Perutelli P, Mori P G
Laboratorio di Ematologia, Istituto G. Gaslini, Genova, Italy.
Haematologica. 1992 Sep-Oct;77(5):421-6.
Glanzmann's thrombasthenia is a rare autosomal recessive bleeding disorder characterized by a quantitative deficiency or a functional abnormality of the major platelet membrane integrin receptor: the glycoprotein (GP) IIb/IIIa complex. The GPIIb/IIIa complex functions as a platelet receptor for fibrinogen, von Willebrand factor, fibronectin and vitronectin; therefore it plays an important role in platelet adhesion and aggregation. Thrombasthenic platelets are severely deficient in GPIIb/IIIa content or function, and fail to aggregate and form the hemostatic plug at the site of vessel injury. On the other hand, heterozygous subjects (having about half the number of normal GPIIb/IIIa complexes) do not show bleeding problems. It has been demonstrated that a molecular defect affecting one of the two GP coding genes is sufficient to determine a contemporary deficit of both GPIIb and GPIIIa, and hence the thrombasthenic phenotype. Up to now, few molecular abnormalities giving rise to Glanzmann's thrombasthenia have been characterized. Large rearrangements within the GPIIb or GPIIIa coding genes appear to be unusual, whereas small modifications in the nucleotide sequence of the coding regions occur with higher frequency.
Glanzmann血小板无力症是一种罕见的常染色体隐性出血性疾病,其特征是主要血小板膜整合素受体即糖蛋白(GP)IIb/IIIa复合物存在数量缺陷或功能异常。GPIIb/IIIa复合物作为纤维蛋白原、血管性血友病因子、纤连蛋白和玻连蛋白的血小板受体发挥作用;因此它在血小板黏附和聚集中起重要作用。血小板无力症患者的血小板严重缺乏GPIIb/IIIa含量或功能,无法在血管损伤部位聚集并形成止血栓。另一方面,杂合子个体(拥有约一半数量的正常GPIIb/IIIa复合物)不会出现出血问题。已经证明,影响两个GP编码基因之一的分子缺陷足以导致GPIIb和GPIIIa同时缺乏,从而导致血小板无力症表型。到目前为止,很少有导致Glanzmann血小板无力症的分子异常被鉴定出来。GPIIb或GPIIIa编码基因内的大片段重排似乎不常见,而编码区核苷酸序列的小修饰出现频率更高。