Literature DB >> 5676523

Adenine phosphoribosyltransferase deficiency: a previously undescribed genetic defect in man.

W N Kelley, R I Levy, F M Rosenbloom, J F Henderson, J E Seegmiller.   

Abstract

A deficiency of adenine phosphoribosyltransferase (A-PRTase) is described in four members in three generations of one family. A-PRTase is coded by an autosome and the mutants described in this report are heterozygotes for this enzyme defect. The level of enzyme activity in these heterozygotes was inappropriately low, ranging from 21 to 37% of normal rather than the expected 50% of normal. Examination of various physical and chemical properties of the A-PRTase obtained from the mutant heterozygotes failed to reveal differences from the normal enzyme. These patients have no discernable abnormality in uric acid production despite the finding that patients with a deficiency of a closely related enzyme, hypoxanthine-guanine phosphoribosyltransferase, invariably produce excessive quantities of uric acid. A relationship of the A-PRTase deficiency to the disturbance in lipoprotein metabolism observed in the propositus has not been firmly established. Possible manifestations of the homozygous form of this enzyme deficiency will require identification of such individuals in the future.

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Year:  1968        PMID: 5676523      PMCID: PMC297392          DOI: 10.1172/JCI105913

Source DB:  PubMed          Journal:  J Clin Invest        ISSN: 0021-9738            Impact factor:   14.808


  14 in total

1.  HYPERTRIGLYCERIDEMIA IN GOUT.

Authors:  E B FELDMAN; S L WALLACE
Journal:  Circulation       Date:  1964-04       Impact factor: 29.690

2.  Enzymatic deficiencies of purine nucleotide synthesis in the human erythrocyte.

Authors:  B A LOWY; M K WILLIAMS; I M LONDON
Journal:  J Biol Chem       Date:  1962-05       Impact factor: 5.157

3.  A specific enzyme defect in gout associated with overproduction of uric acid.

Authors:  W N Kelley; F M Rosenbloom; J F Henderson; J E Seegmiller
Journal:  Proc Natl Acad Sci U S A       Date:  1967-06       Impact factor: 11.205

Review 4.  Fat transport in lipoproteins--an integrated approach to mechanisms and disorders.

Authors:  D S Fredrickson; R I Levy; R S Lees
Journal:  N Engl J Med       Date:  1967-01-26       Impact factor: 91.245

5.  Studies on the control of pyrimidine biosynthesis in human diploid cell strains, I. Effect of 6-azauridine on cellular phenotype.

Authors:  L Pinsky; R S Krooth
Journal:  Proc Natl Acad Sci U S A       Date:  1967-04       Impact factor: 11.205

6.  Blood-uric-acid levels in familial hypercholesterolaemia.

Authors:  J Jensen; D H Blankenhorn; V Kornerup
Journal:  Lancet       Date:  1966-02-05       Impact factor: 79.321

7.  Total inhibition of hepatic beta-lipoprotein production in the rat by orotic acid.

Authors:  H G Windmueller; R I Levy
Journal:  J Biol Chem       Date:  1967-05-10       Impact factor: 5.157

8.  Gout, hyperlipidemia, and diabetes interrelationships.

Authors:  D Berkowitz
Journal:  JAMA       Date:  1966-07-11       Impact factor: 56.272

9.  X-linked recessive inheritance of a syndrome of mental retardation with hyperuricemia.

Authors:  S L Shapiro; G L Sheppard; F E Dreifuss; D S Newcombe
Journal:  Proc Soc Exp Biol Med       Date:  1966-06

10.  Enzyme defect associated with a sex-linked human neurological disorder and excessive purine synthesis.

Authors:  J E Seegmiller; F M Rosenbloom; W N Kelley
Journal:  Science       Date:  1967-03-31       Impact factor: 47.728

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  35 in total

Review 1.  Heberden oration 1979: human aberrations of purine metabolism and their significance for rheumatology.

Authors:  J E Seegmiller
Journal:  Ann Rheum Dis       Date:  1980-04       Impact factor: 19.103

2.  An infant with nephrolithiasis and renal failure: Answers.

Authors:  Kazuyuki Ueno; Masaki Shimizu; Tatsuya Kubo; Noboru Igarashi; Kiyoshi Hatasaki
Journal:  Pediatr Nephrol       Date:  2015-04-11       Impact factor: 3.714

Review 3.  Diagnosis and management of non-calcium-containing stones in the pediatric population.

Authors:  Saritha Ranabothu; Ari P Bernstein; Beth A Drzewiecki
Journal:  Int Urol Nephrol       Date:  2018-05-30       Impact factor: 2.370

4.  Contributions of Lesch-Nyhan syndrome to the understanding of purine metabolism.

Authors:  J E Seegmiller
Journal:  J Inherit Metab Dis       Date:  1989       Impact factor: 4.982

Review 5.  PRPS1 mutations: four distinct syndromes and potential treatment.

Authors:  Arjan P M de Brouwer; Hans van Bokhoven; Sander B Nabuurs; Willem Frans Arts; John Christodoulou; John Duley
Journal:  Am J Hum Genet       Date:  2010-04-09       Impact factor: 11.025

6.  Rapid method for the diagnosis of partial adenine phosphoribosyltransferase deficiencies causing 2,8-dihydroxyadenine urolithiasis.

Authors:  F Takeuchi; K Matsuta; T Miyamoto; S Enomoto; S Fujimori; I Akaoka; N Kamatani; K Nishioka
Journal:  Hum Genet       Date:  1985       Impact factor: 4.132

7.  Selection of human cells having two different types of mutations in individual cells (genetic/artificial mutants). Application to the diagnosis of the heterozygous state for a type of adenine phosphoribosyltransferase deficiency.

Authors:  N Kamatani; S Kuroshima; C Terai; K Kawai; K Mikanagi; K Nishioka
Journal:  Hum Genet       Date:  1987-06       Impact factor: 4.132

8.  Human adenine phosphoribosyltransferase: characterization from subjects with a deficiency of enzyme activity.

Authors:  T E O'Toole; J M Wilson; M H Gault; W N Kelley
Journal:  Biochem Genet       Date:  1983-12       Impact factor: 1.890

9.  Human adenine phosphoribosyltransferase. Identification of allelic mutations at the nucleotide level as a cause of complete deficiency of the enzyme.

Authors:  Y Hidaka; T D Palella; T E O'Toole; S A Tarlé; W N Kelley
Journal:  J Clin Invest       Date:  1987-11       Impact factor: 14.808

10.  Comparison between various strategies for the disease-gene mapping using linkage disequilibrium analyses: studies on adenine phosphoribosyltransferase deficiency used as an example.

Authors:  Shin-Ichi Kuno; Atsuo Taniguchi; Akira Saito; Sanae Tsuchida-Otsuka; Naoyuki Kamatani
Journal:  J Hum Genet       Date:  2004-07-28       Impact factor: 3.172

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