Primary intraosseous carcinoma of the maxilla arising from an odontogenic keratocyst: a case report and review of the literature

Gonçalves, J.Maria.; Marola, L.Henrique.Godoi.; Modolo, F.; Vieira, D.Serafin.Couto.; de Los Santos, G.Philippi.; León, J.Esquiche.; Gondak, Rério.

General Dentistry 67(6): 26-32

2019


ISSN/ISBN: 0363-6771
PMID: 31658021
Document Number: 699363
A 47-year-old man was referred for treatment of a painful lesion of 5 months' duration located on the left side of the maxilla. A small perforation in the buccal cortex was observed during the intraoral examination. Cone beam computed tomography (CBCT) showed an extensive, well-delimited radiolucent lesion extending from the alveolar ridge to the nasal cavity. An incisional biopsy was performed, and a cystic lesion consistent with an odontogenic keratocyst (OKC) was observed microscopically. The initial treatment option was decompression to be followed by enucleation. However, 3 months after decompression of the lesion, the patient returned because there was a significant increase in the size of the perforation. A destructive lytic lesion that involved the left side of the maxilla and crossed the midline was evident in the CBCT. The examination of a second incisional biopsy specimen showed epithelial neoplasia comprising islands and projections toward the surface. There was abundant keratin deposition, resulting in the formation of pearls and plugs. A diagnosis of primary intraosseous carcinoma arising from an OKC was confirmed, and the patient underwent a maxillectomy. After 1 year of follow-up, there were no signs of recurrence.

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