A Japanese case with Frasier syndrome caused by the splice junction mutation of WT1 gene

被引:11
作者
Okuhara, K [1 ]
Tajima, T [1 ]
Nakae, J [1 ]
Sasaki, S [1 ]
Tochimaru, H [1 ]
Abe, S [1 ]
Fujieda, K [1 ]
机构
[1] Hokkaido Univ, Sch Med, Dept Pediat, Kita Ku, Sapporo, Hokkaido 0608628, Japan
关键词
WT1; gene; Frasier syndrome; gonadoblastoma; splice junction mutation;
D O I
10.1507/endocrj.46.639
中图分类号
R5 [内科学];
学科分类号
1002 ; 100201 ;
摘要
The Wilms' tumor suppressor gene, WT1, plays an important role in the development of the urogenital system and also subsequent normal function of this system. Recently, the splice mutations in intron 9 of WT1 gene have been detected in Frasier syndrome, which is characterized by streak gonads, pseudohermaphroditism, slowly progressive nephropathy and frequent development of gonadoblastoma. Here to elucidate the molecular basis in a Japanese patient of Frasier syndrome, WT1 gene was analyzed by polymerase-chain-reaction (PCR) and direct sequencing. We identified the splice junction mutation in intron 9 of WT1, which is recognized as a mutation hot-spot in intron 9. This finding concludes that 1) the mutation in intron 9 might be the cause of Frasier syndrome, and 2) the mutation hot-spot in Japanese and Caucasian patients is similar.
引用
收藏
页码:639 / 642
页数:4
相关论文
共 50 条
  • [41] WT1, A WILMS-TUMOR GENE
    BARD, JBL
    ARMSTRONG, JF
    BICKMORE, WA
    EXPERIMENTAL NEPHROLOGY, 1993, 1 (04): : 218 - 223
  • [42] Posttransplant recurrence of proteinuria in a case of focal segmental glomerulosclerosis associated with WT1 mutation
    Ghiggeri, G. M.
    Aucella, F.
    Caridi, G.
    Bisceglia, L.
    Ghio, L.
    Gigante, M.
    Perfumo, F.
    Carraro, M.
    Gesualdo, L.
    AMERICAN JOURNAL OF TRANSPLANTATION, 2006, 6 (09) : 2208 - 2211
  • [43] A child with isolated nephrotic syndrome and WT1 mutation presenting as a 46, XY phenotypic male
    Yang, Yonghui
    Feng, Dongning
    Huang, Jun
    Nie, Xiaojing
    Yu, Zihua
    EUROPEAN JOURNAL OF PEDIATRICS, 2013, 172 (01) : 127 - 129
  • [44] A high incidence of WT1 abnormality in bilateral Wilms tumours in Japan, and the penetrance rates in children with WT1 germline mutation
    Kaneko, Y.
    Okita, H.
    Haruta, M.
    Arai, Y.
    Oue, T.
    Tanaka, Y.
    Horie, H.
    Hinotsu, S.
    Koshinaga, T.
    Yoneda, A.
    Ohtsuka, Y.
    Taguchi, T.
    Fukuzawa, M.
    BRITISH JOURNAL OF CANCER, 2015, 112 (06) : 1121 - 1133
  • [45] A WT1 exon 1 mutation in a child diagnosed with Denys-Drash syndrome
    Little, S
    Hanks, S
    King-Underwood, L
    Picton, S
    Cullinane, C
    Rapley, E
    Rahman, N
    Pritchard-Jones, K
    PEDIATRIC NEPHROLOGY, 2005, 20 (01) : 81 - 85
  • [46] The podocyte as a target: cyclosporin A in the management of the nephrotic syndrome caused by WT1 mutations
    Constantinos J. Stefanidis
    Uwe Querfeld
    European Journal of Pediatrics, 2011, 170 : 1377 - 1383
  • [47] The podocyte as a target: cyclosporin A in the management of the nephrotic syndrome caused by WT1 mutations
    Stefanidis, Constantinos J.
    Querfeld, Uwe
    EUROPEAN JOURNAL OF PEDIATRICS, 2011, 170 (11) : 1377 - 1383
  • [48] Discordant expression of a new WT1 gene mutation in a family with monozygotic twins presenting with congenital nephrotic syndrome
    Filip Fencl
    Michal Malina
    Veronika Stará
    Jakub Zieg
    Dana Mixová
    Tomáš Seeman
    Květa Bláhová
    European Journal of Pediatrics, 2012, 171 : 121 - 124
  • [49] Description of mutation spectrum and polymorphism of Wilms' tumor 1 (WT1) gene in hypospadias patients in the Indonesian population
    Diposarosa, Rizki
    Pamungkas, Kuriniawan O.
    Sribudiani, Yunia
    Herman, Herry
    Suciati, Lita P.
    Rahayu, Nurul S.
    Effendy, Sjarif H.
    JOURNAL OF PEDIATRIC UROLOGY, 2018, 14 (03) : 237.e1 - 237.e7
  • [50] The role of WT1 gene in neuroblastoma
    Wang, Jingfu
    Oue, Takaharu
    Uehara, Shuichiro
    Yamanaka, Hiroaki
    Oji, Yusuke
    Fukuzawa, Masahiro
    JOURNAL OF PEDIATRIC SURGERY, 2011, 46 (02) : 326 - 331