Literature DB >> 7112549

A female case of the Leach-Nyhan syndrome.

K Hara, S Kashiwamata, N Ogasawara, H Ohishi, R Natsume, T Yamanaka, S Hakamada, S Miyazaki, K Watanabe.   

Abstract

The classical Lesch-Nyhan syndrome has the deficiency of hypoxanthine-guanine phosphoribosyltransferase (HGPRT) activity as the result of mutation in the structural gene for the enzyme located on the X chromosome and is believed to be of X-linked recessive or sex-linked mode of inheritance. This is the first report of a girl who showed typical clinical features and biochemical characteristics of the classical Lesch-Nyhan syndrome. Her mother was not a heterozygote for a deficiency of HGPRT. Possible genetic mechanisms responsible for this case were discussed.

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Year:  1982        PMID: 7112549     DOI: 10.1620/tjem.137.275

Source DB:  PubMed          Journal:  Tohoku J Exp Med        ISSN: 0040-8727            Impact factor:   1.848


  4 in total

1.  Regional brain volume abnormalities in Lesch-Nyhan disease and its variants: a cross-sectional study.

Authors:  David J Schretlen; Mark Varvaris; Tiffany E Ho; Tracy D Vannorsdall; Barry Gordon; James C Harris; H A Jinnah
Journal:  Lancet Neurol       Date:  2013-12       Impact factor: 44.182

Review 2.  Delineation of the motor disorder of Lesch-Nyhan disease.

Authors:  H A Jinnah; Jasper E Visser; James C Harris; Alfonso Verdu; Laura Larovere; Irene Ceballos-Picot; Pedro Gonzalez-Alegre; Vladimir Neychev; Rosa J Torres; Olivier Dulac; Isabelle Desguerre; David J Schretlen; Kenneth L Robey; Gabor Barabas; Bastiaan R Bloem; William Nyhan; Raquel De Kremer; Gary E Eddey; Juan G Puig; Stephen G Reich
Journal:  Brain       Date:  2006-03-20       Impact factor: 13.501

Review 3.  Genotype-phenotype correlations in neurogenetics: Lesch-Nyhan disease as a model disorder.

Authors:  Rong Fu; Irene Ceballos-Picot; Rosa J Torres; Laura E Larovere; Yasukazu Yamada; Khue V Nguyen; Madhuri Hegde; Jasper E Visser; David J Schretlen; William L Nyhan; Juan G Puig; Patrick J O'Neill; H A Jinnah
Journal:  Brain       Date:  2013-08-22       Impact factor: 13.501

Review 4.  Hypoxanthine-guanine phosophoribosyltransferase (HPRT) deficiency: Lesch-Nyhan syndrome.

Authors:  Rosa J Torres; Juan G Puig
Journal:  Orphanet J Rare Dis       Date:  2007-12-08       Impact factor: 4.123

  4 in total

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