Primary Intraosseous Odontogenic Carcinoma Arising from a Dentigerous Cyst

Abstract

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.

Primary intraosseous carcinoma arising in dentigerous cyst: Case report

“…Dentigerous cysts occur within the age range of 20–30 years and squamous cell carcinoma arising in dentigerous cysts are often diagnosed in age range of 50–60 years with a mean age of 57 years [ 3 ]. Therefore, we can suggest that PIOC have a relatively slow mode of development, as is observed in the case discussed here.…”

“…PIOC arising in dentigerous cyst is extremely rare. After cross-checking the last 3 reviews on the subject, only 35 cases have been reported from 1958 to 2015 [ [2] , [3] , [4] ]. In addition two other cases were published in 2017 and 2020 in English literature [ 5 , 6 ].…”

“…PIOC arising in an odontogenic cyst is a rare observation with an incident rate of 0,3 to 2% [ 3 ]. Only a hundred cases have been published [ 8 ].…”

“…The mandible is more frequently affected (86.7%) [ 4 ] with a predilection for the lower 3rd molar [ 3 ] as occurred in this case. Clinically, PIOC arising in the epithelium of a dentigerous cyst are mostly painless.…”

“…The extent of the carcinoma should always determine the therapeutic approach. When carcinoma is in situ, further surgery may be postponed albeit with regular clinical and radiological follow-ups [ 3 , 8 ]. When carcinoma is infiltrating, a surgical revision is indicated.…”