Gitschier J, Wood W I, Tuddenham E G, Shuman M A, Goralka T M, Chen E Y, Lawn R M
Nature. 1985;315(6018):427-30. doi: 10.1038/315427a0.
The most common inherited bleeding disorder in man, haemophilia A, is caused by defect in factor VIII, a component in the blood coagulation pathway. The X-chromosome-linked disease almost certainly stems from a heterogeneous collection of genetic lesions. Because, without proper treatment, haemophilia can be a fatal disease, new mutations are necessary to account for its constant frequency in the population. In addition, haemophilia A displays a wide range of severity, and some 15% of haemophiliacs generate high levels of antibodies against factor VIII ('inhibitor patients'). The present work elucidates the molecular genetic basis of haemophilia in some individuals. Using the recently cloned factor VIII gene as a probe, we have identified two different nonsense point mutations in the factor VIII gene of haemophiliacs, as well as two different partial deletions of the gene. Our survey of 92 haemophiliacs indicates no firm correlation between antibody (inhibitor) production and gross gene defects.
人类最常见的遗传性出血性疾病——甲型血友病,是由血液凝固途径中的一个成分即凝血因子VIII的缺陷所引起的。这种X染色体连锁疾病几乎肯定源于一系列不同的基因损伤。由于未经适当治疗的血友病可能是一种致命疾病,因此需要新的突变来解释其在人群中的恒定发病率。此外,甲型血友病表现出广泛的严重程度,约15%的血友病患者会产生高水平的抗凝血因子VIII抗体(“抑制剂患者”)。目前的研究阐明了部分个体中血友病的分子遗传基础。我们以最近克隆的凝血因子VIII基因作为探针,在血友病患者的凝血因子VIII基因中鉴定出两种不同的无义点突变以及该基因的两种不同的部分缺失。我们对92名血友病患者的调查表明,抗体(抑制剂)产生与总体基因缺陷之间没有确凿的相关性。