Paranasal sinus osteomas are benign osteogenic lesions, often diagnosed incidentally on computed tomography (CT). Their etiology is unknown, and they most commonly involve the frontal and ethmoid sinus. We present an exceedingly rare localization of osteoma within the cavity of a concha bullosa of the middle turbinate in a 38-year-old woman with frontal headaches and right-sided facial pain. The patient was treated by partial turbinectomy with tumor excision and preservation of the lateral middle turbinate mucosa. The patient was free of pain following surgery. We report the case and imaging and review the pertinent literature. To our knowledge, this is the first case of massive involvement of the cavity of a pneumatized middle turbinate by an osteoma. Solitary osteoma within the concha bullosa cavity should be included in the differential diagnosis of rhinogenic headaches as well as in the differential diagnosis of extraordinary pathologic entities within the pneumatized middle turbinate.