Annual hearing screening in children with osteogenesis imperfecta: Results from the first five years in glasgow

IF 16.4 1区化学 Q1 CHEMISTRY, MULTIDISCIPLINARY

Accounts of Chemical Research Pub Date : 2024-09-07 DOI:10.1016/j.ijporl.2024.112096

Emmett Lui , Owen Conlan , Karen Hunter , Avril Mason , Haytham Kubba

{"title":"Annual hearing screening in children with osteogenesis imperfecta: Results from the first five years in glasgow","authors":"Emmett Lui , Owen Conlan , Karen Hunter , Avril Mason , Haytham Kubba","doi":"10.1016/j.ijporl.2024.112096","DOIUrl":null,"url":null,"abstract":"<div><h3>Background</h3><p>Hearing loss is common in people with osteogenesis imperfecta (OI), although exactly how common is unknown. The prevalence of hearing loss in children with OI has been reported to be anything from 0 to 77 %. Brittle Bone Society guidelines suggest that, unless there are ear symptoms, children with OI should have their hearing tested every three years starting at age three. There is limited evidence to support this recommendation. We postulate that annual hearing screening would be easier to manage and would have a worthwhile pick-up rate.</p></div><div><h3>Methods</h3><p>In March 2019 we began a programme of annual hearing screening for all children (ages 0–16) with OI. We collected data on age, genotype, otoscopy findings, tympanometry findings, audiometric test results and subsequent outcomes for the first five years of our programme (2019–2024).</p></div><div><h3>Results</h3><p>Nineteen children with OI participated in the screening programme. Only one abnormality was found: a unilateral mild hearing impairment with a type B tympanogram, suggesting middle ear effusion. This was present in year 2 of the programme but resolved by year 3.</p></div><div><h3>Conclusion</h3><p>The screening programme has a low pickup rate (5 %) for new otological problems in the paediatric population. However, we believe that the low cost and small workload associated with the screening programme justifies continuing it until further conclusions can be drawn.</p></div>","PeriodicalId":1,"journal":{"name":"Accounts of Chemical Research","volume":null,"pages":null},"PeriodicalIF":16.4000,"publicationDate":"2024-09-07","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"https://www.sciencedirect.com/science/article/pii/S0165587624002507/pdfft?md5=9ab3ba5a8da76a5638b9cc24d2ab3ca8&pid=1-s2.0-S0165587624002507-main.pdf","citationCount":"0","resultStr":null,"platform":"Semanticscholar","paperid":null,"PeriodicalName":"Accounts of Chemical Research","FirstCategoryId":"3","ListUrlMain":"https://www.sciencedirect.com/science/article/pii/S0165587624002507","RegionNum":1,"RegionCategory":"化学","ArticlePicture":[],"TitleCN":null,"AbstractTextCN":null,"PMCID":null,"EPubDate":"","PubModel":"","JCR":"Q1","JCRName":"CHEMISTRY, MULTIDISCIPLINARY","Score":null,"Total":0}

引用次数: 0

Abstract

Background

Hearing loss is common in people with osteogenesis imperfecta (OI), although exactly how common is unknown. The prevalence of hearing loss in children with OI has been reported to be anything from 0 to 77 %. Brittle Bone Society guidelines suggest that, unless there are ear symptoms, children with OI should have their hearing tested every three years starting at age three. There is limited evidence to support this recommendation. We postulate that annual hearing screening would be easier to manage and would have a worthwhile pick-up rate.

Methods

In March 2019 we began a programme of annual hearing screening for all children (ages 0–16) with OI. We collected data on age, genotype, otoscopy findings, tympanometry findings, audiometric test results and subsequent outcomes for the first five years of our programme (2019–2024).

Results

Nineteen children with OI participated in the screening programme. Only one abnormality was found: a unilateral mild hearing impairment with a type B tympanogram, suggesting middle ear effusion. This was present in year 2 of the programme but resolved by year 3.

Conclusion

The screening programme has a low pickup rate (5 %) for new otological problems in the paediatric population. However, we believe that the low cost and small workload associated with the screening programme justifies continuing it until further conclusions can be drawn.

查看原文本刊更多论文

成骨不全症儿童的年度听力筛查：格拉斯哥头五年的结果

背景听力损失在成骨不全症（OI）患者中很常见，但具体有多常见尚不清楚。据报道，成骨不全症儿童听力损失的发生率从 0% 到 77% 不等。脆骨协会的指导方针建议，除非有耳部症状，否则患有成骨不全症的儿童应从三岁开始每三年接受一次听力测试。支持这一建议的证据有限。我们推测，每年进行一次听力筛查将更易于管理，而且筛查率也会更高。方法2019 年 3 月，我们开始对所有患有 OI 的儿童（0-16 岁）进行年度听力筛查。我们收集了该计划前五年（2019-2024 年）的年龄、基因型、耳镜检查结果、鼓室测量结果、听力测试结果和后续结果等数据。仅发现了一项异常：单侧轻度听力障碍，鼓室图为 B 型，提示中耳积液。结论：筛查计划在儿童中发现新耳科问题的比例较低（5%）。不过，我们认为，筛查项目成本低、工作量小，因此在得出进一步结论之前，有理由继续实施该项目。

本文章由计算机程序翻译，如有差异，请以英文原文为准。

求助全文

约1分钟内获得全文求助全文

来源期刊

Accounts of Chemical Research 化学-化学综合

CiteScore

31.40

自引率

1.10%

发文量

312

审稿时长

2 months

期刊介绍： Accounts of Chemical Research presents short, concise and critical articles offering easy-to-read overviews of basic research and applications in all areas of chemistry and biochemistry. These short reviews focus on research from the author’s own laboratory and are designed to teach the reader about a research project. In addition, Accounts of Chemical Research publishes commentaries that give an informed opinion on a current research problem. Special Issues online are devoted to a single topic of unusual activity and significance. Accounts of Chemical Research replaces the traditional article abstract with an article "Conspectus." These entries synopsize the research affording the reader a closer look at the content and significance of an article. Through this provision of a more detailed description of the article contents, the Conspectus enhances the article's discoverability by search engines and the exposure for the research.