Stefani C. Fontana, Ashley Borgstadt, Garth R. Fraga, Alan R. Reeves, Brian T. Andrews

Angiolymphoid hyperplasia with eosinophilia (ALHE) is a rare, benign microvascular proliferation tumor. The etiology of ALHE is unknown, though some hypothesize it occurs in reaction to focal trauma. This study presents a case of AHLE within a vascular malformation, its treatment, and a review of the literature. A retrospective case report was performed with a 4-year follow-up. Medical records including clinic notes, radiography, operative reports, pathology, and long-term follow-up were reviewed. Radiologic imaging revealed a vascular malformation with highly tortuous, corkscrew-shaped arterial vessels. Histology revealed midsized vessels with plump, epithelialized endothelium and focal areas of lymphocytic infiltrate punctuated with eosinophils, consistent with ALHE. This case demonstrates a unique presentation of ALHE in association with vascular malformation. Initial physical exam lacked the classic dermatologic presentation of clusters of red-to-brown nodules; however, the corkscrew vessels lined by epithelized endothelial cells and eosinophilic infiltrate were pathognomonic for ALHE.