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通过外显子组测序鉴定的 SUCLA2 基因中的一个新的纯合突变。

A novel homozygous mutation in SUCLA2 gene identified by exome sequencing.

机构信息

Unit of Molecular Neurogenetics, Fondazione Istituto Neurologico Carlo Besta, Istituto di Ricovero e Cura a Carattere Scientifico, via Temolo 4, 20126 Milan, Italy.

出版信息

Mol Genet Metab. 2012 Nov;107(3):403-8. doi: 10.1016/j.ymgme.2012.08.020. Epub 2012 Sep 7.

Abstract

Mitochondrial disorders with multiple mitochondrial respiratory chain (MRC) enzyme deficiency and depletion of mitochondrial DNA (mtDNA) are autosomal recessive conditions due to mutations in several nuclear genes necessary for proper mtDNA maintenance. In this report, we describe two Italian siblings presenting with encephalomyopathy and mtDNA depletion in muscle. By whole exome-sequencing and prioritization of candidate genes, we identified a novel homozygous missense mutation in the SUCLA2 gene in a highly conserved aminoacid residue. Although a recurrent mutation in the SUCLA2 gene is relatively frequent in the Faroe Islands, mutations in other populations are extremely rare. In contrast with what has been reported in other patients, methyl-malonic aciduria, a biomarker for this genetic defect, was absent in our proband and very mildly elevated in her affected sister. This report demonstrates that next-generation technologies, particularly exome-sequencing, are user friendly, powerful means for the identification of disease genes in genetically and clinically heterogeneous inherited conditions, such as mitochondrial disorders.

摘要

线粒体疾病伴多种线粒体呼吸链(MRC)酶缺乏和线粒体 DNA(mtDNA)耗竭是由于几个核基因的突变导致的常染色体隐性疾病,这些核基因对于 mtDNA 的正常维持是必需的。在本报告中,我们描述了两名意大利同胞的病例,他们表现为肌肉脑肌病和 mtDNA 耗竭。通过全外显子组测序和候选基因的优先级排序,我们在一个高度保守的氨基酸残基中发现了 SUCLA2 基因的新型纯合错义突变。虽然 SUCLA2 基因中的反复突变在法罗群岛较为常见,但在其他人群中极为罕见。与其他患者的报告不同,甲基丙二酸血症是这种遗传缺陷的生物标志物,在我们的先证者中不存在,而在其受影响的姐妹中则非常轻微升高。本报告表明,下一代技术,特别是外显子组测序,是用于鉴定遗传和临床异质性遗传疾病(如线粒体疾病)中疾病基因的便捷、强大的手段。

https://cdn.ncbi.nlm.nih.gov/pmc/blobs/59f1/3490101/0ac17438338c/gr1.jpg

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