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线粒体疾病

Mitochondrial diseases.

作者信息

Nonaka I

机构信息

Division of Ultrastructural Research, National Center of Neurology and Psychiatry (NCNP), Toyko, Japan.

出版信息

Curr Opin Neurol Neurosurg. 1992 Oct;5(5):622-32.

PMID:1392136
Abstract

With the discovery of mitochondrial DNA (mtDNA) mutations in different neuromuscular disorders, investigations now seek to clarify how the mutant mtDNA induces biochemical and morphologic defects. In one of the most important approaches human mutant mtDNA is transferred into cells that lack mtDNA to examine the relationship between the amount of mutant mtDNA and defects in cell growth, respiration and enzyme activities. The resulting cells are 'cybrids'; these clonal cells contain the heteroplasmic mutant and normal mtDNA from patients with mitochondrial diseases. The mitochondria become functionally defective when the amount of mutant mtDNA exceeds a certain threshold, which differs from mutation to mutation: 60 to 70% in chronic progressive external ophthalmoplegia (CPEO) and probably 95% in the syndromes of mitochondrial encephalopathy, myopathy, lactic acidosis, and stroke-like episodes (MELAS), and myoclonic epilepsy with ragged red fibers (MERRF). This threshold effect may explain the tissue-specific patterns of clinical expression.

摘要

随着在不同神经肌肉疾病中线粒体DNA(mtDNA)突变的发现,目前的研究旨在阐明突变的mtDNA如何引发生化和形态学缺陷。在最重要的方法之一中,人类突变mtDNA被转入缺乏mtDNA的细胞中,以研究突变mtDNA的数量与细胞生长、呼吸和酶活性缺陷之间的关系。由此产生的细胞是“胞质杂种”;这些克隆细胞含有来自线粒体疾病患者的异质性突变和正常mtDNA。当突变mtDNA的数量超过一定阈值时,线粒体就会出现功能缺陷,这个阈值因突变而异:慢性进行性眼外肌麻痹(CPEO)中为60%至70%,线粒体脑肌病、乳酸酸中毒和卒中样发作综合征(MELAS)以及肌阵挛性癫痫伴破碎红纤维(MERRF)中可能为95%。这种阈值效应可能解释了临床表达的组织特异性模式。

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