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凝血因子 XIIIA 亚基错义突变影响因子 XIII 作用各步骤的结构和功能。

Coagulation Factor XIIIA Subunit Missense Mutations Affect Structure and Function at the Various Steps of Factor XIII Action.

作者信息

Thomas Anne, Biswas Arijit, Dodt Johannes, Philippou Helen, Hethershaw Emma, Ensikat Hans Juergen, Ivaskevicius Vytautas, Oldenburg Johannes

机构信息

Institute of Experimental Haematology and Transfusion Medicine, University Clinic Bonn, Bonn, Germany.

Paul Ehrlich Institut, Langen, Germany.

出版信息

Hum Mutat. 2016 Oct;37(10):1030-41. doi: 10.1002/humu.23041. Epub 2016 Aug 21.

Abstract

Inherited defects of coagulation Factor XIII (FXIII) can be categorized into severe and mild forms based on their genotype and phenotype. Heterozygous mutations occurring in F13A1 and F13B genes causing mild FXIII deficiency have been reported only in the last few years primarily because the mild FXIII deficiency patients are often asymptomatic unless exposed to some kind of a physical trauma. However, unlike mutations causing severe FXIII deficiency, many of these mutations have not been comprehensively characterized based on expression studies. In our current article, we have transiently expressed 16 previously reported missense mutations detected in the F13A1 gene of patients with mild FXIII deficiency and analyzed their respective expression phenotype. Complimentary to expression analysis, we have used in silico analysis to understand and explain some of the in vitro findings. The expression phenotype has been evaluated with a number of expression phenotype determining assays. We observe that the mutations influence different aspects of FXIII function and can be functionally categorized on the basis of their expression phenotype. We identified mutations which even in heterozygous form would have strong impact on the functional status of the protein (namely mutations p.Arg716Gly, p.Arg704Gln, p.Gln602Lys, p.Leu530Pro, p.His343Tyr, p.Pro290Arg, and p.Arg172Gln).

摘要

凝血因子 XIII(FXIII)的遗传性缺陷可根据其基因型和表型分为严重和轻度两种形式。导致轻度 FXIII 缺乏的 F13A1 和 F13B 基因中的杂合突变直到最近几年才被报道,主要是因为轻度 FXIII 缺乏患者通常没有症状,除非受到某种身体创伤。然而,与导致严重 FXIII 缺乏的突变不同,这些突变中的许多尚未根据表达研究进行全面表征。在我们当前的文章中,我们瞬时表达了在轻度 FXIII 缺乏患者的 F13A1 基因中检测到的 16 个先前报道的错义突变,并分析了它们各自的表达表型。作为表达分析的补充,我们使用了计算机分析来理解和解释一些体外研究结果。通过多种表达表型测定方法评估了表达表型。我们观察到这些突变影响 FXIII 功能的不同方面,并且可以根据它们的表达表型在功能上进行分类。我们鉴定出即使以杂合形式存在也会对蛋白质功能状态产生强烈影响的突变(即 p.Arg716Gly、p.Arg704Gln、p.Gln602Lys、p.Leu530Pro、p.His343Tyr、p.Pro290Arg 和 p.Arg172Gln 突变)。

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