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与 Waardenburg 综合征 IV 型相关的 SOX10 基因的一种新变体。

A novel variant of the SOX10 gene associated with Waardenburg syndrome type IV.

机构信息

Luoyang Maternal and Child Health Hospital, Luoyang, 471000, China.

出版信息

BMC Med Genomics. 2023 Jun 26;16(1):147. doi: 10.1186/s12920-023-01572-1.

Abstract

BACKGROUND

Waardenburg syndrome (WS) is a rare genetic disorder characterized by varying degrees of sensorineural hearing loss and accumulated pigmentation in the skin, hair and iris. The syndrome is classified into four types (WS1, WS2, WS3, and WS4), each with different clinical phenotypes and underlying genetic causes. The aim of this study was to identify the pathogenic variant in a Chinese family with Waardenburg syndrome type IV.

METHODS

The patient and his parents underwent a thorough medical examination. We applied whole exome sequencing to identify the causal variant on the patient and other family members.

RESULTS

The patient presented with iris pigmentary abnormality, congenital megacolon and sensorineural hearing loss. The clinical diagnosis of the patient was WS4. The whole exome sequencing (WES) revealed a novel variant (c.452_456dup) in the SOX10 gene, which could be responsible for the observed pathogenic of WS4 in this patient. Our analysis suggests that this variant produces a truncated protein that contributes to the development of the disease. The genetic test confirmed the diagnosis of WS4 in the patient from the studied pedigree.

CONCLUSIONS

This present study demonstrated that genetic test based on WES, an effective alternative to regular clinical examinations, helps diagnose WS4. The newly identified SOX10 gene variant can expand the understanding of WS4.

摘要

背景

瓦登伯格综合征(WS)是一种罕见的遗传性疾病,其特征是程度不同的感觉神经性听力损失和皮肤、头发和虹膜色素沉着。该综合征分为四型(WS1、WS2、WS3 和 WS4),每型具有不同的临床表型和潜在的遗传原因。本研究旨在鉴定一个中国 WS4 型家系的致病变异。

方法

对患者及其父母进行了全面的医学检查。我们应用全外显子组测序鉴定患者和其他家庭成员中的致病变异。

结果

患者表现为虹膜色素异常、先天性巨结肠和感觉神经性听力损失。患者的临床诊断为 WS4。全外显子组测序(WES)显示 SOX10 基因中的一个新变异(c.452_456dup),可能导致该患者 WS4 的发病。我们的分析表明,该变异产生的截短蛋白有助于疾病的发生。遗传测试证实了所研究家系中患者的 WS4 诊断。

结论

本研究表明,基于 WES 的基因测试是常规临床检查的有效替代方法,有助于 WS4 的诊断。新发现的 SOX10 基因突变可以扩展对 WS4 的认识。

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