BACKGROUND: Angiolymphoid hyperplasia with eosinophilia (ALHE) is a rare disease manifested by the proliferation of morphologically distinct endothelial cells. OBJECTIVE: To illustrate by a case report the clinical and varied histopathologic findings of ALHE. METHODS: A 29-year-old woman presented with a clinical picture of ALHE but had several histologic features of angiosarcoma. RESULTS: Management of this patient included repeat biopsies of the lesions, excision of the involved areas, careful histologic examination of the entire specimen, and appropriate follow-up. CONCLUSION: ALHE may present with various histologic features. Knowledge of the spectrum of benign and malignant vascular neoplasms helps manage these challenging cases.

Angiolymphoid hyperplasia with eosinophilia of the tongue: report of a case and review of the literature.Oral Oncol. 2002 Jan;38(1):103-6.

Angiolymphoid hyperplasia with eosinophilia (ALHE) is an uncommon disorder manifested as solitary or multiple nodules in dermis and subcutaneous tissue of head and neck region. ALHE affecting tongue is very rare. We report a 56-year-old man with ALHE of the tongue and review the previously reported cases of ALHE of the tongue.

Angiolymphoid hyperplasia with eosinophilia--report of a case with reference to the vascular changes and cell infiltrates in this disease.Nippon Hifuka Gakkai Zasshi. 1990 Aug;100(9):935-46.

We reported angiolymphoid hyperplasia with eosinophilia (ALHE) in a 47-year-old woman. The most characteristic histological findings were epithelioid endothelial cells with intracytoplasmic vacuoles, and remarkable arterio-venous (A-V) shunts. Mast cells, seen in the lesions, had no special relationship with eosinophils in their distribution. Degranulation of mast cells was not seen. Electron microscopic study revealed abundant cytoplasmic organellae and fine filaments in epithelioid endothelial cells. Immunohistochemical study revealed mixed infiltration of T and B lymphocytes, and polymorphous T-cell population in the lesions. These findings suggest that active vascular proliferations take place in the lesions of ALHE, that the eosinophilic infiltrate and mast cells may be independent of each other, and that the lymphocytic infiltrate probably is a reactive process. The vascular changes seen in Kimura's disease are characterized by a proliferation of capillaries with swollen, but not epithelioid, endothelial cells and vessels with lamellated adventitia. A-V shunts are not observed in its lesions. Therefore ALHE and Kimura's disease should be considered different entities.