Document Detail


Patients with X-linked lymphoproliferative disease due to BIRC4 mutation have normal invariant natural killer T-cell populations.
MedLine Citation:
PMID:  19398375     Owner:  NLM     Status:  MEDLINE    
Abstract/OtherAbstract:
Human invariant natural killer T cells (iNKT cells) are a unique population of T cells that express an invariantly rearranged T-cell receptor (TCR) composed of TCRValpha24 and TCRVbeta11 chains which recognize glycosphingolipid antigens presented by the CD1d molecule. iNKT cells are absent in patients with X-linked lymphoproliferative disease (XLP) due to SH2D1A mutation, and are reported to be decreased in patients with XLP due to BIRC4 mutations. However, mice deficient in the BIRC4 gene product, X-linked Inhibitor of Apoptosis (XIAP), have normal iNKT cell populations. Because of this, we studied iNKT cell populations in 6 patients with XLP due to BIRC4 mutations, with comparison to 103 pediatric and adult normal control samples. We found that iNKT cells constitute 0.008%-1.176% of normal peripheral blood T cells, and that iNKT cell populations were normal or increased in patients with BIRC4 mutations. We conclude that XLP due to BIRC4 mutation is not associated with decreased populations of iNKT cells, and that XIAP is likely not a requirement for iNKT cell development.
Authors:
Rebecca A Marsh; Joyce Villanueva; Mi-Ok Kim; Kejian Zhang; Dan Marmer; Kimberly A Risma; Michael B Jordan; Jack J Bleesing; Alexandra H Filipovich
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Publication Detail:
Type:  Journal Article; Research Support, N.I.H., Extramural; Research Support, Non-U.S. Gov't     Date:  2009-04-23
Journal Detail:
Title:  Clinical immunology (Orlando, Fla.)     Volume:  132     ISSN:  1521-7035     ISO Abbreviation:  Clin. Immunol.     Publication Date:  2009 Jul 
Date Detail:
Created Date:  2009-06-08     Completed Date:  2009-07-08     Revised Date:  2010-09-27    
Medline Journal Info:
Nlm Unique ID:  100883537     Medline TA:  Clin Immunol     Country:  United States    
Other Details:
Languages:  eng     Pagination:  116-23     Citation Subset:  IM    
Affiliation:
Division of Hematology/Oncology, Cincinnati Children's Hospital Medical Center, Cincinnati, Ohio 45229, USA. Rebecca.Marsh@cchmc.org
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MeSH Terms
Descriptor/Qualifier:
Adolescent
Adult
Child
Child, Preschool
DNA Mutational Analysis
Flow Cytometry
Humans
Lymphoproliferative Disorders / blood,  genetics,  immunology*
Mutation*
Natural Killer T-Cells / cytology,  immunology*,  metabolism
X-Linked Inhibitor of Apoptosis Protein / genetics*
Grant Support
ID/Acronym/Agency:
1R03AI079797-01/AI/NIAID NIH HHS; R03 AI079797-01/AI/NIAID NIH HHS
Chemical
Reg. No./Substance:
0/X-Linked Inhibitor of Apoptosis Protein; 0/XIAP protein, human
Comments/Corrections

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