Malignant tumors arising from dentigerous cysts are classified as primary squamous cell carcinoma derived from an odontogenic cyst or as odontogenic carcinoma according to the 2005 WHO classification and are extremely rare. We report a malignant tumor arising from a dentigerous cyst in the right maxillary anterior teeth , together with a literature review. The patient was a 75-year-old man who visited a hospital with complaining of discomfort in the lingual part of the right maxillary anterior teeth. On panoramic radiography and plain computed tomography (CT), dentigerous cyst, keratocystic odontogenic tumor or ameloblastoma was suspected. The extirpated material was histopathologically diagnosed as an odontogenic carcinoma (in situ) arising from the dentigerous cyst. Postoperative ultrasonography (US) and contrast enhanced CT revealed no metastasis to the cervical lymph nodes. The patient is currently being followed up without resection or anticancer drug administration. Neither local recurrence nor metastases were observed 18 month after surgery.
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