Mannucci P M, Lombardi R, Bader R, Horellou M H, Finazzi G, Besana C, Conard J, Samama M
Blood. 1984 Sep;64(3):614-21.
In seven patients with acquired von Willebrand's disease (AvWD) associated with lymphoproliferative disorders or benign monoclonal gammopathies, the platelet contents of von Willebrand factor antigen and ristocetin cofactor (vWF:Ag and vWF:RiCof, respectively) were normal. All the multimers of vWF:Ag could be seen in the 1.6% SDS-agarose gel electrophoresis patterns of plasma and platelet lysates. Infusion of 1-deamino-8-D-arginine vasopressin (DDAVP) augmented plasma levels of vWF:Ag and vWF:RiCof of all patients and corrected prolonged bleeding times (BT). However, compared with patients with congenital vWD type I and comparable degrees of baseline abnormalities treated in the same way, vWF:Ag and vWF:RiCof were increased less and cleared more rapidly from plasma and the BT remained normal for a shorter period of time. These studies provide evidence that these AvWD patients have qualitatively normal vWF in plasma, but at lower concentrations, that vWF in platelets is normal both qualitatively and quantitatively, and that cellular vWF can be rapidly released into plasma by DDAVP to correct the hemostatic abnormalities. However, vWF is removed rapidly from plasma, making the correction more transient than in congenital vWD type I.
在7例与淋巴增殖性疾病或良性单克隆丙种球蛋白病相关的获得性血管性血友病(AvWD)患者中,血管性血友病因子抗原(vWF:Ag)和瑞斯托霉素辅因子(分别为vWF:RiCof)的血小板含量正常。在血浆和血小板裂解物的1.6% SDS-琼脂糖凝胶电泳图谱中可看到vWF:Ag的所有多聚体。输注1-去氨基-8-D-精氨酸加压素(DDAVP)可提高所有患者血浆中vWF:Ag和vWF:RiCof的水平,并纠正延长的出血时间(BT)。然而,与以相同方式治疗的先天性I型血管性血友病患者及具有相当程度基线异常的患者相比,vWF:Ag和vWF:RiCof的升高幅度较小,从血浆中清除得更快,且BT在较短时间内保持正常。这些研究表明,这些AvWD患者血浆中的vWF质量正常,但浓度较低,血小板中的vWF在质量和数量上均正常,并且细胞内vWF可通过DDAVP迅速释放到血浆中以纠正止血异常。然而,vWF从血浆中迅速清除,使得这种纠正比先天性I型血管性血友病更短暂。