Osteoid osteoma is an osteoblastic benign bone tumor more frequent in long bones of young male patients. It is the third most commonly diagnosed benign bone tumor and has distinctive symptomatology, nocturnal pain that relieves with nonsteroidal anti-inflammatory drugs. Nowadays, total resection is the preferred management. In the present paper, an unusual variant in the maxilla of a female elderly patient without previous symptomatology is exposed; it was surgically removed without signs of reappearance in 12 months of follow-up. Although it is not a common site of appearance, the manifestation of this tumor in the skull bones seems to be associated with a different pattern in contrast to the skeletal type as can be seen in the present case and others previously reported. The authors consider due to the clinical similarity between this and other tumors that it should be taken into consideration for future diagnosis dilemma.