Document Detail


Bazex-Dupré-Christol syndrome in a 1-year-old boy and his mother.
MedLine Citation:
PMID:  18304168     Owner:  NLM     Status:  MEDLINE    
Abstract/OtherAbstract:
Bazex-Dupré-Christol syndrome is a rare genodermatosis with cancer predisposition, characterized by follicular atrophoderma, multiple milia, congenital hypotrichosis, hypohidrosis and basal cell malformations that include nevoid basal cell carcinomas of early onset. We present two patients with this syndrome, a 1-year-old boy with diffuse scalp and eyebrows alopecia, milia papules on the face, ears, trunk, and limbs. Hypohidrosis was observed on his trunk and head. His 16-year-old mother had identical changes since childhood, with hair fragility, and multiple atrophic "ice pick" follicular depressions on the dorsa of her hands. She also had a basal cell carcinoma on her face. Microscopic examination of hairs from the mother revealed abnormalities such as diameter irregularities, broken shafts, trichorrexis nodosa and pili bifurcatti. Pili bifurcatti is an uncommon hair shaft dysplasia that has not before been observed in Bazex-Dupré-Christol syndrome.
Authors:
Andrezza Camarinha Napolitano Barcelos; Marcello Menta Simonsen Nico
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Publication Detail:
Type:  Case Reports; Journal Article    
Journal Detail:
Title:  Pediatric dermatology     Volume:  25     ISSN:  1525-1470     ISO Abbreviation:  Pediatr Dermatol     Publication Date:    2008 Jan-Feb
Date Detail:
Created Date:  2008-02-28     Completed Date:  2008-04-07     Revised Date:  2009-03-03    
Medline Journal Info:
Nlm Unique ID:  8406799     Medline TA:  Pediatr Dermatol     Country:  United States    
Other Details:
Languages:  eng     Pagination:  112-3     Citation Subset:  IM    
Affiliation:
Department of Dermatology, Faculty of Medicine, University of São Paulo, São Paulo, Brazil. zza@ig.com.br
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MeSH Terms
Descriptor/Qualifier:
Acrodermatitis / diagnosis,  genetics
Adolescent
Basal Cell Nevus Syndrome / diagnosis,  genetics*
Carcinoma, Basal Cell / diagnosis,  pathology*
Cell Transformation, Neoplastic
Female
Heterozygote*
Humans
Hypohidrosis / diagnosis,  genetics
Hypotrichosis / diagnosis,  genetics
Infant
Male
Mothers
Pedigree
Precancerous Conditions / pathology*
Prognosis
Skin Neoplasms / diagnosis,  genetics*
Syndrome

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


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