title: A programme of studies including assessment of diagnostic accuracy of school hearing screening tests and a cost-effectiveness model of school entry hearing screening programmes.
abstract: Identification of permanent hearing impairment at the earliest possible age is crucial to maximise the development of speech and language. Universal newborn hearing screening identifies the majority of the 1 in 1000 children born with a hearing impairment, but later onset can occur at any time and there is no optimum time for further screening. A universal but non-standardised school entry screening (SES) programme is in place in many parts of the UK but its value is questioned.
  
  
  
  To evaluate the diagnostic accuracy of hearing screening tests and the cost-effectiveness of the SES programme in the UK.
  
  
  
  Systematic review, case-control diagnostic accuracy study, comparison of routinely collected data for services with and without a SES programme, parental questionnaires, observation of practical implementation and cost-effectiveness modelling.
  
  
  
  Second- and third-tier audiology services; community.
  
  
  
  Children aged 4-6 years and their parents.
  
  
  
  Diagnostic accuracy of two hearing screening devices, referral rate and source, yield, age at referral and cost per quality-adjusted life-year.
  
  
  
  The review of diagnostic accuracy studies concluded that research to date demonstrates marked variability in the design, methodological quality and results. The pure-tone screen (PTS) (Amplivox, Eynsham, UK) and HearCheck (HC) screener (Siemens, Frimley, UK) devices had high sensitivity (PTS ≥ 89%, HC ≥ 83%) and specificity (PTS ≥ 78%, HC ≥ 83%) for identifying hearing impairment. The rate of referral for hearing problems was 36% lower with SES (Nottingham) relative to no SES (Cambridge) [rate ratio 0.64, 95% confidence interval (CI) 0.59 to 0.69; p < 0.001]. The yield of confirmed cases did not differ between areas with and without SES (rate ratio 0.82, 95% CI 0.63 to 1.06; p = 0.12). The mean age of referral did not differ between areas with and without SES for all referrals but children with confirmed hearing impairment were older at referral in the site with SES (mean age difference 0.47 years, 95% CI 0.24 to 0.70 years; p < 0.001). Parental responses revealed that the consequences to the family of the referral process are minor. A SES programme is unlikely to be cost-effective and, using base-case assumptions, is dominated by a no screening strategy. A SES programme could be cost-effective if there are fewer referrals associated with SES programmes or if referrals occur more quickly with SES programmes.
  
  
  
  A SES programme using the PTS or HC screener is unlikely to be effective in increasing the identified number of cases with hearing impairment and lowering the average age at identification and is therefore unlikely to represent good value for money. This finding is, however, critically dependent on the results of the observational study comparing Nottingham and Cambridge, which has limitations. The following are suggested: systematic reviews of the accuracy of devices used to measure hearing at school entry; characterisation and measurement of the cost-effectiveness of different approaches to the ad-hoc referral system; examination of programme specificity as opposed to test specificity; further observational comparative studies of different programmes; and opportunistic trials of withdrawal of SES programmes.
  
  
  
  Current Controlled Trials ISRCTN61668996.
  
  
  
  This project was funded by the NIHR Health Technology Assessment programme and will be published in full in Health Technology Assessment; Vol. 20, No. 36. See the NIHR Journals Library website for further project information.
authors.affiliation: National Institute for Health Research, Nottingham Hearing Biomedical Research Unit, Hearing and Otology Group, Division of Clinical Neuroscience, School of Medicine, University of Nottingham, Nottingham, UK.
authors.forename: Heather
authors.initials: H
authors.lastname: Fortnum
authors.affiliation: National Institute for Health Research, Collaborations for Leadership in Applied Health Research and Care South West Peninsula, University of Exeter Medical School, Exeter, UK.
authors.forename: Obioha C
authors.initials: OC
authors.lastname: Ukoumunne
authors.affiliation: Institute of Health Research, University of Exeter Medical School, Exeter, UK.
authors.forename: Chris
authors.initials: C
authors.lastname: Hyde
authors.affiliation: Institute of Health Research, University of Exeter Medical School, Exeter, UK.
authors.forename: Rod S
authors.initials: RS
authors.lastname: Taylor
authors.affiliation: National Institute for Health Research, Nottingham Hearing Biomedical Research Unit, Hearing and Otology Group, Division of Clinical Neuroscience, School of Medicine, University of Nottingham, Nottingham, UK.
authors.forename: Mara
authors.initials: M
authors.lastname: Ozolins
authors.affiliation: National Institute for Health Research, Nottingham Hearing Biomedical Research Unit, Hearing and Otology Group, Division of Clinical Neuroscience, School of Medicine, University of Nottingham, Nottingham, UK.
authors.forename: Sam
authors.initials: S
authors.lastname: Errington
authors.affiliation: National Institute for Health Research, Collaborations for Leadership in Applied Health Research and Care South West Peninsula, University of Exeter Medical School, Exeter, UK.
authors.forename: Zhivko
authors.initials: Z
authors.lastname: Zhelev
authors.affiliation: Optimity Advisors, Kemp House, London, UK.
authors.forename: Clive
authors.initials: C
authors.lastname: Pritchard
authors.affiliation: Nottingham Audiology Services, Nottingham University Hospitals, Nottingham, UK.
authors.forename: Claire
authors.initials: C
authors.lastname: Benton
authors.affiliation: Cambridgeshire Community Services, Community Child Health, Ida Darwin Hospital, Fulbourn, Cambridge, UK.
authors.forename: Joanne
authors.initials: J
authors.lastname: Moody
authors.affiliation: Peninsula Clinical Trials Unit, Plymouth University Peninsula Schools of Medicine and Dentistry, Plymouth, UK.
authors.forename: Laura
authors.initials: L
authors.lastname: Cocking
authors.affiliation: Parent representative, Nottingham, UK.
authors.forename: Julian
authors.initials: J
authors.lastname: Watson
authors.affiliation: Optimity Advisors, Kemp House, London, UK.
authors.forename: Sarah
authors.initials: S
authors.lastname: Roberts
mesh_terms: D000367:Age Factors; D002648:Child; D002675:Child, Preschool; D003362:Cost-Benefit Analysis; D006311:Hearing Disorders / Q000175:diagnosis*; D006320:Hearing Tests / Q000191:economics*; D006801:Humans; D008403:Mass Screening / Q000191:economics / Q000458:organization & administration* / Q000706:statistics & numerical data*; D012017:Referral and Consultation / Q000706:statistics & numerical data; D012572:School Health Services / Q000191:economics / Q000458:organization & administration* / Q000706:statistics & numerical data; D012680:Sensitivity and Specificity; D012959:Socioeconomic Factors; D013673:Technology Assessment, Biomedical; D006113:United Kingdom