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一个中国听力损失家系中LARS2基因的新型复合杂合突变

Novel compound heterozygous mutations in the LARS2 gene in a Chinese family with hearing loss.

作者信息

Lu Mengyi, Zhou Kai, Yang Xiuyun, Lin Lin, Lu Lixiang, Qin Yujie, Zhou Ni, Li Lingbo

机构信息

Department of Otolaryngology & Head and Neck, Liuzhou Worker's Hospital of Guangxi Zhuang Autonomous Region, 156 Heping Road, Liuzhou, 545007, China.

出版信息

Neurogenetics. 2025 Jan 7;26(1):17. doi: 10.1007/s10048-024-00797-1.

Abstract

BACKGROUND

Mutations in the LARS2 gene are correlated with Perrault syndrome, a rare autosomal recessive genetic disorder, that is typically characterized by sensorineural hearing loss and ovarian insufficiency.

METHODS

Whole-exome sequencing and mutational analysis were employed to identify hearing loss-causing genes in a Chinese family from the Guangxi Zhuang Autonomous Region. Clinical phenotypes, audiological data, and color Doppler ultrasound of the family were collected, and a series of computer software were used to analyze the impact of genetic variations on protein structure and function.

RESULTS

Novel compound heterozygous LARS2 variants, c.604G > A and c.703C > T, were linked to hearing loss in the family, the latter of which has not been reported in any public database. The proband and her brother in this family presented with hearing loss, while the parents had normal hearing. Additionally, the c.703C > T mutation is a nonsense mutation, leading to a significant loss of amino acids, while the c.604G > A mutation affects the secondary structure and side-chain structure of the protein.

CONCLUSION

These mutations expand the LARS2 mutation spectrum and provide a basis for the genetic diagnosis of Perrault syndrome and related hearing loss.

摘要

背景

LARS2基因的突变与佩罗特综合征相关,佩罗特综合征是一种罕见的常染色体隐性遗传病,其典型特征是感音神经性听力损失和卵巢功能不全。

方法

采用全外显子组测序和突变分析来鉴定来自广西壮族自治区的一个中国家庭中导致听力损失的基因。收集该家庭的临床表型、听力学数据和彩色多普勒超声检查结果,并使用一系列计算机软件分析基因变异对蛋白质结构和功能的影响。

结果

新发现的复合杂合LARS2变异体c.604G>A和c.703C>T与该家庭的听力损失有关,其中后者在任何公共数据库中均未被报道。该家庭中的先证者及其兄弟出现听力损失,而其父母听力正常。此外,c.703C>T突变是一种无义突变,导致氨基酸大量缺失,而c.604G>A突变影响蛋白质的二级结构和侧链结构。

结论

这些突变扩展了LARS2突变谱,为佩罗特综合征及相关听力损失的基因诊断提供了依据。

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