Document Detail


Molecular pathogenesis in Diamond-Blackfan anemia.
MedLine Citation:
PMID:  20882441     Owner:  NLM     Status:  MEDLINE    
Abstract/OtherAbstract:
Diamond-Blackfan anemia (DBA) is a congenital anemia and a broad spectrum of developmental abnormalities that presents soon after birth. The anemia is due to a failure of erythropoiesis with normal platelet and myeloid lineages. Approximately 10-20% of DBA cases are inherited. Genetic studies have identified heterozygous mutations in at least one of eight ribosomal protein genes in up to 50% of cases. Mutations in RPL5 and RPL11 are at a high risk for developing malformation. Especially, mutations in RPL5 are associated with multiple physical abnormalities, including cleft lip/plate and thumb and heart anomalies. Recently, the 5q- syndrome, a subtype of myelodysplastic syndrome characterized by a defect in erythroid differentiation, is caused by a somatically acquired deletion of chromosome 5q, which results in haploinsufficiency of RPS14. These data indicate that abnormalities in ribosome function are broadly implicated in both congenital and acquired bone marrow failure syndrome in humans.
Authors:
Etsuro Ito; Yuki Konno; Tsutomu Toki; Kiminori Terui
Publication Detail:
Type:  Journal Article; Research Support, Non-U.S. Gov't; Review     Date:  2010-09-30
Journal Detail:
Title:  International journal of hematology     Volume:  92     ISSN:  1865-3774     ISO Abbreviation:  Int. J. Hematol.     Publication Date:  2010 Oct 
Date Detail:
Created Date:  2010-10-06     Completed Date:  2011-01-28     Revised Date:  -    
Medline Journal Info:
Nlm Unique ID:  9111627     Medline TA:  Int J Hematol     Country:  United States    
Other Details:
Languages:  eng     Pagination:  413-8     Citation Subset:  IM    
Affiliation:
Department of Pediatrics, Hirosaki University Graduate School of Medicine, Aomori, Japan. eturou@cc.hirosaki-u.ac.jp
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MeSH Terms
Descriptor/Qualifier:
Anemia, Diamond-Blackfan / diagnosis,  genetics*,  pathology,  physiopathology*
Animals
Chromosome Deletion
Chromosomes, Human, Pair 5 / genetics
Erythropoiesis
Humans
Mutation
Ribosomal Proteins / genetics
Ribosomes / genetics,  pathology
Chemical
Reg. No./Substance:
0/Ribosomal Proteins

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