Literature DB >> 21178275

The Wiskott-Aldrich syndrome: The actin cytoskeleton and immune cell function.

Michael P Blundell1, Austen Worth, Gerben Bouma, Adrian J Thrasher.   

Abstract

Wiskott-Aldrich syndrome (WAS) is a rare X-linked recessive primary immunodeficiency characterised by immune dysregulation, microthrombocytopaenia, eczema and lymphoid malignancies. Mutations in the WAS gene can lead to distinct syndrome variations which largely, although not exclusively, depend upon the mutation. Premature termination and deletions abrogate Wiskott-Aldrich syndrome protein (WASp) expression and lead to severe disease (WAS). Missense mutations usually result in reduced protein expression and the phenotypically milder X-linked thrombocytopenia (XLT) or attenuated WAS [1-3]. More recently however novel activating mutations have been described that give rise to X-linked neutropenia (XLN), a third syndrome defined by neutropenia with variable myelodysplasia [4-6]. WASP is key in transducing signals from the cell surface to the actin cytoskeleton, and a lack of WASp results in cytoskeletal defects that compromise multiple aspects of normal cellular activity including proliferation, phagocytosis, immune synapse formation, adhesion and directed migration.

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Year:  2010        PMID: 21178275      PMCID: PMC3835520          DOI: 10.3233/DMA-2010-0735

Source DB:  PubMed          Journal:  Dis Markers        ISSN: 0278-0240            Impact factor:   3.434


  25 in total

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2.  Low T Cell Numbers Resembling T-B+ SCID in a Patient with Wiskott-Aldrich Syndrome and the Outcome of Two Hematopoietic Stem Cell Transplantations.

Authors:  Deniz Cagdas; Selin Aytac; Barış Kuskonmaz; Tadashi Ariga; Mirjam van der Burg; Duygu Uckan Cetinkaya; Özden Sanal; İlhan Tezcan
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Review 3.  DOCK8 deficiency: Insights into pathophysiology, clinical features and management.

Authors:  Catherine M Biggs; Sevgi Keles; Talal A Chatila
Journal:  Clin Immunol       Date:  2017-06-15       Impact factor: 3.969

4.  Mutant huntingtin impairs immune cell migration in Huntington disease.

Authors:  Wanda Kwan; Ulrike Träger; Dimitrios Davalos; Austin Chou; Jill Bouchard; Ralph Andre; Aaron Miller; Andreas Weiss; Flaviano Giorgini; Christine Cheah; Thomas Möller; Nephi Stella; Katerina Akassoglou; Sarah J Tabrizi; Paul J Muchowski
Journal:  J Clin Invest       Date:  2012-11-19       Impact factor: 14.808

Review 5.  Myotonic dystrophy kinase-related Cdc42-binding kinases (MRCK), the ROCK-like effectors of Cdc42 and Rac1.

Authors:  Zhuoshen Zhao; Ed Manser
Journal:  Small GTPases       Date:  2015-06-19

6.  Stem cell factor programs the mast cell activation phenotype.

Authors:  Tomonobu Ito; Daniel Smrž; Mi-Yeon Jung; Geethani Bandara; Avanti Desai; Šárka Smržová; Hye Sun Kuehn; Michael A Beaven; Dean D Metcalfe; Alasdair M Gilfillan
Journal:  J Immunol       Date:  2012-04-23       Impact factor: 5.422

Review 7.  Inborn errors of immunity with atopic phenotypes: A practical guide for allergists.

Authors:  Riccardo Castagnoli; Vassilios Lougaris; Giuliana Giardino; Stefano Volpi; Lucia Leonardi; Francesco La Torre; Silvia Federici; Stefania Corrente; Bianca Laura Cinicola; Annarosa Soresina; Caterina Cancrini; Gian Luigi Marseglia; Fabio Cardinale
Journal:  World Allergy Organ J       Date:  2021-02-22       Impact factor: 4.084

8.  A case of familial X-linked thrombocytopenia with a novel WAS gene mutation.

Authors:  Eu Kyoung Lee; Yeun-Joo Eem; Nack-Gyun Chung; Myung Shin Kim; Dae Chul Jeong
Journal:  Korean J Pediatr       Date:  2013-06-21

9.  CD47: A Cell Surface Glycoprotein Which Regulates Multiple Functions of Hematopoietic Cells in Health and Disease.

Authors:  Per-Arne Oldenborg
Journal:  ISRN Hematol       Date:  2013-01-21

10.  Use of zinc-finger nucleases to knock out the WAS gene in K562 cells: a human cellular model for Wiskott-Aldrich syndrome.

Authors:  Miguel G Toscano; Per Anderson; Pilar Muñoz; Gema Lucena; Marién Cobo; Karim Benabdellah; Philip D Gregory; Michael C Holmes; Francisco Martin
Journal:  Dis Model Mech       Date:  2013-01-11       Impact factor: 5.758

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