Literature DB >> 19082760

Mutations of the Wiskott-Aldrich Syndrome Protein affect protein expression and dictate the clinical phenotypes.

Hans D Ochs1.   

Abstract

Mutations of the Wiskott-Aldrich Syndrome Protein (WASP) are responsible for classic Wiskott-Aldrich Syndrome (WAS), X-linked thrombocytopenia (XLT), and in rare instances congenital X-linked neutropenia (XLN). WASP is a regulator of actin polymerization in hematopoietic cells with well-defined functional domains that are involved in cell signaling and cell locomotion, immune synapse formation, and apoptosis. Mutations of WASP are located throughout the gene and either inhibit or disregulate normal WASP function. Analysis of a large patient population demonstrates a strong phenotype-genotype correlation. Classic WAS occurs when WASP is absent, XLT when mutated WASP is expressed and XLN when missense mutations occur in the Cdc42-binding site. However, because there are exceptions to this rule it is difficult to predict the long-term prognosis of a given affected boy solely based on the analysis of WASP expression.

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Year:  2009        PMID: 19082760     DOI: 10.1007/s12026-008-8084-3

Source DB:  PubMed          Journal:  Immunol Res        ISSN: 0257-277X            Impact factor:   2.829


  34 in total

1.  WASP confers selective advantage for specific hematopoietic cell populations and serves a unique role in marginal zone B-cell homeostasis and function.

Authors:  Lisa S Westerberg; Miguel A de la Fuente; Fredrik Wermeling; Hans D Ochs; Mikael C I Karlsson; Scott B Snapper; Luigi D Notarangelo
Journal:  Blood       Date:  2008-09-04       Impact factor: 22.113

2.  Wiskott-Aldrich syndrome. An immunologic deficiency disease involving the afferent limb of immunity.

Authors:  M D Cooper; H P Chae; J T Lowman; W Krivit; R A Good
Journal:  Am J Med       Date:  1968-04       Impact factor: 4.965

3.  The Wiskott-Aldrich syndrome. A disorder with a possible defect in antigen processing or recognition.

Authors:  R M Blaese; W Strober; R S Brown; T A Waldmann
Journal:  Lancet       Date:  1968-05-18       Impact factor: 79.321

4.  Defective actin reorganization and polymerization of Wiskott-Aldrich T cells in response to CD3-mediated stimulation.

Authors:  M D Gallego; M Santamaría; J Peña; I J Molina
Journal:  Blood       Date:  1997-10-15       Impact factor: 22.113

5.  Impaired dendritic-cell homing in vivo in the absence of Wiskott-Aldrich syndrome protein.

Authors:  Sofia de Noronha; Samantha Hardy; Joanna Sinclair; Michael P Blundell; Jessica Strid; Oliver Schulz; Jörg Zwirner; Gareth E Jones; David R Katz; Christine Kinnon; Adrian J Thrasher
Journal:  Blood       Date:  2004-10-19       Impact factor: 22.113

6.  Missense mutations of the WASP gene cause intermittent X-linked thrombocytopenia.

Authors:  Lucia D Notarangelo; Cinzia Mazza; Silvia Giliani; Chiara D'Aria; Francesca Gandellini; Chiara Ravelli; Maria Grazia Locatelli; David L Nelson; Hans D Ochs; Luigi D Notarangelo
Journal:  Blood       Date:  2002-03-15       Impact factor: 22.113

7.  The Wiskott-Aldrich syndrome protein acts downstream of CD2 and the CD2AP and PSTPIP1 adaptors to promote formation of the immunological synapse.

Authors:  Karen Badour; Jinyi Zhang; Fabio Shi; Mary K H McGavin; Vik Rampersad; Lynne A Hardy; Deborah Field; Katherine A Siminovitch
Journal:  Immunity       Date:  2003-01       Impact factor: 31.745

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

Authors:  Angela Gismondi; Loredana Cifaldi; Cinzia Mazza; Silvia Giliani; Silvia Parolini; Stefania Morrone; Jordan Jacobelli; Elisabetta Bandiera; Luigi Notarangelo; Angela Santoni
Journal:  Blood       Date:  2004-03-04       Impact factor: 22.113

9.  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

10.  The Cdc42/Rac interactive binding region motif of the Wiskott Aldrich syndrome protein (WASP) is necessary but not sufficient for tight binding to Cdc42 and structure formation.

Authors:  M G Rudolph; P Bayer; A Abo; J Kuhlmann; I R Vetter; A Wittinghofer
Journal:  J Biol Chem       Date:  1998-07-17       Impact factor: 5.157

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  15 in total

1.  Modified lentiviral LTRs allow Flp recombinase-mediated cassette exchange and in vivo tracing of "factor-free" induced pluripotent stem cells.

Authors:  Johannes Kuehle; Soeren Turan; Tobias Cantz; Dirk Hoffmann; Julia D Suerth; Tobias Maetzig; Daniela Zychlinski; Christoph Klein; Doris Steinemann; Christopher Baum; Juergen Bode; Axel Schambach
Journal:  Mol Ther       Date:  2014-01-17       Impact factor: 11.454

2.  Clinical experience in T cell deficient patients.

Authors:  Theresa S Cole; Andrew J Cant
Journal:  Allergy Asthma Clin Immunol       Date:  2010-05-13       Impact factor: 3.406

3.  Combined de-novo mutation and non-random X-chromosome inactivation causing Wiskott-Aldrich syndrome in a female with thrombocytopenia.

Authors:  Boonchai Boonyawat; Santhosh Dhanraj; Fahad Al Abbas; Bozana Zlateska; Eyal Grunenbaum; Chaim M Roifman; Leslie Steele; Stephen Meyn; Victor Blanchette; Stephen W Scherer; Sabina Swierczek; Josef Prchal; Qili Zhu; Troy R Torgerson; Hans D Ochs; Yigal Dror
Journal:  J Clin Immunol       Date:  2013-08-14       Impact factor: 8.317

4.  Quality of Life of Patients with Wiskott Aldrich Syndrome and X-Linked Thrombocytopenia: a Study of the Primary Immune Deficiency Consortium (PIDTC), Immune Deficiency Foundation, and the Wiskott-Aldrich Foundation.

Authors:  Ami J Shah; Robert Sokolic; Brent Logan; Ziyan Yin; Sumathi Iyengar; Chris Scalchunes; Christina Mangurian; Michael Albert; Morton J Cowan
Journal:  J Clin Immunol       Date:  2019-10-16       Impact factor: 8.317

Review 5.  Neutropenia in primary immunodeficiency.

Authors:  Robert Sokolic
Journal:  Curr Opin Hematol       Date:  2013-01       Impact factor: 3.284

6.  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

7.  Intermittent low platelet counts hampering diagnosis of X-linked thrombocytopenia in children: report of two unrelated cases and a novel mutation in the gene coding for the Wiskott-Aldrich syndrome protein.

Authors:  Samuel Souza Medina; Lúcia Helena Siqueira; Marina Pereira Colella; Gabriela Goes Yamaguti-Hayakawa; Bruno Kosa Lino Duarte; Maria Marluce Dos Santos Vilela; Margareth Castro Ozelo
Journal:  BMC Pediatr       Date:  2017-06-22       Impact factor: 2.125

8.  A risk factor analysis of outcomes after unrelated cord blood transplantation for children with Wiskott-Aldrich syndrome.

Authors:  Zhanna Shekhovtsova; Carmem Bonfim; Annalisa Ruggeri; Samantha Nichele; Kristin Page; Amal AlSeraihy; Francisco Barriga; José Sánchez de Toledo Codina; Paul Veys; Jaap Jan Boelens; Karin Mellgren; Henrique Bittencourt; Tracey O'Brien; Peter J Shaw; Alicja Chybicka; Fernanda Volt; Federica Giannotti; Eliane Gluckman; Joanne Kurtzberg; Andrew R Gennery; Vanderson Rocha
Journal:  Haematologica       Date:  2017-03-02       Impact factor: 9.941

9.  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

Review 10.  TCR Signaling Abnormalities in Human Th2-Associated Atopic Disease.

Authors:  Joshua D Milner
Journal:  Front Immunol       Date:  2018-04-16       Impact factor: 7.561

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