Abstract
A 47-year-old man was referred to treatment of a lesion located on the left maxilla with 5 months of evolution. Intraoral examination revealed a perforation in the vestibular cortical bone. The computerized tomography (CT) showed an extensive, well-delineated, radiolucent lesion, extending from the alveolar ridge up to the nasal cavity. Microscopically, a cystic lesion consistent with odontogenic keratocyst (OKC) was observed. Decompression to posterior enucleation was performed. After 3 months, the patient returned presenting with a significant increase of the vestibular cortical perforation. A destructive lytic lesion involving the maxilla and surpassing the medium line was evident on the CT. The second incisional biopsy presented as an epithelial neoplasia composed of islands and projections toward the surface with abundant keratin deposition forming pearls and keratin plugging. Diagnosis of primary intraosseous carcinoma arising from an OKC was confirmed and the patient underwent to a maxillectomy. After 1 year of follow-up, the patient remains without recurrence.
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