CEDNIK syndrome with phenotypic variability

Pediatr Dermatol. 2022 Jul;39(4):650-652. doi: 10.1111/pde.14961. Epub 2022 Mar 1.

Abstract

CEDNIK syndrome is a rare autosomal recessive syndrome characterized by cerebral dysgenesis, neuropathy, ichthyosis, and keratoderma of which 25 cases from 19 families have been reported to date. It is a progressive neurodegenerative disorder caused by the loss-of-function pathogenic variant of the SNAP29 gene encoding a member of the SNARE family of proteins. We describe two female siblings from a Syrian parent-related family with CEDNIK syndrome due to homozygous pathogenic variant in SNAP29 [c.223delG(p.Val75Serf*28)]. Palmoplantar keratoderma, reported as a cardinal sign in CEDNIK syndrome, was absent in both patients as of the last follow-up, and one of our patients had a verrucous venous malformation, a finding that has not been previously reported.

Keywords: CEDNIK syndrome; palmoplantar keratoderma; phenotypic variability.

MeSH terms

  • Biological Variation, Population
  • Female
  • Humans
  • Keratoderma, Palmoplantar* / diagnosis
  • Keratoderma, Palmoplantar* / genetics
  • Neurocutaneous Syndromes
  • Qb-SNARE Proteins / genetics
  • Qc-SNARE Proteins* / genetics

Substances

  • Qb-SNARE Proteins
  • Qc-SNARE Proteins

Supplementary concepts

  • Cerebral dysgenesis, neuropathy, ichthyosis, and palmoplantar keratoderma syndrome