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Invasive aspergillosis is a rare disease and is often misdiagnosed. The clinical course is quite aggressive and it is a potentially fatal disease. We report a case of invasive aspergillosis involving the dura mater and optic nerves which was successfully treated with voriconazole, even though the patient had residual monocular blindness. An 86-year-old Japanese man complained of developing loss of vision in his left eye while taking oral fluconazole prescribed by an otolaryngologist for mycosis of the left maxillary sinus. He was referred to our hospital. At the first visit, he already had no light perception in the left eye, with decreased ocular motility in all directions and orbital apex syndrome. His corrected distance visual acuity (CDVA) in the right eye was 20/25 with enlargement of Mariotte's blind spot. Magnetic resonance imaging revealed inflammation around both optic nerves that also involved the dura mater. His antifungal therapy was changed to intravenous voriconazole. Although his right CDVA temporarily declined to 20/50, it improved to 20/16 by 10 months after the initiation of treatment. Maxillary sinus biopsy detected Aspergillus. Invasive aspergillosis progresses rapidly and aggressively. The present case highlights the importance of early diagnosis and selection of an appropriate antifungal agent. [ABSTRACT FROM AUTHOR]