Literature DB >> 21727700

Ectodermal dysplasia-skin fragility syndrome.

Vijay S Adhe1, Atul M Dongre, Uday S Khopkar.   

Abstract

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.

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Year:  2011        PMID: 21727700     DOI: 10.4103/0378-6323.82415

Source DB:  PubMed          Journal:  Indian J Dermatol Venereol Leprol        ISSN: 0378-6323            Impact factor:   2.545


  2 in total

1.  A plakophilin-1 gene mutation in an egyptian family with ectodermal dysplasia-skin fragility syndrome.

Authors:  Ebtesam M Abdalla; Cristina Has
Journal:  Mol Syndromol       Date:  2014-11-28

2.  Ectodermal Dysplasia-Skin Fragility Syndrome: A Rare Case Report.

Authors:  Subhash Kashyap; Vinay Shanker; Neelam Sharma
Journal:  Indian J Dermatol       Date:  2015 Jul-Aug       Impact factor: 1.494

  2 in total

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