Literature DB >> 113502

[Mucopolysaccharidesis Type VII resulting from beta-glucuronidase deficiency. Report of one family].

P Guibaud, I Maire, R Goddon, G Teyssier, M T Zabot, G Mandon.   

Abstract

The patient is a north african female, fourth born child in a family with consanguinity. Facial dysmorphia, clubfeet, swollen extremities and heel borne ponctuate calcifications are observed soon after birth. beta glucuronidase activity is very low in serum, leukocytes and skin fibroblasts. At 18 months, gorwth and psychomotor development are normal. Flat facies and dorsolumbar cyphosis are striking. There is no clinical sign of storage disease, neither ocular or cytologic (blood, bone-marrow) abnormalities. Squeletal abnormalities are predominant on cervical and lumbar column and pelvis. Urinary excretion of chondroitin 4 sulfate and chondroitin 6 sulfate is increased. A 4 year old sister is affected. Facial dysmorphia, mild squeletal abnormalities are observed. Again, growth is normal and there is no symptom of storage disease. Enzymic expression of parent heterozygotism is marked in serum studies, but less marked in leukocytes and fibroblasts. The last two children are heterozygotes. At the time of a 5th pregnancy, enzymic activity studies of amniotic fluid and amniotic cells have shown that the foetus was an inaffected female. This child was normal at birth.

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Year:  1979        PMID: 113502

Source DB:  PubMed          Journal:  J Genet Hum        ISSN: 0021-7743


  3 in total

1.  Phenotypic expression in mucopolysaccharidosis VII.

Authors:  P L Bernsen; R A Wevers; F J Gabreëls; K J Lamers; A E Sonnen; J H Stekhoven
Journal:  J Neurol Neurosurg Psychiatry       Date:  1987-06       Impact factor: 10.154

2.  Molecular analysis of patients with beta-glucuronidase deficiency presenting as hydrops fetalis or as early mucopolysaccharidosis VII.

Authors:  R Vervoort; M R Islam; W S Sly; M T Zabot; W J Kleijer; A Chabas; A Fensom; E P Young; I Liebaers; W Lissens
Journal:  Am J Hum Genet       Date:  1996-03       Impact factor: 11.025

3.  Clinical course of sly syndrome (mucopolysaccharidosis type VII).

Authors:  Adriana M Montaño; Ngu Lock-Hock; Robert D Steiner; Brett H Graham; Marina Szlago; Robert Greenstein; Mercedes Pineda; Antonio Gonzalez-Meneses; Mahmut Çoker; Dennis Bartholomew; Mark S Sands; Raymond Wang; Roberto Giugliani; Alfons Macaya; Gregory Pastores; Anastasia K Ketko; Fatih Ezgü; Akemi Tanaka; Laila Arash; Michael Beck; Rena E Falk; Kaustuv Bhattacharya; José Franco; Klane K White; Grant A Mitchell; Loreta Cimbalistiene; Max Holtz; William S Sly
Journal:  J Med Genet       Date:  2016-02-23       Impact factor: 6.318

  3 in total

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