Fournier D J, Kabral A, Castaldi P A, Berndt M C
Department of Medicine, University of Sydney, Westmead Hospital, N.S.W., Australia.
Thromb Haemost. 1989 Nov 24;62(3):977-83.
Glanzmann's thrombasthenia is a congenital bleeding abnormality characterized by absent platelet aggregation due to the failure of fibrinogen to bind to activated thrombasthenic platelets. In the majority of cases, this defect is caused by the absence or marked reduction of a specific fibrinogen-binding aggregation receptor, the GP IIb/IIIa complex. E.T., an 18-year-old female with a life-long history of bleeding and easy bruising, had the normal clinical features of Glanzmann's thrombasthenia. Surprisingly, sodium dodecyl sulphate-polyacrylamide gel electrophoresis of her platelets showed no apparent abnormality of the GP IIb/IIIa complex. Control platelets washed in the presence of 2 mM EDTA and control and patient platelets washed in the presence of 2 mM calcium ions showed normal reactivity with anti-GP IIb, anti-GP IIIa, and anti-GP IIb/IIIa complex specific monoclonal antibodies as evaluated by flow cytometry. In contrast, patient's platelets washed in the presence of 2 mM EDTA reacted with anti-GP IIb, anti-GP IIIa, but not with the complex-specific monoclonal antibodies. The increased susceptibility of the patient's GP IIb/IIIa complex to EDTA dissociation was confirmed by crossed immunoelectrophoresis (CIE). CIE analysis further indicated that the patient's GP IIb/IIIa complex did not bind fibrinogen. The combined results suggest that this patient has Glanzmann's thrombasthenia due to an abnormal association of the GP IIb/IIIa complex which results in the failure of the complex to bind fibrinogen.
Glanzmann血小板无力症是一种先天性出血异常疾病,其特征是由于纤维蛋白原无法与活化的血小板无力症血小板结合,导致血小板聚集缺失。在大多数情况下,这种缺陷是由特定的纤维蛋白原结合聚集受体GP IIb/IIIa复合物缺失或显著减少引起的。E.T.是一名18岁女性,有终生出血和易瘀伤病史,具有Glanzmann血小板无力症的正常临床特征。令人惊讶的是,对她的血小板进行十二烷基硫酸钠-聚丙烯酰胺凝胶电泳显示,GP IIb/IIIa复合物没有明显异常。通过流式细胞术评估,在2 mM EDTA存在下洗涤的对照血小板以及在2 mM钙离子存在下洗涤的对照和患者血小板,与抗GP IIb、抗GP IIIa和抗GP IIb/IIIa复合物特异性单克隆抗体反应正常。相比之下,在2 mM EDTA存在下洗涤的患者血小板与抗GP IIb、抗GP IIIa反应,但不与复合物特异性单克隆抗体反应。交叉免疫电泳(CIE)证实了患者的GP IIb/IIIa复合物对EDTA解离的敏感性增加。CIE分析进一步表明,患者的GP IIb/IIIa复合物不结合纤维蛋白原。综合结果表明,该患者患有Glanzmann血小板无力症,原因是GP IIb/IIIa复合物异常结合,导致该复合物无法结合纤维蛋白原。