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PMID: 29293 Published · ppublish English Comparative Study Journal Article

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

Pentchev PG, Brady RO, Blair HE, Britton DE, Sorrell SH

Abstract

Glucocerebrosidase was purified 26,000-fold from spleens from normal humans and from patients with Gaucher disease (Gaucher spleens). The specific activities of the purified normal and mutant enzymes with glucocerebroside as substrate were 8.5 X 10(5) and 5.4 X 10(4) nmol/mg of protein per hr, respectively. The ratio of enzymatic activities was constant throughout the isolation procedure. The two enzymes appeared to be similar by other parameters such as substrate affinity, heat lability, and pH optimum. Immunotitration with glucocerebrosidase antiserum showed equivalent quantities of crossreacting material in extracts of normal and Gaucher spleens. These data strongly suggest that the genetic basis of Gaucher disease is a strucutral mutation of glucocerebrosidase. The results of sodium dodecyl sulfate gel electrophporesis also indicate that there are differences between the normal and the Gaucher disease enzyme.

MeSH Terms
Electrophoresis, Agar Gel Gaucher Disease/enzymology Glucosidases/metabolism Glucosylceramidase/metabolism Hot Temperature Humans Hydrogen-Ion Concentration Spleen/enzymology
Chemicals
Glucosidases Glucosylceramidase
Authors & Affiliations
5 authors, click to expand affiliations / ORCID
Pentchev P G
Brady R O
Blair H E
Britton D E
Sorrell S H
References (14)
14 references, click to expand
  1. Purification and properties of a heat-stable glucocerebrosidase activating factor from control and Gaucher spleen.
    J Biol Chem. 1977 Jan 25;252(2):563-73 PMID: 13071
  2. beta-Glucoside hydrolase activity of normal and glucosylceramidotic cultured human skin fibroblasts.
    J Biol Chem. 1977 Feb 10;252(3):825-9 PMID: 14134
  3. METABOLISM OF GLUCOCEREBROSIDES. II. EVIDENCE OF AN ENZYMATIC DEFICIENCY IN GAUCHER'S DISEASE.
    Biochem Biophys Res Commun. 1965 Jan 18;18:221-5 PMID: 14282020
  4. Protein measurement with the Folin phenol reagent.
    J Biol Chem. 1951 Nov;193(1):265-75 PMID: 14907713
  5. Enzyme replacement therapy in Gaucher's disease: large-scale purification of glucocerebrosidase suitable for human administration.
    Proc Natl Acad Sci U S A. 1977 Aug;74(8):3560-3 PMID: 269414
  6. Autolysis of glycoproteins in rat kidney lysosomes in vitro. Effects on the isoelectric focusing behaviour of glycoproteins, arylsulphatase and beta-glucuronidase.
    Biochem J. 1974 Aug;141(2):527-35 PMID: 4455220
  7. Infantile (type II) Gaucher's disease: in utero diagnosis and fetal pathology.
    J Pediatr. 1972 Dec;81(6):1134-9 PMID: 4674572
  8. The effect of a heat-stable factor in human spleen on glucocerebrosidase and acid -glucosidase activities.
    Biochim Biophys Acta. 1973 Feb 28;297(2):491-6 PMID: 4705477
  9. Isolation and characterization of glucocerebrosidase from human placental tissue.
    J Biol Chem. 1973 Aug 10;248(15):5256-61 PMID: 4768898
  10. Chronic Gaucher's disease: heat-resistance of leukocyte glucocerebrosidase in relation to some clinical parameters.
    Biomedicine. 1973 Aug 10;19(8):345-8 PMID: 4774111
  11. 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
  12. Demonstration of a deficiency of glucocerebroside-cleaving enzyme in Gaucher's disease.
    J Clin Invest. 1966 Jul;45(7):1112-5 PMID: 5338605
  13. Cleavage of structural proteins during the assembly of the head of bacteriophage T4.
    Nature. 1970 Aug 15;227(5259):680-5 PMID: 5432063
  14. Leukocyte mitosis: suppression in vitro associated with acute infectious hepatitis.
    Science. 1967 Jan 6;155(3758):80-1 PMID: 6015564
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
1978-08-00
Pages
3970-3
Language
English
Region
United States
NLM ID
7505876
PMCID
PMC392911
Subset
IM
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