To report a case of rhino-orbito-cerebral aspergillosis successfully treated with a combination of amphotericin B, posaconazole and amphotericin B irrigation.

A 59-year-old male with hypertension, diabetes mellitus and hyperlipidemia was admitted to our neurology department for left facial paresthesia, pain and consulted to ophthalmology for left eyeball pain. His visual acuity was no light perception in the left eye, and fundus examination showed papilledema and a cherry-red spot. Left exophthalmos and complete ptosis with ophthalmoplegia were also observed. Orbital computed tomography revealed left maxillary and ethmoid sinusitis, and nasal endoscopic examination revealed a black eschar adjacent to the middle turbinate. Subsequent biopsy suggested mucormycosis. The patient was immediately treated with a combination of amphotericin B and posaconazole. In addition, left endoscopic sinus surgery was performed and aspergillosis was histopathologically confirmed. The patient underwent amphotericin B irrigation for 5 days after canula insertion up to orbital apex. The patient survived for 18 months and is still alive.

A combination of amphotericin B, posaconazole and amphotericin B irrigation using the canula through the orbital apex may be helpful in treating patients with rhino-orbito-cerebral aspergillosis who refuse orbital exenteration.