Document Detail


Cutaneous findings in a new syndrome of autosomal recessive ectodermal dysplasia with corkscrew hairs.
MedLine Citation:
PMID:  1479096     Owner:  NLM     Status:  MEDLINE    
Abstract/OtherAbstract:
BACKGROUND: The association of hair shaft abnormalities with the phenotypic findings of a new, distinct form of an autosomal recessive syndrome of ectodermal dysplasia was present in 27 patients from seven families. OBJECTIVE: Our purpose was to present the cutaneous findings that characterize this syndrome with particular attention given to the hair shaft abnormalities. METHODS: Multiple field visits were used to gather data on phenotypic findings and prospectively evaluate their prevalence. RESULTS: Corkscrew hair, an exaggeration of pili torti, represents the most striking feature of this syndrome. Prominent cutaneous findings include scalp keloids, follicular plugging, keratosis pilaris, xerosis, eczema, palmoplantar keratodermia, cutaneous syndactyly, onychodysplasia, and conjunctival neovascularization. Other features include typical facies, anteverted pinnae, malar hypoplasia, cleft lip and palate, and dental abnormalities. CONCLUSION: A syndrome characterized by pili torti and corkscrew hairs, previously reported in only one patient, can be recognized.
Authors:
W Abramovits-Ackerman; T Bustos; V Simosa-Leon; L Fernandez; M Ramella
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Publication Detail:
Type:  Journal Article    
Journal Detail:
Title:  Journal of the American Academy of Dermatology     Volume:  27     ISSN:  0190-9622     ISO Abbreviation:  J. Am. Acad. Dermatol.     Publication Date:  1992 Dec 
Date Detail:
Created Date:  1993-02-08     Completed Date:  1993-02-08     Revised Date:  2004-11-17    
Medline Journal Info:
Nlm Unique ID:  7907132     Medline TA:  J Am Acad Dermatol     Country:  UNITED STATES    
Other Details:
Languages:  eng     Pagination:  917-21     Citation Subset:  IM    
Affiliation:
Family Practice (Dermatology), University of Texas Health Sciences Center, San Antonio.
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MeSH Terms
Descriptor/Qualifier:
Adolescent
Adult
Aged
Child
Child, Preschool
Ectodermal Dysplasia / genetics*
Female
Hair / abnormalities*,  pathology
Humans
Infant
Male
Middle Aged
Prospective Studies
Syndrome
Comments/Corrections
Comment In:
J Am Acad Dermatol. 1994 Feb;30(2 Pt 1):289-90   [PMID:  8141890 ]

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


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