Literature DB >> 144057

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

R A Pfeiffer, H Kresse, N Bäumer, E Sattinger.   

Abstract

beta-glucuronidase deficiency in fibroblasts, leucocytes and in serum and increased urinary excretion of mucopolysaccharides were found in a girl, now 13 years old, who exhibits some features of a mucopolysaccharidosis such as moderate mental deficiency, craniofacial dysmorphism, a short neck, protruding sternum, vertebral deformities and corneal clouding. Coarse granulations were found in her leucocytes. The liver and spleen are not enlarged and there is no gingival hyperplasia. Additional features, hitherto undescribed, are hydronephrosis and defective ossification of the medial carpal and tarsal bones. Low enzyme activity in the parents and a normal brother suggests heterozygosity.

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Year:  1977        PMID: 144057     DOI: 10.1007/bf00442197

Source DB:  PubMed          Journal:  Eur J Pediatr        ISSN: 0340-6199            Impact factor:   3.183


  14 in total

1.  Uridine diphospho-N-acetylgalactosamine-4-sulfate sulfohydrolase activity of human arylsulfatase B and its deficiency in the Maroteaux-Lamy syndrome.

Authors:  A L Fluharty; R L Stevens; D Fung; S Peak; H Kihara
Journal:  Biochem Biophys Res Commun       Date:  1975-01-02       Impact factor: 3.575

2.  Allelism, non-allelism, and genetic compounds among the mucopolysaccharidoses.

Authors:  V A McKusick; R R Howell; I E Hussels; E F Neufeld; R E Stevenson
Journal:  Lancet       Date:  1972-05-06       Impact factor: 79.321

3.  Familial osteolysis of the carpal and tarsal bones.

Authors:  J Gluck; J J Miller
Journal:  J Pediatr       Date:  1972-09       Impact factor: 4.406

4.  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

5.  Beta glucuronidase deficiency: report of clinical, radiologic, and biochemical features of a new mucopolysaccharidosis.

Authors:  W S Sly; B A Quinton; W H McAlister; D L Rimoin
Journal:  J Pediatr       Date:  1973-02       Impact factor: 4.406

6.  Hereditary osteolysis. A clinical, radiological and chemical study.

Authors:  E Kohler; D Babbitt; B Huizenga; T A Good
Journal:  Radiology       Date:  1973-07       Impact factor: 11.105

7.  Essential osteolysis with nephropathy. A review of the literature and case report of an unusual syndrome.

Authors:  J S Torg; H H Steel
Journal:  J Bone Joint Surg Am       Date:  1968-12       Impact factor: 5.284

8.  Alpha-L-iduronidase activity in cultured skin fibroblasts and amniotic fluid cells.

Authors:  C W Hall; E F Neufeld
Journal:  Arch Biochem Biophys       Date:  1973-10       Impact factor: 4.013

9.  Variation in the phenotypic expression of beta-glucuronidase deficiency.

Authors:  A L Beaudet; N M DiFerrante; G D Ferry; B L Nichols; C E Mullins
Journal:  J Pediatr       Date:  1975-03       Impact factor: 4.406

10.  Beta-glucuronidase deficiency (mucopolysaccharidosis type VII).

Authors:  A L Beaudet; N M Ferrante; G D Ferry; B L Nichols
Journal:  Birth Defects Orig Artic Ser       Date:  1974
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  8 in total

1.  A new case of mucopolysaccharidosis VII presenting as non immune hydrops fetalis.

Authors:  Elisabeth Walter-Nicolet; Thameur Rakza; Laurent Storme; Catherine Vaillant; Eric Magnenant; Robin Cremer; Caroline Thumerelle; Dries Dobbelaere
Journal:  Eur J Pediatr       Date:  2003-05-14       Impact factor: 3.183

2.  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

3.  Elevation of glycosaminoglycans in the amniotic fluid of a fetus with mucopolysaccharidosis VII.

Authors:  Francyne Kubaski; Ana Carolina Brusius-Facchin; Robert W Mason; Pravin Patel; Maira G Burin; Kristiane Michelin-Tirelli; Rejane Gus Kessler; Fernanda Bender; Sandra Leistner-Segal; Carolina A Moreno; Denise P Cavalcanti; Roberto Giugliani; Shunji Tomatsu
Journal:  Prenat Diagn       Date:  2017-03-12       Impact factor: 3.050

4.  Behavioral consequences of bone marrow transplantation in the treatment of murine mucopolysaccharidosis type VII.

Authors:  L Bastedo; M S Sands; D T Lambert; M A Pisa; E Birkenmeier; P L Chang
Journal:  J Clin Invest       Date:  1994-09       Impact factor: 14.808

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

Authors:  I Maire; G Mandon; M T Zabot; M Mathieu; P Guibaud
Journal:  J Inherit Metab Dis       Date:  1980       Impact factor: 4.982

6.  Presentation of mucopolysaccharidosis VII (beta-glucuronidase deficiency) in infancy.

Authors:  H E Hoyme; K L Jones; M C Higginbottom; J S O'Brien
Journal:  J Med Genet       Date:  1981-06       Impact factor: 6.318

7.  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

Review 8.  Mucopolysaccharidoses: early diagnostic signs in infants and children.

Authors:  Cinzia Galimberti; Annalisa Madeo; Maja Di Rocco; Agata Fiumara
Journal:  Ital J Pediatr       Date:  2018-11-16       Impact factor: 2.638

  8 in total

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