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Two for two: Dual therapy with erlotinib and acitretin for twins with severe keratoderma in Olmsted syndrome. Pediatr Dermatol 2023;40(4):735-737

Date

01/30/2023

Pubmed ID

36709954

DOI

10.1111/pde.15264

Scopus ID

2-s2.0-85147304534 (requires institutional sign-in at Scopus site)   4 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.

Author List

Butala S, Phan S, Siegel DH, Carlberg V, Paller AS

Author

Valerie M. Carlberg MD Associate Professor in the Dermatology department at Medical College of Wisconsin




MESH terms used to index this publication - Major topics in bold

Acitretin
Child, Preschool
ErbB Receptors
Erlotinib Hydrochloride
Humans
Keratoderma, Palmoplantar
Pain