Literature DB >> 1779649

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

A Chabas1, M L Giros, A Guardiola.   

Abstract

A phenotypically normal mother of a mucopolysaccharidosis VII child, is reported with an unusually low beta-glucuronidase activity. Low enzyme activity was systemic (6-10% of controls) and residual beta-glucuronidase in leukocytes had an apparently normal Km value. [35S]sulphate incorporation and chase assays in fibroblasts gave values similar to control cells. A normal excretion pattern of glycosaminoglycan was found in this woman's urine. Low enzymatic activity can be related to a non-pathological 'pseudodeficiency' allele for beta-glucuronidase; this woman appears to be an apparent compound heterozygote for this allele and mucopolysaccharidosis VII. Her next pregnancy was monitored by chorionic villus sampling and a heterozygous fetus was suspected. These studies stress the need for complete enzyme investigations of obligate carriers for mucopolysaccharidoses in order to prevent difficulties at prenatal analysis.

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Year:  1991        PMID: 1779649     DOI: 10.1007/bf01800472

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


  26 in total

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5.  Apparent deficiency of hexosaminidase A in healthy members of a family with Tay-Sachs disease.

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6.  Absence of hexosaminidase A and B in a normal adult.

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10.  Prenatal diagnosis of mucopolysaccharidosis I: A special difficulty arising from an unusually low enzyme activity in mother's cells.

Authors:  R Gatti; C Borrone; M Filocamo; N Pannone; P Di Natale
Journal:  Prenat Diagn       Date:  1985 Mar-Apr       Impact factor: 3.050

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  7 in total

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7.  Lysosomal and network alterations in human mucopolysaccharidosis type VII iPSC-derived neurons.

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  7 in total

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