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<p>Abstract</p> <p>Introduction</p> <p>Oral melanoacanthoma is a rare, benign pigmented lesion characterized clinically by the sudden appearance and rapid growth of a macular brown-black lesion and histologically by acanthosis of the superficial epithelium and proliferation of dendritic melanocytes.</p> <p>Case presentation</p> <p>We present a case report of oral melanoacanthoma in a 24-year-old Asian Indian man. He presented with an intra-oral brown macular lesion on the left buccal mucosa with a duration of one and a half months. Microscopic examination revealed acanthosis of stratified squamous surface epithelium and dendritic melanocytes diffusely distributed in the epithelium; the Masson-Fontana silver impregnation technique was used to demonstrate the dendritic melanocytes. Based on the history, clinical features and histological presentation, the lesion was diagnosed as melanoacanthoma.</p> <p>Conclusion</p> <p>This is the first reported instance of oral melanoacanthoma in the Indian sub-continent. This report details the course of the lesion from diagnosis to its resolution. Melanoacanthoma must be differentiated from other intra-oral pigmented lesions and biopsy may be required to rule out melanoma.</p>