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在中国共济失调毛细血管扩张症患者中鉴定出 12 种新型 Atm 突变。

Twelve novel Atm mutations identified in Chinese ataxia telangiectasia patients.

机构信息

Department of Medical Genetics, Peking University Health Science Center, Beijing, China.

出版信息

Neuromolecular Med. 2013 Sep;15(3):536-40. doi: 10.1007/s12017-013-8240-3. Epub 2013 Jun 27.

DOI:10.1007/s12017-013-8240-3
PMID:23807571
原文链接:https://pmc.ncbi.nlm.nih.gov/articles/PMC3732755/
Abstract

Ataxia telangiectasia (A-T) is an autosomal recessive disease characterized mainly by progressive cerebellar ataxia, oculocutaneous telangiectasia, and immunodeficiency. This disease is caused by mutations of the ataxia telangiectasia mutated (Atm) gene. More than 500 Atm mutations that are responsible for A-T have been identified so far. However, there have been very few A-T cases reported in China, and only two Chinese A-T patients have undergone Atm gene analysis. In order to systemically investigate A-T in China and map their Atm mutation spectrum, we recruited eight Chinese A-T patients from six unrelated families nationwide. Using direct sequencing of genomic DNA and the multiplex ligation-dependent probe amplification, we identified twelve pathogenic Atm mutations, including one missense, four nonsense, five frameshift, one splicing, and one large genomic deletion. All the Atm mutations we identified were novel, and no homozygous mutation and founder-effect mutation were found. These results suggest that Atm mutations in Chinese populations are diverse and distinct largely from those in other ethnic areas.

摘要

共济失调毛细血管扩张症(A-T)是一种常染色体隐性遗传病,主要表现为进行性小脑共济失调、眼-皮肤毛细血管扩张和免疫缺陷。该病由共济失调毛细血管扩张症突变基因(Atm)突变引起。迄今为止,已发现超过 500 种导致 A-T 的 Atm 突变。然而,中国报道的 A-T 病例非常少,仅有两名中国 A-T 患者进行过 Atm 基因分析。为了系统地研究中国的 A-T 并绘制其 Atm 突变谱,我们从全国六个无关家庭招募了 8 名 A-T 患者。我们通过对基因组 DNA 的直接测序和多重连接依赖性探针扩增,鉴定了 12 种致病性 Atm 突变,包括 1 种错义突变、4 种无义突变、5 种移码突变、1 种剪接突变和 1 种大片段缺失。我们鉴定的所有 Atm 突变均为新突变,未发现纯合突变和热点突变。这些结果表明,中国人群的 Atm 突变与其他种族地区的突变存在显著差异。

https://cdn.ncbi.nlm.nih.gov/pmc/blobs/ed86/3732755/2f92a273939b/12017_2013_8240_Fig1_HTML.jpg
https://cdn.ncbi.nlm.nih.gov/pmc/blobs/ed86/3732755/2f92a273939b/12017_2013_8240_Fig1_HTML.jpg
https://cdn.ncbi.nlm.nih.gov/pmc/blobs/ed86/3732755/2f92a273939b/12017_2013_8240_Fig1_HTML.jpg

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本文引用的文献

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Eur J Hum Genet. 2012 Mar;20(3):305-12. doi: 10.1038/ejhg.2011.196. Epub 2011 Nov 9.
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Functional characterization and targeted correction of ATM mutations identified in Japanese patients with ataxia-telangiectasia.鉴定日本共济失调毛细血管扩张症患者中 ATM 突变的功能特征和靶向校正。
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中国的共济失调毛细血管扩张症:一例新型变异病例报告及文献综述
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