Fibrous dysplasia (FD) commonly affects the craniofacial structures including the orbit. Rarely, growth involving the orbital apex can cause intra-orbital or intracanalicular optic nerve compression leading to permanent vision loss. Any sign of optic nerve compression is an indication for surgical resection. We report a case of compressive optic neuropathy secondary to craniofacial FD that was treated with combined endoscopic and trans-orbital resection as well as orbital wall and facial reconstruction, a novel technique for optimal management. Surgical challenges including avoiding thermal and mechanical injury to the optic nerve, reduced access to the posterior medial orbital wall, and meticulous reconstruction of the medial and inferior wall were overcome by the combined approach. A successful functional and aesthetic outcome was achieved post-operatively with evidence of visual improvement.