Veyradier A, Jumilly A L, Ribba A S, Obert B, Houllier A, Meyer D, Girma J P
INSERM U143, Hôpital de Bicêtre, Le Kremlin-Bicêtre, France.
Thromb Haemost. 1999 Jul;82(1):134-9.
Among the numerous variants of vWD, no patient with an abnormal vWF binding to GPIIb/IIIa has been described to date. To search for such potential variants, we developed a two-site assay for measuring the binding of purified GPIIb/IIIa to vWF in biological fluids and we used it to study a large series of plasmas from various types of von Willebrand disease (vWD) and recombinant vWF (rvWF). vWF in plasma or rvWF in culture medium was immobilized onto anti-vWF monoclonal antibodies (MoAb)-coated wells of microtiter plates. After incubation with either unlabeled GPIIb/IIIa and a 125I-anti-GPIIb/IIIa MoAb or 125I-GPIIb/IIIa, binding curves and binding isotherms were respectively established. Normal pool plasma and wild-type rvWF were used as reference samples. We tested plasmas from 85 normal subjects, 115 patients with different types of vWD (64 type 1, 2 type 3, 9 type 2A, 4 type 2M, 16 type 2B, 15 type 2N, 3 type IID and 2 acquired forms) and 50 patients with various bleeding disorders. Four mutated rvWF with 2A (Glu875Lys and Pro885Ser) or 2B (Dupl.Met540 and Val551Phe) substitutions and one rvWF mutated in the RGD domain of the C-terminal part of vWF-subunit (Asp1746Gly) were also studied. Among the various samples tested, only rvWF Asp1746Gly had no affinity for GPIIb/IIIa. In contrast, GPIIb/IIIa similarly bound to the other vWF, independently of the proteic environment, the factor VIII level, the degree of multimerization or the mutation of vWF. Our results indicate that subjects with an abnormal vWF binding to GPIIb/IIIa are probably rare and difficult to target for a specific screening.
在众多血管性血友病(vWD)变体中,迄今为止尚未有vWF与糖蛋白IIb/IIIa(GPIIb/IIIa)结合异常的患者被报道。为了寻找此类潜在变体,我们开发了一种双位点检测法,用于测定生物体液中纯化的GPIIb/IIIa与vWF的结合,并使用该方法研究了来自各种类型血管性血友病(vWD)和重组vWF(rvWF)的大量血浆样本。血浆中的vWF或培养基中的rvWF被固定在包被有抗vWF单克隆抗体(MoAb)的微孔板孔中。在用未标记的GPIIb/IIIa和125I标记的抗GPIIb/IIIa MoAb或125I标记的GPIIb/IIIa孵育后,分别建立结合曲线和结合等温线。正常混合血浆和野生型rvWF用作参考样本。我们检测了85名正常受试者、115名不同类型vWD患者(64例1型、2例3型、9例2A型、4例2M型、16例2B型、15例2N型、3例IID型和2例获得性形式)以及50名各种出血性疾病患者的血浆。还研究了四种具有2A(Glu875Lys和Pro885Ser)或2B(Dupl.Met540和Val551Phe)替代的突变rvWF以及一种在vWF亚基C末端RGD结构域发生突变(Asp1746Gly)的rvWF。在测试的各种样本中,只有rvWF Asp1746Gly对GPIIb/IIIa没有亲和力。相比之下,GPIIb/IIIa与其他vWF的结合类似,与蛋白质环境、因子VIII水平、多聚化程度或vWF的突变无关。我们的结果表明,vWF与GPIIb/IIIa结合异常的受试者可能很少见,并且难以进行针对性的特异性筛查。