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Defective expression of the interleukin-2/interleukin-15 receptor beta subunit leads to a natural killer cell-deficient form of severe combined immunodeficiency.
- Source :
-
Blood [Blood] 2001 Aug 01; Vol. 98 (3), pp. 877-9. - Publication Year :
- 2001
-
Abstract
- Development of T and natural killer (NK) cells is critically dependent on cytokine signaling, and defects in cytokine receptor complex subunits have been shown to result in severe combined immunodeficiency (SCID) syndromes in humans and in murine models. An infant boy had typical clinical features of SCID and was found to lack NK cells in his peripheral circulation. Molecular analysis did not reveal abnormalities in his gammac or JAK-3 genes, and he was investigated for defects in the interleukin-15 (IL-15) receptor complex because functional IL-15 signaling is essential for NK cell development. Expression of the IL-2R/IL-15Rbeta chain was significantly reduced in the patient's peripheral blood mononuclear cells (PBMCs) by immunoblot, flow cytometry, and Northern blot analysis. Furthermore, IL-2 stimulation of PBMCs showed only minimal tyrosine phosphorylation of JAK-3. These data demonstrate that defects in IL-2R/1L-15Rbeta expression can lead to a unique NK-deficient SCID immunophenotype. (Blood. 2001;98:877-879)
- Subjects :
- Exons genetics
Humans
Immunophenotyping
Infant, Newborn
Interleukin-2 Receptor beta Subunit
Male
Polymorphism, Single-Stranded Conformational
Protein Subunits
Receptors, Interleukin metabolism
Receptors, Interleukin-15
Receptors, Interleukin-2 metabolism
Sequence Analysis, DNA
Severe Combined Immunodeficiency blood
Severe Combined Immunodeficiency metabolism
Signal Transduction
Killer Cells, Natural pathology
Severe Combined Immunodeficiency etiology
Subjects
Details
- Language :
- English
- ISSN :
- 0006-4971
- Volume :
- 98
- Issue :
- 3
- Database :
- MEDLINE
- Journal :
- Blood
- Publication Type :
- Academic Journal
- Accession number :
- 11468191
- Full Text :
- https://doi.org/10.1182/blood.v98.3.877