Angiolymphoid hyperplasia with eosinophilia (ALHE) is a benign angioproliferative disorder with an uncertain pathogenesis.1 The treatment of ALHE includes surgical excision, destructive therapies, and medical therapies, many of which are often ineffective.2 In this article, we describe the clinical response to medications targeting eosinophilic inflammation in the treatment of ALHE, including the novel use of dupilumab, which was associated with complete resolution.