Document Detail


A novel mutation in a Fijian boy with Shwachman-Diamond syndrome.
MedLine Citation:
PMID:  19816210     Owner:  NLM     Status:  MEDLINE    
Abstract/OtherAbstract:
Shwachman-Diamond syndrome (SDS) is an autosomal recessive disorder characterized by pancreatic insufficiency, bone marrow dysfunction, and metaphyseal chondrodysplasia. SDS is associated with mutations in the Shwachman-Bodian-Diamond Syndrome gene, with 90% of reported mutations in exon 2. We present a Fijian boy with SDS who has a novel A>G substitution in exon 1 of the Shwachman-Bodian-Diamond Syndrome gene that has not been reported in the literature. This patient's unique clinical course includes the presence of a cleft lip and episodic hypoglycemia. SDS lacks a clear genotype-phenotype correlation, as is showed by the heterogeneity in its clinical presentation.
Authors:
Aimee R Newman; Billur Moghaddam; Janet M Yoon
Publication Detail:
Type:  Case Reports; Journal Article    
Journal Detail:
Title:  Journal of pediatric hematology/oncology     Volume:  31     ISSN:  1536-3678     ISO Abbreviation:  J. Pediatr. Hematol. Oncol.     Publication Date:  2009 Nov 
Date Detail:
Created Date:  2009-11-06     Completed Date:  2009-12-08     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:  847-9     Citation Subset:  IM    
Affiliation:
Department of Pediatrics, University of California, Davis Medical Center, Sacramento, CA, USA.
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MeSH Terms
Descriptor/Qualifier:
Abnormalities, Multiple / genetics*,  pathology
Fiji
Humans
Infant, Newborn
Male
Mutation, Missense*
Proteins / genetics*
Chemical
Reg. No./Substance:
0/Proteins; 0/SBDS protein, human

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


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