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[一名眼皮肤白化病患者的TYR基因变异分析]

[Analysis of TYR gene variant in a patient with Oculocutaneous albinism].

作者信息

Jin Xiaolei, Xie Hanbing, Wang Ping, Yang Shuo, Mai Jingqun, Xiao Xiao, Liu Shanling

机构信息

Department of Rehabilitation Medicine, Children's Department of Medical Genetics/Prenatal Diagnosis Center, West China Second Hospital, Sichuan University, MoE Key Laboratory of Birth Defects and Related Maternal and Child Diseases, Chengdu, Sichuan 610041, China.

出版信息

Zhonghua Yi Xue Yi Chuan Xue Za Zhi. 2025 Mar 10;42(3):349-354. doi: 10.3760/cma.j.cn511374-20240928-00512.

Abstract

OBJECTIVE

To explore the genetic basis of a patient with suspected Oculocutaneous albinism (OCA).

METHODS

An OCA patient presented at the West China Second Hospital of Sichuan University and his mother were selected as the study subjects. Peripheral blood samples were collected for the extraction of, genomic DNA, and whole exome sequencing (WES) was carried out. Candidate variants were verified through specific primer amplification, Sanger sequencing, and agarose gel electrophoresis. Bioinformatic analysis and pathogenicity rating were conducted on the candidate variants. This study has been approved by the Medical Ethics Committee of West China Second Hospital (No. 2024-228).

RESULTS

Genetic testing revealed that the patient had harbored variants in exon 1 of the TYR gene, including a c.157G>T (p.G53C) missense variant and a c.609dup (p.A204fs) frameshifting variant. Specific primer amplification and Sanger sequencing, combined with agarose gel electrophoresis, confirmed that these are compound heterozygous variants. Based on the guidelines from the ACMG, the c.157G>T was rated as likely pathogenic, and c.609dup was rated as pathogenic. Alphafold3 predicted that the variant proteins had significant structural changes.

CONCLUSION

The patient was diagnosed with OCA due to compound heterozygous variants of the TYR gene. Discovery of the c.609dup variant has enriched the mutational spectrum of OCA and provided a basis for genetic counseling and prenatal diagnosis for this patient.

摘要

目的

探讨一名疑似眼皮肤白化病(OCA)患者的遗传基础。

方法

选取一名在四川大学华西第二医院就诊的OCA患者及其母亲作为研究对象。采集外周血样本用于提取基因组DNA,并进行全外显子组测序(WES)。通过特异性引物扩增、Sanger测序和琼脂糖凝胶电泳对候选变异进行验证。对候选变异进行生物信息学分析和致病性评级。本研究已获得华西第二医院医学伦理委员会批准(编号:2024 - 228)。

结果

基因检测显示该患者在TYR基因第1外显子存在变异,包括一个c.157G>T(p.G53C)错义变异和一个c.609dup(p.A204fs)移码变异。特异性引物扩增、Sanger测序结合琼脂糖凝胶电泳证实这些为复合杂合变异。根据美国医学遗传学与基因组学学会(ACMG)的指南,c.157G>T被评为可能致病,c.609dup被评为致病。Alphafold3预测变异蛋白有显著的结构变化。

结论

该患者因TYR基因的复合杂合变异被诊断为OCA。c.609dup变异的发现丰富了OCA的突变谱,为该患者的遗传咨询和产前诊断提供了依据。

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