Intraosseous venous malformations are rare benign lesions that account for approximately 0.5% to 1% of all osseous tumors. Involvement of the facial skeleton is rather uncommon, with the mandible and maxilla most frequently involved. In the current study, we report a case of an intraosseous venous malformation in a 52-year-old male patient with a history of mandibular keratocystic odontogenic tumor. The patient presented with an asymptomatic bony lesion in the mandible. Treatment involved surgical excision. Histopathologic examination of the excised specimen revealed an intraosseous venous malformation. Twelve months postoperatively, there was no evidence of recurrence.