Background: Myoepithelial cells are widely present in the breast, and their hyperplasia may result in a spectrum of disease ranging from myoepitheliosis to myoepithelial carcinoma. Case Report: A 46-year-old woman presented with a palpable mass in her right breast. Mammography and ultrasonography showed a lesion in the upper quadrant of the right breast with spiculated borders and shape. Excisional biopsy showed adenomyoepithelial adenosis. Conclusions: Although considered benign, adenomyoepithelial lesions tend to recur due to inadequate excision. Therefore, possibility of recurrence and even metastasis should be considered during follow-up of patients with a diagnosis of adenomyoepithelial lesions.