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来自不同地理区域的两个有族裔关联的伊朗聋人家庭中,TMC1基因非编码外显子3发生颠换突变;奠基者效应的证据。

A transversion mutation in non-coding exon 3 of the TMC1 gene in two ethnically related Iranian deaf families from different geographical regions; evidence for founder effect.

作者信息

Davoudi-Dehaghani Elham, Zeinali Sirous, Mahdieh Nejat, Shirkavand Atefeh, Bagherian Hamideh, Tabatabaiefar Mohammad Amin

机构信息

Department of Molecular Medicine, Biotechnology Research Center, Pasteur Institute of Iran, Pasteur St., Tehran, Iran.

出版信息

Int J Pediatr Otorhinolaryngol. 2013 May;77(5):821-6. doi: 10.1016/j.ijporl.2013.02.021. Epub 2013 Mar 21.

Abstract

OBJECTIVES

Transmembrane channel-like 1 (TMC1) gene is a member of the transmembrane channel-like (TMC) gene family that encodes an integral membrane protein of the inner ear. It is suggested that mutation in this gene is one of the main causes of autosomal recessive non-syndromic hearing loss (ARNSHL) in different populations. The aim of this study was to determine the contribution of the TMC1 gene mutations in causing hearing loss in Iran.

METHODS

In total 54 unrelated Iranian families containing 159 affected individuals with ARNSHL detected by audiometric and otologic examinations were analyzed. Haplotype analysis of all members of 45 GJB2- & GJB6-negative families, using four microsatellite markers linked to DFNB7/11 was performed.

RESULTS

Co-segregation of hearing loss with all investigated markers for the DFNB7/11 locus was found in one family. DNA sequencing of all coding and non-coding exons and intron boundaries of the TMC1 gene identified c.-258A>C mutation in non-coding exon 3 only in individuals with hearing loss. This mutation has been previously reported in another Iranian family (G9) that share similar ethnicity. This variant was not detected in 300 ethnically matched healthy controls.

CONCLUSIONS

These results increase the probability that this nucleotide variation may be a pathogenic mutation. This study showed that the ethnicity may be more useful than geographical location to design research strategy for determining which genes should be considered when a heterogeneous disorder is under investigation.

摘要

目的

跨膜通道样蛋白1(TMC1)基因是跨膜通道样(TMC)基因家族的成员,该家族编码一种内耳的整合膜蛋白。有研究表明,该基因突变是不同人群常染色体隐性非综合征性听力损失(ARNSHL)的主要原因之一。本研究旨在确定TMC1基因突变在伊朗人群听力损失病因中的作用。

方法

对54个无血缘关系的伊朗家庭进行分析,这些家庭中有159名经听力测定和耳科检查确诊为ARNSHL的患者。使用与DFNB7/11连锁的四个微卫星标记,对45个GJB2和GJB6基因阴性家庭的所有成员进行单倍型分析。

结果

在一个家庭中发现听力损失与DFNB7/11位点的所有研究标记共分离。对TMC1基因所有编码和非编码外显子以及内含子边界进行DNA测序,仅在听力损失患者中发现非编码外显子3中的c.-258A>C突变。该突变先前在另一个具有相似种族背景的伊朗家庭(G9)中也有报道。在300名种族匹配的健康对照中未检测到该变异。

结论

这些结果增加了这种核苷酸变异可能是致病突变的可能性。本研究表明,在设计研究策略以确定在研究异质性疾病时应考虑哪些基因时,种族因素可能比地理位置更有用。

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