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复杂II型先天性代谢缺陷——罕见的人类线粒体疾病

Inborn errors of complex II--unusual human mitochondrial diseases.

作者信息

Rustin Pierre, Rötig Agnès

机构信息

Unité de Recherches sur les Handicaps Génétiques de l'Enfant (INSERM U-393), Tour Lavoisier, Hôpital Necker-Enfants Malades, 149, rue de Sèvres, F-75743 Cedex 15, Paris, France.

出版信息

Biochim Biophys Acta. 2002 Jan 17;1553(1-2):117-22. doi: 10.1016/s0005-2728(01)00228-6.

Abstract

The succinate dehydrogenase consists of only four subunits, all nuclearly encoded, and is part of both the respiratory chain and the Krebs cycle. Mutations in the four genes encoding the subunits of the mitochondrial respiratory chain succinate dehydrogenase have been recently reported in human and shown to be associated with a wide spectrum of clinical presentations. Although a comparatively rare deficiency in human, molecularly defined succinate dehydrogenase deficiency has already been found to cause encephalomyopathy in childhood, optic atrophy or tumor in adulthood. Because none of the typical housekeeping genes encoding this respiratory chain complex is known to present tissue-specific isoforms, the tissue-specific involvement represents a quite intriguing question, which is mostly addressed in this review. A differential impairment of electron flow through the respiratory chain, handling of oxygen, and/or metabolic blockade possibly associated with defects in the different subunits that can be advocated to account for tissue-specific involvement is discussed.

摘要

琥珀酸脱氢酶仅由四个亚基组成,全部由细胞核编码,并且是呼吸链和三羧酸循环的一部分。最近在人类中报道了编码线粒体呼吸链琥珀酸脱氢酶亚基的四个基因发生突变,并显示与广泛的临床表现相关。尽管在人类中相对罕见,但分子定义的琥珀酸脱氢酶缺乏症已被发现可导致儿童期脑肌病、成年期视神经萎缩或肿瘤。由于编码这种呼吸链复合物的典型管家基因均未发现存在组织特异性同工型,因此组织特异性受累是一个相当有趣的问题,本综述主要探讨了这一问题。本文讨论了通过呼吸链的电子流、氧气处理和/或代谢阻断的差异损伤,这些损伤可能与不同亚基的缺陷有关,可被认为是组织特异性受累的原因。

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