Literature DB >> 1550586

Intermediate form of mucopolysaccharidosis type II (Hunter disease): a C1327 to T substitution in the iduronate sulfatase gene.

K Sukegawa1, S Tomatsu, K Tamai, M Ikeda, T Sasaki, M Masue, S Fukuda, Y Yamada, T Orii.   

Abstract

Hunter disease, an X-linked recessive lysosomal storage disorder, is caused by a deficiency in iduronate sulfatase activity. Sequence analysis of mRNA of fibroblasts of an intermediate phenotype patient showed a single C1327 to T nucleotide transition. This mutation resulted in a substitution of termination codon for normal arginine at position 443 of the peptide sequence. Expression studies with this abnormal cDNA in fibroblasts from the patient revealed a loss of enzymatic activity and instability of the mutated protein. We posturate that this mutation is probably the cause of the intermediate form of Hunter disease.

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Year:  1992        PMID: 1550586     DOI: 10.1016/0006-291x(92)90555-y

Source DB:  PubMed          Journal:  Biochem Biophys Res Commun        ISSN: 0006-291X            Impact factor:   3.575


  8 in total

1.  Germline and somatic mosaicism in a female carrier of Hunter disease.

Authors:  R Froissart; I Maire; V Bonnet; T Levade; D Bozon
Journal:  J Med Genet       Date:  1997-02       Impact factor: 6.318

2.  Mutational and structural analysis of Japanese patients with mucopolysaccharidosis type II.

Authors:  Tomomi Kato; Zenichiro Kato; Izumi Kuratsubo; Noboru Tanaka; Tabito Ishigami; Jun-Ichi Kajihara; Kazuko Sukegawa-Hayasaka; Koji Orii; Koji Isogai; Toshiyuki Fukao; Nobuyuki Shimozawa; Tadao Orii; Naomi Kondo; Yasuyuki Suzuki
Journal:  J Hum Genet       Date:  2005-08-30       Impact factor: 3.172

3.  Mutation analysis in the iduronate-2-sulphatase gene in 43 Japanese patients with mucopolysaccharidosis type II (Hunter disease).

Authors:  K Isogai; K Sukegawa; S Tomatsu; T Fukao; X Q Song; Y Yamada; S Fukuda; T Orii; N Kondo
Journal:  J Inherit Metab Dis       Date:  1998-02       Impact factor: 4.982

Review 4.  Review: the immunochemical analysis of enzyme from mucopolysaccharidoses patients.

Authors:  D A Brooks
Journal:  J Inherit Metab Dis       Date:  1993       Impact factor: 4.982

5.  Molecular analysis of the iduronate-2-sulfatase gene in Thai patients with Hunter syndrome.

Authors:  S Keeratichamroen; J R Ketudat Cairns; D Wattanasirichaigoon; P Wasant; L Ngiwsara; P Suwannarat; S Pangkanon; J Kuptanon; P Tanpaiboon; T Rujirawat; S Liammongkolkul; J Svasti
Journal:  J Inherit Metab Dis       Date:  2008-05-20       Impact factor: 4.982

6.  Mucopolysaccharidosis type II (Hunter disease): 13 gene mutations in 52 Japanese patients and carrier detection in four families.

Authors:  Y Yamada; S Tomatsu; K Sukegawa; Y Suzuki; N Kondo; J J Hopwood; T Orii
Journal:  Hum Genet       Date:  1993-09       Impact factor: 4.132

7.  Molecular diagnosis of mucopolysaccharidosis type II (Hunter syndrome) by automated sequencing and computer-assisted interpretation: toward mutation mapping of the iduronate-2-sulfatase gene.

Authors:  J J Jonsson; E L Aronovich; S E Braun; C B Whitley
Journal:  Am J Hum Genet       Date:  1995-03       Impact factor: 11.025

8.  Processing of iduronate 2-sulphatase in human fibroblasts.

Authors:  R Froissart; G Millat; M Mathieu; D Bozon; I Maire
Journal:  Biochem J       Date:  1995-07-15       Impact factor: 3.857

  8 in total

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