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PMID: 1460030 Published · ppublish English Comparative Study Journal Article Research Support, Non-U.S. Gov't Research Support, U.S. Gov't, P.H.S.

Affected paroxysmal nocturnal hemoglobinuria T lymphocytes harbor a common defect in assembly of N-acetyl-D-glucosamine inositol phospholipid corresponding to that in class A Thy-1- murine lymphoma mutants.

The Journal of biological chemistry ·Vol. 267 ·No. 35 ·1992-12-15 ·Pages 25347-51

Armstrong C, Schubert J, Ueda E, Knez JJ, Gelperin D, Hirose S, Silber R, Hollan S, Schmidt RE, Medof ME

Abstract

Deficient expression of glycoinositol phospholipid (GPI) anchored proteins in affected paroxysmal nocturnal hemoglobinuria (PNH) cells has been traced to a defect in GPI anchor assembly. In a previous study (Schubert, J., Schmidt, R. E., and Medof, M. E. (1993) J. Biol. Chem., in press) we characterized the biosynthesis of putative Man-containing GPI anchor precursors in normal peripheral blood lymphocytes and investigated assembly of these intracellular GPI intermediates in CD48- affected and CD48+ unaffected T and natural killer cell lines of PNH patients. We found that affected T cells from five patients exhibited a uniform defect in which dolichol-phosphoryl-Man was synthesized but no GPI mannolipids were expressed. In this study, membranes of patients' affected T cells were labeled with UDP-[3H]GlcNAc to evaluate earlier steps in GPI synthesis, and intact cells were fused to Thy-1- murine lymphoma mutants harboring different defects in early GPI assembly to test for the presence of corresponding or complementary lesions. In all cases, affected cell membranes failed to assemble GlcNAc-inositol phospholipid, the initial precursor of GPI anchor structures, and the intact cells failed to complement class A mutants while complementing other classes. Affected polymorphonuclear leukocytes from three additional patients of different origin were then labeled with [3H]Man and the labeling patterns found to correspond to those obtained with the T lymphocytes. Taken together the data indicate that the genetic lesion in PNH cells resides in a DNA element which: 1) encodes a product required for the synthesis of GlcNAc-inositol phospholipid, 2) corresponds to that altered in class A Thy-1- murine lymphoma mutants, and 3) is commonly affected in different patients.

MeSH Terms
Acetylglucosamine/metabolism Animals Antigens, CD/genetics CD48 Antigen Cell Membrane/metabolism Dolichol Monophosphate Mannose/metabolism Glycosylphosphatidylinositols/metabolism Hemoglobinuria, Paroxysmal/genetics,immunology,metabolism Humans Killer Cells, Natural/immunology Lymphoma/genetics,immunology,metabolism Mannose/blood Mice Mutation Neutrophils/immunology,metabolism Trypanosoma brucei brucei/metabolism Uridine Diphosphate N-Acetylglucosamine/metabolism
Chemicals
Antigens, CD CD48 Antigen CD48 protein, human Cd48 protein, mouse Glycosylphosphatidylinositols Uridine Diphosphate N-Acetylglucosamine Dolichol Monophosphate Mannose Mannose Acetylglucosamine
Authors & Affiliations
10 authors, click to expand affiliations / ORCID
Armstrong C
Institute of Pathology, Case Western Reserve University, Cleveland, Ohio 44106.
Schubert J
Ueda E
Knez J J
Gelperin D
Hirose S
Silber R
Hollan S
Schmidt R E
Medof M E
Article Info
Journal
The Journal of biological chemistry
Abbr.
J Biol Chem
ISSN
0021-9258
Published
1992-12-15
Pages
25347-51
Language
English
Region
United States
NLM ID
2985121R
Subset
IM
Grants
NIAID NIH HHS · AI23598 · United States
NIDDK NIH HHS · P01DK38181 · United States
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