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下一代测序有助于遗传诊断,并改善临床实践中听力损失患者的管理。

Next-generation sequencing facilitates genetic diagnosis and improves the management of patients with hearing loss in clinical practice.

机构信息

Medical Genetic Center, Guangdong Women and Children Hospital, Guangzhou, Guangdong, 510010, China; Maternal and Children Metabolic-Genetic Key Laboratory, Guangdong Women and Children Hospital, Guangzhou, Guangdong, 510010, China.

Medical Genetic Center, Guangdong Women and Children Hospital, Guangzhou, Guangdong, 510010, China.

出版信息

Int J Pediatr Otorhinolaryngol. 2022 Oct;161:111258. doi: 10.1016/j.ijporl.2022.111258. Epub 2022 Jul 31.

DOI:10.1016/j.ijporl.2022.111258
PMID:35939872
Abstract

BACKGROUND

Hearing loss (HL) is a prevalent sensorineural disorder, and is among the most etiologically heterogeneous disorders. With the advent of next-generation sequencing (NGS) technologies, hundreds of candidate genes can be analyzed simultaneously in a cost-effective manner.

METHODS

Ninety-four patients from 87 families diagnosed with non-syndromic or syndromic HL were enrolled. A custom-designed HL panel and clinical exome sequencing (CES) were applied to explore molecular etiology in the cohort, and the efficacy of the two panels was examined.

RESULTS

The etiologic diagnosis for HL has been identified for 36 out of 87 probands (41.4%), 28 with an autosomal recessive (AR) inheritance pattern and 8 with an autosomal dominant (AD) pattern. Candidate variants in 18 different genes were identified in the study cohort, 10 with AR inheritance pattern and 8 with AD pattern. Fourteen of the variants identified in the study were novel.

CONCLUSIONS

The custom-designed HL panel covers almost all known HL-associated genes, and can be used as an effective clinical diagnostic platform; CES evaluates all exons related to clinical symptoms, and is also suitable for clinical diagnosis of HL. Next-generation sequencing facilitates genetic diagnosis and improves the management of patients with HL in the clinical practice.

摘要

背景

听力损失(HL)是一种常见的感觉神经性疾病,也是病因学上最具异质性的疾病之一。随着下一代测序(NGS)技术的出现,可以以具有成本效益的方式同时分析数百个候选基因。

方法

招募了 94 名来自 87 个家庭的被诊断为非综合征或综合征性 HL 的患者。应用定制的 HL 面板和临床外显子组测序(CES)来探索队列中的分子病因,并检查两个面板的功效。

结果

87 名先证者中有 36 名(41.4%)确定了 HL 的病因诊断,其中 28 名呈常染色体隐性(AR)遗传模式,8 名呈常染色体显性(AD)遗传模式。在研究队列中鉴定出 18 个不同基因的候选变异,其中 10 个具有 AR 遗传模式,8 个具有 AD 遗传模式。在研究中鉴定出的 14 个变异是新的。

结论

定制的 HL 面板几乎涵盖了所有已知的与 HL 相关的基因,可作为有效的临床诊断平台;CES 评估与临床症状相关的所有外显子,也适用于 HL 的临床诊断。下一代测序有助于遗传诊断,并改善 HL 患者的临床管理。

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Int J Pediatr Otorhinolaryngol. 2022 Oct;161:111258. doi: 10.1016/j.ijporl.2022.111258. Epub 2022 Jul 31.
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引用本文的文献

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Case report of a novel mutation in the TNC gene in Chinese patients with nonsyndromic hearing loss.中国非综合征型听力损失患者 TNC 基因新突变的病例报告。
Medicine (Baltimore). 2024 Apr 19;103(16):e37702. doi: 10.1097/MD.0000000000037702.
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Exome sequencing in fetuses with short long bones detected by ultrasonography: A retrospective cohort study.
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