Thirteen Synchronous Multifocal Calcifying Epithelial Odontogenic Tumors (CEOT): Case Report and Review of the Literature

Background: Calcifying epithelial odontogenic tumor (CEOT, Pindborg tumor) is a rare, benign, locally aggressive neoplasm of the jaws that accounts for approximately 1% of all odontogenic tumors. It was first defined by Pindborg in 1955 and has been reported approximately 350 times in the literature; 7 reported multiple (up to 4) synchronous lesions. Materials and Methods: We report an individual with the largest number of CEOTs reported to date and provide a literature review of multifocal CEOT cases. Results: A 30-year-old male presented to the Oral and Maxillofacial Surgery Department of the West Virginia University School of Dentistry (WVU SoD) to extract multiple impacted teeth previous to construction of a complete denture. A pantograph showed 15 impacted teeth, almost all associated with well-demarcated cyst-like radiolucencies, some with small, ill-defined radiopaque flecks. Microscopically, the lesions showed sheets and strands of polygonal epithelial cells with eosinophilic cytoplasm. Spread throughout the epithelium and connective tissue were small, spherical, amorphous, pale purplish calcifications. Each lesion was similar and consistent with a diagnosis of CEOT. Conclusion: We report a patient with 13 independent CEOTs scattered throughout all quadrants. This case represents the largest number of Pindborg tumors or any other type of odontogenic tumor yet reported in a single individual. Published by Elsevier Inc. on behalf of The American Association of Oral and Maxillofacial Surgeons.