Literature DB >> 20008191

New insights into the biology of Wiskott-Aldrich syndrome (WAS).

Adrian J Thrasher1.   

Abstract

The Wiskott-Aldrich syndrome (WAS) is an X-linked immunodeficiency disease with a characteristic clinical phenotype that includes thrombocytopenia with small platelets, eczema, recurrent infections due to immunodeficiency, and an increased incidence of autoimmune manifestations and malignancies. The identification of the molecular defect in the WAS gene has broadened the clinical spectrum of disease to include chronic or intermittent X-linked thrombocytopenia (XLT), a relatively mild form of WAS, and X-linked neutropenia (XLN) due to an arrest of myelopoiesis. The pathophysiological mechanisms relate to defective actin polymerization in hematopoietic cells as a result of deficient or dysregulated activity of the WAS protein (WASp). The severity of disease is variable and somewhat predictable from genotype. Treatment strategies therefore range from conservative through to early definitive intervention by using allogeneic hematopoietic stem cell transplantation and potentially somatic gene therapy. All aspects of the condition from clinical presentation to molecular pathology and basic cellular mechanisms have been reviewed recently.

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Year:  2009        PMID: 20008191     DOI: 10.1182/asheducation-2009.1.132

Source DB:  PubMed          Journal:  Hematology Am Soc Hematol Educ Program        ISSN: 1520-4383


  13 in total

1.  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
Journal:  J Clin Immunol       Date:  2016-11-30       Impact factor: 8.317

2.  Molecular analysis of Arp2/3 complex activation in cells.

Authors:  Brian J Galletta; Anders E Carlsson; John A Cooper
Journal:  Biophys J       Date:  2012-11-20       Impact factor: 4.033

3.  Additional diverse findings expand the clinical presentation of DOCK8 deficiency.

Authors:  Ozden Sanal; Huie Jing; Tuba Ozgur; Deniz Ayvaz; Dara M Strauss-Albee; Sibel Ersoy-Evans; Ilhan Tezcan; Gulten Turkkani; Helen F Matthews; Goknur Haliloglu; Aysel Yuce; Bilgehan Yalcin; Ozay Gokoz; Kader K Oguz; Helen C Su
Journal:  J Clin Immunol       Date:  2012-04-04       Impact factor: 8.317

Review 4.  Germline Genetic Predisposition to Hematologic Malignancy.

Authors:  Elissa Furutani; Akiko Shimamura
Journal:  J Clin Oncol       Date:  2017-02-13       Impact factor: 44.544

5.  Human genome-wide association and mouse knockout approaches identify platelet supervillin as an inhibitor of thrombus formation under shear stress.

Authors:  Leonard C Edelstein; Elizabeth J Luna; Ian B Gibson; Molly Bray; Ying Jin; Altaf Kondkar; Srikanth Nagalla; Nacima Hadjout-Rabi; Tara C Smith; Daniel Covarrubias; Stephen N Jones; Firdos Ahmad; Moritz Stolla; Xianguo Kong; Zhiyou Fang; Wolfgang Bergmeier; Chad Shaw; Suzanne M Leal; Paul F Bray
Journal:  Circulation       Date:  2012-05-01       Impact factor: 29.690

6.  Long-term outcome and lineage-specific chimerism in 194 patients with Wiskott-Aldrich syndrome treated by hematopoietic cell transplantation in the period 1980-2009: an international collaborative study.

Authors:  Daniele Moratto; Silvia Giliani; Carmem Bonfim; Evelina Mazzolari; Alain Fischer; Hans D Ochs; Andrew J Cant; Adrian J Thrasher; Morton J Cowan; Michael H Albert; Trudy Small; Sung-Yun Pai; Elie Haddad; Antonella Lisa; Sophie Hambleton; Mary Slatter; Marina Cavazzana-Calvo; Nizar Mahlaoui; Capucine Picard; Troy R Torgerson; Lauri Burroughs; Adriana Koliski; Jose Zanis Neto; Fulvio Porta; Waseem Qasim; Paul Veys; Kristina Kavanau; Manfred Hönig; Ansgar Schulz; Wilhelm Friedrich; Luigi D Notarangelo
Journal:  Blood       Date:  2011-06-09       Impact factor: 22.113

7.  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

Review 8.  Meganucleases and other tools for targeted genome engineering: perspectives and challenges for gene therapy.

Authors:  George Silva; Laurent Poirot; Roman Galetto; Julianne Smith; Guillermo Montoya; Philippe Duchateau; Frédéric Pâques
Journal:  Curr Gene Ther       Date:  2011-02       Impact factor: 4.391

9.  Wiskott-Aldrich Syndrome Interacting Protein Deficiency Uncovers the Role of the Co-receptor CD19 as a Generic Hub for PI3 Kinase Signaling in B Cells.

Authors:  Selina Jessica Keppler; Francesca Gasparrini; Marianne Burbage; Shweta Aggarwal; Bruno Frederico; Raif S Geha; Michael Way; Andreas Bruckbauer; Facundo D Batista
Journal:  Immunity       Date:  2015-10-06       Impact factor: 31.745

10.  Whole Wiskott‑Aldrich syndrome protein gene deletion identified by high throughput sequencing.

Authors:  Xiangling He; Runying Zou; Bing Zhang; Yalan You; Yang Yang; Xin Tian
Journal:  Mol Med Rep       Date:  2017-08-31       Impact factor: 2.952

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