DiMauro S, Bonilla E, Zeviani M, Servidei S, DeVivo D C, Schon E A
H. Houston Merritt Clinical Research Center for Muscular Dystrophy and Related Diseases, Columbia-Presbyterian Medical Center, New York, NY 10032.
J Inherit Metab Dis. 1987;10 Suppl 1:113-28. doi: 10.1007/BF01812852.
The mitochondrial myopathies or encephalomyopathies with known biochemical defects can be divided into 5 groups: (1) defects of mitochondrial transport, such as CPT deficiency or carnitine deficiencies; (2) defects of substrate utilization, such as PDHC deficiency or defects of beta-oxidation; (3) defects of the Krebs cycle, such as fumarase deficiency; (4) defects of oxidation-phosphorylation coupling, such as Luft disease, and (5) defects of the respiratory chain. These disorders are reviewed, with particular emphasis on the defects of the respiratory chain. Defects of complex I, III and IV show remarkable clinical and biochemical heterogeneity. All 3 complexes contain some subunits encoded by mtDNA and others encoded by nuclear DNA. At least some of the cytoplasmically made subunits appear to be tissue specific and may be developmentally regulated, thus explaining the genetic heterogeneity of these disorders.
已知存在生化缺陷的线粒体肌病或脑肌病可分为5组:(1)线粒体转运缺陷,如肉碱棕榈酰转移酶缺乏症或肉碱缺乏症;(2)底物利用缺陷,如丙酮酸脱氢酶复合体缺乏症或β氧化缺陷;(3)三羧酸循环缺陷,如延胡索酸酶缺乏症;(4)氧化磷酸化偶联缺陷,如 Luft 病;以及(5)呼吸链缺陷。本文对这些疾病进行了综述,特别强调了呼吸链缺陷。复合体I、III和IV的缺陷表现出显著的临床和生化异质性。所有这3个复合体都包含一些由线粒体DNA编码的亚基和其他由核DNA编码的亚基。至少一些在细胞质中合成的亚基似乎具有组织特异性,并且可能受到发育调控,从而解释了这些疾病的遗传异质性。