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一名患有严重迟发性抑郁症和家族性进行性外眼肌麻痹患者的多个组织中线粒体DNA的缺失

Multiple deletions of mitochondrial DNA in several tissues of a patient with severe retarded depression and familial progressive external ophthalmoplegia.

作者信息

Suomalainen A, Majander A, Haltia M, Somer H, Lönnqvist J, Savontaus M L, Peltonen L

机构信息

Department of Human Molecular Genetics, National Public Health Institute, Helsinki, Finland.

出版信息

J Clin Invest. 1992 Jul;90(1):61-6. doi: 10.1172/JCI115856.

Abstract

Multiple deletions of mitochondrial DNA (mtDNA) have recently been reported in familial progressive external ophthalmoplegia (PEO), in a case of progressive encephalomyopathy, and in inherited recurrent myoglobinuria. The inheritance of familial PEO has been autosomal dominant, which indicates that a mutation in an unknown nuclear gene results in several mtDNA deletions of different sizes in these patients. We report a patient with autosomal dominant PEO, whose major clinical symptom, however, was severe retarded depression. The morphological analyses of the tissue samples derived from autopsy showed various abnormalities in the mitochondria in all the tissues studied. The activities of the respiratory chain enzymes encoded by mtDNA were remarkably reduced in the skeletal muscle. The mtDNA analyses confirmed that besides myopathy, this patient had a multisystem disorder with widespread distribution of multiple deletions of mtDNA. The highest percentage of mutated mtDNA was found in the brain, skeletal muscle and the heart, the relative quantity of mutated mtDNA correlating to the severity of the clinical symptoms.

摘要

最近有报道称,在家族性进行性眼外肌麻痹(PEO)、一例进行性脑病以及遗传性复发性肌红蛋白尿中发现了线粒体DNA(mtDNA)的多处缺失。家族性PEO的遗传方式为常染色体显性遗传,这表明在这些患者中,一个未知核基因的突变导致了不同大小的多个mtDNA缺失。我们报告了一名患有常染色体显性PEO的患者,然而其主要临床症状是严重的迟发性抑郁。对尸检获得的组织样本进行的形态学分析显示,在所研究的所有组织中,线粒体均存在各种异常。mtDNA编码的呼吸链酶在骨骼肌中的活性显著降低。mtDNA分析证实,除了肌病外,该患者还患有多系统疾病,mtDNA多处缺失广泛分布。在大脑、骨骼肌和心脏中发现的突变mtDNA比例最高,突变mtDNA的相对数量与临床症状的严重程度相关。

https://cdn.ncbi.nlm.nih.gov/pmc/blobs/c9ca/443063/b94b205d8b10/jcinvest00050-0068-a.jpg

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