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南非混合型卟啉病家族中原卟啉原氧化酶基因缺失(537delAT)的鉴定与特征分析

Identification and characterisation of a deletion (537delAT) in the protoporphyrinogen oxidase gene in a South African variegate porphyria family.

作者信息

Corrigall A V, Hift R J, Hancock V, Meissner D, Davids L, Kirsch R E, Meissner P N

机构信息

MRC/UCT Liver Research Centre, Department of Medicine, University of Cape Town Medical School, Observatory, South Africa.

出版信息

Hum Mutat. 1998;12(6):403-7. doi: 10.1002/(SICI)1098-1004(1998)12:6<403::AID-HUMU6>3.0.CO;2-X.

Abstract

Variegate porphyria is an autosomal dominant disorder of haem metabolism resulting from a partial decrease in protoporphyrinogen oxidase activity. Variegate porphyria is highly prevalent in South Africa, the result of a founder effect now confirmed genetically as a single point mutation (R59W) which has been described in nearly all South African variegate porphyria patients studied. Only two other mutations (H20P, R168C) have been reported in South Africa. We utilised simultaneous, single-stranded conformational polymorphism and heteroduplex analysis, and direct sequencing to identify a further mutation; a 2 bp deletion in exon 6 which results in a premature stop codon 11 codons downstream from the mutation and is the first reported deletion in the protoporphyrinogen oxidase gene in a South African family. The familial segregation of this mutation strongly suggests that it is the disease causing mutation for variegate porphyria in this family. This further evidence for allelic heterogeneity limits the utility of tests for the R59W mutation in the diagnosis of variegate porphyria in South Africa.

摘要

混合型卟啉病是一种常染色体显性遗传性血红素代谢紊乱疾病,由原卟啉原氧化酶活性部分降低所致。混合型卟啉病在南非极为常见,这是一种奠基者效应的结果,现已通过基因鉴定为单一的点突变(R59W),几乎所有接受研究的南非混合型卟啉病患者都存在该突变。在南非仅报告了另外两种突变(H20P、R168C)。我们采用同步单链构象多态性和异源双链分析以及直接测序来鉴定另一种突变;外显子6中有2个碱基对缺失,这导致在突变下游11个密码子处出现一个过早的终止密码子,这是在南非一个家族中原卟啉原氧化酶基因首次报告的缺失。该突变的家族性分离强烈表明它是这个家族中导致混合型卟啉病的致病突变。等位基因异质性的这一进一步证据限制了R59W突变检测在南非混合型卟啉病诊断中的效用。

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