Palmoplantar Keratoderma in Slurp2-Deficient Mice

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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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Clouston syndrome (hidrotic ectodermal dysplasia) is characterized by the clinical triad of nail dystrophy, alopecia and palmoplantar hyperkeratosis. Clouston syndrome is transmitted as an autosomal dominant trait and caused by mutations in the GJB6 gene (13q12), encoding the gap junction protein connexin 30 (C × 30). At present, there is no treatment for the disease and management is purely su...

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ژورنال

عنوان ژورنال: Journal of Investigative Dermatology

سال: 2016

ISSN: 0022-202X

DOI: 10.1016/j.jid.2015.11.003