Peripheral ameloblastoma is an uncommon, extraosseous counterpart of solid ameloblastoma, which occurs in the soft tissues overlying tooth-bearing areas or the alveolar mucosa of the mandible and maxilla. In this paper, the authors report a case of peripheral ameloblastoma located in the maxillary gingiva of a 54-year-old woman and review the literature regarding clinicopathological features, differential diagnosis and therapeutic management of peripheral ameloblastomas.