Literature DB >> 1348358

Multiple sulfatase deficiency: catalytically inactive sulfatases are expressed from retrovirally introduced sulfatase cDNAs.

W Rommerskirch1, K von Figura.   

Abstract

Multiple sulfatase deficiency (MSD) is an inherited lysosomal storage disease characterized by the deficiency of at least seven sulfatases. The basic defect in MSD is thought to be in a post-translational modification common to all sulfatases. In accordance with this concept, RNAs of normal size and amount were detected in MSD fibroblasts for three sulfatases tested. cDNAs encoding arylsulfatase A, arylsulfatase B, or steroid sulfatase were introduced into MSD fibroblasts and fibroblasts with a single sulfatase deficiency by retroviral gene transfer. Infected fibroblasts overexpressed the respective sulfatase polypeptides. While in single-sulfatase-deficiency fibroblasts a concomitant increase of sulfatase activities was observed, MSD fibroblasts expressed sulfatase polypeptides with a severely diminished catalytic activity. From these results we conclude that the mutation in MSD severely decreases the capacity of a co- or post-translational process that renders sulfatases enzymatically active or prevents their premature inactivation.

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Year:  1992        PMID: 1348358      PMCID: PMC48701          DOI: 10.1073/pnas.89.7.2561

Source DB:  PubMed          Journal:  Proc Natl Acad Sci U S A        ISSN: 0027-8424            Impact factor:   11.205


  28 in total

1.  Structure of the arylsulfatase A gene.

Authors:  J Kreysing; K von Figura; V Gieselmann
Journal:  Eur J Biochem       Date:  1990-08-17

2.  A universal retroviral vector for efficient constitutive expression of exogenous genes.

Authors:  C A Boulter; E F Wagner
Journal:  Nucleic Acids Res       Date:  1987-09-11       Impact factor: 16.971

3.  Arylsulfatase A pseudodeficiency: loss of a polyadenylylation signal and N-glycosylation site.

Authors:  V Gieselmann; A Polten; J Kreysing; K von Figura
Journal:  Proc Natl Acad Sci U S A       Date:  1989-12       Impact factor: 11.205

4.  Restoration of arylsulphatase A activity in human-metachromatic-leucodystrophy fibroblasts via retroviral-vector-mediated gene transfer.

Authors:  W Rommerskirch; A L Fluharty; C Peters; K von Figura; V Gieselmann
Journal:  Biochem J       Date:  1991-12-01       Impact factor: 3.857

5.  Synthesis and stability of arylsulfatase A and B in fibroblasts from multiple sulfatase deficiency.

Authors:  F Steckel; A Hasilik; K von Figura
Journal:  Eur J Biochem       Date:  1985-08-15

6.  Purification and properties of arylsulfatase. A from human urine.

Authors:  R L Stevens; A L Fluharty; M H Skokut; H Kihara
Journal:  J Biol Chem       Date:  1975-04-10       Impact factor: 5.157

7.  Complementation of multiple sulfatase deficiency in somatic cell hybrids.

Authors:  K Fedde; A L Horwitz
Journal:  Am J Hum Genet       Date:  1984-05       Impact factor: 11.025

8.  Arysulfatase A modulation with pH in multiple sulfatase deficiency disorder fibroblasts.

Authors:  A L Fluharty; R L Stevens; S D de la Flor; L J Shapiro; H Kihara
Journal:  Am J Hum Genet       Date:  1979-09       Impact factor: 11.025

9.  Phylogenetic conservation of arylsulfatases. cDNA cloning and expression of human arylsulfatase B.

Authors:  C Peters; B Schmidt; W Rommerskirch; K Rupp; M Zühlsdorf; M Vingron; H E Meyer; R Pohlmann; K von Figura
Journal:  J Biol Chem       Date:  1990-02-25       Impact factor: 5.157

10.  Steroid sulfatase. Biosynthesis and processing in normal and mutant fibroblasts.

Authors:  J Conary; A Nauerth; G Burns; A Hasilik; K von Figura
Journal:  Eur J Biochem       Date:  1986-07-01
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  10 in total

1.  Conversion of cysteine to formylglycine: a protein modification in the endoplasmic reticulum.

Authors:  T Dierks; B Schmidt; K von Figura
Journal:  Proc Natl Acad Sci U S A       Date:  1997-10-28       Impact factor: 11.205

2.  SUMF1 mutations affecting stability and activity of formylglycine generating enzyme predict clinical outcome in multiple sulfatase deficiency.

Authors:  Lars Schlotawa; Eva Charlotte Ennemann; Karthikeyan Radhakrishnan; Bernhard Schmidt; Anupam Chakrapani; Hans-Jürgen Christen; Hugo Moser; Beat Steinmann; Thomas Dierks; Jutta Gärtner
Journal:  Eur J Hum Genet       Date:  2011-01-12       Impact factor: 4.246

Review 3.  Pathology and current treatment of neurodegenerative sphingolipidoses.

Authors:  Matthias Eckhardt
Journal:  Neuromolecular Med       Date:  2010-08-22       Impact factor: 3.843

4.  Characterization of iduronate sulphatase mutants affecting N-glycosylation sites and the cysteine-84 residue.

Authors:  G Millat; R Froissart; I Maire; D Bozon
Journal:  Biochem J       Date:  1997-08-15       Impact factor: 3.857

5.  Immortalization and characterization of a cell line exhibiting a severe multiple sulphatase deficiency phenotype.

Authors:  K Nelson; J Bielicki; D S Anson
Journal:  Biochem J       Date:  1997-08-15       Impact factor: 3.857

6.  Rapid degradation of an active formylglycine generating enzyme variant leads to a late infantile severe form of multiple sulfatase deficiency.

Authors:  Lars Schlotawa; Karthikeyan Radhakrishnan; Matthias Baumgartner; Regula Schmid; Bernhard Schmidt; Thomas Dierks; Jutta Gärtner
Journal:  Eur J Hum Genet       Date:  2013-01-16       Impact factor: 4.246

7.  Overexpression of inactive arylsulphatase mutants and in vitro activation by light-dependent oxidation with vanadate.

Authors:  Terri M Christianson; Chris M Starr; Todd C Zankel
Journal:  Biochem J       Date:  2004-09-01       Impact factor: 3.857

8.  Overexpression of N-acetylgalactosamine-4-sulphatase induces a multiple sulphatase deficiency in mucopolysaccharidosis-type-VI fibroblasts.

Authors:  D S Anson; V Muller; J Bielicki; G S Harper; J J Hopwood
Journal:  Biochem J       Date:  1993-09-15       Impact factor: 3.857

Review 9.  Formylglycine, a post-translationally generated residue with unique catalytic capabilities and biotechnology applications.

Authors:  Mason J Appel; Carolyn R Bertozzi
Journal:  ACS Chem Biol       Date:  2015-01-16       Impact factor: 5.100

Review 10.  Multiple Sulfatase Deficiency: A Disease Comprising Mucopolysaccharidosis, Sphingolipidosis, and More Caused by a Defect in Posttranslational Modification.

Authors:  Lars Schlotawa; Laura A Adang; Karthikeyan Radhakrishnan; Rebecca C Ahrens-Nicklas
Journal:  Int J Mol Sci       Date:  2020-05-13       Impact factor: 5.923

  10 in total

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