A Case of Angiolymphoid Hyperplasia with Eosinophilia of Eyelid

Mijung Chi

Purpose: We describe an angiolymphoid hyperplasia with eosinophilia (ALHE) that rarely developed in the eyelid

Methods: A 63 year-old male who had been diagnosed ALHE suspect by biopsy of inguinal mass presented with an eyelid mass of 1 month duration. Light brown, solitary, 1.0 x 0.5cm sized mass involved right upper eyelid. There was not lymphadenopathy but eosinophilia. An excisional biopsy of the mass was carried out for diagnosis and management.

Results: Macroscopic examination of the excised mass revealed a well-defined, smooth, firm, yellowish red color lesion measuring 1.0 x 0.6 x 0.5cm. Histopathology showed the presence of a proliferation of small blood vessels, many of which were lined by enlarged endothelial cells with uniform ovoid nuclei and intracytoplasmic vacuoles. These distinctive endothelial cells were described as having a cobblestone appearance. In addition, a perivascular and interstitial infiltrate composed primarily of lymphocytes and eosinophils was presented. ALHE was finally confirmed with clinical and microscopic examination.

Conclusions: ALHE is an uncommon idiopathic condition that manifests in adults aged 30-50 years as mean 1cm sized isolated nodules in the skin of the head and neck but rare in periocular region. A CBC count reveals eosinophilia in approximately 20% of patients and lymphadenopathy is rare. While Kimura disease, which shows some similarity to ALHE, involves deeper tissues as lymph nodes, salivary glands, and the subcutis of young males and presents lymphadenopathy and eosinophilia frequently. Simple surgical excision is often used but the lesions tend to recur. We report an ALHE and suggest that it should be considered a differential diagnosis of eyelid mass, even though it is rare.