We describe a rare case of proliferative myositis affecting the lateral border of the tongue. The site of the lesion and its gross pathological presentation were highly suggestive of a malignant process. Subsequent biopsy and characteristic histological appearance led to the diagnosis of this benign condition. This is the first reported case of a painful presentation of proliferative myositis involving the tongue. This report serves to remind the head and neck surgeon of the need to obtain histological diagnosis of malignancy before embarking upon radical surgical treatment. We recommend careful follow-up to ensure complete resolution of the lesion.