To report an immunocompetent patient with more than 1-year survival after treatment with voriconazole, despite invasive paranasal sinus aspergillosis involving the orbital apex.

A 74-year-old woman with only preexisting hypertension visited our clinic complaining of pain and immovable left eye that occurred approximately 4 days prior. Computed tomography (CT) and magnetic resonance imaging (MRI) showed a sphenoid sinusitis with suspicious lesion involving the orbital apex of the left eye. A biopsy by functional endoscopic sphenoid surgery (FESS) was performed through the sphenoid sinus, which enabled us to diagnose aspergillosis. The patient had esotropia, ophthalmoplegia, positive RAPD, and ptosis in the left eye. On follow-up, a new MRI showed acute to subacute stage infarction at the left occipital lobe. After the patient was treated with intravenous voriconazole, ptosis, and ophthalmoplegia improved. At the 13-month follow-up, she was alive with no disease recurrence.

Invasive aspergillosis of orbit and cerebrum in healthy patients is a rare clinical entity. In case of central nervous system involvement, the survival outcome is poor with high mortality; however, good results can be obtained by treatment with voriconazole.