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与线粒体脑肌病相关的ATP6基因错义突变m.8914C>T的生化特征

The biochemical characterization of a missense mutation m.8914C>T in ATP6 gene associated with mitochondrial encephalomyopathy.

作者信息

Guo Ya, Zhang Yu, Li Fei, Liu Peipei, Liu Yedan, Yang Chengqing, Song Jie, Zhang Na, Chen Zongbo

机构信息

Pediatric Department of the Affiliated Hospital of Qingdao University, No. 16 Jiangsu Road, Shandong, 266000, PR China.

Ophthalmology Department of Qingdao Municipal Hospital, No. 1 Jiaozhou Road, Shandong, 266000, PR China.

出版信息

Int J Dev Neurosci. 2018 Dec;71:172-174. doi: 10.1016/j.ijdevneu.2018.09.007. Epub 2018 Sep 28.

Abstract

Mutations in ATP6 gene are frequent causes of mitochondrial encephalomyopathies. ATP6 gene encodes one subunit of complexⅤ. The present study described a missense mutation in ATP6 gene in a 8-year-old Chinese boy with mitochondrial encephalomyopathy. We identified one missense mutation in ATP6 gene (m.8914C>T) by mitochondrial DNA sequencing. This mutation altered the amino acid proline in serine, and alterative protein is predicted to be harmful. The mutation load in blood sample of patient is 59.49%. Activity of all mitochondrial complexes in blood are normal, however, the total function of mitochondrial oxidative phosphorylation were declined (including pathwayⅠ, pathwayⅡ and pathwayⅣ). The missense mutation (m.8914C>T) in ATP6 gene could result in abnormal function of complexV and is related with mitochondrial encephalomyopathy.

摘要

ATP6基因突变是线粒体脑肌病的常见病因。ATP6基因编码复合物Ⅴ的一个亚基。本研究描述了一名8岁中国线粒体脑肌病男孩中ATP6基因的一个错义突变。我们通过线粒体DNA测序在ATP6基因中鉴定出一个错义突变(m.8914C>T)。该突变将氨基酸脯氨酸改变为丝氨酸,预测替代蛋白具有有害性。患者血液样本中的突变负荷为59.49%。血液中所有线粒体复合物的活性均正常,然而,线粒体氧化磷酸化的总功能下降(包括途径Ⅰ、途径Ⅱ和途径Ⅳ)。ATP6基因中的错义突变(m.8914C>T)可导致复合物Ⅴ功能异常,并与线粒体脑肌病相关。

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