Document Detail


Temple-Baraitser syndrome: a rare and possibly unrecognized condition.
MedLine Citation:
PMID:  20683999     Owner:  NLM     Status:  MEDLINE    
Abstract/OtherAbstract:
Temple-Baraitser syndrome, previously described in two unrelated patients, is the association of severe mental retardation and abnormal thumbs and great toes. We report two additional unrelated patients with Temple-Baraitser syndrome, review clinical and radiological features of previously reported cases and discuss mode of inheritance. Patients share a consistent pattern of anomalies: hypo or aplasia of the thumb and great toe nails and broadening and/or elongation of the thumbs and halluces, which have a tubular aspect. All patients were born to unrelated parents and occurred as a single occurrence in multiple sibships, suggesting sporadic inheritance from a de novo mutation mechanism. Comparative genomic hybridization in Patients 1, 2 and 3 did not reveal any copy number variations. We confirm that Temple-Baraitser syndrome represents a distinct syndrome, probably unrecognized, possibly caused by a de novo mutation in a not yet identified gene.
Authors:
Adeline Jacquinet; Marion Gérard; Michael T Gabbett; Léon Rausin; Jean-Paul Misson; Björn Menten; Geert Mortier; Lionel Van Maldergem; Alain Verloes; François-Guillaume Debray
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Publication Detail:
Type:  Case Reports; Journal Article    
Journal Detail:
Title:  American journal of medical genetics. Part A     Volume:  152A     ISSN:  1552-4833     ISO Abbreviation:  Am. J. Med. Genet. A     Publication Date:  2010 Sep 
Date Detail:
Created Date:  2010-08-30     Completed Date:  2010-12-15     Revised Date:  -    
Medline Journal Info:
Nlm Unique ID:  101235741     Medline TA:  Am J Med Genet A     Country:  United States    
Other Details:
Languages:  eng     Pagination:  2322-6     Citation Subset:  IM    
Affiliation:
Center for Human Genetics, CHU & University of Liège, Liège, Belgium.
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MeSH Terms
Descriptor/Qualifier:
Abnormalities, Multiple
Adult
Child
Family
Female
Humans
Infant
Inheritance Patterns*
Male
Mental Retardation*
Middle Aged
Rare Diseases / diagnosis*,  genetics
Thumb / abnormalities*
Toes / abnormalities*

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


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