Angiolymphoid Hyperplasia With Eosinophilia Within a Vascular Malformation: Case Report and Review of the Literature. (September 2016)

• Record Type: Journal Article
• Authors: Fontana, Stefani C.; Borgstadt, Ashley; Fraga, Garth R.; Reeves, Alan R.; Andrews, Brian T.
• Is Part Of: Annals of otology, rhinology & laryngology. Volume 125:Number 9(2016:Sep.)
• Page Start: 775
• Page End: 778
• Publication Date: 2016-09
• Subjects: vascular malformation -- angiolymphoid hyperplasia with eosinophilia -- ALHE; Otolaryngology -- Periodicals; 617.51
• DOI: 10.1177/0003489416646796
• Languages: English
• ISSNs: 0003-4894

Abstract

Objectives: 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. Methods: 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. Results: 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. Conclusion: 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.