Chen C I, Federici A B, Cramer E M, Canciani M T, Harrison P, Zheng S, Massé J M, Mannucci P M, Hayward C P
Department of Pathology, McMaster University and the Hamilton Health Sciences Corporation, Ontario, Canada.
Br J Haematol. 1998 Oct;103(1):20-8. doi: 10.1046/j.1365-2141.1998.00943.x.
In normal platelet alpha-granules von Willebrand factor (VWF) is stored with multimerin and factor V in an eccentric electron-lucent zone. Because the platelet stores of VWF are deficient in 'platelet low' type 1 and type 3 von Willebrand disease (VWD), we investigated their electron-lucent zone proteins. The patients with VWD had partial to complete deficiencies of plasma and platelet VWF but normal alpha-granular multimerin and factor V, and normal alpha-granular fibrinogen, thrombospondin-1, fibronectin, osteonectin and P-selectin. In type 3 VWD platelets, alpha-granular electron-lucent zones lacking VWF-associated tubules were identified and multimerin was found in its normal alpha-granular location. These findings indicate that the formation of the electron-lucent zone and the sorting of multimerin to this region occur independent of VWE The isolated abnormalities in VWF suggests a VWF gene mutation is the cause of 'platelet low' type 1 VWD.
在正常血小板α-颗粒中,血管性血友病因子(VWF)与多聚体蛋白和因子Ⅴ一起储存在偏心的电子透亮区。由于在1型和3型血管性血友病(VWD)的“血小板低”状态下,血小板中VWF的储存量不足,我们对其电子透亮区蛋白进行了研究。VWD患者的血浆和血小板VWF部分或完全缺乏,但α-颗粒多聚体蛋白和因子Ⅴ正常,α-颗粒纤维蛋白原、血小板反应蛋白-1、纤连蛋白、骨连接蛋白和P-选择素也正常。在3型VWD血小板中,发现了缺乏VWF相关小管的α-颗粒电子透亮区,且多聚体蛋白位于其正常的α-颗粒位置。这些发现表明,电子透亮区的形成以及多聚体蛋白向该区域的分选独立于VWF发生。VWF的孤立异常表明,VWF基因突变是1型“血小板低”VWD的病因。