一个印度家庭中出现的临床表现各异的新型 SERPINC1 c.119G>A (p.Cys40Tyr) 突变。

IF 1.2 4区 医学 Q4 HEMATOLOGY Blood Coagulation & Fibrinolysis Pub Date : 2024-11-04 DOI:10.1097/MBC.0000000000001333
Kranti Patil, Asha Shah, Gurpreet Saini, Shreyas Tawde, Shruti Kharat, Fiza Jivani, Aniket Kamble, Shrimati Shetty
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引用次数: 0

摘要

众所周知,SERPINC1 基因突变导致的遗传性抗凝血酶(AT)缺乏症是血栓性疾病中最严重的一种。我们报告了一个遗传性抗凝血酶缺乏症家族中的三位成员,他们的 SERPINC1 第 2 外显子都有一个新的突变,即 c.119 G>A (p.Cys40Tyr)。两个兄弟分别在 18 岁和 21 岁时出现急性肺血栓栓塞症(PTE),而他们 58 岁的父亲至今未出现任何血栓发作。PolyPhen-2、SIFT 和 MutationTaster 对该变异体进行了校内预测,发现该变异体具有高度损伤性。临床外显子组测序结果显示,除了 SERPINE1 -844 G>A 变异在两个受影响的兄弟中为同基因状态外,没有发现任何强共同遗传的血栓性疾病基因,而父亲则是该变异的杂合子。在没有其他已知环境诱发因素的情况下,不能排除 SERPINE1 变体对两个兄弟姐妹临床表现的叠加效应。
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A novel SERPINC1 c.119G>A (p.Cys40Tyr) mutation with variable clinical expression in an Indian family.

Hereditary antithrombin (AT) deficiency due to mutations in SERPINC1 is known to be the most severe form of thrombophilia. We report three members in a family with hereditary AT deficiency with a novel mutation in exon 2 of SERPINC1, that is c.119 G>A (p.Cys40Tyr). Two brothers presented with acute pulmonary thromboembolism (PTE) at 18 and 21 years of age, whereas their 58-year-old father did not have any thrombotic episode till date. The in-silico prediction of the variant was found to be highly damaging by PolyPhen-2, SIFT and MutationTaster. Clinical exome sequencing did not show any strong coinherited thrombophilia genes, except SERPINE1 -844 G>A variant in homozygous state in the two affected brothers as compared to the father who was heterozygous for this variant. The additive effect of SERPINE1 variant in the clinical expression in two siblings cannot be ruled out, in the absence of any other known environmental triggering factors.

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来源期刊
CiteScore
1.90
自引率
0.00%
发文量
111
审稿时长
4-8 weeks
期刊介绍: Blood Coagulation & Fibrinolysis is an international fully refereed journal that features review and original research articles on all clinical, laboratory and experimental aspects of haemostasis and thrombosis. The journal is devoted to publishing significant developments worldwide in the field of blood coagulation, fibrinolysis, thrombosis, platelets and the kininogen-kinin system, as well as dealing with those aspects of blood rheology relevant to haemostasis and the effects of drugs on haemostatic components
期刊最新文献
Variation among venous thromboembolism risk assessment tools for postcesarean patients: a retrospective cohort study. Is the combination of two automated rapid assays for diagnosis of heparin-induced thrombocytopenia necessary? A novel SERPINC1 c.119G>A (p.Cys40Tyr) mutation with variable clinical expression in an Indian family. A novel γ-chain mutation p.Asp318His in a Chinese family with dysfibrinogenemia. Molecular mechanism analysis of a family with hereditary coagulation FXI deficiency caused by compound heterozygous mutations.
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