A Novel Deletion Mutation of the F8 Gene for Hemophilia A

被引:3
作者
Wang, Jingwei [1 ,2 ]
Gu, Jian [1 ]
Chen, Hongbing [3 ]
Wu, Qian [2 ]
Xiong, Liang [1 ]
Qiao, Bin [1 ]
Zhang, Yan [1 ]
Xiao, Hongjun [4 ]
Tong, Yongqing [1 ,2 ]
机构
[1] Wuhan Univ, Renmin Hosp, Dept Clin Lab, Wuhan 430060, Peoples R China
[2] Wuhan Univ, Clin Mol Diag Inst, Wuhan 430060, Peoples R China
[3] Wuhan Univ, Renmin Hosp, Dept Pulm & Crit Care Med, Wuhan 430060, Peoples R China
[4] Wuhan Univ, Med Vocat & Tech Sch, Wuhan 430060, Peoples R China
关键词
hemophilia A; coagulation factor FVIII; gene mutation; bleeding disorder; FACTOR-VIII; PRENATAL-DIAGNOSIS; RECOMMENDATION; INHIBITOR; CARRIER; DOMAIN;
D O I
10.3390/diagnostics12112876
中图分类号
R5 [内科学];
学科分类号
1002 ; 100201 ;
摘要
Background: Hemophilia A (HA) is an X-linked recessive blood coagulation disorder caused by a variety of abnormalities in F8 gene, resulting in the absence of impaired molecule production of factor VIII (FVIII) in the plasma. The genetic testing of the F8 gene encoding FVIII is used for confirmation of HA diagnosis, which significantly reduced serious complications of this disease and, ultimately, increased life expectancy. Methods: Sanger sequencing was performed in F8 gene exons of the suspected patients with blood coagulation-related indicators. Results: A novel F8 indel variant c.6343delC, p.Leu2115SerfsTer28 in exon 22 of the F8 gene was identified in the suspected families. The infant with this novel variant appeared the symptom of minor bleeding and oral cavity bleeding, and decreased activity of FVIII, which is consistent with that of F8 deleterious variants. The 3'D protein structural analysis of the novel variant shows a change in FVIII protein stability, which may be responsible for the pathogenesis of HA. Conclusions: A novel deleterious variant was identified in our case, which expands the F8 variants spectrum. Our result is helpful for HA diagnosis and benefits carrier detection and prenatal diagnosis. Our study also reveals that mutation screening of the F8 gene should be necessary for HA suspected patients.
引用
收藏
页数:10
相关论文
共 50 条
  • [1] Detection of F8 mutations in carriers and patients with severe hemophilia A. Identification of a novel mutation
    Lopez-Vasquez, Lucia
    Albanez, Silvia
    Pestana, Carolina
    Porco, Antonietta
    INVESTIGACION CLINICA, 2016, 57 (04): : 377 - 387
  • [2] Characterization of a genetically engineered mouse model of hemophilia A with complete deletion of the F8 gene
    Chao, B. N.
    Baldwin, W. H.
    Healey, J. F.
    Parker, E. T.
    Shafer-Weaver, K.
    Cox, C.
    Jiang, P.
    Kanellopoulou, C.
    Lollar, P.
    Meeks, S. L.
    Lenardo, M. J.
    JOURNAL OF THROMBOSIS AND HAEMOSTASIS, 2016, 14 (02) : 346 - 355
  • [3] A frameshift deletion in F8 associated with hemophilia A in Labrador Retriever dogs
    Hytonen, Marjo K. K.
    Viitanen, Sanna
    Hundi, Sruthi
    Donner, Jonas
    Lohi, Hannes
    Kaukonen, Maria
    ANIMAL GENETICS, 2023, 54 (05) : 606 - 612
  • [4] Factor 8 (F8) gene mutation profile of Turkish hemophilia A patients with inhibitors
    Fidanci, Inang D.
    Kavakli, Kaan
    Ucar, Canan
    Timur, Cetin
    Meral, Adalet
    Kilinc, Yurdanur
    Sayilang, Huelya
    Kazanci, Elif
    Caglayan, S. Hande
    BLOOD COAGULATION & FIBRINOLYSIS, 2008, 19 (05) : 383 - 388
  • [5] F8 gene inversion and duplication cause no obvious hemophilia A phenotype
    Li, Shaoying
    He, Jianchun
    Chu, Liming
    Ren, Shuai
    He, Wenzhi
    Ma, Xiaoyan
    Wang, Yanchao
    Zhang, Mincong
    Kong, Lingyin
    Liang, Bo
    Li, Qing
    FRONTIERS IN GENETICS, 2023, 14
  • [6] Identification and functional characterization of a novel splicing variant in the F8 coagulation gene causing severe hemophilia A
    Fama, Rosella
    Borroni, Ester
    Zanolini, Diego
    Merlin, Simone
    Bruscaggin, Valentina
    Walker, Gillian E.
    Olgasi, Cristina
    Babu, Deepak
    Giacchello, Jacopo Agnelli
    Valeri, Federica
    Giordano, Mara
    Borchiellini, Alessandra
    Follenzi, Antonia
    JOURNAL OF THROMBOSIS AND HAEMOSTASIS, 2020, 18 (05) : 1050 - 1064
  • [7] Hemophilia A: different phenotypes may be explained by multiple and variable effects of the causative mutation in the F8 gene
    Castaman, Giancarlo
    HAEMATOLOGICA, 2018, 103 (02) : 195 - 196
  • [8] F8 gene mutation profile in Indian hemophilia A patients: Identification of 23 novel mutations and factor VIII inhibitor risk association
    Pinto, Patricia
    Ghosh, Kanjaksha
    Shetty, Shrimati
    MUTATION RESEARCH-FUNDAMENTAL AND MOLECULAR MECHANISMS OF MUTAGENESIS, 2016, 786 : 27 - 33
  • [9] Whole F8 gene sequencing identified pathogenic structural variants in the remaining unsolved patients with severe hemophilia A
    Jourdy, Yohann
    Chatron, Nicolas
    Fretigny, Mathilde
    Zawadzki, Christophe
    Lienhart, Anne
    Stieltjes, Natalie
    Rohrlich, Pierre -Simon
    Thauvin-Robinet, Christel
    Volot, Fabienne
    Hamida, Yasmine Ferhat
    Hariti, Ghania
    Leuci, Alexandre
    Dargaud, Yesim
    Sanlaville, Damien
    Vinciguerra, Christine
    JOURNAL OF THROMBOSIS AND HAEMOSTASIS, 2024, 22 (06) : 1616 - 1626
  • [10] An aberrant F8 intron 1 inversion with concomitant large duplication and deletion in a Chinese severe hemophilia A patient
    Wang, Xiong
    Wang, Hongmei
    Tan, Haowen
    Liu, Xiu-Ping
    Li, Huijun
    HEMATOLOGY, 2021, 26 (01) : 53 - 57