Suppr超能文献

m.3460G>A 与 Leber 遗传性视神经病变六家中国家系的分子特征

Molecular characterization of six Chinese families with m.3460G>A and Leber hereditary optic neuropathy.

机构信息

Key Laboratory of Animal Models and Human Disease Mechanisms of Chinese Academy of Sciences & Yunnan Province, Kunming Institute of Zoology, Kunming, Yunnan, China.

出版信息

Neurogenetics. 2010 Jul;11(3):349-56. doi: 10.1007/s10048-010-0236-7. Epub 2010 Mar 16.

Abstract

The primary mutation m.3460G>A occurs with a very low frequency (approximately 1%) in Chinese patients with Leber hereditary optic neuropathy (LHON). Up to now, there is no comprehensive study of Chinese patients harboring this mutation. We characterized six unrelated probands with m.3460G>A in this study, which were identified from 1,626 patients with LHON or suspected with LHON. The overall penetrance of LHON (25.6% [10/39]) in four pedigrees with m.3460G>A was substantially lower than those families with m.11778G>A (33.3% [619/1859]) as reported in our previous study. Intriguingly, family Le688 with a heteroplasmic m.3460G>A presented a lower penetrance (12.5%) than the other three families with a homoplasmic mutation. There is an elevated gender bias (affected male to affected female = 4:1) in the four families with m.3460G>A compared to those LHON families with m.11778G>A (2.4:1). Complete mtDNA sequencing indicated that the six matrilines belonged to haplogroups B4d1, F2, A5b, M12a, D4b2b, and D4b2, respectively. We did not identify any potential secondary mutation(s) that will affect or be associated with the penetrance of LHON in the six probands by using an evolutionary analysis and protein secondary-structure prediction. Taken together, our results suggested that the m.3460G>A mutation occurred multiple times in Chinese LHON patients. The heteroplasmic status of mutation m.3460G>A might influence the penetrance of LHON in family Le688.

摘要

主要突变 m.3460G>A 在患有莱伯遗传性视神经病变(LHON)的中国患者中的出现频率非常低(约 1%)。到目前为止,还没有针对携带这种突变的中国患者的综合研究。我们在这项研究中对 6 个不相关的 m.3460G>A 先证者进行了特征描述,这些先证者是从 1626 名 LHON 患者或疑似 LHON 的患者中发现的。在我们之前的研究中报道,m.11778G>A(33.3% [619/1859])的四个家系中,m.3460G>A 的 LHON 总体外显率(25.6% [10/39])明显低于 m.11778G>A 的家系。有趣的是,具有异质性 m.3460G>A 的家族 Le688 的外显率(12.5%)低于其他三个具有同质性突变的家族。与 m.11778G>A 的 LHON 家系相比(2.4:1),m.3460G>A 的四个家系中存在较高的性别偏倚(受累男性与受累女性=4:1)。通过进化分析和蛋白质二级结构预测,我们没有发现任何潜在的次要突变(s)会影响或与六个先证者的 LHON 外显率相关。综上所述,我们的结果表明,m.3460G>A 突变在中国 LHON 患者中多次发生。突变 m.3460G>A 的异质性状态可能会影响家族 Le688 中 LHON 的外显率。

文献AI研究员

20分钟写一篇综述,助力文献阅读效率提升50倍。

立即体验

用中文搜PubMed

大模型驱动的PubMed中文搜索引擎

马上搜索

文档翻译

学术文献翻译模型,支持多种主流文档格式。

立即体验