Literature DB >> 21906397

Prolonged survival after splenectomy in Wiskott-Aldrich syndrome: a case report.

Kostas N Syrigos1, Nektaria Makrilia, Jeffrey Neidhart, Michael Moutsos, Sotirios Tsimpoukis, Maria Kiagia, Muhammad W Saif.   

Abstract

Wiskott-Aldrich syndrome is a rare X-linked immunodeficiency disorder that is characterized by a variable clinical phenotype. Matched donor bone marrow transplantation is currently the only curative therapeutic option. We present the case of a 24-year-old male who was diagnosed at the age of seven with Wiskott-Aldrich syndrome. He did not respond to intravenous gammaglobulin and he experienced recurrent pulmonary infections despite prophylactic antibiotics. The patient had no matched donor. At the age of nine, he was submitted to splenectomy and his platelet count was normalized. Fifteen years later, the patient remains asymptomatic with a normal platelet count. He is still receiving prophylactic antibiotics and no bleeding episodes or septic complications have been reported. This case demonstrates that splenectomy can represent a safe therapeutic option in selected WAS patients, provided that there is a tight follow-up program, patient education and adherence to guidelines regarding post-splenectomy prophylaxis.

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Year:  2011        PMID: 21906397      PMCID: PMC3179709          DOI: 10.1186/1824-7288-37-42

Source DB:  PubMed          Journal:  Ital J Pediatr        ISSN: 1720-8424            Impact factor:   2.638


  17 in total

1.  An international study examining therapeutic options used in treatment of Wiskott-Aldrich syndrome.

Authors:  Mary Ellen Conley; Delphine Saragoussi; Luigi Notarangelo; Amos Etzioni; Jean Laurent Casanova
Journal:  Clin Immunol       Date:  2003-12       Impact factor: 3.969

Review 2.  Clinical spectrum, pathophysiology and treatment of the Wiskott-Aldrich syndrome.

Authors:  Michael H Albert; Luigi D Notarangelo; Hans D Ochs
Journal:  Curr Opin Hematol       Date:  2011-01       Impact factor: 3.284

3.  X-linked thrombocytopenia (XLT) due to WAS mutations: clinical characteristics, long-term outcome, and treatment options.

Authors:  Michael H Albert; Tanja C Bittner; Shigeaki Nonoyama; Lucia Dora Notarangelo; Siobhan Burns; Kohsuke Imai; Teresa Espanol; Anders Fasth; Isabelle Pellier; Gabriele Strauss; Tomohiro Morio; Benjamin Gathmann; Jeroen G Noordzij; Cristina Fillat; Manfred Hoenig; Michaela Nathrath; Alfons Meindl; Philipp Pagel; Uwe Wintergerst; Alain Fischer; Adrian J Thrasher; Bernd H Belohradsky; Hans D Ochs
Journal:  Blood       Date:  2010-02-19       Impact factor: 22.113

4.  Stem cell transplantation for the Wiskott-Aldrich syndrome: a single-center experience confirms efficacy of matched unrelated donor transplantation.

Authors:  S-Y Pai; D DeMartiis; C Forino; S Cavagnini; A Lanfranchi; S Giliani; D Moratto; C Mazza; F Porta; L Imberti; L D Notarangelo; E Mazzolari
Journal:  Bone Marrow Transplant       Date:  2006-10-02       Impact factor: 5.483

Review 5.  Ten years of gene therapy for primary immune deficiencies.

Authors:  Alessandro Aiuti; Maria Grazia Roncarolo
Journal:  Hematology Am Soc Hematol Educ Program       Date:  2009

6.  Splenectomy and/or bone marrow transplantation in the management of the Wiskott-Aldrich syndrome: long-term follow-up of 62 cases.

Authors:  C A Mullen; K D Anderson; R M Blaese
Journal:  Blood       Date:  1993-11-15       Impact factor: 22.113

7.  Immunologic studies before and after splenectomy in a patient with the Wiskott-Aldrich syndrome.

Authors:  A P Knutsen; W F Rosse; T R Kinney; R H Buckley
Journal:  J Clin Immunol       Date:  1981-01       Impact factor: 8.317

8.  Update of guidelines for the prevention and treatment of infection in patients with an absent or dysfunctional spleen.

Authors:  J M Davies; R Barnes; D Milligan
Journal:  Clin Med (Lond)       Date:  2002 Sep-Oct       Impact factor: 2.659

9.  Long-term outcome following hematopoietic stem-cell transplantation in Wiskott-Aldrich syndrome: collaborative study of the European Society for Immunodeficiencies and European Group for Blood and Marrow Transplantation.

Authors:  Hulya Ozsahin; Marina Cavazzana-Calvo; Luigi D Notarangelo; Ansgar Schulz; Adrian J Thrasher; Evelina Mazzolari; Mary A Slatter; Francoise Le Deist; Stephane Blanche; Paul Veys; Anders Fasth; Robbert Bredius; Petr Sedlacek; Nico Wulffraat; Juan Ortega; Carsten Heilmann; Anne O'Meara; Jacek Wachowiak; Krzysztof Kalwak; Susanne Matthes-Martin; Tayfun Gungor; Aydan Ikinciogullari; Paul Landais; Andrew J Cant; Wilhelm Friedrich; Alain Fischer
Journal:  Blood       Date:  2007-09-27       Impact factor: 22.113

Review 10.  Wiskott-Aldrich Syndrome: Immunodeficiency resulting from defective cell migration and impaired immunostimulatory activation.

Authors:  Gerben Bouma; Siobhan O Burns; Adrian J Thrasher
Journal:  Immunobiology       Date:  2009-07-22       Impact factor: 3.144

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  1 in total

1.  Inherited Thrombocytopenia with a Different Type of Gene Mutation: A Brief Literature Review and Two Case Studies.

Authors:  Mohammad Taghi Arzanian
Journal:  Iran J Pediatr       Date:  2016-07-18       Impact factor: 0.364

  1 in total

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