Literature DB >> 2109145

Immunoquantification of the low abundance lysosomal enzyme N-acetylgalactosamine 4-sulphatase.

D A Brooks1, P A McCourt, G J Gibson, J J Hopwood.   

Abstract

The low abundance lysosomal enzyme N-acetylgalactosamine 4-sulphatase (4-sulphatase) has been quantified using a microimmunopurification step and a monoclonal-based ELISA detection system. The assay is similar in principle to a two-site ELISA but uses a single monoclonal antibody against one epitope to bind 4-sulphatase in two separate assay steps. The sensitivity of this assay is sufficient to allow the quantification of 4-sulphatase in human cultured skin fibroblasts derived from normal controls and patients deficient in 4-sulphatase activity (mucopolysaccharidosis type VI or Maroteaux-Lamy syndrome). The results obtained suggest a range of mucopolysaccharidosis type VI or 4-sulphatase deficient mutants, from those expressing little or no quantifiable 4-sulphatase protein to those examples with quantifiable levels of 4-sulphatase protein which is enzymically inactive. Phenotypic variability in patients with a 4-sulphatase deficiency may therefore be partially attributed to a range of protein expressions. The method should allow the determination of 4-sulphatase specific activity in mucopolysaccharidosis type VI patients.

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Year:  1990        PMID: 2109145     DOI: 10.1007/bf01799338

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


  18 in total

1.  Studies on glucosaminidase. 4. The fluorimetric assay of N-acetyl-beta-glucosaminidase.

Authors:  D H LEABACK; P G WALKER
Journal:  Biochem J       Date:  1961-01       Impact factor: 3.857

Review 2.  Mucopolysaccharidoses.

Authors:  J Muenzer
Journal:  Adv Pediatr       Date:  1986

3.  Cleavage of structural proteins during the assembly of the head of bacteriophage T4.

Authors:  U K Laemmli
Journal:  Nature       Date:  1970-08-15       Impact factor: 49.962

4.  The immunochemistry of solid-phase sandwich enzyme-linked immunosorbent assays.

Authors:  J E Butler; J H Peterman; M Suter; S E Dierks
Journal:  Fed Proc       Date:  1987-06

5.  Enzymic diagnosis of the genetic mucopolysaccharide storage disorders.

Authors:  H Kresse; K von Figura; U Klein; J Glössl; E Paschke; R Pohlmann
Journal:  Methods Enzymol       Date:  1982       Impact factor: 1.600

6.  alpha-L-Iduronidase deficiency in mucopolysaccharidosis type I against a radiolabelled sulfated disaccharide substrate derived from dermatan sulfate.

Authors:  V J Muller; J J Hopwood
Journal:  Clin Genet       Date:  1984-11       Impact factor: 4.438

7.  Human alpha-L-iduronidase. 1. Purification, monoclonal antibody production, native and subunit molecular mass.

Authors:  P R Clements; D A Brooks; G T Saccone; J J Hopwood
Journal:  Eur J Biochem       Date:  1985-10-01

8.  Human N-acetylgalactosamine-4-sulphate sulphatase. Purification, monoclonal antibody production and native and subunit Mr values.

Authors:  G J Gibson; G T Saccone; D A Brooks; P R Clements; J J Hopwood
Journal:  Biochem J       Date:  1987-12-15       Impact factor: 3.857

9.  Genetic heterogeneity in acid alpha-glucosidase deficiency.

Authors:  N G Beratis; G U LaBadie; K Hirschhorn
Journal:  Am J Hum Genet       Date:  1983-01       Impact factor: 11.025

10.  Clinical findings in 12 patients with MPS IV A (Morquio's disease). Further evidence for heterogeneity. Part I: Clinical and biochemical findings.

Authors:  J Nelson; D Broadhead; J Mossman
Journal:  Clin Genet       Date:  1988-02       Impact factor: 4.438

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  6 in total

1.  Enzyme replacement therapy in a feline model of Maroteaux-Lamy syndrome.

Authors:  A C Crawley; D A Brooks; V J Muller; B A Petersen; E L Isaac; J Bielicki; B M King; C D Boulter; A J Moore; N L Fazzalari; D S Anson; S Byers; J J Hopwood
Journal:  J Clin Invest       Date:  1996-04-15       Impact factor: 14.808

Review 2.  Review: the immunochemical analysis of enzyme from mucopolysaccharidoses patients.

Authors:  D A Brooks
Journal:  J Inherit Metab Dis       Date:  1993       Impact factor: 4.982

3.  A specific fluorogenic assay for N-acetylgalactosamine-4-sulphatase activity using immunoadsorption.

Authors:  D A Brooks; G J Gibson; P A McCourt; J J Hopwood
Journal:  J Inherit Metab Dis       Date:  1991       Impact factor: 4.982

4.  Analysis of N-acetylgalactosamine-4-sulfatase protein and kinetics in mucopolysaccharidosis type VI patients.

Authors:  D A Brooks; P A McCourt; G J Gibson; L J Ashton; M Shutter; J J Hopwood
Journal:  Am J Hum Genet       Date:  1991-04       Impact factor: 11.025

5.  Correction of human mucopolysaccharidosis type-VI fibroblasts with recombinant N-acetylgalactosamine-4-sulphatase.

Authors:  D S Anson; J A Taylor; J Bielicki; G S Harper; C Peters; G J Gibson; J J Hopwood
Journal:  Biochem J       Date:  1992-06-15       Impact factor: 3.857

6.  Two site-directed mutations abrogate enzyme activity but have different effects on the conformation and cellular content of the N-acetylgalactosamine 4-sulphatase protein.

Authors:  D A Brooks; D A Robertson; C Bindloss; T Litjens; D S Anson; C Peters; C P Morris; J J Hopwood
Journal:  Biochem J       Date:  1995-04-15       Impact factor: 3.857

  6 in total

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