Document Detail


A unique clinical presentation of X-linked lymphoproliferative syndrome with a novel mutation in SH2D1A and review of the literature.
MedLine Citation:
PMID:  20975587     Owner:  NLM     Status:  MEDLINE    
Abstract/OtherAbstract:
X-linked lymphoproliferative syndrome is a well-described syndrome often characterized by progression to fatal infectious mononucleosis. Many mutations of the SH2D1A gene have been identified in patients with X-linked lymphoproliferative syndrome. These mutations are often associated with either decreased or impaired function of the protein product, signaling lymphocytic activation molecule-associated protein. We describe a patient with a novel missense mutation in SH2D1A. We report on his unique presentation, clinical course and subsequent successful treatment with a matched unrelated donor bone marrow transplant.
Authors:
Michael J Eckrich; Elizabeth Yang; Jennifer Domm; Richard Ho; Cassie Calder; Becky Manes; Jack Bleesing; Haydar Frangoul
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Publication Detail:
Type:  Case Reports; Journal Article; Review    
Journal Detail:
Title:  Journal of pediatric hematology/oncology     Volume:  33     ISSN:  1536-3678     ISO Abbreviation:  J. Pediatr. Hematol. Oncol.     Publication Date:  2011 Jan 
Date Detail:
Created Date:  2010-12-23     Completed Date:  2011-02-18     Revised Date:  2011-10-06    
Medline Journal Info:
Nlm Unique ID:  9505928     Medline TA:  J Pediatr Hematol Oncol     Country:  United States    
Other Details:
Languages:  eng     Pagination:  e39-42     Citation Subset:  IM    
Affiliation:
Department of Pediatrics, Division of Hematology/Oncology, Vanderbilt University, School of Medicine, USA.
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MeSH Terms
Descriptor/Qualifier:
Adolescent
Bone Marrow Transplantation
Humans
Intracellular Signaling Peptides and Proteins / genetics*
Lymphoproliferative Disorders / genetics*,  therapy
Male
Mutation, Missense
Tissue Donors
Chemical
Reg. No./Substance:
0/Intracellular Signaling Peptides and Proteins; 0/SH2D1A protein, human

From MEDLINE®/PubMed®, a database of the U.S. National Library of Medicine


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