Myelolipoma is a benign tumor consists of mature adipocyte and hematopoietic elements. Mostly, this tumor locates in adrenal gland and it also can be found in extra-adrenal area. However, intraosseous myelolipoma is extremely rare. The etiology of this tumor is unclear but some hypothesis proposed that altered mesenchymal stem cell functioning and hormonal events act together in the pathogenesis of myelolipoma. With radiographic view, myelolipoma shows similar characteristics of other fat-containing tumors. The histopathologic examination is necessary for the definite diagnosis. This case report includes a patient with intraosseous myelolipoma of maxilla and mandible and literature review about the clinical, histopathologic, and radiologic features of myelolipoma.