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PMID: 4322125 Published · ppublish English Journal Article

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

Science (New York, N.Y.) ·Vol. 171 ·No. 3972 ·1971-02-19 ·Pages 689-91

McDonald JA, Kelley WN

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.

MeSH Terms
Athetosis/genetics Child Chorea/genetics Compulsive Behavior Diphosphates/metabolism Erythrocytes/enzymology Guanine/metabolism Humans Hypoxanthines/metabolism Intellectual Disability Kinetics Male Molecular Biology Mutation Purine-Pyrimidine Metabolism, Inborn Errors/enzymology Self Mutilation Transferases/blood,metabolism Uric Acid/blood
Chemicals
Diphosphates Hypoxanthines Uric Acid Guanine Transferases
Authors & Affiliations
2 authors, click to expand affiliations / ORCID
McDonald J A
Kelley W N
Article Info
Journal
Science (New York, N.Y.)
Abbr.
Science
ISSN
0036-8075
Published
1971-02-19
Pages
689-91
Language
English
Region
United States
NLM ID
0404511
Subset
IM
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