Literature DB >> 6456376

The mucopolysaccharidoses: biochemistry and clinical symptoms.

H Kresse, M Cantz, K von Figura, J Glössl, E Paschke.   

Abstract

The mucopolysaccharidoses are a group of genetic diseases which are characterized by an excessive intralysosomal accumulation of partially degraded mucopolysaccharides. This storage is caused by the inactivity of one of eleven enzymes that are required for the degradation of the different types of mucopolysaccharides. There is a rough correlation between phenotype and chemical nature of the storage material. Similar clinical pictures, however, may be caused by an inactivity of different enzymes. Conversely, different clinical expressions of the defect of a single enzyme may be attributed to allelic mutations. The recent development of specific assay procedures for the respective enzymes allows 1. an early genotype-specific diagnosis of affected patients, 2. prenatal diagnosis of the metabolic defect in families at risk, 3. to prognosticate the course of the disease at least in some instances, and 4. genetic counseling for members of affected families. At present, there is no specific therapy. Attempts of enzyme replacement therapy are still at an experimental stage.

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Year:  1981        PMID: 6456376     DOI: 10.1007/bf01721920

Source DB:  PubMed          Journal:  Klin Wochenschr        ISSN: 0023-2173


  53 in total

1.  Iduronate sulfatase analysis of hair roots for identification of Hunter syndrome heterozygotes.

Authors:  T Yutaka; A L Fluharty; R L Stevens; H Kihara
Journal:  Am J Hum Genet       Date:  1978-11       Impact factor: 11.025

2.  A Rare Disease in Two Brothers.

Authors:  C Hunter
Journal:  Proc R Soc Med       Date:  1917

3.  Hurler/Scheie genetic compound (mucopolysaccharidosis IH/IS) in Japanese brothers.

Authors:  T Kajii; I Matsuda; T Osawa; H Katsunuma; T Ichida
Journal:  Clin Genet       Date:  1974       Impact factor: 4.438

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.  Enzymatic phosphorylation of lysosomal enzymes in the presence of UDP-N-acetylglucosamine. Absence of the activity in I-cell fibroblasts.

Authors:  A Hasilik; A Waheed; K von Figura
Journal:  Biochem Biophys Res Commun       Date:  1981-02-12       Impact factor: 3.575

6.  Sanfilippo A syndrome: sulfamidase deficiency in cultured skin fibroblasts and liver.

Authors:  R Matalon; A Dorfman
Journal:  J Clin Invest       Date:  1974-10       Impact factor: 14.808

7.  Sanfilippo syndrome type C: deficiency of acetyl-CoA:alpha-glucosaminide N-acetyltransferase in skin fibroblasts.

Authors:  U Klein; H Kresse; K von Figura
Journal:  Proc Natl Acad Sci U S A       Date:  1978-10       Impact factor: 11.205

8.  The desulphation of hexosamine sulphates by arylsulphatase B.

Authors:  A A Farooqui
Journal:  Experientia       Date:  1976-10-15

9.  Multiple deficiency of mucopolysaccharide sulfatases in mucosulfatidosis.

Authors:  R Basner; K von Figura; J Glössl; U Klein; H Kresse; W Mlekusch
Journal:  Pediatr Res       Date:  1979-12       Impact factor: 3.756

10.  Arylsulfatase B deficiency in Maroteaux-Lamy syndrome cultured fibroblasts.

Authors:  A L Fluharty; R L Stevens; D L Sanders; H Kihara
Journal:  Biochem Biophys Res Commun       Date:  1974-07-24       Impact factor: 3.575

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  4 in total

Review 1.  Proteoglycans in health and disease: structures and functions.

Authors:  A R Poole
Journal:  Biochem J       Date:  1986-05-15       Impact factor: 3.857

2.  Urinary screening for disorders of heteroglycan metabolism. Results of 10 years experience with a comprehensive system.

Authors:  A C Sewell
Journal:  Klin Wochenschr       Date:  1988-01-15

3.  Lysosomal storage of sulfated glycosaminoglycans in renal interstitial cells of rats treated with tilorone.

Authors:  R Lullmann-Rauch
Journal:  Cell Tissue Res       Date:  1987-12       Impact factor: 5.249

Review 4.  Heparan Sulfate, Mucopolysaccharidosis IIIB and Sulfur Metabolism Disorders.

Authors:  Marta Kaczor-Kamińska; Kamil Kamiński; Maria Wróbel
Journal:  Antioxidants (Basel)       Date:  2022-03-30
  4 in total

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