Two for two: Dual therapy with erlotinib and acitretin for twins with severe keratoderma in Olmsted syndrome

Sneha Butala, Sheshanna Phan, Dawn H. Siegel, Valerie Carlberg, Amy S. Paller*

*Corresponding author for this work

Research output: Contribution to journalArticlepeer-review

1 Scopus citations

Abstract

Olmsted syndrome (OS) is a rare genetic disorder, characterized by painful palmoplantar keratoderma (PPK), periorificial and intertriginous hyperkeratoses, and alopecia. Fewer than 75 cases have been described. Variants in TRPV3 result in constitutive activation of transient receptor potential vanilloid 3, leading to increased epidermal growth factor receptor (EGFR) signaling, palmoplantar epidermal hyperproliferation, and exquisite lesional pain. We describe pre-school aged twins with OS with partial improvement from oral erlotinib, an EGFR inhibitor, but dramatic reduction of their persistent palmoplantar thickening and pain from adding acitretin.

Original languageEnglish (US)
Pages (from-to)735-737
Number of pages3
JournalPediatric dermatology
Volume40
Issue number4
DOIs
StatePublished - Jul 1 2023

Keywords

  • Olmsted syndrome
  • acitretin
  • erlotinib
  • keratoderma
  • pediatric dermatology

ASJC Scopus subject areas

  • Dermatology
  • Pediatrics, Perinatology, and Child Health

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