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PMID: 240271 Published · ppublish English Journal Article Research Support, U.S. Gov't, P.H.S.

Characterization of Hex S, the major residual beta hexosaminidase activity in type O Gm2 gangliosidosis (Sandhoff-Jatzkewitz disease).

American journal of human genetics ·Vol. 27 ·No. 5 ·1975-09-00 ·Pages 639-50

Ikonne JU, Rattazzi MC, Desnick RJ

Abstract

Hex S, the major residual beta hexosaminidase activity present in tissues, fluids, and cultured skin fibroblasts of patients with type 0 GM2 gangliosidosis, was isolated and characterized biochemically and immunologically. when appropriate tissue homogenates were tested by electrophoresis on cellulose acetate gels, hex S as well as hex C, the corresponding minor beta hexosaminidase component found in normal visceral tissues, migrated with greater anodic mobilities than hex A. However, a small but reproducible electrophoretic difference was observed between partially purified hex S and hex C components. Hex S and hex C had slightly higher apparent molecular weights than those of hex A or hex G; no major differences were found between hex S and hex A in thermostability, pH optimum, or kinetic properties. Hex S, like hex C from placenta, reacted with an antiserum directed towards the unique antigenic determinants alpha of hex A, indicating that hex S, hex C, and hex A share a common antigenic determinant. No reactivity of hex S was detected with an antiserum directed toward the common antigenic determinant beta of hex A and hex B. These results suggest that further biochemical and immunologic characterization of hex S and elucidation of its relationships with hex A, hex B, and hex C may significantly contribute to the understanding of the molecular defects in the GM2 gangliosidoses.

MeSH Terms
Brain Chemistry Chromatography, Ion Exchange Electrophoresis Enzyme Inhibitors Gangliosidoses/genetics Hexosaminidases/analysis Humans Hydrogen-Ion Concentration Kidney/analysis Liver/analysis Lung/analysis Molecular Weight Myocardium/analysis Spleen/analysis Syndrome
Chemicals
Enzyme Inhibitors Hexosaminidases
Authors & Affiliations
3 authors, click to expand affiliations / ORCID
Ikonne J U
Rattazzi M C
Desnick R J
References (24)
24 references, click to expand
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Article Info
Journal
American journal of human genetics
Abbr.
Am J Hum Genet
ISSN
0002-9297
Published
1975-09-00
Pages
639-50
Language
English
Region
United States
NLM ID
0370475
PMCID
PMC1762835
Subset
IM
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