Literature DB >> 1528024

Mouse models of hypoxanthine phosphoribosyltransferase deficiency.

D J Williamson1, M L Hooper, D W Melton.   

Abstract

Lesch--Nyhan syndrome is an X-linked disease caused by the deficiency of hypoxanthine phosphoribosyltransferase, an enzyme involved in the purine salvage pathways. It is characterized by severe gout, choreoathetosis, self-mutilatory behaviour and mental retardation. The derivation of mice genetically deficient in this enzyme may help to elucidate the pathogenesis of the neurological abnormality where previously models using drug administration to mimic the disorder have had to suffice.

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Year:  1992        PMID: 1528024     DOI: 10.1007/bf01799622

Source DB:  PubMed          Journal:  J Inherit Metab Dis        ISSN: 0141-8955            Impact factor:   4.982


  24 in total

Review 1.  Altering the genome by homologous recombination.

Authors:  M R Capecchi
Journal:  Science       Date:  1989-06-16       Impact factor: 47.728

2.  Monoamine deficiency in a transgenic (Hprt-) mouse model of Lesch-Nyhan syndrome.

Authors:  S B Dunnett; D J Sirinathsinghji; R Heavens; D C Rogers; M R Kuehn
Journal:  Brain Res       Date:  1989-11-06       Impact factor: 3.252

3.  Lesch-Nyhan syndrome: absence of the mutant enzyme in erythrocytes of a heterozygote for both normal and mutant hypoxanthine-guanine phosphoribosyl transferase.

Authors:  J A McDonald; W N Kelley
Journal:  Biochem Genet       Date:  1972-02       Impact factor: 1.890

4.  Germ line transmission and expression of a corrected HPRT gene produced by gene targeting in embryonic stem cells.

Authors:  S Thompson; A R Clarke; A M Pow; M L Hooper; D W Melton
Journal:  Cell       Date:  1989-01-27       Impact factor: 41.582

5.  A potential animal model for Lesch-Nyhan syndrome through introduction of HPRT mutations into mice.

Authors:  M R Kuehn; A Bradley; E J Robertson; M J Evans
Journal:  Nature       Date:  1987 Mar 19-25       Impact factor: 49.962

Review 6.  A dopamine deficiency model of Lesch-Nyhan disease--the neonatal-6-OHDA-lesioned rat.

Authors:  G R Breese; H E Criswell; G E Duncan; R A Mueller
Journal:  Brain Res Bull       Date:  1990-09       Impact factor: 4.077

Review 7.  Animal models of Lesch-Nyhan syndrome.

Authors:  H A Jinnah; F H Gage; T Friedmann
Journal:  Brain Res Bull       Date:  1990-09       Impact factor: 4.077

8.  Sprouting of striatal serotonin nerve terminals following selective lesions of nigro-striatal dopamine neurons in neonatal rat.

Authors:  J Luthman; B Bolioli; T Tsutsumi; A Verhofstad; G Jonsson
Journal:  Brain Res Bull       Date:  1987-08       Impact factor: 4.077

9.  Behavioral and neurochemical evaluation of a transgenic mouse model of Lesch-Nyhan syndrome.

Authors:  S Finger; R P Heavens; D J Sirinathsinghji; M R Kuehn; S B Dunnett
Journal:  J Neurol Sci       Date:  1988-09       Impact factor: 3.181

10.  Hypoxanthine phosphoribosyl transferase deficiency, haematopoiesis and fertility in the mouse.

Authors:  J D Ansell; K Samuel; D G Whittingham; C E Patek; K Hardy; A H Handyside; K W Jones; A L Muggleton-Harris; A H Taylor; M L Hooper
Journal:  Development       Date:  1991-06       Impact factor: 6.868

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  1 in total

Review 1.  CAG-polyglutamine-repeat mutations: independence from gene context.

Authors:  J M Ordway; J A Cearley; P J Detloff
Journal:  Philos Trans R Soc Lond B Biol Sci       Date:  1999-06-29       Impact factor: 6.237

  1 in total

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