Fortnum Heather, Ukoumunne Obioha C, Hyde Chris, Taylor Rod S, Ozolins Mara, Errington Sam, Zhelev Zhivko, Pritchard Clive, Benton Claire, Moody Joanne, Cocking Laura, Watson Julian, Roberts Sarah
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.
National Institute for Health Research, Collaborations for Leadership in Applied Health Research and Care South West Peninsula, University of Exeter Medical School, Exeter, UK.
Health Technol Assess. 2016 May;20(36):1-178. doi: 10.3310/hta20360.
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.
尽早识别永久性听力障碍对于最大限度地促进言语和语言发展至关重要。新生儿听力普遍筛查可识别出每1000名出生时患有听力障碍儿童中的大多数,但迟发性听力障碍可能在任何时候出现,且没有进行进一步筛查的最佳时机。英国许多地区实施了一项普遍但未标准化的入学听力筛查(SES)计划,但其价值受到质疑。
评估英国听力筛查测试的诊断准确性以及SES计划的成本效益。
系统评价、病例对照诊断准确性研究、对有无SES计划的服务常规收集数据进行比较、家长问卷调查、实际实施观察以及成本效益建模。
二级和三级听力学服务;社区。
4至6岁儿童及其父母。
两种听力筛查设备的诊断准确性、转诊率及来源、确诊率、转诊年龄以及每质量调整生命年的成本。
对诊断准确性研究的综述得出结论,迄今为止的研究表明,在设计、方法质量和结果方面存在显著差异。纯音筛查仪(PTS)(英国伊恩舍姆的Amplivox公司)和听力检查仪(HC)筛查仪(英国弗林利的西门子公司)在识别听力障碍方面具有较高的敏感性(PTS≥89%,HC≥83%)和特异性(PTS≥78%,HC≥83%)。与没有SES计划的地区(剑桥)相比,实施SES计划的地区(诺丁汉)听力问题转诊率低36%[率比0.64,95%置信区间(CI)0.59至0.69;p<0.001]。有无SES计划的地区确诊病例的确诊率无差异(率比0.82,95%CI 0.63至1.06;p=0.12)。对于所有转诊病例,有无SES计划地区的平均转诊年龄无差异,但在实施SES计划的地区,确诊听力障碍的儿童转诊时年龄较大(平均年龄差0.47岁,95%CI 0.24至0.70岁;p<0.001)。家长反馈显示,转诊过程对家庭的影响较小。SES计划不太可能具有成本效益,基于基本假设,其被无筛查策略所主导。如果与SES计划相关的转诊较少,或者SES计划能更快地进行转诊,那么SES计划可能具有成本效益。
使用PTS或HC筛查仪的SES计划不太可能有效增加已识别的听力障碍病例数量并降低平均确诊年龄,因此不太可能物有所值。然而,这一发现严重依赖于比较诺丁汉和剑桥的观察性研究结果,而该研究存在局限性。建议如下:对用于入学听力测量的设备准确性进行系统评价;对临时转诊系统不同方法的成本效益进行描述和测量;考察计划特异性而非测试特异性;对不同计划进行进一步的观察性比较研究;以及对取消SES计划进行机会性试验。
当前对照试验ISRCTN61668996。
本项目由英国国家卫生研究院卫生技术评估计划资助,将在《卫生技术评估》全文发表;第20卷,第36期。有关更多项目信息,请参见英国国家卫生研究院期刊图书馆网站。