Novel ACTG1 mutation causing autosomal dominant non-syndromic hearing impairment in a Chinese family

被引:25
作者
Liu, Ping [1 ]
Li, Hu [2 ]
Ren, Xiang [1 ]
Mao, Haiyan [2 ]
Zhu, Qihui [1 ]
Zhu, Zhengfeng [2 ]
Yang, Rong [1 ]
Yuan, Wenlin [2 ]
Liu, Jingyu [1 ]
Wang, Qing [1 ]
Liu, Mugen [1 ]
机构
[1] Huazhong Univ Sci & Technol, Coll Life Sci & Technol, Ctr Human Genome Res, Key Lab Mol Biophys Minist Educ, Wuhan 430074, Peoples R China
[2] Fourth Peoples Hosp Yancheng, Jiangsu 224000, Peoples R China
基金
中国国家自然科学基金;
关键词
non-syndromic hearing impairment (NSHI); ACTG1; linkage; sequence analysis; restriction fragment length polymorphism (RFLP) analysis;
D O I
10.1016/S1673-8527(08)60075-2
中图分类号
Q5 [生物化学]; Q7 [分子生物学];
学科分类号
071010 ; 081704 ;
摘要
The gamma-actin (ACTG1) gene is a cytoplasmic nonmuscle actin gene, which encodes a major cytoskeletal protein in the sensory hair cells of the cochlea. Mutations in ACTG1 were found to cause autosomal dominant, progressive, sensorineural hearing loss linked to the DFNA 20/26 locus on chromosome 17q25.3 in European and American families, respectively. In this study, a novel missense mutation (c.364A>G; p.I122V) co-segregated with the affected individuals in the family and did not exist in the unaffected family members and 150 unrelated normal controls. The alteration of residue Ile122 was predicted to damage its interaction with actin-binding proteins, which may cause disruption of hair cell organization and function. These findings strongly suggested that the I122V mutation in ACTG1 caused autosomal dominant non-syndromic hearing impairment in a Chinese family and expanded the spectrum of ACTG1 mutations causing hearing loss.
引用
收藏
页码:553 / 558
页数:6
相关论文
共 15 条
  • [1] Hereditary deafness and phenotyping in humans
    Bitner-Glindzicz, M
    [J]. BRITISH MEDICAL BULLETIN, 2002, 63 : 73 - 94
  • [2] Effects of human deafness γ-actin mutations (DFNA20/26) on actin function
    Bryan, Keith E.
    Wen, Kuo-Kuang
    Zhu, Mei
    Rendtorff, Nanna Dahl
    Feldkamp, Michael
    Tranebjaerg, Lisbeth
    Friderici, Karen H.
    Rubenstein, Peter A.
    [J]. JOURNAL OF BIOLOGICAL CHEMISTRY, 2006, 281 (29) : 20129 - 20139
  • [3] THE STRUCTURE OF THE CUTICULAR PLATE, AN INVIVO ACTIN GEL
    DEROSIER, DJ
    TILNEY, LG
    [J]. JOURNAL OF CELL BIOLOGY, 1989, 109 (06) : 2853 - 2867
  • [4] Localization of a calcium sensitive binding site for gelsolin on actin subdomain 1: Implication for severing process
    Feinberg, J
    Lebart, MC
    Benyamin, Y
    Roustan, C
    [J]. BIOCHEMICAL AND BIOPHYSICAL RESEARCH COMMUNICATIONS, 1997, 233 (01) : 61 - 65
  • [5] INTERACTIONS BETWEEN ACTIN-FILAMENTS AND BETWEEN ACTIN-FILAMENTS AND MEMBRANES IN QUICK-FROZEN AND DEEPLY ETCHED HAIR-CELLS OF THE CHICK EAR
    HIROKAWA, N
    TILNEY, LG
    [J]. JOURNAL OF CELL BIOLOGY, 1982, 95 (01) : 249 - 261
  • [6] Hofer D, 1997, J CELL SCI, V110, P765
  • [7] ATOMIC MODEL OF THE ACTIN FILAMENT
    HOLMES, KC
    POPP, D
    GEBHARD, W
    KABSCH, W
    [J]. NATURE, 1990, 347 (6288) : 44 - 49
  • [8] Functional specificity of actin isoforms
    Khaitlina, SY
    [J]. INTERNATIONAL REVIEW OF CYTOLOGY - A SURVEY OF CELL BIOLOGY, VOL. 202, 2001, 202 : 35 - 98
  • [9] DETERMINATION OF THE ALPHA-ACTININ-BINDING SITE ON ACTIN-FILAMENTS BY CRYOELECTRON MICROSCOPY AND IMAGE-ANALYSIS
    MCGOUGH, A
    WAY, M
    DEROSIER, D
    [J]. JOURNAL OF CELL BIOLOGY, 1994, 126 (02) : 433 - 443
  • [10] A novel missense mutation in ACTG1 causes dominant deafness in a Norwegian DFNA20/26 family, but ACTG1 mutations are not frequent among families with hereditary hearing impairment
    Rendtorff, Nanna D.
    Zhu, Mei
    Fagerheim, Toril
    Antal, Torben L.
    Jones, MaryPat
    Teslovich, Tanya M.
    Gillanders, Elizabeth M.
    Barmada, Michael
    Teig, Erik
    Trent, Jeffrey M.
    Friderici, Karen H.
    Stephan, Dietrich A.
    Tranebjaerg, Lisbeth
    [J]. EUROPEAN JOURNAL OF HUMAN GENETICS, 2006, 14 (10) : 1097 - 1105