Literature DB >> 6957882

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

E I Ginns, R O Brady, S Pirruccello, C Moore, S Sorrell, F S Furbish, G J Murray, J Tager, J A Barranger.   

Abstract

Multiple molecular forms of beta-glucocerebrosidase that permit discrimination between neurologic and non-neurologic phenotypes of Gaucher disease have been identified radioimmunologically in fibroblasts and human brain tissue. In normal human fibroblasts these forms have been shown by NaDodSO4/polyacrylamide gel electrophoresis to have apparent Mr of 63,000 (form A1), 61,000 (form A2), and 56,000 (form B). The Mr 63,000 form may be a precursor of the Mr 56,000 form. Non-neurologic Gaucher disease (type 1) fibroblasts and normal brain tissue are characteristic in that they contain only one major immunoreactive protein, the Mr 56,000 form. In contrast, fibroblast extracts and brain tissue from neurologic Gaucher disease phenotypes contain only the higher molecular weight forms A1 and A2. These data and the low residual activity of the enzyme in all the variants of Gaucher disease suggest that the mutations of beta-glucocerebrosidase are allelic and involve the active site.

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Year:  1982        PMID: 6957882      PMCID: PMC346953          DOI: 10.1073/pnas.79.18.5607

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


  31 in total

1.  beta-Glucoside hydrolase activity of normal and glucosylceramidotic cultured human skin fibroblasts.

Authors:  O T Mueller; A Rosenberg
Journal:  J Biol Chem       Date:  1977-02-10       Impact factor: 5.157

2.  A rapid and sensitive method for the quantitation of microgram quantities of protein utilizing the principle of protein-dye binding.

Authors:  M M Bradford
Journal:  Anal Biochem       Date:  1976-05-07       Impact factor: 3.365

3.  Neuropathic Gaucher's disease with normal 4-methylumbelliferyl-beta-glucosidase activity in the liver.

Authors:  M Owada; T Sakiyama; T Kitagawa
Journal:  Pediatr Res       Date:  1977-05       Impact factor: 3.756

4.  Acid beta-glucosidase and the diagnosis of Gaucher's disease in liver and spleen.

Authors:  J Butterworth; D M Broadhead
Journal:  Clin Chim Acta       Date:  1978-08-01       Impact factor: 3.786

5.  Gaucher disease: isolation and comparison of normal and mutant glucocerebrosidase from human spleen tissue.

Authors:  P G Pentchev; R O Brady; H E Blair; D E Britton; S H Sorrell
Journal:  Proc Natl Acad Sci U S A       Date:  1978-08       Impact factor: 11.205

6.  Gaucher's disease. Factors affecting the 4-methylum-belliferyl-beta-D-glucosidase activity of cultured skin fibroblasts.

Authors:  J Butterworth; D M Broadhead
Journal:  Clin Genet       Date:  1978-08       Impact factor: 4.438

7.  Blood glucosylceramide levels in Gaucher's disease and its distribution amongst lipoprotein fractions.

Authors:  G Dawson; J Y Oh
Journal:  Clin Chim Acta       Date:  1977-02-15       Impact factor: 3.786

8.  Deficiency of steroid beta-glucosidase in Gaucher disease.

Authors:  J N Kanfer; S S Raghavan; R A Mumford; R S Labow; D G Williamson; D S Layne
Journal:  Biochem Biophys Res Commun       Date:  1975-11-17       Impact factor: 3.575

9.  The diagnosis of the adult type of Gaucher's disease and its carrier state by demonstration of deficiency of beta-glucosidase activity in peripheral blood leukocytes.

Authors:  E Beutler; W Kuhl
Journal:  J Lab Clin Med       Date:  1970-11

10.  Enzyme replacement therapy in Gaucher's disease: large-scale purification of glucocerebrosidase suitable for human administration.

Authors:  F S Furbish; H E Blair; J Shiloach; P G Pentchev; R O Brady
Journal:  Proc Natl Acad Sci U S A       Date:  1977-08       Impact factor: 11.205

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  33 in total

1.  A quantitative immunoelectronmicroscopic study on soluble, membrane-associated and membrane-bound lysosomal enzymes in human intestinal epithelial cells.

Authors:  R Willemsen; R Brünken; C W Sorber; A T Hoogeveen; H A Wisselaar; J M Van Dongen; A J Reuser
Journal:  Histochem J       Date:  1991-10

2.  The clinical, molecular, and pathological characterisation of a family with two cases of lethal perinatal type 2 Gaucher disease.

Authors:  E Sidransky; N Tayebi; B K Stubblefield; W Eliason; A Klineburgess; G P Pizzolato; J N Cox; J Porta; A Bottani; C D DeLozier-Blanchet
Journal:  J Med Genet       Date:  1996-02       Impact factor: 6.318

3.  Comparative study on glucocerebrosidase in spleens from patients with Gaucher disease.

Authors:  J M Aerts; W E Donker-Koopman; S Brul; S Van Weely; M C Sa Miranda; J A Barranger; J M Tager; A W Schram
Journal:  Biochem J       Date:  1990-07-01       Impact factor: 3.857

4.  Genetic heterogeneity in type 1 Gaucher disease: multiple genotypes in Ashkenazic and non-Ashkenazic individuals.

Authors:  S Tsuji; B M Martin; J A Barranger; B K Stubblefield; M E LaMarca; E I Ginns
Journal:  Proc Natl Acad Sci U S A       Date:  1988-04       Impact factor: 11.205

5.  Gene mapping and leader polypeptide sequence of human glucocerebrosidase: implications for Gaucher disease.

Authors:  E I Ginns; P V Choudary; S Tsuji; B Martin; B Stubblefield; J Sawyer; J Hozier; J A Barranger
Journal:  Proc Natl Acad Sci U S A       Date:  1985-10       Impact factor: 11.205

6.  A method for enrichment of hybrid somatic cells: complementation studies in certain lysosomal enzymopathies.

Authors:  P V Nelson; W F Carey
Journal:  J Inherit Metab Dis       Date:  1985       Impact factor: 4.982

7.  Gaucher disease: the effects of phosphatidylserine on glucocerebrosidase from normal and Gaucher fibroblasts.

Authors:  F Y Choy
Journal:  Hum Genet       Date:  1984       Impact factor: 4.132

8.  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

9.  Gaucher disease: heterologous expression of two alleles associated with neuronopathic phenotypes.

Authors:  M E Grace; A Berg; G S He; L Goldberg; M Horowitz; G A Grabowski
Journal:  Am J Hum Genet       Date:  1991-09       Impact factor: 11.025

10.  Glucocerebrosidase "processing" and gene expression in various forms of Gaucher disease.

Authors:  E Beutler; W Kuhl; J Sorge
Journal:  Am J Hum Genet       Date:  1985-11       Impact factor: 11.025

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