Primary intraosseous hemangiomas of the maxillofacial region are rare lesions that comprise less than 1% of all osseous tumors. A review of the literature on primary intraosseous hemangiomas of the facial bones revealed a limited number of publications, much of which was largely limited to case reports. This case report summarizes the workup and surgical treatment of a 37-year-old female with a primary intraosseous hemangiomas of the left maxillary bone. The image, histology, treatment, and literature are reviewed.