Management Issues in the Treatment of an Ameloblastoma with an Atypical Presentation.

Benign ameloblastoma (BA) is the most common tumor arising from the odontogenic epithelium. Surgical resection with adequate margins is the mainstay of treatment due to the high index of tumor recurrence when not completely excised. Although locoregional spread has been described in the literature, it is very uncommon. We describe the treatment and follow-up of a 22-year-old woman with multiple recurrences and locoregional spread of a mandibular ameloblastoma who was referred to our center after several tumor resections with subsequent reconstructions. After a complete macroscopical removal of a new recurrence, the mandible was primarily reconstructed. A local homolateral recurrence and a second lesion in the contralateral maxilla were detected after 1-year follow-up and accordingly treated. After 4 years the patient showed a new tumor in the temporal fossa and was reoperated. The histopathology was consistent with a BA in all cases. Even though it is rare, locoregional spread of BA has been reported previously. Recurrences discovered during follow-up may require further resections. A close follow-up is mandatory, and treatment of these cases may result demanding requiring a multiple team approach, including oncologists and radiotherapists.