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人类凝血因子IX基因供体剪接连接处的点突变导致的乙型血友病。

Haemophilia B caused by a point mutation in a donor splice junction of the human factor IX gene.

作者信息

Rees D J, Rizza C R, Brownlee G G

出版信息

Nature. 1985;316(6029):643-5. doi: 10.1038/316643a0.

Abstract

Haemophilia B (Christmas disease) is an inherited, recessive, sex-linked, haemorrhagic condition caused by a defect in the intrinsic clotting factor IX. This disease occurs in males at a frequency of approximately 1 in 30,000. Patients differ in the severity of their clinical symptoms, and variation in the clotting activity and in the concentration of factor IX antigen in their plasma has been demonstrated. There is probably heterogeneity in the molecular defects of the factor IX gene causing the disease. Here we study a severely affected, antigen-negative patient, and show that the only significant sequence difference from the normal factor IX gene is a point mutation changing the obligatory GT to a TT within the donor splice junction of exon f. We infer that this change is the cause of the disease in this individual. In addition, we have used oligodeoxynucleotide probes specific for this mutation to demonstrate the feasibility of carrier detection and prenatal diagnosis for relatives of the patient.

摘要

血友病B(克里斯马斯病)是一种遗传性、隐性、性连锁出血性疾病,由内源性凝血因子IX缺陷引起。这种疾病在男性中的发病率约为三万分之一。患者临床症状的严重程度各不相同,并且已证明其血浆中凝血活性和因子IX抗原浓度存在差异。导致该疾病的因子IX基因的分子缺陷可能存在异质性。在此,我们研究了一名严重患病的抗原阴性患者,结果表明,与正常因子IX基因唯一显著的序列差异是一个点突变,该突变使外显子f供体剪接连接处的必需GT变为TT。我们推断这种变化是该个体患病的原因。此外,我们使用了针对此突变的寡脱氧核苷酸探针,证明了对该患者亲属进行携带者检测和产前诊断的可行性。

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