Literature DB >> 4322125

Lesch-Nyhan syndrome: altered kinetic properties of mutant enzyme.

J A McDonald, W N Kelley.   

Abstract

Hypoxanthine-guanine phosphoribosyltransferase is virtually inactive in erythrocytes from patients with the classical Lesch-Nyhan syndrome. In one such patient, activity of this enzyme ranged from 8 to 34 percent of normal in erythrocytes when assayed with a very high concentration of magnesium 5-phosphoribosyl-1-pyrophosphate. In addition, the mutant enzyme exhibited sigmoidal kinetics with this substrate as well as an increased Michaelis constant for both guanine and hypoxanthine. These findings provide the first evidence for genetic heterogeneity within the group of patients with the Lesch-Nyhan syndrome.

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Year:  1971        PMID: 4322125     DOI: 10.1126/science.171.3972.689

Source DB:  PubMed          Journal:  Science        ISSN: 0036-8075            Impact factor:   47.728


  31 in total

1.  Clinical quiz. Elevated urinary uric acid excretion (UUAE).

Authors:  M J Schoeneman
Journal:  Pediatr Nephrol       Date:  1990-03       Impact factor: 3.714

2.  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

3.  Electrophoretic properties of hypoxanthine-guanine phosphoribosyl transferase in erythrocytes of subjects with Lesch-Nyhan syndrome.

Authors:  B Bakay; W L Nyhan
Journal:  Biochem Genet       Date:  1972-04       Impact factor: 1.890

4.  Heterogeneity in the biochemical characteristics of red blood cell hypoxanthine-guanine phosphoribosyl transferase from two unrelated patients with the Lesch-Nyhan syndrome.

Authors:  B J Richardson; D L Ryckman; L M Komarnicki; J L Hamerton
Journal:  Biochem Genet       Date:  1973-06       Impact factor: 1.890

5.  Hypoxanthine-guanine phosphoribosyltransferase deficiency in three brothers with gout: characterization of a variant, HPRTEdinburgh, having altered isoelectric point, increased thermal lability and normal levels of messenger RNA.

Authors:  F F Snyder; J E Joyce; T Carter-Edwards; R Joshi; H L Rylance; R C Wallace; G Nuki
Journal:  J Inherit Metab Dis       Date:  1989       Impact factor: 4.982

Review 6.  Attenuated variants of Lesch-Nyhan disease.

Authors:  H A Jinnah; Irene Ceballos-Picot; Rosa J Torres; Jasper E Visser; David J Schretlen; Alfonso Verdu; Laura E Laróvere; Chung-Jen Chen; Antonello Cossu; Chien-Hui Wu; Radhika Sampat; Shun-Jen Chang; Raquel Dodelson de Kremer; William Nyhan; James C Harris; Stephen G Reich; Juan G Puig
Journal:  Brain       Date:  2010-02-22       Impact factor: 13.501

7.  Human hypoxanthine-guanine phosphoribosyltransferase. Demonstration of structural variants in lymphoblastoid cells derived from patients with a deficiency of the enzyme.

Authors:  J M Wilson; B W Baugher; P M Mattes; P E Daddona; W N Kelley
Journal:  J Clin Invest       Date:  1982-03       Impact factor: 14.808

8.  Molecular basis of hypoxanthine-guanine phosphoribosyltransferase deficiency in a patient with the Lesch-Nyhan syndrome.

Authors:  J M Wilson; W N Kelley
Journal:  J Clin Invest       Date:  1983-05       Impact factor: 14.808

9.  Expression of purine overproduction in a series of 8-azaguanine-resistant diploid human lymphoblast lines.

Authors:  J E Lever; G Nuki; J E Seegmiller
Journal:  Proc Natl Acad Sci U S A       Date:  1974-07       Impact factor: 11.205

Review 10.  Hypoxanthine-guanine phosphoribosyl transferase deficiency.

Authors:  C H de Bruyn
Journal:  Hum Genet       Date:  1976-02-29       Impact factor: 4.132

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