A novel pathogenic variant in the corneodesmosin gene causing generalized inflammatory peeling skin syndrome with marked eosinophilia and trichorrhexis invaginata.

Pediatr Dermatol

Department of Dermatology, Starship Children's Health, Auckland District Health Board, Auckland, New Zealand.

Published: March 2022

AI Article Synopsis

  • Generalized inflammatory peeling skin syndrome (PSS) is a rare genetic skin disorder linked to mutations in the corneodesmosin gene, leading to excessive skin shedding.
  • An infant case of PSS was reported, presenting with severe skin symptoms just two days after birth, including abnormal skin peeling and redness.
  • Genetic testing confirmed a complete deletion of the corneodesmosin gene, thus supporting the diagnosis of this specific type of skin syndrome.

Article Abstract

Generalized inflammatory peeling skin syndrome (PSS) is a rare autosomal recessive genodermatosis caused by loss-of-function disease-causing variants of the corneodesmosin gene (CDSN), resulting in excessive shedding of the superficial layers of the epidermis. We describe a case of generalized inflammatory PSS in an infant, presenting at day two of life with ichthyosiform erythroderma and superficial peeling of the skin. Hair microscopy showed trichorrhexis invaginata. Normal amounts of skin LEKT1, a product of SPINK5 on immunohistochemical staining excluded a diagnosis of Netherton syndrome. Genetic analysis revealed a homozygous novel complete CDSN deletion, estimated 4.6 kb in size, supporting the diagnosis of generalized inflammatory PSS.

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Source
http://www.ncbi.nlm.nih.gov/pmc/articles/PMC9305742PMC
http://dx.doi.org/10.1111/pde.14939DOI Listing

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