Literature DB >> 6796758

Beta-glucuronidase deficiency: enzyme studies in an affected family and prenatal diagnosis.

I Maire, G Mandon, M T Zabot, M Mathieu, P Guibaud.   

Abstract

A beta-glucuronidase deficiency found in serum, leukocytes and fibroblasts and an increased [35S]sulphate incorporation in fibroblasts led us to diagnose two cases of type VII mucopolysaccharidosis in one family. In spite of the wide distribution of activities in serum from controls, decreased beta-glucuronidase activity allowed us to demonstrate the heterozygous status of the parents and two other children. Following these studies, and antenatal diagnosis was performed when the mother was pregnant again; amniotic fluid and cultured amniotic cells were used for enzyme activity determination. A heterozygous fetus was suspected and confirmed after birth. The reliability of various biological materials for enzymatic diagnosis and existence of genetic variants in the normal population are discussed.

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Year:  1980        PMID: 6796758     DOI: 10.1007/BF01799071

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


  29 in total

1.  Human beta-glucuronidase. II. Fate of infused human placental beta-glucuronidase in the rat.

Authors:  D Achord; F Brot; A Gonzalez-Noriega; W Sly; P Stahl
Journal:  Pediatr Res       Date:  1977-07       Impact factor: 3.756

2.  Assignment of the structural gene for human beta glucuronidase to chromosome 7 and tetrameric association of subunits in the enzyme molecule.

Authors:  C J Chern; C M Croce
Journal:  Am J Hum Genet       Date:  1976-07       Impact factor: 11.025

3.  Different forms of rat beta-glucuronidase with rapid and slow clearance following intravenous injection: selective serum enhancement of slow clearance forms by organophosphate compounds.

Authors:  P Stahl; B Mandell; J S Rodman; P Schlesinger; S Lang
Journal:  Arch Biochem Biophys       Date:  1975-10       Impact factor: 4.013

4.  The abnormalities of lysosomal enzymes in mucopolysacc- haridoses.

Authors:  F Van Hoof; H G Hers
Journal:  Eur J Biochem       Date:  1968-12

5.  A -glucuronidase deficiency mucopolysaccharidosis: studies in cultured fibroblasts.

Authors:  C W Hall; M Cantz; E F Neufeld
Journal:  Arch Biochem Biophys       Date:  1973-03       Impact factor: 4.013

6.  Enzyme therapy. V. In vivo fate of erythrocyte-entrapped beta-glucuronidase in beta-glucuronidase-deficient mice.

Authors:  S R Thorpe; M B Fiddler; R J Desnick
Journal:  Pediatr Res       Date:  1975-12       Impact factor: 3.756

7.  Assignment of a structural gene for beta-glucuronidase to human chromosome C7.

Authors:  K H Grzeschik
Journal:  Somatic Cell Genet       Date:  1976-09

8.  Enzyme therapy. VI: Comparative in vivo fates and effects on lysosomal integrity of enzyme entrapped in negatively and positively charged liposomes.

Authors:  L D Steger; R J Desnick
Journal:  Biochim Biophys Acta       Date:  1977-02-04

9.  Beta-glucuronidase deficiency in a girl with unusual clinical features.

Authors:  R A Pfeiffer; H Kresse; N Bäumer; E Sattinger
Journal:  Eur J Pediatr       Date:  1977-10-12       Impact factor: 3.183

10.  Human beta-glucuronidase: assignment of the structural gene to chromosome 7 using somatic cell hybrids.

Authors:  P A Lalley; J A Brown; R L Eddy; L L Haley; M G Byers; A P Goggin; T B Shows
Journal:  Biochem Genet       Date:  1977-04       Impact factor: 1.890

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  2 in total

1.  Low beta-glucuronidase activity in a healthy member of a family with mucopolysaccharidosis VII.

Authors:  A Chabas; M L Giros; A Guardiola
Journal:  J Inherit Metab Dis       Date:  1991       Impact factor: 4.982

Review 2.  Comparative biology of test species.

Authors:  E J Calabrese
Journal:  Environ Health Perspect       Date:  1988-04       Impact factor: 9.031

  2 in total

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