Angiolymphoid Hyperplasia with Eosinophilia of Orbit in Young Male

Abstract

Angiolymphoid hyperplasia with eosinophilia (ALHE) is an uncommon benign clinical entity characterized by the presence of a variable number of papules, plaques or nodules of the dermis and subcutaneous tissues. ALHE shows a predilection for the head and neck area. Orbital involvement is unusual. A 20‑year‑old male presented with a 4‑month history of swelling of the upper lid of right eye. Magnetic resonance imaging orbit revealed altered signal density seen in supero‑medial aspect of right eyeball with adjacent inflammatory changes. The patient underwent anterior orbitotomy and excision biopsy of the mass under general anesthesia. Histopathology of the mass showed vascular proliferation and chronic inflammation accompanied by infiltration of eosinophils, which confirmed the diagnosis of ALHE. The patient recovered both functionally and cosmetically and had no signs of recurrence after 6 months. Angiolymphoid hyperplasia with eosinophilia with orbital involvement in males is a rare clinical entity, and further work is required to accurately describe its incidence, etiology and presentation. ALHE can be diagnosed and differentiated from Kimura’s disease (KD) on histopathological grounds. The presence of vascular hyperplasia with plump of endothelial cells protruding into the lumen is the most important discriminator in establishing the diagnosis of ALHE. Such distinction is crucial for the patient because ALHE is not associated with any of the systemic manifestations present in KD.

Keywords: Angiolymphoid hyperplasia, eosinophilia, orbit