A nineteen years old male patient showed a cystic lesion in left maxillary canine to premolar area (#23-#25). This lesion was asymptomatic, and found during his routine radiological check in local clinic. In the radiological observation the cystic lesion showed round radiolucent image containing many calcified bodies which were usually small but irregular in shape, expanding tumorously and resulted in the displacement of canine and second premolar in the absence of first premolar. The lesion was surgically enucleated, and a cystic fibrous tissue containing abnormal teeth was removed and examined pathologically. With the histological observation of tumorous odontogenic epithelium including many ghost cells, which were closely associated with abortive teeth, the lesion was finally diagnosed as CCOT associated with complex odontoma. The ghost cells of CCOT was strongly positive for β-catenin, GADD45, and LC3, and slightly positive for MMP-9, while they were rarely positive for BCL2, Wnt1, HSP-70, and p38. Therefore, it was presumed that the ghost cells of CCOT might undergo dormant cell state through altered cytodifferentiation stimulated by severe growth arrest, DNA damage signaling, and abundant autophage formation.

Calcifying cystic odontogenic tumor (CCOT), also known as calcifying odontogenic cyst or Gorin cyst is a rare developmental lesion which arises from odontogenic epithelium. It has been classified as a benign odontogenic neoplasm related to odontogenic apparatus by the World Health Organization. CCOT may be associated with other odontogenic tumors, most frequently odontoma, a finding that is a rare event in other types of odontogenic cysts or tumors. This report describes a case of hybrid odontogenic tumor composed of CCOT and ameloblastic fibroma-odontoma of the impacted right maxillary canine region that occurred in a 14-year-old female.