJ, Critchley JA, See Tai S, Buckingham K, Westley D, Harridge SDR, et al. No. 11 Interferon alfa (pegylated and non-pegylated) and ribavirin for the treatment of mild chronic hepatitis C: a systematic review and economic evaluation. By Shepherd J, Jones J, Hartwell D, Davidson P, Price A, Waugh N.

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No. 12 Systematic review and economic evaluation of bevacizumab and cetuximab for the treatment of metastatic colorectal cancer. By Tappenden P, Jones R, Paisley S, Carroll C.

No. 16 Additional therapy for young children with spastic cerebral palsy: a randomised controlled trial. By Weindling AM, Cunningham CC, Glenn SM, Edwards RT, Reeves DJ. No. 17 Screening for type 2 diabetes: literature review and economic modelling. By Waugh N, Scotland G, McNamee P, Gillett M, Brennan A, Goyder E, et al. No. 18 The effectiveness and cost-effectiveness of cinacalcet for secondary hyperparathyroidism in end-stage renal disease patients on dialysis: a systematic review and economic evaluation. By Garside R, Pitt M, Anderson R, Mealing S, Roome C, Snaith A, et al. No. 19 The clinical effectiveness and cost-effectiveness of gemcitabine for metastatic breast cancer: a systematic review and economic evaluation. By Takeda AL, Jones J, Loveman E, Tan SC, Clegg AJ. No. 20 A systematic review of duplex ultrasound, magnetic resonance angiography and computed tomography angiography for the diagnosis and assessment of symptomatic, lower limb peripheral arterial disease. By Collins R, Cranny G, Burch J, Aguiar-Ibáñez R, Craig D, Wright K, et al.

No. 21 The clinical effectiveness and costeffectiveness of treatments for children with idiopathic steroid-resistant nephrotic syndrome: a systematic review. By Colquitt JL, Kirby J, Green C, Cooper K, Trompeter RS.

No. 24 The clinical effectiveness and cost of repetitive transcranial magnetic stimulation versus electroconvulsive therapy in severe depression: a multicentre pragmatic randomised controlled trial and economic analysis. By McLoughlin DM, Mogg A, Eranti S, Pluck G, Purvis R, Edwards D, et al. No. 25 A randomised controlled trial and economic evaluation of direct versus indirect and individual versus group modes of speech and language therapy for children with primary language impairment. By Boyle J, McCartney E, Forbes J, O’Hare A. No. 26 Hormonal therapies for early breast cancer: systematic review and economic evaluation. By Hind D, Ward S, De Nigris E, Simpson E, Carroll C, Wyld L. No. 27 Cardioprotection against the toxic effects of anthracyclines given to children with cancer: a systematic review. By Bryant J, Picot J, Levitt G, Sullivan I, Baxter L, Clegg A. No. 28 Adalimumab, etanercept and infliximab for the treatment of ankylosing spondylitis: a systematic review and economic evaluation. By McLeod C, Bagust A, Boland A, Dagenais P, Dickson R, Dundar Y, et al.

Health Technology Assessment 2007; Vol. 11: No. 32

No. 29 Prenatal screening and treatment strategies to prevent group B streptococcal and other bacterial infections in early infancy: cost-effectiveness and expected value of information analyses. By Colbourn T, Asseburg C, Bojke L, Philips Z, Claxton K, Ades AE, et al.

No. 31 A randomised controlled trial of postoperative radiotherapy following breast-conserving surgery in a minimum-risk older population. The PRIME trial. By Prescott RJ, Kunkler IH, Williams LJ, King CC, Jack W, van der Pol M, et al.

No. 32 Current practice, accuracy, effectiveness and cost-effectiveness of the school entry hearing screen. By Bamford J, Fortnum H, Bristow K, Smith J, Vamvakas G, Davies L, et al.

No. 30 Clinical effectiveness and costeffectiveness of bone morphogenetic proteins in the non-healing of fractures and spinal fusion: a systematic review. By Garrison KR, Donell S, Ryder J, Shemilt I, Mugford M, Harvey I, et al.

Health Technology Assessment 2007; Vol. 11: No. 32

Health Technology Assessment Programme Director, Professor Tom Walley, Director, NHS HTA Programme, Department of Pharmacology & Therapeutics, University of Liverpool

Deputy Director, Professor Jon Nicholl, Director, Medical Care Research Unit, University of Sheffield, School of Health and Related Research

Prioritisation Strategy Group Members Chair, Professor Tom Walley, Director, NHS HTA Programme, Department of Pharmacology & Therapeutics, University of Liverpool

Professor Bruce Campbell, Consultant Vascular & General Surgeon, Royal Devon & Exeter Hospital Professor Robin E Ferner, Consultant Physician and Director, West Midlands Centre for Adverse Drug Reactions, City Hospital NHS Trust, Birmingham

Dr Edmund Jessop, Medical Adviser, National Specialist, Commissioning Advisory Group (NSCAG), Department of Health, London

Dr Ron Zimmern, Director, Public Health Genetics Unit, Strangeways Research Laboratories, Cambridge

Professor Jon Nicholl, Director, Medical Care Research Unit, University of Sheffield, School of Health and Related Research

HTA Commissioning Board Members Programme Director, Professor Tom Walley, Director, NHS HTA Programme, Department of Pharmacology & Therapeutics, University of Liverpool

Professor Deborah Ashby, Professor of Medical Statistics, Department of Environmental and Preventative Medicine, Queen Mary University of London

Chair, Professor Jon Nicholl, Director, Medical Care Research Unit, University of Sheffield, School of Health and Related Research

Professor Ann Bowling, Professor of Health Services Research, Primary Care and Population Studies, University College London

Deputy Chair, Dr Andrew Farmer, University Lecturer in General Practice, Department of Primary Health Care, University of Oxford

Professor John Cairns, Professor of Health Economics, Public Health Policy, London School of Hygiene and Tropical Medicine, London

Dr Jeffrey Aronson, Reader in Clinical Pharmacology, Department of Clinical Pharmacology, Radcliffe Infirmary, Oxford

Professor Nicky Cullum, Director of Centre for Evidence Based Nursing, Department of Health Sciences, University of York Professor Jon Deeks, Professor of Health Statistics, University of Birmingham

Professor Jenny Donovan, Professor of Social Medicine, Department of Social Medicine, University of Bristol Professor Freddie Hamdy, Professor of Urology, University of Sheffield Professor Allan House, Professor of Liaison Psychiatry, University of Leeds Professor Sallie Lamb, Director, Warwick Clinical Trials Unit, University of Warwick Professor Stuart Logan, Director of Health & Social Care Research, The Peninsula Medical School, Universities of Exeter & Plymouth Professor Miranda Mugford, Professor of Health Economics, University of East Anglia Dr Linda Patterson, Consultant Physician, Department of Medicine, Burnley General Hospital

Professor Ian Roberts, Professor of Epidemiology & Public Health, Intervention Research Unit, London School of Hygiene and Tropical Medicine Professor Mark Sculpher, Professor of Health Economics, Centre for Health Economics, Institute for Research in the Social Services, University of York Professor Kate Thomas, Professor of Complementary and Alternative Medicine, University of Leeds Professor David John Torgerson, Director of York Trial Unit, Department of Health Sciences, University of York Professor Hywel Williams, Professor of Dermato-Epidemiology, University of Nottingham

185 Current and past membership details of all HTA ‘committees’ are available from the HTA website (www.hta.ac.uk)

Health Technology Assessment Programme

Diagnostic Technologies & Screening Panel Members Chair, Dr Ron Zimmern, Director of the Public Health Genetics Unit, Strangeways Research Laboratories, Cambridge

Ms Norma Armston, Freelance Consumer Advocate, Bolton Professor Max Bachmann, Professor of Health Care Interfaces, Department of Health Policy and Practice, University of East Anglia Professor Rudy Bilous Professor of Clinical Medicine & Consultant Physician, The Academic Centre, South Tees Hospitals NHS Trust Ms Dea Birkett, Service User Representative, London

Dr Paul Cockcroft, Consultant Medical Microbiologist and Clinical Director of Pathology, Department of Clinical Microbiology, St Mary's Hospital, Portsmouth Professor Adrian K Dixon, Professor of Radiology, University Department of Radiology, University of Cambridge Clinical School Dr David Elliman, Consultant in Community Child Health, Islington PCT & Great Ormond Street Hospital, London Professor Glyn Elwyn, Research Chair, Centre for Health Sciences Research, Cardiff University, Department of General Practice, Cardiff Professor Paul Glasziou, Director, Centre for Evidence-Based Practice, University of Oxford

Dr Jennifer J Kurinczuk, Consultant Clinical Epidemiologist, National Perinatal Epidemiology Unit, Oxford Dr Susanne M Ludgate, Clinical Director, Medicines & Healthcare Products Regulatory Agency, London Mr Stephen Pilling, Director, Centre for Outcomes, Research & Effectiveness, Joint Director, National Collaborating Centre for Mental Health, University College London Mrs Una Rennard, Service User Representative, Oxford Dr Phil Shackley, Senior Lecturer in Health Economics, Academic Vascular Unit, University of Sheffield

Dr Margaret Somerville, Director of Public Health Learning, Peninsula Medical School, University of Plymouth Dr Graham Taylor, Scientific Director & Senior Lecturer, Regional DNA Laboratory, The Leeds Teaching Hospitals Professor Lindsay Wilson Turnbull, Scientific Director, Centre for MR Investigations & YCR Professor of Radiology, University of Hull Professor Martin J Whittle, Clinical Co-director, National Co-ordinating Centre for Women’s and Childhealth Dr Dennis Wright, Consultant Biochemist & Clinical Director, The North West London Hospitals NHS Trust, Middlesex

Pharmaceuticals Panel Members Chair, Professor Robin Ferner, Consultant Physician and Director, West Midlands Centre for Adverse Drug Reactions, City Hospital NHS Trust, Birmingham

Professor Imti Choonara, Professor in Child Health, Academic Division of Child Health, University of Nottingham Professor John Geddes, Professor of Epidemiological Psychiatry, University of Oxford Mrs Barbara Greggains, Non-Executive Director, Greggains Management Ltd

Ms Anne Baileff, Consultant Nurse in First Contact Care, Southampton City Primary Care Trust, University of Southampton

Dr Bill Gutteridge, Medical Adviser, National Specialist Commissioning Advisory Group (NSCAG), London Mrs Sharon Hart, Consultant Pharmaceutical Adviser, Reading

Dr Jonathan Karnon, Senior Research Fellow, Health Economics and Decision Science, University of Sheffield Dr Yoon Loke, Senior Lecturer in Clinical Pharmacology, University of East Anglia Ms Barbara Meredith, Lay Member, Epsom Dr Andrew Prentice, Senior Lecturer and Consultant Obstetrician & Gynaecologist, Department of Obstetrics & Gynaecology, University of Cambridge

Dr Martin Shelly, General Practitioner, Leeds Mrs Katrina Simister, Assistant Director New Medicines, National Prescribing Centre, Liverpool Dr Richard Tiner, Medical Director, Medical Department, Association of the British Pharmaceutical Industry, London

Dr Frances Rotblat, CPMP Delegate, Medicines & Healthcare Products Regulatory Agency, London

186 Current and past membership details of all HTA ‘committees’ are available from the HTA website (www.hta.ac.uk)

Health Technology Assessment 2007; Vol. 11: No. 32

Therapeutic Procedures Panel Members Chair, Professor Bruce Campbell, Consultant Vascular and General Surgeon, Department of Surgery, Royal Devon & Exeter Hospital

Professor Matthew Cooke, Professor of Emergency Medicine, Warwick Emergency Care and Rehabilitation, University of Warwick Mr Mark Emberton, Senior Lecturer in Oncological Urology, Institute of Urology, University College Hospital

Dr Mahmood Adil, Deputy Regional Director of Public Health, Department of Health, Manchester

Professor Paul Gregg, Professor of Orthopaedic Surgical Science, Department of General Practice and Primary Care, South Tees Hospital NHS Trust, Middlesbrough

Dr Aileen Clarke, Consultant in Public Health, Public Health Resource Unit, Oxford

Ms Maryann L Hardy, Lecturer, Division of Radiography, University of Bradford

Dr Simon de Lusignan, Senior Lecturer, Primary Care Informatics, Department of Community Health Sciences, St George’s Hospital Medical School, London Dr Peter Martin, Consultant Neurologist, Addenbrooke’s Hospital, Cambridge

Dr John C Pounsford, Consultant Physician, Directorate of Medical Services, North Bristol NHS Trust Dr Karen Roberts, Nurse Consultant, Queen Elizabeth Hospital, Gateshead

Professor Neil McIntosh, Edward Clark Professor of Child Life & Health, Department of Child Life & Health, University of Edinburgh

Dr Vimal Sharma, Consultant Psychiatrist/Hon. Senior Lecturer, Mental Health Resource Centre, Cheshire and Wirral Partnership NHS Trust, Wallasey

Professor Jim Neilson, Professor of Obstetrics and Gynaecology, Department of Obstetrics and Gynaecology, University of Liverpool

Professor Scott Weich, Professor of Psychiatry, Division of Health in the Community, University of Warwick

Disease Prevention Panel Members Chair, Dr Edmund Jessop, Medical Adviser, National Specialist Commissioning Advisory Group (NSCAG), London

Dr Elizabeth Fellow-Smith, Medical Director, West London Mental Health Trust, Middlesex Mr Ian Flack, Director PPI Forum Support, Council of Ethnic Minority Voluntary Sector Organisations, Stratford Dr John Jackson, General Practitioner, Newcastle upon Tyne

Mrs Sheila Clark, Chief Executive, St James’s Hospital, Portsmouth Mr Richard Copeland, Lead Pharmacist: Clinical Economy/Interface, Wansbeck General Hospital, Northumberland

Professor Yi Mien Koh, Director of Public Health and Medical Director, London NHS (North West London Strategic Health Authority), London Ms Jeanett Martin, Director of Clinical Leadership & Quality, Lewisham PCT, London Dr Chris McCall, General Practitioner, Dorset

Mrs Veronica James, Chief Officer, Horsham District Age Concern, Horsham

Dr David Pencheon, Director, Eastern Region Public Health Observatory, Cambridge

Professor Mike Kelly, Director, Centre for Public Health Excellence, National Institute for Health and Clinical Excellence, London

Dr Ken Stein, Senior Clinical Lecturer in Public Health, Director, Peninsula Technology Assessment Group, University of Exeter, Exeter

Dr Carol Tannahill, Director, Glasgow Centre for Population Health, Glasgow Professor Margaret Thorogood, Professor of Epidemiology, University of Warwick, Coventry Dr Ewan Wilkinson, Consultant in Public Health, Royal Liverpool University Hospital, Liverpool

187 Current and past membership details of all HTA ‘committees’ are available from the HTA website (www.hta.ac.uk)

Health Technology Assessment Programme

Expert Advisory Network Members Professor Douglas Altman, Professor of Statistics in Medicine, Centre for Statistics in Medicine, University of Oxford

Professor Carol Dezateux, Professor of Paediatric Epidemiology, London Dr Keith Dodd, Consultant Paediatrician, Derby

Professor John Bond, Director, Centre for Health Services Research, University of Newcastle upon Tyne, School of Population & Health Sciences, Newcastle upon Tyne

Mr John Dunning, Consultant Cardiothoracic Surgeon, Cardiothoracic Surgical Unit, Papworth Hospital NHS Trust, Cambridge

Professor Andrew Bradbury, Professor of Vascular Surgery, Solihull Hospital, Birmingham

Mr Jonothan Earnshaw, Consultant Vascular Surgeon, Gloucestershire Royal Hospital, Gloucester

Mr Shaun Brogan, Chief Executive, Ridgeway Primary Care Group, Aylesbury Mrs Stella Burnside OBE, Chief Executive, Regulation and Improvement Authority, Belfast Ms Tracy Bury, Project Manager, World Confederation for Physical Therapy, London Professor Iain T Cameron, Professor of Obstetrics and Gynaecology and Head of the School of Medicine, University of Southampton Dr Christine Clark, Medical Writer & Consultant Pharmacist, Rossendale Professor Collette Clifford, Professor of Nursing & Head of Research, School of Health Sciences, University of Birmingham, Edgbaston, Birmingham Professor Barry Cookson, Director, Laboratory of Healthcare Associated Infection, Health Protection Agency, London Dr Carl Counsell, Clinical Senior Lecturer in Neurology, Department of Medicine & Therapeutics, University of Aberdeen Professor Howard Cuckle, Professor of Reproductive Epidemiology, Department of Paediatrics, Obstetrics & Gynaecology, University of Leeds Dr Katherine Darton, Information Unit, MIND – The Mental Health Charity, London

Professor Martin Eccles, Professor of Clinical Effectiveness, Centre for Health Services Research, University of Newcastle upon Tyne Professor Pam Enderby, Professor of Community Rehabilitation, Institute of General Practice and Primary Care, University of Sheffield Professor Gene Feder, Professor of Primary Care Research & Development, Centre for Health Sciences, Barts & The London Queen Mary’s School of Medicine & Dentistry, London

Professor Stan Kaye, Cancer Research UK Professor of Medical Oncology, Section of Medicine, Royal Marsden Hospital & Institute of Cancer Research, Surrey Dr Duncan Keeley, General Practitioner (Dr Burch & Ptnrs), The Health Centre, Thame Dr Donna Lamping, Research Degrees Programme Director & Reader in Psychology, Health Services Research Unit, London School of Hygiene and Tropical Medicine, London Mr George Levvy, Chief Executive, Motor Neurone Disease Association, Northampton Professor James Lindesay, Professor of Psychiatry for the Elderly, University of Leicester, Leicester General Hospital Professor Julian Little, Professor of Human Genome Epidemiology, Department of Epidemiology & Community Medicine, University of Ottawa

Mr Leonard R Fenwick, Chief Executive, Newcastle upon Tyne Hospitals NHS Trust

Professor Rajan Madhok, Consultant in Public Health, South Manchester Primary Care Trust, Manchester

Mrs Gillian Fletcher, Antenatal Teacher & Tutor and President, National Childbirth Trust, Henfield

Professor Alexander Markham, Director, Molecular Medicine Unit, St James’s University Hospital, Leeds

Professor Jayne Franklyn, Professor of Medicine, Department of Medicine, University of Birmingham, Queen Elizabeth Hospital, Edgbaston, Birmingham Dr Neville Goodman, Consultant Anaesthetist, Southmead Hospital, Bristol Professor Robert E Hawkins, CRC Professor and Director of Medical Oncology, Christie CRC Research Centre, Christie Hospital NHS Trust, Manchester Professor Allen Hutchinson, Director of Public Health & Deputy Dean of ScHARR, Department of Public Health, University of Sheffield Professor Peter Jones, Professor of Psychiatry, University of Cambridge, Cambridge

Professor Alistaire McGuire, Professor of Health Economics, London School of Economics

Professor Chris Price, Visiting Professor in Clinical Biochemistry, University of Oxford Professor William Rosenberg, Professor of Hepatology and Consultant Physician, University of Southampton, Southampton Professor Peter Sandercock, Professor of Medical Neurology, Department of Clinical Neurosciences, University of Edinburgh Dr Susan Schonfield, Consultant in Public Health, Hillingdon PCT, Middlesex Dr Eamonn Sheridan, Consultant in Clinical Genetics, Genetics Department, St James’s University Hospital, Leeds Professor Sarah Stewart-Brown, Professor of Public Health, University of Warwick, Division of Health in the Community Warwick Medical School, LWMS, Coventry Professor Ala Szczepura, Professor of Health Service Research, Centre for Health Services Studies, University of Warwick Dr Ross Taylor, Senior Lecturer, Department of General Practice and Primary Care, University of Aberdeen Mrs Joan Webster, Consumer member, HTA – Expert Advisory Network

Dr Peter Moore, Freelance Science Writer, Ashtead Dr Andrew Mortimore, Public Health Director, Southampton City Primary Care Trust, Southampton Dr Sue Moss, Associate Director, Cancer Screening Evaluation Unit, Institute of Cancer Research, Sutton Mrs Julietta Patnick, Director, NHS Cancer Screening Programmes, Sheffield Professor Robert Peveler, Professor of Liaison Psychiatry, Royal South Hants Hospital, Southampton

188 Current and past membership details of all HTA ‘committees’ are available from the HTA website (www.hta.ac.uk)

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Health Technology Assessment 2007; Vol. 11: No. 32 School entry hearing screen

Feedback The HTA Programme and the authors would like to know your views about this report.

Health Technology Assessment 2007; Vol. 11: No. 32

August 2007

Health Technology Assessment NHS R&D HTA Programme www.hta.ac.uk ISSN 1366-5278

HTA

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