Phenotypic Variability with SLURP1 Mutations and Diffuse Palmoplantar Keratoderma

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Diffuse epidermolytic palmoplantar keratoderma (Unna-Thost-).

DESCRIPTION We report the case of a 28-year-old man presenting to our hospital with refractory diffuse hyperkeratosis of palms and soles. He reported first appearance in early childhood. His first-born daughter also developed hyperkeratosis on palms and soles directly after birth. No other family member seems to be affected by similar skin lesions. Clinically, our patient presented diffuse hype...

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Palmoplantar keratoderma along with neuromuscular and metabolic phenotypes in Slurp1-deficient mice

Mutations in SLURP1 cause mal de Meleda, a rare palmoplantar keratoderma (PPK). SLURP1 is a secreted protein that is expressed highly in keratinocytes but has also been identified elsewhere (e.g., spinal cord neurons). Here, we examined Slurp1-deficient mice (Slurp1(-/-)) created by replacing exon 2 with β-gal and neo cassettes. Slurp1(-/-) mice developed severe PPK characterized by increased k...

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Desmoplakin mutations with palmoplantar keratoderma, woolly hair and cardiomyopathy.

Mutations in genes encoding for desmosomal components are associated with a broad spectrum of phenotypes comprising skin and hair abnormalities and account for 45-50% of cases of arrhythmogenic right ventricular cardiomyopathy. Today, more than 120 dominant and recessive desmoplakin (DSP) gene mutations have been reported to be associated with skin, hair and/or heart defects. Here we report on ...

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Dear Editor, Clouston syndrome, also called hidrotic ectodermal dysplasia (HED), is an autosomal dominant ectodermal dysplasia characterized by a clinical triad of onychodystrophy, generalized hypotrichosis, and palmoplantar keratoderma (1). Herein we report the case of a 24-year-old male with the distinctive clinical triad associated with multiple epidermoid cysts, which probably reflects the ...

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ژورنال

عنوان ژورنال: Acta Dermato Venereologica

سال: 2020

ISSN: 0001-5555

DOI: 10.2340/00015555-3404