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两名患有隐性营养不良性大疱性表皮松解症的患病兄弟姐妹中VII型胶原蛋白的错义突变。

A missense mutation in type VII collagen in two affected siblings with recessive dystrophic epidermolysis bullosa.

作者信息

Christiano A M, Greenspan D S, Hoffman G G, Zhang X, Tamai Y, Lin A N, Dietz H C, Hovnanian A, Uitto J

机构信息

Department of Dermatology, Jefferson Medical College, Thomas Jefferson University, Philadelphia, Pennsylvania 19107.

出版信息

Nat Genet. 1993 May;4(1):62-6. doi: 10.1038/ng0593-62.

Abstract

Recessive dystrophic epidermolysis bullosa is a severe mutilating genodermatosis. Previous ultrastructural demonstrations of altered anchoring fibrils, and recent genetic linkage analyses have suggested that type VII collagen, the major component of anchoring fibrils, is a candidate gene. We have identified a homozygous methionine-to-lysine mutation in two affected siblings, while their unaffected mother and half-brother are heterozygous carriers. The mutation resides in a highly conserved region of the C-terminus of type VII collagen, strongly suggesting that it is the cause of the disease in this family.

摘要

隐性营养不良性大疱性表皮松解症是一种严重致残的遗传性皮肤病。以往对锚定原纤维改变的超微结构研究以及最近的基因连锁分析表明,作为锚定原纤维主要成分的VII型胶原蛋白是一个候选基因。我们在两名患病同胞中发现了一个纯合的甲硫氨酸到赖氨酸的突变,而他们未患病的母亲和同父异母的兄弟是杂合携带者。该突变位于VII型胶原蛋白C末端的一个高度保守区域,强烈提示它是这个家族中该病的病因。

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