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Abstract:Background: Adenomyoepithelioma is an uncommon neoplasm that occurs almost exclusively in the breast and rarely described at other sites. It is a biphasic tumor that arises from both myoepithelial and epithelial cells.Case: We present a case of cutaneous adenomyoepithelioma in a 25 year old female patient presenting with a nodular lesion over the right cheek. Histopathological examination showed presence of dermal nodules composed of scattered tubules lined by cuboidal epithelial cells in a background sheet of myoepithelial cells. The epithelial and myoepithelial components were confirmed immunohistochemically using cytokeratin (CK) and smooth muscle antigen (SMA). Conclusion:Primary cutaneous adenomyoepithelioma is an uncommon benign tumor with a good prognosis. However, failure to achieve a free resection margin may result in local recurrence or rarely, malignant transformation. Therefore, it is important to make an accurate pathologic diagnosis followed by complete excision