Literature DB >> 2495719

Posttranslational processing of human lysosomal acid beta-glucosidase: a continuum of defects in Gaucher disease type 1 and type 2 fibroblasts.

J E Bergmann1, G A Grabowski.   

Abstract

The major processing steps in the maturation of the lysosomal hydrolase, acid beta-glucosidase, were examined in fibroblasts from normal individuals and from patients with types 1 and 2 Gaucher disease. In pulse-chase studies with normal fibroblasts, remodeling of N-linked oligosaccharides resulted in the temporal appearance of three molecular-weight forms of acid beta-glucosidase. An initial 64-kDa form, containing high mannose-type oligosaccharide side chains, was processed quantitatively, within 24 h, to a sialylated 69-kDa form. During the subsequent 96 h, some of the 69-kDa form is processed to 59 kDa. Glycosidase digestion studies revealed that the increase in the apparent molecular weight of the normal enzyme from 64 kDa to 69 kDa resulted primarily from the addition to sialic acid residues in the Golgi apparatus. The polypeptide backbone of both the 64-kDa and 69-kDa forms was 55.3 kDa. Processing of acid beta-glucosidase in fibroblasts from three of four type 1 (nonneuronopathic) Ashkenazi Jewish Gaucher disease patients was nearly normal. With fibroblasts from one Ashkenazi Jewish and three non-Jewish type 1 as well as from two type 2 (acute neuronopathic) Gaucher disease patients, only a 64-kDa form of acid beta-glucosidase was detected. Inefficient and incomplete processing to the 69-kDa form was found in one type 2 cell line (GM2627). These results indicate that no firm correlation exists between the type or degree of abnormal processing of acid beta-glucosidase in fibroblasts and the phenotype of Gaucher disease.

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Year:  1989        PMID: 2495719      PMCID: PMC1715631     

Source DB:  PubMed          Journal:  Am J Hum Genet        ISSN: 0002-9297            Impact factor:   11.025


  32 in total

1.  Primary structure of the vesicular stomatitis virus polymerase (L) gene: evidence for a high frequency of mutations.

Authors:  M Schubert; G G Harmison; E Meier
Journal:  J Virol       Date:  1984-08       Impact factor: 5.103

2.  Nucleotide sequences of the mRNA's encoding the vesicular stomatitis virus G and M proteins determined from cDNA clones containing the complete coding regions.

Authors:  J K Rose; C J Gallione
Journal:  J Virol       Date:  1981-08       Impact factor: 5.103

3.  Complementation analysis in Gaucher disease using single cell microassay techniques. Evidence for a single "Gaucher gene".

Authors:  R A Gravel; A Leung
Journal:  Hum Genet       Date:  1983       Impact factor: 4.132

4.  Immunological and catalytic quantitation of splenic glucocerebrosidase from the three clinical forms of Gaucher disease.

Authors:  P G Pentchev; B Neumeyer; L Svennerholm; C G Groth; R O Brady
Journal:  Am J Hum Genet       Date:  1983-07       Impact factor: 11.025

5.  Pathology of the Gaucher cell.

Authors:  J L Parkin; R D Brunning
Journal:  Prog Clin Biol Res       Date:  1982

6.  Mutations of glucocerebrosidase: discrimination of neurologic and non-neurologic phenotypes of Gaucher disease.

Authors:  E I Ginns; R O Brady; S Pirruccello; C Moore; S Sorrell; F S Furbish; G J Murray; J Tager; J A Barranger
Journal:  Proc Natl Acad Sci U S A       Date:  1982-09       Impact factor: 11.205

7.  Biochemical studies in a patient with subacute neuropathic Gaucher disease without visceral glucosylceramide storage.

Authors:  D A Wenger; S Roth; T Kudoh; W D Grover; S H Tucker; E M Kaye; M D Ullman
Journal:  Pediatr Res       Date:  1983-05       Impact factor: 3.756

8.  Biosynthesis and maturation of arylsulfatase B in normal and mutant cultured human fibroblasts.

Authors:  F Steckel; A Hasilik; K von Figura
Journal:  J Biol Chem       Date:  1983-12-10       Impact factor: 5.157

9.  Nucleotide sequences of the mRNA's encoding the vesicular stomatitis virus N and NS proteins.

Authors:  C J Gallione; J R Greene; L E Iverson; J K Rose
Journal:  J Virol       Date:  1981-08       Impact factor: 5.103

10.  Effect of microtubule assembly status on the intracellular processing and surface expression of an integral protein of the plasma membrane.

Authors:  A A Rogalski; J E Bergmann; S J Singer
Journal:  J Cell Biol       Date:  1984-09       Impact factor: 10.539

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  27 in total

1.  Histone deacetylase inhibitors prevent the degradation and restore the activity of glucocerebrosidase in Gaucher disease.

Authors:  Jie Lu; Chunzhang Yang; Masako Chen; Donald Y Ye; Russell R Lonser; Roscoe O Brady; Zhengping Zhuang
Journal:  Proc Natl Acad Sci U S A       Date:  2011-12-12       Impact factor: 11.205

2.  Pharmacological chaperones facilitate the post-ER transport of recombinant N370S mutant β-glucocerebrosidase in plant cells: evidence that N370S is a folding mutant.

Authors:  Gholamreza Babajani; Michael B Tropak; Don J Mahuran; Allison R Kermode
Journal:  Mol Genet Metab       Date:  2012-04-26       Impact factor: 4.797

3.  Production of active human glucocerebrosidase in seeds of Arabidopsis thaliana complex-glycan-deficient (cgl) plants.

Authors:  Xu He; Jason D Galpin; Michael B Tropak; Don Mahuran; Thomas Haselhorst; Mark von Itzstein; Daniel Kolarich; Nicolle H Packer; Yansong Miao; Liwen Jiang; Gregory A Grabowski; Lorne A Clarke; Allison R Kermode
Journal:  Glycobiology       Date:  2011-11-07       Impact factor: 4.313

4.  Recombinant human acid beta-glucosidase stored in tobacco seed is stable, active and taken up by human fibroblasts.

Authors:  Serena Reggi; Stefano Marchetti; Tamara Patti; Francesca De Amicis; Roberta Cariati; Bruno Bembi; Corrado Fogher
Journal:  Plant Mol Biol       Date:  2005-01       Impact factor: 4.076

5.  The iminosugar isofagomine increases the activity of N370S mutant acid beta-glucosidase in Gaucher fibroblasts by several mechanisms.

Authors:  Richard A Steet; Stephen Chung; Brandon Wustman; Allan Powe; Hung Do; Stuart A Kornfeld
Journal:  Proc Natl Acad Sci U S A       Date:  2006-08-31       Impact factor: 11.205

6.  Decreased glucocerebrosidase activity in Gaucher disease parallels quantitative enzyme loss due to abnormal interaction with TCP1 and c-Cbl.

Authors:  Jie Lu; Jeffrey Chiang; Rajiv R Iyer; Eli Thompson; Christine R Kaneski; David S Xu; Chunzhang Yang; Masako Chen; Richard J Hodes; Russell R Lonser; Roscoe O Brady; Zhengping Zhuang
Journal:  Proc Natl Acad Sci U S A       Date:  2010-11-22       Impact factor: 11.205

7.  The studies on substrate, product and inhibitor binding to a wild-type and neuronopathic form of human acid-beta-glucosidase.

Authors:  Igor Z Zubrzycki; Agnieszka Borcz; Magdalena Wiacek; Wojciech Hagner
Journal:  J Mol Model       Date:  2007-08-23       Impact factor: 1.810

8.  Participation of asparagine 370 and glutamine 235 in the catalysis by acid beta-glucosidase: the enzyme deficient in Gaucher disease.

Authors:  Benjamin Liou; Gregory A Grabowski
Journal:  Mol Genet Metab       Date:  2009-02-13       Impact factor: 4.797

Review 9.  Current issues in enzyme therapy for Gaucher disease.

Authors:  G A Grabowski
Journal:  Drugs       Date:  1996-08       Impact factor: 9.546

10.  Complex alleles of the acid beta-glucosidase gene in Gaucher disease.

Authors:  T Latham; G A Grabowski; B D Theophilus; F I Smith
Journal:  Am J Hum Genet       Date:  1990-07       Impact factor: 11.025

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