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中国线粒体疾病的临床和遗传谱:一项多中心回顾性横断面研究。

The clinical and genetic spectrum of mitochondrial diseases in China: A multicenter retrospective cross-sectional study.

机构信息

Department of Neurology, Peking University First Hospital, Beijing, China.

Department of Neurology, Qilu Hospital of Shandong University, Jinan, China.

出版信息

Clin Genet. 2024 Dec;106(6):733-744. doi: 10.1111/cge.14605. Epub 2024 Aug 9.

Abstract

Mitochondrial diseases (MtDs) present diverse clinical phenotypes, yet large-scale studies are hindered by their rarity. This retrospective, multicenter study, conducted across five Chinese hospitals' neurology departments from 2009 to 2019, aimed to address this gap. Nationwide, 1351 patients were enrolled, with a median onset age of 14.0 (18.5) years. The predominant phenotype was mitochondrial encephalomyopathy, lactic acidosis, and stroke-like episodes (MELAS) (45.0%). Mitochondrial DNA (mtDNA) mutations were prevalent (87.4%), with m.3243A>G being the most common locus (48.7%). Meanwhile, POLG mutations in nuclear DNA (nDNA) accounted for 16.5%. Comparative analysis based on age groups (with a cut-off at 14 years) revealed the highest prevalence of MELAS, with Leigh syndrome (LS) and chronic progressive external ophthalmoplegia (CPEO) being the second most common phenotypes in junior and senior groups, respectively. Notably, the most commonly mutated nuclear genes varied across age groups. In conclusion, MELAS predominated in this Chinese MtD cohort, underscored by m.3243A>G and POLG as principal mtDNA mutations and pathogenic nuclear genes. The phenotypic and genotypic disparities observed among different age cohorts highlight the complex nature of MtDs.

摘要

线粒体疾病(MtD)呈现出多样的临床表型,但由于其罕见性,大规模的研究受到了阻碍。本项回顾性、多中心研究于 2009 年至 2019 年在五家中国医院的神经科进行,旨在填补这一空白。全国范围内共纳入 1351 例患者,中位发病年龄为 14.0(18.5)岁。主要表型为线粒体脑肌病、乳酸酸中毒和卒中样发作(MELAS)(45.0%)。线粒体 DNA(mtDNA)突变较为常见(87.4%),最常见的突变位点为 m.3243A>G(48.7%)。同时,核 DNA(nDNA)中的 POLG 突变占 16.5%。基于年龄组(以 14 岁为界)的对比分析显示,MELAS 的患病率最高,Leigh 综合征(LS)和慢性进行性眼外肌麻痹(CPEO)分别是青少年和老年组的第二和第三常见表型。值得注意的是,不同年龄组最常突变的核基因也不同。总之,MELAS 在本项中国 MtD 队列中占主导地位,其主要的 mtDNA 突变和致病性核基因分别为 m.3243A>G 和 POLG。不同年龄组之间观察到的表型和基因型差异突显了 MtD 的复杂性。

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