A Case Report on Maxillary Ameloblastic Fibroma in a 55-Year-Old Man

Abstract

Ameloblastic Fibroma (AF) accounts for only 2% of all odontogenic tumors.1 It has been shown to be more common in males than females.2 A malignant transformation of this tumor is known as ameloblastic fibrosarcoma. To the best of our knowledge, there are no published reports on AF in the Philippines, with only one report of its malignant counterpart, ameloblastic fibrosarcoma.3 We present a rare case of AF with an unusual presentation and discuss its rarity, pathogenesis, histologic features and management. CASE REPORT A 55-year-old Filipino man consulted at our tertiary government hospital for a one-year history of a gradually enlarging maxillary alveolar ridge mass extending to the hard palate. Upon first noticing the mass a year prior, he consulted with a local dentist and an intraoral examination revealed a smooth swelling on the right maxillary alveolar ridge between the first and second maxillary premolar, approximately measuring 2 cm x 2 cm x 1 cm. The mass was noted to extend to the hard palate. (Figure 1A) He was advised consultation with an ENT surgeon for further evaluation and management but he did not comply. In the interim, the mass gradually enlarged. Upon first consultation at ITRMC, intraoral examination revealed a 6 cm x 4 cm x 2 cm soft, non-friable, ulcerative mass with irregular borders on the right maxillary alveolar ridge extending to the hard palate. (Figure 1B) The mass was malodorous, easily bled on manipulation and had violaceous areas. A punch biopsy revealed AF. Past medical history was unremarkable. A contrast-enhanced computed tomography (CECT) scan of the paranasal sinuses (PNS) revealed a 6.7 cm x 8.7 cm x 6.7 cm expansile, lobulated, mixed attenuating, heterogeneously enhancing mass in the midface. The bulk of the mass involved the right maxilla but extended to involve the entire hard palate and most of the left maxillary sinus. (Figure 2) The lesion was associated with osteolytic destruction and abutted the floor of the right orbit and bilateral zygomaticomaxillary buttresses. The inferior and posterior portions of the nasal septum were also eroded. (Figure 3) Given the clinical and radiographic features, odontogenic myxoma and ameloblastoma were considered as differential diagnoses as they may present as a painless, slow-growing, multiloculated mass with bony expansions.4,5 Oral cavity squamous cell carcinoma was also considered. The mass was excised via a right subtotal maxillectomy and left inferior maxillectomy using a Weber-Ferguson approach (Figure 4) and a temporary surgical obturator was fitted. Post-operative recovery was unremarkable and the patient was discharged after five days. Once the surgical site healed he was fitted with an acrylic surgical obturator. Final histopathological report was AF, compatible with the pre-operative biopsy result. (Figure 5) On follow up three months post-surgery, the patient was able to eat and speak with ease and no tumor recurrence was observed. One year and three weeks later, there was still no recurrence noted on inspection of the surgical defect, his speech was hypernasal but understandable, and he had no difficulty communicating and eating.

Keywords

• odontogenic tumors, ameloblastic fibroma, ameloblastic fibrosarcoma, maxillectomy