A 62-year-old woman who was prescribed prednisolone and cyclosporine for uveitis developed right blepharoptosis, diplopia and hypesthesia of superficial sensation of the trigeminal nerve V1 domain. Preserved right visual acuity led to a diagnosis of superior orbital fissure syndrome. We could not find any abnormalities on computed tomography except for right maxillary sinusitis associated with calcification. No infectious agents were detected in the blood or cerebrospinal fluid. We concluded that maxillary aspergillus, which was proven histopathologically after surgery, caused superior orbital fissure syndrome through the ophthalmic veins. Surgical excision of the lesion and voriconazole administration resulted in complete remission. To the best of our knowledge, there have been no other reports of superior orbital fissure syndrome caused by maxillary aspergillus sinusitis. The calcified sinusitis suggested fungal infections, which allowed us to avoid using corticosteroids.