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Inverted ductal papilloma is an extremely rare disease, which is a benign salivary gland tumor classified as the ductal papilloma. It is characterized by hyperplasia of ductal epithelial cells near the orifice with intraluminal papillary projections. Clinically, it is presented as a painless submucosal nodular lesion with benign nature. Herein, we present a 76-year-old man with a tumor in upper lip. Surgical excision was performed and the histological diagnosis was inverted ductal papilloma with no detection of human papillomavirus. Due to its rarity, the exact etipathogenesis of inverted ductal papilloma remains unclear. The possible etiology and clinicopathologic features of this tumor are presented with a review of literature.
