Document Detail


Congenital heart defects in two siblings in an Axenfeld-Rieger syndrome family.
MedLine Citation:
PMID:  20179581     Owner:  NLM     Status:  MEDLINE    
Abstract/OtherAbstract:
Axenfeld-Rieger syndrome is a genetically heterogeneous, autosomal dominant disorder characterized by anomalies of the anterior segment of the eye, face, teeth, and umbilicus. Many other extraocular findings, including congenital heart defects, have been reported in association with this syndrome. It has been suggested by some investigators that the coexistence of Axenfeld-Rieger syndrome and congenital heart defects is not a chance event but it represents a distinct entity. We report a family in which four members in three generations have typical ocular features of Axenfeld-Rieger syndrome. Two of them, who are siblings, also have congenital heart defects. The congenital heart defect was bicuspid aortic valve anomaly with severe stenosis and mild regurgitation in one sibling and ostium secundum atrial septal defect in the other. To our knowledge, the combination of congenital heart defects with Axenfeld-Rieger syndrome in siblings has not been reported previously. Our observation further strengthens the notion that Axenfeld-Rieger syndrome associated with congenital heart defects is not a chance event.
Authors:
Mehmet Necdet Akkus; Atilla Argin
Publication Detail:
Type:  Case Reports; Journal Article    
Journal Detail:
Title:  Clinical dysmorphology     Volume:  19     ISSN:  1473-5717     ISO Abbreviation:  Clin. Dysmorphol.     Publication Date:  2010 Apr 
Date Detail:
Created Date:  2010-03-10     Completed Date:  2010-05-26     Revised Date:  -    
Medline Journal Info:
Nlm Unique ID:  9207893     Medline TA:  Clin Dysmorphol     Country:  England    
Other Details:
Languages:  eng     Pagination:  56-61     Citation Subset:  IM    
Affiliation:
Departments of Cardiology, Mersin University School of Medicine, Turkey. mnakkus@hotmail.com
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MeSH Terms
Descriptor/Qualifier:
Abnormalities, Multiple / pathology*
Adult
Aortic Valve / pathology,  ultrasonography
Eye Abnormalities / complications*
Family
Female
Heart Defects, Congenital / complications*,  ultrasonography
Humans
Infant
Infant, Newborn
Male
Pregnancy
Siblings*
Syndrome
Young Adult

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


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