Ruggeri Z M, Mannucci P M, Lombardi R, Federici A B, Zimmerman T S
Blood. 1982 Jun;59(6):1272-8.
We have studied the modifications in the multimeric composition of plasma factor VIII/von Willebrand factor and the bleeding time response following administration of 1-Deamino-[8-D-arginine]-Vasopressin (DDAVP) to patients with different subtypes of von Willebrand's disease. In type I, all multimers were present in plasma in the resting state, though they were decreased in concentration. Administration of DDAVP resulted in an increased concentration of these forms as well as the appearance of larger forms than were previously present. There was concomitant correction of the bleeding time. In type IIA, large multimers were absent in the resting state, and although DDAVP induced an average threefold increase in the plasma concentration of factor VIII/von Willebrand factor, the larger multimers did not appear and the bleeding time, although shortened, was not corrected. In contrast, the larger multimers that were also absent from type IIB plasma in the resting state rapidly appeared following DDAVP administration. However, their appearance was transitory and the bleeding time, as in IIA patients, was shortened but not corrected. The characteristic multimeric composition of platelet factor VIII/von Willebrand factor in given subtypes predicted the alteration in plasma factor VIII/von Willebrand factor induced by DDAVP. These studies provide evidence that the different subtypes of von Willebrand's disease represent distinct abnormalities of factor VIII/von Willebrand factor. They also suggest that complete hemostatic correction following DDAVP can be routinely expected only in type I von Willebrand's disease, and only if factor VIII/von Willebrand factor can be raised to normal levels.
我们研究了血管性血友病不同亚型患者在给予1-去氨基-[8-D-精氨酸]-血管加压素(DDAVP)后血浆因子VIII/血管性血友病因子多聚体组成的变化以及出血时间的反应。在I型中,所有多聚体在静息状态下都存在于血浆中,尽管其浓度降低。给予DDAVP导致这些形式的浓度增加,并且出现了比以前更大的形式。同时出血时间得到纠正。在IIA型中,静息状态下不存在大的多聚体,尽管DDAVP使因子VIII/血管性血友病因子的血浆浓度平均增加了三倍,但更大的多聚体并未出现,出血时间虽然缩短,但未得到纠正。相比之下,静息状态下IIB型血浆中也不存在的更大的多聚体在给予DDAVP后迅速出现。然而,它们的出现是短暂的,并且与IIA型患者一样,出血时间缩短但未得到纠正。特定亚型中血小板因子VIII/血管性血友病因子的特征性多聚体组成预测了DDAVP诱导的血浆因子VIII/血管性血友病因子的变化。这些研究提供了证据,表明血管性血友病的不同亚型代表了因子VIII/血管性血友病因子的不同异常。它们还表明,只有在I型血管性血友病中,并且只有当因子VIII/血管性血友病因子能够升高到正常水平时,才可以常规预期DDAVP后完全止血纠正。