Document Detail


Ectrodactyly-Ectodermal Dysplasia-Clefting Syndrome Associated With p63 Mutation and an Uncommon Phenotype.
MedLine Citation:
PMID:  20180707     Owner:  NLM     Status:  In-Data-Review    
Abstract/OtherAbstract:
Abstract Ectrodactyly-ectodermal dysplasia-clefting syndrome is an uncommon disorder that includes a clinical spectrum of limb, facial, ocular, internal ear, and urogenital malformations. The disease is caused by heterozygous mutations in the 3q27-29 located p63 gene. In this paper we describe a 17-year-old girl affected by ectrodactyly-ectodermal dysplasia-clefting syndrome with a de novo p63 mutation that predicts a heterozygous missense substitution (arginine to tryptophan substitution caused by a cytosine to thymine transition) at the amino acid 304 (R304W) of the p63 DNA-binding domain. Scattered freckles on face, legs, and abdominal region, an uncommon feature associated with this syndrome, were recognized in our patient. The clinical features and genotype-phenotype correlation with previous p63 mutations related to the syndrome are discussed and compared with those observed in our patient. This case expands the phenotypic spectrum of ectrodactyly-ectodermal dysplasia-clefting syndrome.
Authors:
Lívia Máris Ribeiro Paranaíba; Hercílio Martelli-Júnior; Roseli Teixeira de Miranda; Andréia Bufalino; Ruy Camargo Abdo Filho; Ricardo D Coletta
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Publication Detail:
Type:  Journal Article    
Journal Detail:
Title:  The Cleft palate-craniofacial journal : official publication of the American Cleft Palate-Craniofacial Association     Volume:  47     ISSN:  1545-1569     ISO Abbreviation:  Cleft Palate Craniofac. J.     Publication Date:  2010 Sep 
Date Detail:
Created Date:  2010-09-08     Completed Date:  -     Revised Date:  -    
Medline Journal Info:
Nlm Unique ID:  9102566     Medline TA:  Cleft Palate Craniofac J     Country:  United States    
Other Details:
Languages:  eng     Pagination:  544-7     Citation Subset:  D; IM    
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