Literature DB >> 29086100

Clinical Manifestations and Pathophysiological Mechanisms of the Wiskott-Aldrich Syndrome.

Fabio Candotti1,2.   

Abstract

The Wiskott-Aldrich syndrome (WAS) is a rare X-linked disorder originally described by Dr. Alfred Wiskott in 1937 and Dr. Robert Aldrich in 1954 as a familial disease characterized by infections, bleeding tendency, and eczema. Today, it is well recognized that the syndrome has a wide clinical spectrum ranging from mild, isolated thrombocytopenia to full-blown presentation that can be complicated by life-threatening hemorrhages, immunodeficiency, atopy, autoimmunity, and cancer. The pathophysiology of classic and emerging features is being elucidated by clinical studies, but remains incompletely defined, which hinders the application of targeted therapies. At the same time, progress of hematopoietic stem cell transplantation and gene therapy offer optimistic prospects for treatment options aimed at the replacement of the defective lymphohematopoietic system that have the potential to provide a cure for this rare and polymorphic disease.

Entities:  

Keywords:  Thrombocytopenia; X-chromosome; atopy; autoimmunity; immunodeficiency

Mesh:

Substances:

Year:  2017        PMID: 29086100     DOI: 10.1007/s10875-017-0453-z

Source DB:  PubMed          Journal:  J Clin Immunol        ISSN: 0271-9142            Impact factor:   8.542


  136 in total

1.  Wiskott-Aldrich syndrome protein is necessary for efficient IgG-mediated phagocytosis.

Authors:  R Lorenzi; P M Brickell; D R Katz; C Kinnon; A J Thrasher
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2.  A unique presentation of Wiskott-Aldrich syndrome in relation to platelet size.

Authors:  Payal D Patel; Joy M Samanich; William B Mitchell; Deepa Manwani
Journal:  Pediatr Blood Cancer       Date:  2011-02-10       Impact factor: 3.167

3.  Phenotypic perturbation of B cells in the Wiskott-Aldrich syndrome.

Authors:  J Y Park; A Shcherbina; F S Rosen; A P Prodeus; E Remold-O'Donnell
Journal:  Clin Exp Immunol       Date:  2005-02       Impact factor: 4.330

4.  Flow cytometric analysis of platelets from children with the Wiskott-Aldrich syndrome reveals defects in platelet development, activation and structure.

Authors:  J W Semple; K A Siminovitch; M Mody; Y Milev; A H Lazarus; J F Wright; J Freedman
Journal:  Br J Haematol       Date:  1997-06       Impact factor: 6.998

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Journal:  Thromb Haemost       Date:  2014-12-18       Impact factor: 5.249

6.  Eltrombopag use in a patient with Wiskott-Aldrich syndrome.

Authors:  Maria Gabelli; Antonio Marzollo; Lucia Dora Notarangelo; Giuseppe Basso; Maria Caterina Putti
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7.  A multiinstitutional survey of the Wiskott-Aldrich syndrome.

Authors:  K E Sullivan; C A Mullen; R M Blaese; J A Winkelstein
Journal:  J Pediatr       Date:  1994-12       Impact factor: 4.406

8.  Intrinsic dendritic cell abnormalities in Wiskott-Aldrich syndrome.

Authors:  M Binks; G E Jones; P M Brickell; C Kinnon; D R Katz; A J Thrasher
Journal:  Eur J Immunol       Date:  1998-10       Impact factor: 5.532

9.  Mutations of the Wiskott-Aldrich Syndrome Protein (WASP): hotspots, effect on transcription, and translation and phenotype/genotype correlation.

Authors:  Yinzhu Jin; Cinzia Mazza; Jacinda R Christie; Silvia Giliani; Maurilia Fiorini; Patrizia Mella; Francesca Gandellini; Donn M Stewart; Qili Zhu; David L Nelson; Luigi D Notarangelo; Hans D Ochs
Journal:  Blood       Date:  2004-07-29       Impact factor: 22.113

10.  Aberrant glycosylation of IgA in Wiskott-Aldrich syndrome and X-linked thrombocytopenia.

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Journal:  J Allergy Clin Immunol       Date:  2012-10-26       Impact factor: 10.793

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