Psammomatoid juvenile ossifying fibroma (PJOF) is a benign fibro-osseous lesion that mainly affects the paranasal sinuses and periorbital bones. It may cause significant esthetic and functional impairment. Herein, we describe the diagnosis and surgical approach of an extensive PJOF arising in the frontal sinus of a young male. After complete lesion removal and histopathological confirmation, the bone defect was repaired with a customized polymethylmethacrylate implant. PJOF may present aggressive clinical behavior. The excision of extensive PJOF in the orbitofrontal area can result in significant esthetic defects. Polymethacrylate implants restore functionally and esthetically the involved area.