Document Detail


Dyskeratosis congenita.
MedLine Citation:
PMID:  19327580     Owner:  NLM     Status:  MEDLINE    
Abstract/OtherAbstract:
Dyskeratosis congenita (DC) is an inherited bone marrow failure syndrome characterized clinically by the triad of abnormal nails, reticular skin pigmentation, and oral leukoplakia, and is associated with high risk of developing aplastic anemia, myelodysplastic syndrome, leukemia, and solid tumors. Patients have very short germline telomeres, and approximately half have mutations in one of six genes encoding proteins that maintain telomere function. Accurate diagnosis of DC is critical to ensure proper clinical management, because patients who have DC and bone marrow failure do not respond to immunosuppressive therapy and may have increased morbidity and mortality associated with hematopoietic stem cell transplantation.
Authors:
Sharon A Savage; Blanche P Alter
Publication Detail:
Type:  Journal Article; Research Support, N.I.H., Intramural    
Journal Detail:
Title:  Hematology/oncology clinics of North America     Volume:  23     ISSN:  1558-1977     ISO Abbreviation:  Hematol. Oncol. Clin. North Am.     Publication Date:  2009 Apr 
Date Detail:
Created Date:  2009-03-30     Completed Date:  2009-05-26     Revised Date:  2010-09-23    
Medline Journal Info:
Nlm Unique ID:  8709473     Medline TA:  Hematol Oncol Clin North Am     Country:  United States    
Other Details:
Languages:  eng     Pagination:  215-31     Citation Subset:  IM    
Affiliation:
Clinical Genetics Branch, Division of Cancer Epidemiology and Genetics, National Cancer Institute, National Institutes of Health, 6120 Executive Boulevard, Rockville, MD 20852, USA. savagesh@mail.nih.gov
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MeSH Terms
Descriptor/Qualifier:
Bone Marrow Diseases / complications,  therapy
Dyskeratosis Congenita / complications*,  diagnosis*,  genetics,  therapy
Hematopoietic Stem Cell Transplantation
Humans
Immunosuppression
Telomere / pathology*
Grant Support
ID/Acronym/Agency:
Z01 CP010144-09/CP/NCI NIH HHS
Comments/Corrections

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