Ruggeri Z M, Nilsson I M, Lombardi R, Holmberg L, Zimmerman T S
J Clin Invest. 1982 Nov;70(5):1124-7. doi: 10.1172/jci110700.
A variant of von Willebrand's disease has been identified in which sodium dodecyl sulfate agarose electrophoresis provides evidence that the von Willebrand factor present is structurally abnormal. Rather than the repeating triplet seen in normal subjects and in patients with the IIA and IIB variants, a repeating doublet was present in the propositus. None of the bands had the same mobility as bands in normal subjects or previously described von Willebrand's disease patients. The larger multimers of von Willebrand factor were lacking both from plasma and platelets, and did not appear in the circulation after infusion of 1-deamino-[8-D-arginine]-vasopressin. There was a marked increase in the concentration of the smallest multimer in the propositus and his phenotypically normal children, indicating that this abnormality of von Willebrand factor is inherited in an autosomal-recessive manner.
已鉴定出一种血管性血友病的变体,其中十二烷基硫酸钠琼脂糖电泳提供证据表明所存在的血管性血友病因子在结构上异常。与正常受试者以及患有IIA和IIB变体的患者中所见的重复三联体不同,先证者中存在重复双峰。没有一条带与正常受试者或先前描述的血管性血友病患者的带具有相同的迁移率。血管性血友病因子的较大多聚体在血浆和血小板中均不存在,并且在输注1-去氨基-[8-D-精氨酸]-血管加压素后未出现在循环中。先证者及其表型正常的子女中最小多聚体的浓度显著增加,表明这种血管性血友病因子的异常以常染色体隐性方式遗传。