Ginsburg D, Konkle B A, Gill J C, Montgomery R R, Bockenstedt P L, Johnson T A, Yang A Y
Howard Hughes Medical Institute, University of Michigan Medical School, Ann Arbor 48109-0650.
Proc Natl Acad Sci U S A. 1989 May;86(10):3723-7. doi: 10.1073/pnas.86.10.3723.
von Willebrand disease (vWD), the most common inherited bleeding disorder in humans, can result from either a quantitative or a qualitative defect in the adhesive glycoprotein, von Willebrand factor (vWF). Molecular studies of vWD have been limited by the large size of the vWF gene and difficulty in obtaining the vWF mRNA from patients. By use of an adaptation of the polymerase chain reaction, vWF mRNA was amplified and sequenced from peripheral blood platelets. A silent vWF allele was identified, resulting from a cis defect in vWF mRNA transcription or processing. In two type IIA vWD patients, two different but adjacent missense mutations were identified, the locations of which may identify an important vWF functional domain. Expression in heterologous cells of recombinant vWF containing one of these latter mutations reproduced the characteristic structural abnormality seen in type IIA vWD plasma.
血管性血友病(vWD)是人类最常见的遗传性出血性疾病,它可能由黏附糖蛋白血管性血友病因子(vWF)的数量或质量缺陷引起。vWD的分子研究受到vWF基因的大尺寸以及从患者获取vWF mRNA的困难的限制。通过使用聚合酶链反应的一种改良方法,从外周血血小板中扩增并测序了vWF mRNA。鉴定出一个沉默的vWF等位基因,这是由vWF mRNA转录或加工中的顺式缺陷导致的。在两名IIA型vWD患者中,鉴定出两个不同但相邻的错义突变,其位置可能确定一个重要的vWF功能域。含有这些突变之一的重组vWF在异源细胞中的表达重现了IIA型vWD血浆中所见的特征性结构异常。