Home LiteratureArticle Details
PMID: 6593712 Published · ppublish English Journal Article Research Support, U.S. Gov't, P.H.S.

Cross-reacting material in Gaucher disease fibroblasts.

Beutler E, Kuhl W, Sorge J

Abstract

Glucocerebrosidase is the enzyme that is deficient in Gaucher diseases. Four monoclonal antibodies reacting with at least two different epitopes of this enzyme have been produced. The amounts of glucocerebrosidase in fibroblasts of patients with all three types of Gaucher disease were investigated by radioiodinating two of the antibodies and measuring their binding to fibroblast extracts immobilized on nitrocellulose filters. The amount of glucocerebrosidase antigen was decreased in all cases of Gaucher disease, particularly in the fibroblasts of patients with the more severe neuronopathic forms of the disorder, types II and III. The catalytic activity was reduced to a greater extent than the amount of antigen in all cases, so that the specific activity of the residual enzyme was found to be diminished. Although measurements in individual cases were quite reproducible and the amount of antigen detected by monoclonal antibodies reacting with different epitopes was quite similar, there was considerable variation between patients. This finding is consistent with the apparent within-type genetic heterogeneity of Gaucher disease, even within the Ashkenazi Jewish population in which it is most prevalent.

MeSH Terms
Antibodies, Monoclonal Cross Reactions Fibroblasts/enzymology Gaucher Disease/enzymology,immunology Glucosidases/immunology Glucosylceramidase/immunology Humans
Chemicals
Antibodies, Monoclonal Glucosidases Glucosylceramidase
Authors & Affiliations
3 authors, click to expand affiliations / ORCID
Beutler E
Kuhl W
Sorge J
References (12)
12 references, click to expand
  1. Gaucher's disease: deficiency of 'acid' -glucosidase and reconstitution of enzyme activity in vitro.
    Proc Natl Acad Sci U S A. 1971 Nov;68(11):2810-3 PMID: 5288260
  2. Enzymic iodination. A probe for accessible surface proteins of normal and neoplastic lymphocytes.
    Biochem J. 1971 Oct;124(5):921-7 PMID: 5131013
  3. Solubilization of glucocerebrosidase from human placenta and demonstration of a phospholipid requirement for its catalytic activity.
    Biochem Biophys Res Commun. 1976 Aug 23;71(4):1048-53 PMID: 971299
  4. Gaucher's disease in an asymptomatic 72-year-old.
    JAMA. 1977 Jun 6;237(23):2529 PMID: 576973
  5. Gaucher disease: isolation and comparison of normal and mutant glucocerebrosidase from human spleen tissue.
    Proc Natl Acad Sci U S A. 1978 Aug;75(8):3970-3 PMID: 29293
  6. Leukocyte beta-glucosidase in homozygotes and heterozygotes for Gaucher disease.
    Am J Hum Genet. 1980 Mar;32(2):158-73 PMID: 6770675
  7. Mutations of glucocerebrosidase: discrimination of neurologic and non-neurologic phenotypes of Gaucher disease.
    Proc Natl Acad Sci U S A. 1982 Sep;79(18):5607-10 PMID: 6957882
  8. Immunological and catalytic quantitation of splenic glucocerebrosidase from the three clinical forms of Gaucher disease.
    Am J Hum Genet. 1983 Jul;35(4):621-8 PMID: 6881138
  9. Determination of Gaucher's disease phenotypes with monoclonal antibody.
    Clin Chim Acta. 1983 Jul 15;131(3):283-7 PMID: 6883722
  10. Monoclonal antibodies against human beta-glucocerebrosidase.
    Eur J Biochem. 1983 Aug 15;134(3):585-9 PMID: 6192991
  11. Complementation analysis in Gaucher disease using single cell microassay techniques. Evidence for a single "Gaucher gene".
    Hum Genet. 1983;65(2):112-6 PMID: 6418635
  12. Protein measurement with the Folin phenol reagent.
    J Biol Chem. 1951 Nov;193(1):265-75 PMID: 14907713
Article Info
Journal
Proceedings of the National Academy of Sciences of the United States of America
Abbr.
Proc Natl Acad Sci U S A
ISSN
0027-8424
Published
1984-10-00
Pages
6506-10
Language
English
Region
United States
NLM ID
7505876
PMCID
PMC391953
Subset
IM
Grants
NIADDK NIH HHS · AM 25696 · United States
Analysis Services
Analysis Services

Contact

No. 2 Wenbo Road, Zhangqiu District, Jinan, Shandong

Qilu Normal University · Genelibs Bioinformatics Lab

750 Shunhua Rd, Jinan

2F, Bldg F, University Science Park

Tel: 0531-88819269

WeChat Official Account

Follow our WeChat subscription account for real-time updates and the latest in medical and biological research.


Business Email

E-mail: [email protected]