Literature DB >> 14612668

WHIM syndrome: a genetic disorder of leukocyte trafficking.

A Virginia Gulino1.   

Abstract

PURPOSE OF REVIEW: WHIM syndrome (the association of warts, hypogammaglobulinemia, recurrent bacterial infections, and 'myelokathexis') is a rare congenital form of neutropenia associated with an unusual immune disorder involving hypogammaglonulinemia and abnormal susceptibility to warts. In this review, we describe the clinical, laboratory and genetic features of WHIM syndrome. RECENT
FINDINGS: The identification of chemokine receptor CXCR4 as the causative gene of WHIM syndrome yields new interest in the study of this disease as a model for the comprehension of CXCR4 biology in humans and highlights the importance of the chemokine network for inducing effective immune responses and governing leukocyte trafficking.
SUMMARY: CXCR4 participates in several biological processes (bone marrow hematopoiesis, cardiogenesis, angiogenesis, neurogenesis) and is implicated in different clinical pathologic conditions (WHIM, HIV infection, tumor metastatization, autoimmunity). Pharmacologic agents that modulate CXCR4 expression/function are already available and promise a wide range of future clinical applications.

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Year:  2003        PMID: 14612668     DOI: 10.1097/00130832-200312000-00005

Source DB:  PubMed          Journal:  Curr Opin Allergy Clin Immunol        ISSN: 1473-6322


  20 in total

1.  WHIM syndrome myelokathexis reproduced in the NOD/SCID mouse xenotransplant model engrafted with healthy human stem cells transduced with C-terminus-truncated CXCR4.

Authors:  Toshinao Kawai; Uimook Choi; Lanise Cardwell; Suk See DeRavin; Nora Naumann; Narda L Whiting-Theobald; Gilda F Linton; Jaehyun Moon; Philip M Murphy; Harry L Malech
Journal:  Blood       Date:  2006-08-31       Impact factor: 22.113

Review 2.  Regulation of CXCR4 signaling.

Authors:  John M Busillo; Jeffrey L Benovic
Journal:  Biochim Biophys Acta       Date:  2006-11-10

Review 3.  Mobilization of hematopoietic stem and progenitor cells using inhibitors of CXCR4 and VLA-4.

Authors:  M P Rettig; G Ansstas; J F DiPersio
Journal:  Leukemia       Date:  2011-09-02       Impact factor: 11.528

Review 4.  Genetics on a WHIM.

Authors:  Omar Al Ustwani; Razelle Kurzrock; Meir Wetzler
Journal:  Br J Haematol       Date:  2013-09-20       Impact factor: 6.998

5.  WHIM Syndrome Caused by Waldenström's Macroglobulinemia-Associated Mutation CXCR4 (L329fs).

Authors:  Qian Liu; Catherina Pan; Lizbeeth Lopez; Jiliang Gao; Daniel Velez; Sandra Anaya-O'Brien; Jean Ulrick; Patricia Littel; John S Corns; Donald T Ellenburg; Harry L Malech; Philip M Murphy; David H McDermott
Journal:  J Clin Immunol       Date:  2016-04-08       Impact factor: 8.317

6.  Potential large animal models for gene therapy of human genetic diseases of immune and blood cell systems.

Authors:  Thomas R Bauer; Rima L Adler; Dennis D Hickstein
Journal:  ILAR J       Date:  2009

Review 7.  WHIM syndrome: congenital immune deficiency disease.

Authors:  Toshinao Kawai; Harry L Malech
Journal:  Curr Opin Hematol       Date:  2009-01       Impact factor: 3.284

Review 8.  Structure-based drug design: from nucleic acid to membrane protein targets.

Authors:  Magdalena M Dailey; Chayanendu Hait; Patrick A Holt; Jon M Maguire; Jason B Meier; M Clarke Miller; Luigi Petraccone; John O Trent
Journal:  Exp Mol Pathol       Date:  2009-01-31       Impact factor: 3.362

9.  Transcription factor Gfi-1 induced by G-CSF is a negative regulator of CXCR4 in myeloid cells.

Authors:  Maria De La Luz Sierra; Paola Gasperini; Peter J McCormick; Jinfang Zhu; Giovanna Tosato
Journal:  Blood       Date:  2007-06-27       Impact factor: 22.113

10.  Impaired recruitment of Grk6 and beta-Arrestin 2 causes delayed internalization and desensitization of a WHIM syndrome-associated CXCR4 mutant receptor.

Authors:  Peter J McCormick; Marta Segarra; Paola Gasperini; A Virginia Gulino; Giovanna Tosato
Journal:  PLoS One       Date:  2009-12-01       Impact factor: 3.240

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