We report what is, to our best knowledge, the first case of chronic recurrent multifocal osteomyelitis (CRMO) in which the frontal and sphenoid bones were involved. Characterized by a prolonged and fluctuating course of osteomyelitis at different sites, CRMO is self-limited, although sequelae can occur. The diagnosis is one of exclusion. It is important to publish cases like this, because the recognition of CRMO can prevent aggressive surgical and medical treatment.