Primary intraosseous carcinoma, NOS (PIOC) is a rare central jaw carcinoma, not classified as any other type of carcinoma, with a suggested odontogenic origin. Here, we present 2 additional cases of PIOC. A 39-year-old man presented with an irregular asymptomatic swelling in the anterior upper vestibule and palate and palpable fixed lymph nodes. Periapical radiography analysis revealed an ill-defined unilocular radiolucency in the periradicular region of endodontically treated maxillary incisors with irregular root resorption. The second case involved a 35-year-old man presenting with an asymptomatic, unilocular, ill-defined radiolucency superimposed on the mandibular molars roots, with loss of lamina dura. Microscopically, both the cases showed small islands of neoplastic epithelial proliferation with cellular atypia and keratinization, confirming the final diagnosis of PIOC. Patients were referred to an oncology service. Clinicians should consider PIOC when evaluating an ill-defined radiolucency in tooth-bearing areas of gnathic bones of young adults.