Document Detail


Ectodermal dysplasia-skin fragility syndrome.
MedLine Citation:
PMID:  21727700     Owner:  NLM     Status:  In-Data-Review    
Abstract/OtherAbstract:
Ectodermal dysplasia-skin fragility (EDSF) syndrome is a rare and first described inherited disorder of desmosomes. It occurs due to loss-of-function mutations in PKP1 gene resulting in poorly formed desmosomes and loss of desmosomal and epidermal integrity. We report a case of a 2-year-old Indian male child who presented with palmoplantar hyperkeratosis with fissuring, short, sparse, and easily pluckable scalp hair, nail dystrophy, and multiple erosions over the skin. Skin biopsy showed epidermal hyperplasia with widening of intercellular spaces. His developmental milestones were delayed but intelligence was normal. Echocardiography, X-ray chest, and electrocardiogram were normal. Very few cases of this syndrome have been reported in the literature. We consider this as the first case report from India.
Authors:
Vijay S Adhe; Atul M Dongre; Uday S Khopkar
Publication Detail:
Type:  Journal Article    
Journal Detail:
Title:  Indian journal of dermatology, venereology and leprology     Volume:  77     ISSN:  0973-3922     ISO Abbreviation:  Indian J Dermatol Venereol Leprol     Publication Date:    2011 Jul-Aug
Date Detail:
Created Date:  2011-07-05     Completed Date:  -     Revised Date:  -    
Medline Journal Info:
Nlm Unique ID:  7701852     Medline TA:  Indian J Dermatol Venereol Leprol     Country:  India    
Other Details:
Languages:  eng     Pagination:  503-6     Citation Subset:  IM    
Affiliation:
Department of Dermatology, Seth GS Medical College and King Edward Memorial Hospital, Mumbai, Maharashtra, India.
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