Primary intraosseous carcinoma (PIOC) or primary intraosseous squamous cell carcinoma is a rare and unique tumor, which occurs within the jawbones without any connection with the oral mucosa. Etiology of PIOCs is not clear, although they are presumed to be arising from the odontogenic epithelial rests or from direct transformation of odontogenic epithelium. The present case report is of a 56-year-old male patient with the complaint of swelling in the edentulous region of 44, which on radiographic evaluation and histopathological examination is diagnosed as de novo PIOC.