Takahashi H, Nagayama R, Hattori A, Ihzumi T, Tsukada T, Shibata A
Am J Hematol. 1981;10(1):89-99. doi: 10.1002/ajh.2830100113.
Two cases of von Willebrand disease (vWD) associated with familial thrombocytopenia were reported. The proband (daughter) and her father showed thrombocytopenia with large platelets and decreased von Willebrand factor activity (VIIIR:WF). Factor VIII procoagulant activity (VIII:C) and factor VIII-related antigen (VIIIR:AG) were normal, but both patients revealed an increased ristocetin-induced platelet aggregation and a qualitative abnormality of the factor VIII protein, which was characterized by fast electrophoretic mobility of VIIIR:AG and an abnormal elution of factor VIII-related activities on Sepharose 2B. DDAVP was hemostatically effective even in this thrombocytopenic patient undergoing a dental extraction.
报告了2例与家族性血小板减少症相关的血管性血友病(vWD)病例。先证者(女儿)及其父亲表现为血小板减少伴大血小板以及血管性血友病因子活性(VIIIR:WF)降低。凝血因子VIII促凝活性(VIII:C)和凝血因子VIII相关抗原(VIIIR:AG)正常,但两名患者均显示瑞斯托霉素诱导的血小板聚集增加以及凝血因子VIII蛋白的定性异常,其特征为VIIIR:AG电泳迁移速度快以及在琼脂糖2B上凝血因子VIII相关活性洗脱异常。去氨加压素即使对这位接受拔牙手术的血小板减少症患者也有止血效果。