We present a case of a 20-year-old woman presenting initially with an asymptomatic palatal swelling. Radiographic examination showed a cyst at the right maxilla with bucco-lingual expansion and perforation of palatal bone. Incisional biopsy was carried out via a buccal approach and the result revealed a benign odontogenic cyst, in keeping with radicular cyst. The patient was then scheduled for cyst enucleation. During the procedure, it was found that the palatal lesion was unrelated to the maxillary cyst. Incisional biopsy of the palatal mass was carried out and revealed a low-grade mucoepidermoid carcinoma. The patient then had a partial maxillectomy with fibula flap reconstruction. There was no recurrence at postoperative 1 year follow-up and she was rehabilitated with dental implants.
In the recent World Health Organization classification of odontogenic tumours, desmoplastic ameloblastoma has been characterized as a variant of ameloblastoma, with specific clinical, radiographical, and histological features. Till date, 145 cases have been reported in Japanese, Chinese, Malaysian, Western, and African populations, with very few cases described in Indians. Here, we report five cases in the Indian population. The male to female ratio was 3:2. The mean age at diagnosis was 33.2 years. Four of the tumours were located in the maxilla, in the anterior premolar region. The lone mandibular tumour was located anteriorly, crossing the midline. Three of the tumours had a mixed radiologic appearance with poorly defined borders. Unilocular change was seen in one of them. Two tumours presented as unilocular radiolucencies with specks of radiopacities and well-circumscribed borders. Histologically, irregular odontogenic islands, with a stretched-out 'kite-tail' appearance, were seen in a dense desmoplastic stroma. The peripheral layer of the epithelial islands was made up of flattened cells and the inner core was made up of spindle-shaped and, in some instances, squamous-shaped cells. In two cases, odontogenic epithelium in the form of follicles, typical of solid/multicystic ameloblastoma, was seen and these were typed as 'hybrid' variants. All the cases were treated by resection.