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[携带m.14484T>C突变的中国家系中Leber遗传性视神经病变的线粒体DNA单倍群及变异分析]

[The analysis of mitochondrial DNA haplogroups and variants for Leber's hereditary optic neuropathy in Chinese families carrying the m.14484T >C mutation].

作者信息

Meng Xiangjuan, Zhu Jinping, Gao Min, Zhang Sai, Zhao Fuxin, Zhang Juanjuan, Liu Xiaoling, Wei Qiping, Tong Yi, Zhang Minglian, Qu Jia, Guan Minxin

机构信息

Zhejiang Provincial Key Laboratory of Medical Genetics, Attardi Institute of Mitochondrial Biomedicine, Wenzhou Medical University, Wenzhou 325035, China; School of Ophthalmology and Optometry, Wenzhou Medical University, Wenzhou 325027, China.

School of Ophthalmology and Optometry, Wenzhou Medical University, Wenzhou 325027, China.

出版信息

Yi Chuan. 2014 Apr;36(4):336-45.

Abstract

The m.14484T>C mutation in mitochondrial ND6 gene (MT-ND6) is a primary mutation underlying the development of Leber's hereditary optic neuropathy (LHON) , but by itself not enough to cause visual loss. To explore the role of mitochondrial haplogroups on the expression of LHON for the people carrying the m.14484T>C mutation, we performed systematic and extended mutational screening of MT-ND6 gene in a cohort of 1177 Han Chinese patients with LHON. A total of 67 affected subjects carried the homoplasmic m.14484T>C mutation, accounting for 5.7% of this LHON population. The penetrances of optic neuropathy among 51 pedigrees carrying the m.14484T>C mutation ranged from 5.6% to 100.0%, with the average of 21.5%. The sequence analysis of entire mitochondrial genomes of 51 probands exhibited distinct sets of polymorphisms belonging to 18 Eastern Asian haplogroups. The frequencies of haplogroup A and haplogroup F were sig-nificantly less in the LHON mtDNA samples than those in 106 Chinese controls. On the other hand, the haplogroup M10a accounted for 9.8% of the patient's mtDNA samples but was absent in 106 Chinese controls. Strikingly, the average pene-trance (46.13%) of optic neuropathy for the pedigrees carrying mitochondrial haplogroup M10a was higher than those car-rying other mtDNA haplogroups. These observations indicated that mitochondrial haplogroup M10a may increase the risk of visual loss.

摘要

线粒体ND6基因(MT-ND6)中的m.14484T>C突变是Leber遗传性视神经病变(LHON)发生的主要突变,但仅靠其自身不足以导致视力丧失。为了探究线粒体单倍群对携带m.14484T>C突变人群中LHON表达的作用,我们对1177例汉族LHON患者进行了MT-ND6基因的系统和扩展突变筛查。共有67例受影响的受试者携带纯合的m.14484T>C突变,占该LHON人群的5.7%。在携带m.14484T>C突变的51个家系中,视神经病变的外显率范围为5.6%至100.0%,平均为21.5%。对51例先证者的整个线粒体基因组进行序列分析,发现了属于18个东亚单倍群的不同多态性集合。LHON线粒体DNA样本中单倍群A和单倍群F的频率显著低于106例中国对照。另一方面,单倍群M10a在患者的线粒体DNA样本中占9.8%,但在106例中国对照中不存在。令人惊讶的是,携带线粒体单倍群M10a的家系中视神经病变的平均外显率(46.13%)高于携带其他线粒体DNA单倍群的家系。这些观察结果表明,线粒体单倍群M10a可能会增加视力丧失的风险。

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