Estrogen receptors and the response to sex hormones in angiolymphoid hyperplasia with eosinophilia

Abstract

Background: Angiolymphoid hyperplasia (ALH) with eosinophilia is a benign rare tumor, characterized by marked proliferation of endothelial cells. The tumors are associated with extensive infiltrate of lymphocytes, histiocytes, and eosinophils, and occur on the head and neck of young adults. A variety of treatments have been attempted with frequent recurrences. We report two cases of ALH with eosinophilia that seemed to be dependent on sex hormones.

Observation: The first case is a patient with ALH that resolved after stopping treatment with birth control pills. Biopsy specimens of the tumor demonstrated increased level of estrogen and progesterone receptors compared with her normal skin. The second case is a patient with a previous lesion of ALH with eosinophilia, who during pregnancy had new lesions develop and whose primary lesion increased in size. All lesions decreased to half their original sizes after pregnancy.

Conclusion: Both of these cases suggest a role for hyperestrogen states with the presence of hormonal receptors. The response to sex hormones could contribute to the pathogenesis of the disease and may offer future alternative treatment modalities.