Lipomas can occur anywhere in the body where fat cells are present; however, intraosseous lipomas are rare. Although solitary lesions have already been reported in the gnathic bones, to the best of our knowledge, this is the first case of bilateral intraosseous lipoma. A 62-year-old woman was referred for evaluation of a swelling on both maxillary tuberosities. The radiographic examination showed a mixed radiolucent-radiopaque image with ill-defined borders on the right side of the maxilla, and an ill-defined radiolucency on the left side. Histologically, both sides revealed numerous mature adipocytes surrounded by immature bone and dystrophic calcification. The patient remains under follow-up and free of disease for 8 months. Due to the rarity of the intraosseous lipomas in the jaws, a literature review of the published cases was performed jointly with this unique case report.