Primary intraosseous carcinoma arising from an odontogenic cyst: a case report and review of the Japanese cases.

A rare case of primary intraosseous carcinoma (PIOC) arising from an odontogenic cyst in a 58-year-old man is reported. Clinical and radiological examinations revealed an odontogenic cyst of the maxilla. Histopathologically, the lesion was composed of a cyst with a parakeratotic epithelial lining and well-differentiated squamous cell carcinoma, showing continuity between them without a connection to the oral mucosa. Twenty-eight well-documented Japanese cases of Type-1 PIOC, including the present case, were reviewed. The mean age of the 28 patients was 56.1 years, and the male to female ratio was 1.8:1.0. Compared with currently reported Japanese reviews of Type-3, foreign Type-1 and Type-3, there were no significant differences in mean patient age and sexual predominance, and no racial difference. The pathogenesis of Type-1 PIOC is also discussed.