Mannucci P M, Lombardi R, Federici A B, Dent J A, Zimmerman T S, Ruggeri Z M
Blood. 1986 Jul;68(1):269-74.
A patient with a lifelong bleeding disorder was diagnosed as having Type II von Willebrand disease. The larger multimers of von Willebrand factor were absent from her plasma but present in platelets. A high-resolution electrophoretic technique was used to study the complex structure of individual von Willebrand factor multimers. In normal plasma, each multimer could be resolved into five bands: a more intense central one and four less intense, two moving faster and two slower than the central band. In normal platelets, each multimer could also be resolved into five bands. The central one had a mobility similar to that in plasma, whereas the four satellite bands had a mobility that differed from that of the corresponding plasma bands. In the patient, platelet von Willebrand factor antigen content and ristocetin cofactor activity were normal, and von Willebrand factor showed the same structure of individual multimers as seen in normal platelets. On the other hand, plasma von Willebrand factor antigen and ristocetin cofactor activity were decreased, and the structure of individual von Willebrand factor multimers was different from that of normal plasma and similar to that seen in normal and patient's platelets. After infusion of 1-deamino-8-D-arginine vasopressin, the largest von Willebrand factor multimers, as well as new satellite bands with a mobility similar to those in normal plasma, appeared in the patient plasma, and the levels of von Willebrand factor antigen and ristocetin cofactor activity became normal. Yet no relevant change in the prolonged bleeding time was observed. This new variant of von Willebrand disease, therefore, is characterized by the presence of a dysfunctional von Willebrand factor molecule that exhibits unique structural abnormalities in plasma but appears to be normal in platelets. The designation of Type IIF is proposed for this type of von Willebrand disease in accordance with the terminology that has been previously used.
一名患有终身出血性疾病的患者被诊断为II型血管性血友病。其血浆中缺乏较大的血管性血友病因子多聚体,但血小板中存在。采用高分辨率电泳技术研究单个血管性血友病因子多聚体的复杂结构。在正常血浆中,每个多聚体可分解为五条带:一条较宽的中央带和四条较窄的带,其中两条移动速度比中央带快,两条比中央带慢。在正常血小板中,每个多聚体也可分解为五条带。中央带的迁移率与血浆中的相似,而四条卫星带的迁移率与相应血浆带的不同。该患者血小板血管性血友病因子抗原含量和瑞斯托霉素辅因子活性正常,血管性血友病因子单个多聚体的结构与正常血小板中的相同。另一方面,血浆血管性血友病因子抗原和瑞斯托霉素辅因子活性降低,单个血管性血友病因子多聚体的结构与正常血浆不同,与正常及患者血小板中的相似。输注1-去氨基-8-D-精氨酸加压素后,患者血浆中出现了最大的血管性血友病因子多聚体以及迁移率与正常血浆中相似的新卫星带,血管性血友病因子抗原和瑞斯托霉素辅因子活性水平恢复正常。然而,未观察到延长的出血时间有相关变化。因此,这种新的血管性血友病变体的特征是存在功能失调的血管性血友病因子分子,该分子在血浆中表现出独特的结构异常,但在血小板中似乎正常。根据先前使用的术语,建议将这种类型的血管性血友病命名为IIF型。