Rustin Pierre, Munnich Arnold, Rötig Agnès
Unité de Recherches sur les Handicaps Génétiques de l'Enfant, INSERM U393, Hôpital Necker-Enfants Malades, 149, rue de Sèvres, Paris 75015, France.
Eur J Hum Genet. 2002 May;10(5):289-91. doi: 10.1038/sj.ejhg.5200793.
Inherited defects of the mitochondrial succinate dehydrogenase (SDH) in humans are associated with striking variable clinical presentations ranging from early-onset devastating encephalomyopathy to tumour susceptibility in adulthood, or optic atrophy in the elderly. Although different genes encoding the four subunits of the SDH have been found mutated in association with these different phenotypes, we propose that the wide clinical spectrum actually originates from the specific roles of the SDH in the respiratory chain and the mitochondria. In particular, beside its function in the Krebs cycle and the respiratory chain, the specific redox properties of the enzyme could confer to the SDH a specific function in superoxide handling.
人类线粒体琥珀酸脱氢酶(SDH)的遗传性缺陷与显著的可变临床表现相关,范围从早发性严重脑肌病到成年期肿瘤易感性,或老年人的视神经萎缩。尽管已发现编码SDH四个亚基的不同基因与这些不同表型相关联发生突变,但我们认为广泛的临床谱实际上源于SDH在呼吸链和线粒体中的特定作用。特别是,除了其在三羧酸循环和呼吸链中的功能外,该酶的特定氧化还原特性可能赋予SDH在超氧化物处理中的特定功能。