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PMID: 15001467 Published · ppublish English Journal Article Research Support, Non-U.S. Gov't

Impaired natural and CD16-mediated NK cell cytotoxicity in patients with WAS and XLT: ability of IL-2 to correct NK cell functional defect.

Blood ·Vol. 104 ·No. 2 ·2004-07-15 ·Pages 436-43

Gismondi A, Cifaldi L, Mazza C, Giliani S, Parolini S, Morrone S, Jacobelli J, Bandiera E, Notarangelo L, Santoni A

Abstract

In this study we show that Wiskott-Aldrich syndrome protein (WASp), a critical regulator of actin cytoskeleton that belongs to the Scar/WAVE family, plays a crucial role in the control of natural killer (NK) cell cytotoxicity. Analysis of NK cell numbers and cytotoxic activity in patients carrying different mutations in the WASP coding gene indicated that although the percentage of NK cells was normal or increased, natural cytotoxicity and antibody-mediated NK cell cytotoxicity were inhibited in all patients with the classical WAS phenotype and in most patients carrying mutations associated with the X-linked thrombocytopenia (XLT) phenotype. The inhibition of NK cell-mediated cytotoxicity was associated with the reduced ability of WAS and XLT NK cells to form conjugates with susceptible target cells and to accumulate F-actin on binding. Treatment with interleukin-2 (IL-2) corrected the functional defects of NK cells by affecting their ability to bind to sensitive target cells and to accumulate F-actin. In addition, we provide information on the molecular mechanisms that control WASp function, demonstrating that binding of NK cells to sensitive targets or triggering through CD16 by means of reverse antibody-dependent cellular cytotoxicity (ADCC) rapidly activates Cdc42. We also found that WASp undergoes tyrosine phosphorylation upon CD16 or beta2-integrin engagement on NK cells.

MeSH Terms
Actins/metabolism Antibodies/immunology Child Child, Preschool Chromosomes, Human, X Genetic Linkage Humans In Vitro Techniques Infant Interleukin-2/metabolism Killer Cells, Natural/immunology,metabolism Phosphorylation Proteins/genetics,metabolism Receptors, IgG/metabolism Thrombocytopenia/genetics,immunology,metabolism Tyrosine/metabolism Wiskott-Aldrich Syndrome/immunology,metabolism Wiskott-Aldrich Syndrome Protein cdc42 GTP-Binding Protein/metabolism
Chemicals
Actins Antibodies Interleukin-2 Proteins Receptors, IgG WAS protein, human Wiskott-Aldrich Syndrome Protein Tyrosine cdc42 GTP-Binding Protein
Authors & Affiliations
10 authors, click to expand affiliations / ORCID
Gismondi Angela
University "La Sapienza," Viale Regina Elena, 324, 00161 Rome, Italy. [email protected]
Cifaldi Loredana
Mazza Cinzia
Giliani Silvia
Parolini Silvia
Morrone Stefania
Jacobelli Jordan
Bandiera Elisabetta
Notarangelo Luigi
Santoni Angela
Article Info
Journal
Blood
Abbr.
Blood
ISSN
0006-4971
Published
2004-07-15
Epub
2004-00-04
Pages
436-43
Language
English
Region
United States
NLM ID
7603509
Subset
IM
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