A 65-year-old male patient, drinker, ex-smoker, was referred to a stomatology clinic for evaluation of a lesion located in the upper alveolar ridge that had appeared 15 days previously. Intraoral examination revealed a reddish sessile nodule with yellowish-white spots. A swelling of hardened consistency in the hard palate was also observed. Radiographic examination and computed tomography showed an expansive bone lesion in the right maxilla and a disruption in the cortical lower margin, connecting the bone lesion to oral mucosal nodule. A biopsy of the nodular lesion was performed and histopathologic examination indicated squamous cell carcinoma (SCC). The patient underwent partial maxillectomy and surgical specimen showed cystic epithelium with malignant transformation, numerous keratin pearl formation, and atypical mitosis. Final diagnosis was a primary intraosseous SCC arising in a residual odontogenic cyst. After 2-year follow-up, the patient has been in good general health without signs of recurrence.
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