Odontogenic Carcinoma with Dentinoid of the Left Maxilla

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Abstract Introduction/Objective Odontogenic carcinoma with dentinoid is a very rare dentinoid producing odontogenic tumor with 10 cases reported in literature, and it is not currently defined by the World Health Organization. It has been described to have a locally aggressive and non-metastatic course with high level of recurrence after excision. Methods/Case Report We report a case of odontogenic carcinoma with dentinoid located in the left maxilla of an otherwise healthy, 22-year-old male. The patient initially endorsed left upper molar tooth pain for 2 months that led him to receive medical attention. Initial examination revealed a 3 x 2 cm firm, non-mobile and non-pulsatile lesion on buccal and lingual surface of the left maxilla with imaging revealing a 2.9 x 2.7 x 2.7 cm mixed radiolucent/radiopaque lesion in the posterior left maxilla with expansion on the buccal and palatal surfaces. Incisional biopsy of the left maxilla (soft tissue) was initially diagnosed as suspicious for osteosarcoma, which was later changed to odontogenic carcinoma with dentinoid. One year later, the mass had increased to 4.5 x 3 x 4 cm in size. Left hemi maxillectomy was performed, and the specimen was diagnosed with odontogenic carcinoma with dentinoid. Previously described histological features of clear cell change, low grade appearance, focal calcification, and dentinoid formation are present in our case. Our case is the first to describe glandular formation of the tumor cells. Results (if a Case Study enter NA) NA Conclusion The histopathological changes of our case reflect previously reported microscopic descriptions for this exceedingly rare tumor that is unclassified by the World Health Organization. It has been reported to occur in both male and female patients without predilection and a wide age range from 14-61 years. To our knowledge, this is the 11th reported case, and the 5th case described in the maxilla. Further studies, including NGS sequencing to determine genetic alterations, will be performed on our case to further elucidate pathophysiological mechanisms. Long term follow up with our patient will be attempted to determine tumor behavior.