Literature DB >> 27787

Multiple glycosidase deficiencies in a case of juvenile (type 3) Gaucher disease.

Y B Chiao, G M Hoyson, S P Peters, R E Lee, W Diven, J V Murphy, R H Glew.   

Abstract

Biochemical investigations were performed on autopsy tissues obtained from an 11-year-old girl who died with the juvenile, subacute neuropathic form of Gaucher disease. In addition to the expected deficiency of glucocerebrosidase activity, extracts of both liver and kidney from this individual displayed a profound (greater than or equal to 90%) deficiency of "soluble" beta-glucosidase, beta-xylosidase, and beta-galactosidase activities. Fibroblasts obtained from this individual also contained markedly reduced levels of beta-xylosidase activity but normal levels of beta-D-fucosidase and beta-galactosidase activity. Because the soluble beta-glucosidase, beta-xylosidase, and a portion of the beta-galactosidase activities from control human liver all cochromatographed on a gel filtration column of Sephadex G-200, it is suggested that these activities all reside in a single enzyme, analogous to the situation described in a number of nonhuman, mammalian tissues. This demonstration of multiple glycosidase deficiencies in addition to the deficiency of glucocerebrosidase in a case of subacute neuropathic Gaucher disease suggests that other biochemical aberrations, in addition to a deficiency of glucocerebrosidase, might contribute to pathology in some cases of Gaucher disease.

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Year:  1978        PMID: 27787      PMCID: PMC392571          DOI: 10.1073/pnas.75.5.2448

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


  21 in total

1.  The use of ion-exchange resins in the application of protein samples to gel filtration columns.

Authors:  R R Miller; S P Peters; M S Kuhlenschmidt; R H Glew
Journal:  Anal Biochem       Date:  1976-05-07       Impact factor: 3.365

2.  METABOLISM OF GLUCOCEREBROSIDES. II. EVIDENCE OF AN ENZYMATIC DEFICIENCY IN GAUCHER'S DISEASE.

Authors:  R O BRADY; J N KANFER; D SHAPIRO
Journal:  Biochem Biophys Res Commun       Date:  1965-01-18       Impact factor: 3.575

3.  Phosphatase studies in Gaucher's disease.

Authors:  A C CROCKER; B H LANDING
Journal:  Metabolism       Date:  1960-04       Impact factor: 8.694

4.  Protein measurement with the Folin phenol reagent.

Authors:  O H LOWRY; N J ROSEBROUGH; A L FARR; R J RANDALL
Journal:  J Biol Chem       Date:  1951-11       Impact factor: 5.157

5.  Isolation and characterization of beta-glucosidase from the cytosol of rat kidney cortex.

Authors:  R H Glew; S P Peters; A R Christopher
Journal:  Biochim Biophys Acta       Date:  1976-01-23

6.  Clinical and biochemical aspects of chronic Gaucher's disease.

Authors:  Y Matoth; R Zaizov; J Hoffman; C Klibansky
Journal:  Isr J Med Sci       Date:  1974-12

7.  N-hexyl-O-glucosyl sphingosine, an inhibitor of glucosyl ceramide -glucosidase.

Authors:  J S Erickson; N S Radin
Journal:  J Lipid Res       Date:  1973-03       Impact factor: 5.922

8.  The common identity of five glycosidases in human liver.

Authors:  M A Chester; B Hultberg; P A Ockerman
Journal:  Biochim Biophys Acta       Date:  1976-04-08

9.  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

10.  Progressive myoclonic epilepsy due to Gaucher's disease in an adult.

Authors:  J O King
Journal:  J Neurol Neurosurg Psychiatry       Date:  1975-09       Impact factor: 10.154

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  3 in total

1.  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

2.  Gaucher disease. III. Substrate specificity of glucocerebrosidase and the use of nonlabeled natural substrates for the investigation of patients.

Authors:  F Y Choy; R G Davidson
Journal:  Am J Hum Genet       Date:  1980-09       Impact factor: 11.025

3.  Human glucocerebrosidase catalyses transglucosylation between glucocerebroside and retinol.

Authors:  D J Vanderjagt; D E Fry; R H Glew
Journal:  Biochem J       Date:  1994-06-01       Impact factor: 3.857

  3 in total

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