Wang Anzi, Su Dongyun, Chen Yanrong, Fu Yuhan, Tan Xiaoyan, Luo Jingyuan, Wang Jie, Li Yue, Chen Shu
Department of Hematology.
Department of Neurology.
Blood Coagul Fibrinolysis. 2022 Dec 1;33(8):468-472. doi: 10.1097/MBC.0000000000001145. Epub 2022 Jul 22.
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.
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。