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线粒体遗传学与人类疾病

Mitochondrial genetics and human disease.

作者信息

Grossman L I, Shoubridge E A

机构信息

Center for Molecular Medicine and Genetics, Wayne State University School of Medicine, Detroit, Michigan 48201, USA.

出版信息

Bioessays. 1996 Dec;18(12):983-91. doi: 10.1002/bies.950181208.

Abstract

Mitochondria contain a molecular genetic system to express the 13 protein components of the electron transport system encoded in the mitochondrial genome (mtDNA). Defects in the function of this system result in some diseases, many of which are multisystem disorders, prominently involving highly aerobic, postmitotic tissues. These defects can be caused by large-scale rearrangements of mtDNA, by point mutations, or by nuclear gene mutations resulting in abnormalities in mtDNA. Although any of these mutations would be expected to produce a similar clinical phenotype by compromising oxidative phosphorylation, the surprising and puzzling result is that different clinical phenotypes are generally associated with specific mtDNA mutations. Moreover, the same mutation can produce a distinct clinical phenotype in different individuals or pedigrees. MtDNA rearrangements are also found in aged individuals, but at a subclinical level, suggesting that normal and pathological processes can differ by the effect of genetic or environmental factors on the error rate of mtDNA replication.

摘要

线粒体含有一个分子遗传系统,用于表达线粒体基因组(mtDNA)中编码的电子传递系统的13种蛋白质成分。该系统功能缺陷会导致一些疾病,其中许多是多系统疾病,主要累及高需氧的有丝分裂后组织。这些缺陷可能由mtDNA的大规模重排、点突变或导致mtDNA异常的核基因突变引起。尽管预期这些突变中的任何一种都会通过损害氧化磷酸化产生相似的临床表型,但令人惊讶和困惑的结果是,不同的临床表型通常与特定的mtDNA突变相关。此外,相同的突变在不同个体或家系中可产生不同的临床表型。在老年个体中也发现了mtDNA重排,但处于亚临床水平,这表明正常和病理过程可能因遗传或环境因素对mtDNA复制错误率的影响而有所不同。

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