Literature DB >> 885539

Rapid prenatal diagnosis of HG-PRT deficiency using ultra-microchemical methods.

P Hösli, C H de Bruyn, F J Oerlemans, M Verjaal, R E Nobrega.   

Abstract

A previously developed simple ultramicromethod has been used for the rapid prenatal diagnosis of hypoxanthine-guanine phosphoribosyl transferase (HG-PRT) deficiency. The method is based on the incubation of small numbers of visually selected, lyophilized fibroblasts (in the present study five cells per incubation) with radioactive substrate in an end volume of 0.3 microliter. Fibroblasts derived from the amniotic fluid of a 15-week male fetus in a heterozygote for the X-linked Lesch-Nyhan syndrome showed a severe degree of HG-PRT deficiency. In total 50 fibroblasts were used. The diagnosis was confirmed upon termination of the pregnancy by the demonstration of HG-PRT deficiency in fetal erythrocytes and cultured skin fibroblasts.

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Year:  1977        PMID: 885539     DOI: 10.1007/bf00393582

Source DB:  PubMed          Journal:  Hum Genet        ISSN: 0340-6717            Impact factor:   4.132


  25 in total

1.  Quantitative radiochemical enzyme assays in single cells: purine phosphoribosyl transferase activities in cultured fibroblasts.

Authors:  C H de Bruyn; T L Oei; P Hösli
Journal:  Biochem Biophys Res Commun       Date:  1976-01-26       Impact factor: 3.575

2.  Prospects for prenatal diagnosis of cystic fibrosis: induction of biochemical abnormalities in fibroblasts from patients with cystic fibrosis by a urinary glycoprotein.

Authors:  P Hösli; R P Erickson; E Vogt
Journal:  Biochem Biophys Res Commun       Date:  1976-11-22       Impact factor: 3.575

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

4.  Development of micro HG-PRT activity assay: preliminary complementation studies with Lesch-Nyhan cell strains.

Authors:  P Hösli; C H de BPRUYN; T L Oei
Journal:  Adv Exp Med Biol       Date:  1974       Impact factor: 2.622

5.  Studies on hair roots for carrier detection in hypoxanthine-quanine phosphoribosyl transferase deficiency.

Authors:  C H de Bruyn; T L Oei; B G ter Haar
Journal:  Clin Genet       Date:  1974       Impact factor: 4.438

6.  Clinical features of patients with the "partial" deficiency of the X-linked uricaciduria enzyme.

Authors:  M L Greene
Journal:  Arch Intern Med       Date:  1972-08

7.  Lesch-Nyhan syndrome: preventive control by prenatal diagnosis.

Authors:  J A Boyle; K O Raivio; K H Astrin; J D Schulman; M L Graf; J E Seegmiller; C B Jacobsen
Journal:  Science       Date:  1970-08-14       Impact factor: 47.728

8.  Rarity of X-linked partial hypoxanthine-guanine phosphoribosyltransferase deficiency in a large gouty population.

Authors:  T F Yü; M E Balis; T A Krenitsky; J Dancis; D N Silvers; G B Elion; A B Gutman
Journal:  Ann Intern Med       Date:  1972-02       Impact factor: 25.391

9.  Lesch-Nyhan mutation: prenatal detection with amniotic fluid cells.

Authors:  R DeMars; G Sarto; J S Felix; P Benke
Journal:  Science       Date:  1969-06-13       Impact factor: 47.728

10.  Hypoxanthine-guanine phosphoribosyltransferase deficiency: activity in normal, mutant, and heterozygote-cultured human skin fibroblasts.

Authors:  W Y Fujimoto; J E Seegmiller
Journal:  Proc Natl Acad Sci U S A       Date:  1970-03       Impact factor: 11.205

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  1 in total

1.  An ultramicrochemical test for mycoplasmal contamination of cultured cells.

Authors:  M P Uitendaal; C H DeBruyn; M Hatanaka; P Hösli
Journal:  In Vitro       Date:  1979-02
  1 in total

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