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来自中国东南部的两名凝血因子VII缺乏症患者的表型和基因型特征

Phenotypic and genotypic characterization of two factor VII deficiency patients from southeastern China.

作者信息

Wang Anzi, Su Dongyun, Chen Yanrong, Fu Yuhan, Tan Xiaoyan, Luo Jingyuan, Wang Jie, Li Yue, Chen Shu

机构信息

Department of Hematology.

Department of Neurology.

出版信息

Blood Coagul Fibrinolysis. 2022 Dec 1;33(8):468-472. doi: 10.1097/MBC.0000000000001145. Epub 2022 Jul 22.

Abstract

The congenital factor VII deficiency (FVIID) is a rare autosomal recessive haemorrhagic disease caused by mutations in the F7 gene. The aim of this study was to identify the mutations causing FVII deficiency and explain the genotype-phenotype association in two unrelated Chinese patients. Mutation detection was conducted by sequencing the whole F7 gene coding exons, exon-intron boundaries and the untranslated regions of 3' and 5'. Then, the genetic information was analyzed to predict the structures of the mutated proteins. A total of four different mutations were detected, including three missense mutations (c.64G>A, c.286A>G, and c.722C>A, predicting p.Gly22Ser, p.Arg96Gly, p.Thr241Asn, respectively) and one insertion mutation (c.204_205insCGGC, predicting p. Leu68Argfs ∗ 37), among which two were reported for the first time (p.Arg96Gly, p.Leu68Argfs ∗ 37). Multiple sequence alignments of FVII protein revealed that the residues p.Arg96 and p.Thr241 were highly conserved. The novel missense mutation p.Arg96Gly was determined as damaging with online software Polyphen-2 and SIFT. We investigated two asymptomatic patients diagnosed with severe FVII deficiency and identified two novel mutations (the mutation p.Arg96Gly and p.Leu68Argfs ∗ 37). Identification of the F7 mutations was important for genetic counseling and accurate prediction of the inheritance pattern.

摘要

先天性因子 VII 缺乏症(FVIID)是一种由 F7 基因突变引起的罕见常染色体隐性出血性疾病。本研究的目的是鉴定导致因子 VII 缺乏的突变,并解释两名无关中国患者的基因型 - 表型关联。通过对整个 F7 基因编码外显子、外显子 - 内含子边界以及 3' 和 5' 非翻译区进行测序来进行突变检测。然后,分析遗传信息以预测突变蛋白的结构。共检测到四种不同的突变,包括三个错义突变(c.64G>A、c.286A>G 和 c.722C>A,分别预测 p.Gly22Ser、p.Arg96Gly、p.Thr241Asn)和一个插入突变(c.204_205insCGGC,预测 p.Leu68Argfs∗37),其中两个是首次报道(p.Arg96Gly、p.Leu68Argfs∗37)。因子 VII 蛋白的多序列比对显示,p.Arg96 和 p.Thr241 残基高度保守。在线软件 Polyphen-2 和 SIFT 将新的错义突变 p.Arg96Gly 判定为有害突变。我们研究了两名被诊断为严重因子 VII 缺乏的无症状患者,并鉴定出两个新突变(p.Arg96Gly 和 p.Leu68Argfs∗37)。鉴定 F7 突变对于遗传咨询和准确预测遗传模式很重要。

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