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人类线粒体疾病:答疑解惑与质疑答案

Human mitochondrial diseases: answering questions and questioning answers.

作者信息

Howell N

机构信息

Department of Radiation Oncology, University of Texas Medical Branch, Galveston 77555, USA.

出版信息

Int Rev Cytol. 1999;186:49-116. doi: 10.1016/s0074-7696(08)61051-7.

Abstract

Since the first identification in 1988 of pathogenic mitochondrial DNA (mtDNA) mutations, the mitochondrial diseases have emerged as a major clinical entity. The most striking feature of these disorders is their marked heterogeneity, which extends to their clinical, biochemical, and genetic characteristics. The major mitochondrial encephalomyopathies include MELAS (mitochondrial encephalopathy with lactic acidosis and stroke-like episodes), MERRF (myoclonic epilepsy with ragged red fibers), KSS/CPEO (Kearns-Sayre syndrome/chronic progressive external ophthalmoplegia), and NARP/MILS (neuropathy, ataxia, and retinitis pigmentosum/maternally inherited Leigh syndrome) and they typically present highly variable multisystem defects that usually involve abnormalities of skeletal muscle and/or the CNS. The primary emphasis here is to review recent investigations of these mitochondrial diseases from the standpoint of how the complexities of mitochondrial genetics and biogenesis might determine their varied features. In addition, the mitochondrial encephalomyopathies are compared and contrasted to Leber hereditary optic neuropathy, a mitochondrial disease in which the pathogenic mtDNA mutations produce a more uniform and focal neuropathology. All of these disorders involve, at some level, a mitochondrial respiratory chain dysfunction. Because mitochondrial genetics differs so strikingly from the Mendelian inheritance of chromosomes, recent research on the origin and subsequent segregation and transmission of mtDNA mutations is reviewed.

摘要

自1988年首次鉴定出致病性线粒体DNA(mtDNA)突变以来,线粒体疾病已成为一种主要的临床实体。这些疾病最显著的特征是其明显的异质性,这种异质性延伸到它们的临床、生化和遗传特征。主要的线粒体脑肌病包括MELAS(伴有乳酸性酸中毒和卒中样发作的线粒体脑病)、MERRF(伴有破碎红纤维的肌阵挛性癫痫)、KSS/CPEO(卡恩斯-塞尔综合征/慢性进行性眼外肌麻痹)和NARP/MILS(神经病、共济失调和色素性视网膜炎/母系遗传的 Leigh 综合征),它们通常表现出高度可变的多系统缺陷,通常涉及骨骼肌和/或中枢神经系统异常。这里主要强调从线粒体遗传学和生物发生的复杂性如何决定其多样特征的角度,回顾这些线粒体疾病的近期研究。此外,将线粒体脑肌病与Leber遗传性视神经病变进行比较和对比,Leber遗传性视神经病变是一种线粒体疾病,其中致病性mtDNA突变产生更一致和局限性的神经病理学改变。所有这些疾病在某种程度上都涉及线粒体呼吸链功能障碍。由于线粒体遗传学与染色体的孟德尔遗传有显著差异,因此回顾了关于mtDNA突变的起源以及随后的分离和传递的近期研究。

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