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过氧化物酶体生物发生障碍:遗传学与细胞生物学

Peroxisome biogenesis disorders: genetics and cell biology.

作者信息

Gould S J, Valle D

机构信息

Departments of Biological Chemistry and Cell Biology and Anatomy, The Johns Hopkins University School of Medicine, Baltimore, MD 21205, USA.

出版信息

Trends Genet. 2000 Aug;16(8):340-5. doi: 10.1016/s0168-9525(00)02056-4.

Abstract

Zellweger syndrome, neonatal adrenoleukodystrophy, infantile Refsum disease and rhizomelic chondrodysplasia punctata are progressive disorders characterized by loss of multiple peroxisomal metabolic functions. These diseases are inherited in an autosomal recessive manner, are caused by defects in the import of peroxisomal matrix proteins and are referred to as the peroxisome biogenesis disorders (PBDs). Recent studies have identified the PEX genes that are mutated in 11 of the 12 known complementation groups of PBD patients. This article reviews these advances in PBD genetics and discusses how studies of human PEX genes, their protein products and PBD cell lines are shaping current models of peroxisome biogenesis.

摘要

泽韦格综合征、新生儿肾上腺脑白质营养不良、婴儿型雷夫叙姆病和点状软骨发育不良是进行性疾病,其特征为多种过氧化物酶体代谢功能丧失。这些疾病以常染色体隐性方式遗传,由过氧化物酶体基质蛋白输入缺陷引起,被称为过氧化物酶体生物发生障碍(PBDs)。最近的研究已鉴定出在12个已知的PBD患者互补组中的11个组中发生突变的PEX基因。本文综述了PBD遗传学的这些进展,并讨论了对人类PEX基因、其蛋白质产物和PBD细胞系的研究如何塑造当前的过氧化物酶体生物发生模型。

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