Peretz H, Rosenberg N, Usher S, Graff E, Newman P J, Coller B S, Seligsohn U
Department of Hematology, Chaim Sheba Medical Center, Tel-Hashomer, Israel.
Blood. 1995 Jan 15;85(2):414-20.
Glanzmann's thrombasthenia is a bleeding disorder characterized by a decrease or absence of the functional platelet membrane glycoprotein (GP) complex, GPIIb/IIIa (alpha IIb beta 3). We describe a new deletion-insertion mutation in the GPIIb gene causing type I Glanzmann's thrombasthenia in two siblings of a consanguineous Iranian-Jewish family. The proband's platelets bound more antibodies against the vitronectin receptor-alpha V beta 3 than normal platelets, suggesting a normal GPIIIa (beta 3) gene and a defect in the GPIIb gene. Sequencing of amplified cDNA and genomic DNA fragments showed a 6-bp deletion and 31-bp insertion in exon 25 of the GPIIb gene. The predominant platelet GPIIb mRNA of the proband was a product of the splicing of exon 24 to a cryptic AG acceptor site in the insertion and encoded for deletion of amino acids Leu817-Asn826 and insertion of eight different amino acids. Cotransfection of COS-7 cells with expression vectors containing wild-type GPIIIa cDNA and the mutated GPIIb cDNA failed to produce detectable amounts of GPIIb/IIIa on the surface of the cells. Allele-specific restriction analysis of genomic DNA of family members showed homozygosity for the mutation in the affected siblings, heterozygosity in the parents, and homozygosity for the normal allele in an unaffected sibling. The observed mutation is in a region that is conserved from rodents to humans and has been suggested to be involved in the interaction between GPIIb and GPIIIa when these GPs are complexed in solution.
Glanzmann血小板无力症是一种出血性疾病,其特征是功能性血小板膜糖蛋白(GP)复合物GPIIb/IIIa(αIIbβ3)减少或缺失。我们描述了GPIIb基因中的一种新的缺失-插入突变,该突变在一个伊朗犹太近亲家庭的两个兄弟姐妹中导致I型Glanzmann血小板无力症。先证者的血小板比正常血小板结合更多抗玻连蛋白受体-αVβ3的抗体,提示GPIIIa(β3)基因正常而GPIIb基因存在缺陷。对扩增的cDNA和基因组DNA片段进行测序显示,GPIIb基因第25外显子有一个6bp的缺失和31bp的插入。先证者主要的血小板GPIIb mRNA是外显子24剪接到插入片段中一个隐蔽的AG受体位点的产物,编码缺失氨基酸Leu817-Asn826并插入8种不同的氨基酸。将含有野生型GPIIIa cDNA和突变型GPIIb cDNA的表达载体共转染COS-7细胞,未能在细胞表面产生可检测到的GPIIb/IIIa量。对家庭成员的基因组DNA进行等位基因特异性限制性分析显示,患病兄弟姐妹的突变纯合,父母为杂合,未患病的兄弟姐妹为正常等位基因纯合。观察到的突变位于从啮齿动物到人类都保守的区域,有人提出当这些糖蛋白在溶液中形成复合物时,该区域参与GPIIb和GPIIIa之间的相互作用。