Peripheral ameloblastoma is a very rare tumour with histological characteristics similar to those of the centrally located ameloblastoma. It appears in the gum and in the alveolar process of the jaws. We report a case of tumour of the left maxillary region, near the canine area. Histologically it presented itself with follicles and nests of cells with a trabecular architectural pattern. High mitotic index and clear images of perineural infiltration were present. The diagnosis was peripheral ameloblastoma with malignant aspects. At 12-month follow-up no local recurrence was noted.