Document Detail


The spectra of clinical phenotypes in aplasia cutis congenita and terminal transverse limb defects.
MedLine Citation:
PMID:  19610107     Owner:  NLM     Status:  MEDLINE    
Abstract/OtherAbstract:
The combination of aplasia cutis congenita (ACC) and terminal transverse limb defects (TTLD) is often referred to as the eponymous Adams-Oliver syndrome (AOS). The molecular basis of this disorder remains unknown, although the common occurrence of cardiac and vascular anomalies suggests a primary defect of vasculogenesis. Through the description of three previously unreported affected individuals, ascertained through the Adams-Oliver Syndrome European Consortium, we illustrate the phenotypic variability characteristically observed within extended families with AOS. Taken in combination with a detailed review of the available literature, we provide evidence for distinct clinical entities within the ACC/TTLD spectrum, which may reflect genetic heterogeneity within this spectrum of disorders.
Authors:
Katie M G Snape; Deborah Ruddy; Martin Zenker; Wim Wuyts; Margo Whiteford; Diana Johnson; Wayne Lam; Richard C Trembath
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Publication Detail:
Type:  Case Reports; Journal Article; Research Support, Non-U.S. Gov't; Review    
Journal Detail:
Title:  American journal of medical genetics. Part A     Volume:  149A     ISSN:  1552-4833     ISO Abbreviation:  Am. J. Med. Genet. A     Publication Date:  2009 Aug 
Date Detail:
Created Date:  2009-07-30     Completed Date:  2009-09-30     Revised Date:  -    
Medline Journal Info:
Nlm Unique ID:  101235741     Medline TA:  Am J Med Genet A     Country:  United States    
Other Details:
Languages:  eng     Pagination:  1860-81     Citation Subset:  IM    
Copyright Information:
2009 Wiley-Liss, Inc.
Affiliation:
Guy's and St Thomas' NHS Hospital Trust/Kings College London, NIHR Biomedical Research Centre, UK. richard.trembath@kcl.ac.uk
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MeSH Terms
Descriptor/Qualifier:
Abnormalities, Multiple / pathology
Child
Child, Preschool
Chromosome Aberrations
Diagnosis, Differential
Ectodermal Dysplasia / complications*,  diagnosis,  pathology*
Female
Humans
Infant
Infant, Newborn
Limb Deformities, Congenital / complications*,  diagnosis,  pathology*
Phenotype

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


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