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具有明显隐性遗传的IIA型血管性血友病

Type IIA von Willebrand disease with apparent recessive inheritance.

作者信息

Asakura A, Harrison J, Gomperts E, Abildgaard C

出版信息

Blood. 1987 May;69(5):1419-20.

PMID:3105622
Abstract

Type IIA von Willebrand's Disease (vWD) is the most common type II variant, and all reported cases (56 individuals in 26 families) have had autosomal dominant inheritance. An eight-year-old female with an increased bleeding tendency since infancy was found to have laboratory values typical of type IIA vWD, but her parents and siblings were asymptomatic. With the exception of uniformly decreased levels of ristocetin cofactor in relation to von Willebrand factor antigen, the results of family studies were normal including the presence of large multimeric forms of von Willebrand factor antigen. These findings are consistent with the propositus having the homozygous state of an autosomal recessive trait. Desmopressin infusion in the propositus was followed by a significant increase of factor VIII coagulant and von Willebrand factor antigen but a limited change in ristocetin cofactor with no development of large multimers.

摘要

IIA型血管性血友病(vWD)是最常见的II型变异型,所有报道的病例(26个家族中的56人)均为常染色体显性遗传。一名自婴儿期起出血倾向增加的8岁女性,其实验室检查结果具有IIA型vWD的典型特征,但她的父母和兄弟姐妹均无症状。除了瑞斯托霉素辅因子水平相对于血管性血友病因子抗原因子一致降低外,家族研究结果均正常,包括存在大分子多聚体形式的血管性血友病因子抗原。这些发现与先证者具有常染色体隐性性状的纯合状态一致。先证者输注去氨加压素后,凝血因子VIII和血管性血友病因子抗原显著增加,但瑞斯托霉素辅因子变化有限,且未出现大分子多聚体。

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