Peripheral ameloblastoma (PA) is a rare benign tumor that can occur mostly in the mandibular gingiva of the premolar area, originating from the residual odontogenic epithelium. The patient is usually asymptomatic and the lesion can be an occasional finding during routine intraoral examination. Due to the lack of clinical and radiographic pathognomonic features, the diagnosis is based on histopathological analysis, associated with 3D computed tomography (CT) imaging. Here, we report the case of a middle-aged man showing an asymptomatic, sessile, normochromic papillomatous mass of the lingual alveolar mucosa, in correspondence of tooth 4.4, which was histologically diagnosed as peripheral ameloblastoma. After the complete excision of the lesion, there were no recurrence and no symptoms during the 3-year follow-up. The treatment of choice for PA is a conservative surgical excision, which usually results in a good prognosis, together with a long-term follow-up, necessary to intercept possible recurrence or, more rarely, malignant transformation.