Patients with orbital aspergillosis commonly present with unilateral proptosis and associated sinus disease. However, neither of these manifestations was observed in a 62-year-old woman who had an orbital apex syndrome with visual loss, complete ophthalmoplegia, and corneal hypoesthesia. Pathologic examination of a specimen from a granulomalike mass removed at left frontotemporal craniotomy showed branching hyphae with the characteristic appearance of Aspergillus.