Treatment of Scedosporium apiospermum central nervous system (CNS) infection typically consists of an azole in combination with surgical debridement. This approach requires prolonged treatment and carries a high associated mortality. We present two cases of the successful treatment of S. apiospermum CNS infections with the combination of voriconazole and terbinafine. 1. Case Reports 1.1. Case？？1 An 81-year-old Hispanic woman with a past medical history significant for hypertension, diabetes mellitus, hypothyroidism, and chronic kidney disease presented with symptoms of sinusitis, including facial pain, nasal congestion, and rhinorrhea. The sinusitis was later complicated by the development of left orbital cellulitis. The patient underwent a functional endoscopic sinus surgery (FESS) without evidence of gross infection and was treated with oral levofloxacin. Three weeks later, her orbital cellulitis resolved; however, she developed nausea, vomiting, headaches, left vision loss, diplopia, and local pain. Her eye exam was significant for intact extraocular movements, no nystagmus, and a positive afferent pupillary defect. No periorbital edema or erythema was observed. Visual acuity for her right eye was 20/60 and for her left eye was 20/100. Her basic laboratory results were normal. She was diagnosed with a left orbital complex syndrome with optic neuropathy and underwent another FESS with sinusotomy and debridement. The patient was briefly placed on intravenous (IV) steroids, and she was discharged home on an oral prednisone taper and levofloxacin. However, the patient was readmitted 10 days later with worsening vision loss on the left; she was then only able to perceive light. The intraoperative cultures from her last FESS grew a moderate amount of mold, which was subsequently identified as Scedosporium apiospermum based on its characteristic macroscopic morphology and conidiation. Susceptibility testing of the isolate revealed a voriconazole MIC of 0.5？ug/mL. An orbital MRI revealed mildly elevated signal within the intraorbital segment of the left optic nerve, mild inflammatory changes surrounding the optic nerve sheath complex and the orbital apex, and very mild asymmetric prominence of the left extraocular muscles concerning for myositis (Figure 1). The patient was placed on IV voriconazole at 6？mg/kg every 12 hours for a presumed fungal sinusitis, myositis, and possibly sinus fungal osteomyelitis. After approximately 30 days of IV voriconazole therapy, the patient was transitioned to oral voriconazole and had obtained a voriconazole trough level of
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