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印度队列中线粒体聚合酶 γ 突变的临床病理和分子谱。

Clinico-pathological and Molecular Spectrum of Mitochondrial Polymerase γ Mutations in a Cohort from India.

机构信息

Department of Neuropathology, National Institute of Mental Health and Neurosciences (NIMHANS), Bangalore, India.

Neuromuscular Laboratory, Neurobiology Research Centre (NRC), National Institute of Mental Health and Neurosciences (NIMHANS), Bangalore, India.

出版信息

J Mol Neurosci. 2021 Nov;71(11):2219-2228. doi: 10.1007/s12031-020-01765-8. Epub 2021 Jan 19.

DOI:10.1007/s12031-020-01765-8
PMID:33469851
Abstract

Polymerase γ catalytic subunit (POLG), a nuclear gene, encodes the enzyme responsible for mitochondrial DNA (mtDNA) replication. POLG mutations are a major cause of inherited mitochondrial diseases. They present with varied phenotypes, age of onset, and severity. Reports on POLG mutations from India are limited. Hence, this study aimed to describe the clinico-pathological and molecular observations of POLG mutations. A total of 446 patients with clinical diagnosis of mitochondrial disorders were sequenced for all exons and intron-exon boundaries of POLG. Of these, 19 (4.26%) patients (M:F: 10:9) had POLG mutations. The age of onset ranged from 5 to 55 years with an overlapping phenotypic spectrum. Ptosis, peripheral neuropathy, seizures, and ataxia were the common neurological features observed. The most common clinical phenotype was chronic progressive external ophthalmoplegia (CPEO) and CPEO plus (n = 14). Muscle biopsy showed characteristic features of mitochondrial myopathy in fourteen patients (14/19) and respiratory chain enzyme deficiency in eleven patients (11/19). Multiple mtDNA deletions were seen in 47.36% (9/19) patients. Eight pathogenic POLG variations including two novel variations (p.G132R and p.V1106A) were identified. The common pathogenic mutation identified was p.L304R, being present in eight patients (42.1%) predominantly in the younger age group followed by p.W748S in four patients (21%). To the best of our knowledge, this is the first extensive study from India, highlights the clinico-pathological and molecular spectrum of POLG mutations.

摘要

聚合酶 γ 催化亚基(POLG)是一种核基因,编码负责线粒体 DNA(mtDNA)复制的酶。POLG 突变是遗传性线粒体疾病的主要原因。它们表现出不同的表型、发病年龄和严重程度。来自印度的关于 POLG 突变的报告有限。因此,本研究旨在描述 POLG 突变的临床-病理和分子观察结果。对 446 名临床诊断为线粒体疾病的患者进行了所有外显子和内含子-外显子边界的 POLG 测序。其中,19 名(4.26%)患者(男:女=10:9)存在 POLG 突变。发病年龄从 5 岁到 55 岁不等,具有重叠的表型谱。眼睑下垂、周围神经病、癫痫发作和共济失调是常见的神经特征。最常见的临床表型是慢性进行性眼外肌麻痹(CPEO)和 CPEO 加(n=14)。14 名患者(14/19)的肌肉活检显示出特征性的线粒体肌病,11 名患者(11/19)的呼吸链酶缺乏。47.36%(9/19)的患者存在多个 mtDNA 缺失。鉴定出 8 种致病性 POLG 变异,包括两种新变异(p.G132R 和 p.V1106A)。常见的致病性突变是 p.L304R,在 8 名患者(42.1%)中发现,主要在年龄较小的患者中,其次是在 4 名患者(21%)中发现的 p.W748S。据我们所知,这是印度的第一项广泛研究,强调了 POLG 突变的临床-病理和分子谱。

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