Osteoblastoma is a benign neoplasm and normally it is found in the spine, ribs, pelvis and in the long bones. Osteoblastoma comprises only about 1% of all primary bone tumors. The vertebral column and extremity long bones are the sites that are most frequently involved when osteoblastoma occurs. It is rare to appear in the craniofacial area. In this report, we describe the presentation, diagnosis, and treatment of a 17-year-old girl with maxillary osteoblastoma. We performed the en bloc resection and reconstructed with artificial bone. Eighteen months after initial operation, the patient has remained free of recurrence with no facial asymmetry.