Primary intraosseous carcinoma (PIOC) is a rare malignant odontogenic epithelial neoplasm arising within the jawbone, without connection to the oral mucosa or evidence of pre-existing malignancy. As PIOC often develops from odontogenic cysts, its clinical and radiographic features may closely mimic those of benign cystic lesions, making pre-operative diagnosis difficult and leading to delayed or inadequate treatment. This case report highlights the importance of careful assessment of subtle clinical, radiographic or intra-operative abnormalities, as well as the necessity of routine histopathologic examination.