Ameloblastoma is the commonest odontogenic tumour of epithelial origin with an incidence of one case per two million persons per year. Most patients’ ages range from 30 to 60 years with equal gender distribution. Despite being classified as a benign tumour, it shows locally aggressive features. It was related to inflammation, trauma and nutritional deficiencies. Yet, recent data strongly indicate that unique genetic abnormalities cause the development of ameloblastoma [1,2,3,4].
In the 2017 WHO classification of head and neck tumours, the terminology and classification of ameloblastoma were modified guided by the updates on genetic research enlightened with the studies published earlier which suggested that MAPK pathway mutations are a cornerstone in the pathogenesis of ameloblastoma. Ameloblastoma remained considered as a benign tumour despite its local aggressiveness and recurrence potentiality. The classification is now simple and limited to ameloblastoma, unicystic ameloblastoma, extraosseous ameloblastoma and malignant type. The conventional ameloblastoma is the commonest type followed by the unicystic one [1, 2, 5].
Ameloblastoma usually presents as a painless slowly growing tumour which can become a large and expansile swelling causing tooth loosening and displacement as well as facial asymmetry. CT scan is considered the gold standard for diagnosis [1, 4, 6].
Recurrence of ameloblastoma after surgical resection is not an uncommon event which reaches up to 15% even after adequate surgical resection. It can be attributed to many factors such as histological subtype, genetic characteristics of the tumour and type of surgical resection performed (conservative versus radical). It is advised to attack the primary tumour with a surgical strategy that is not so conservative to prevent recurrence and not so aggressive to prevent quality of life affection. Wide local excision with 1 to 2 cm safety margins is widely accepted. Management of recurrent ameloblastoma is a surgical challenge [1,2,3, 6,7,8]. The clinical presentation in our patient was in the form of two cystic swellings at the operative bed. CT as well as MRI was used in the preoperative evaluation. Surgical resection was challenging, and using two surgical approaches was necessary to achieve adequate resection with free margins.
Recurrence of ameloblastoma can occur in the remaining part of the mandible, surrounding soft tissue and even in the bone used for reconstruction whether in the form of graft, pedicled or free flap [6, 9]. In the presented patient, the recurrence occurred in both the operative bed and the bone used for reconstruction.
A very long time has been reported between initial presentation and recurrence exactly like our patient who had 17 years between the initial presentation and the recurrence. Essaadi et al. [7], de Miranda et al. [10] and Vasan [11] reported a patient with recurrence in the bony graft 33, 30 and 28 years, respectively, after the initial presentation.
