We report herein the unusual case of a 35-year-old woman found to have angiolymphoid hyperplasia with eosinophilia (ALHE) of the breast. The patient was initially referred to our hospital for investigation of a well-circumscribed breast lump, 2.5 cm in diameter, and a clinical diagnosis of fibroadenoma was made. However, fine-needle aspiration cytology of the lump revealed an admixture of lymphocytes and eosinophils. Therefore, the lump was excised and microscopic examination showed features of ALHE. No evidence of recurrence of the lump has been seen in the 2 years since this excision. To the best of our knowledge, this is the first case to be documented of ALHE arising in the breast.