Literature DB >> 6685450

Postmortem observations on beta-glucuronidase deficiency presenting as hydrops fetalis.

D Irani, H S Kim, H El-Hibri, R V Dutton, A Beaudet, D Armstrong.   

Abstract

This study reports a case of type VII mucopolysaccharidosis (beta-glucuronidase deficiency) presenting as lethal hydrops fetalis. Skin fibroblast cultures established postmortem revealed deficient beta-glucuronidase activity. Mucopolysaccharides were stored in various cells of the brain, heart, kidney, liver, and spleen. The stages of maturation of the bones, kidneys, and brain were discrepant, the brain being the least mature organ. A delay in central nervous system maturation may account for psychomotor retardation in some patients with this enzyme deficiency.

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Year:  1983        PMID: 6685450     DOI: 10.1002/ana.410140415

Source DB:  PubMed          Journal:  Ann Neurol        ISSN: 0364-5134            Impact factor:   10.422


  9 in total

1.  A murine model of mucopolysaccharidosis VII. Gross and microscopic findings in beta-glucuronidase-deficient mice.

Authors:  C Vogler; E H Birkenmeier; W S Sly; B Levy; C Pegors; J W Kyle; W G Beamer
Journal:  Am J Pathol       Date:  1990-01       Impact factor: 4.307

2.  Cardiovascular Histopathology of a 11-Year Old with Mucopolysaccharidosis VII Demonstrates Fibrosis, Macrophage Infiltration, and Arterial Luminal Stenosis.

Authors:  Valerie Lew; Louis Pena; Robert Edwards; Raymond Y Wang
Journal:  JIMD Rep       Date:  2017-07-13

3.  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

4.  Mucopolysaccharidosis type VII associated with hydrops fetalis: histopathological and ultrastructural features with genetic implications.

Authors:  A J Molyneux; E Blair; N Coleman; P Daish
Journal:  J Clin Pathol       Date:  1997-03       Impact factor: 3.411

5.  Foamy changes of placental cells in probable beta glucuronidase deficiency associated with hydrops fetalis.

Authors:  J Nelson; B Kenny; D O'Hara; A Harper; D Broadhead
Journal:  J Clin Pathol       Date:  1993-04       Impact factor: 3.411

6.  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

7.  Neuraminidase deficiency presenting as non-immune hydrops fetalis.

Authors:  M Beck; S W Bender; H L Reiter; W Otto; R Bässler; H Dancygier; J Gehler
Journal:  Eur J Pediatr       Date:  1984-12       Impact factor: 3.183

8.  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

9.  Neuromuscular degeneration and locomotor deficit in a Drosophila model of mucopolysaccharidosis VII is attenuated by treatment with resveratrol.

Authors:  Sudipta Bar; Mohit Prasad; Rupak Datta
Journal:  Dis Model Mech       Date:  2018-11-20       Impact factor: 5.758

  9 in total

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