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线粒体DNA中的8344突变:突变DNA比例与临床病理结果之间的比较。

The 8,344 mutation in mitochondrial DNA: a comparison between the proportion of mutant DNA and clinico-pathologic findings.

作者信息

Ozawa M, Goto Y, Sakuta R, Tanno Y, Tsuji S, Nonaka I

机构信息

Department of Laboratory Medicine, National Center of Neurology and Psychiatry (NCNP), Tokyo, Japan.

出版信息

Neuromuscul Disord. 1995 Nov;5(6):483-8. doi: 10.1016/0960-8966(95)00009-c.

Abstract

Ten patients, two men and eight women with mitochondrial encephalomyopathy, had an A-G mutation at nucleotide pair 8,344 in the mitochondrial DNA, the most common genetic defect in myoclonus epilepsy with ragged-red fibers (MERRF). Eight patients had the clinical and pathologic characteristics of MERRF including myoclonus, seizures, cerebellar ataxia and myopathy with ragged-red fibers. Two patients had atypical symptoms such as early onset of fatal cardiac failure and late onset of rapid mental deterioration, respectively. The striking feature in our patients with the 8,344 mutation cardiac involvement and two developed progressive heart failure. In the typical MERRF patients, the proportion of mutant mitochondrial DNA in their skeletal muscles, quantified by a single strand conformation polymorphism analysis, was above 85%. However, there was no significant correlation between clinical severity, histopathological findings and the proportion of mutant mtDNA in muscle biopsy samples, suggesting that non-ragged-red fibers play an important role in the phenotype expression of the mutants.

摘要

10名线粒体脑肌病患者(2名男性,8名女性)的线粒体DNA第8344位核苷酸对发生了A - G突变,这是肌阵挛性癫痫伴破碎红纤维综合征(MERRF)最常见的基因缺陷。8名患者具有MERRF的临床和病理特征,包括肌阵挛、癫痫发作、小脑共济失调以及伴有破碎红纤维的肌病。2名患者分别有非典型症状,如早期出现致命性心力衰竭和晚期出现快速精神衰退。我们的患者中8344突变的显著特征是心脏受累,有2例发展为进行性心力衰竭。在典型的MERRF患者中,通过单链构象多态性分析定量,其骨骼肌中突变线粒体DNA的比例高于85%。然而,临床严重程度、组织病理学发现与肌肉活检样本中突变mtDNA比例之间无显著相关性,这表明非破碎红纤维在突变体的表型表达中起重要作用。

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