Rhino-orbital-cerebral mucormycosis (ROCM) infections are rare and usually occur in patients with diabetes, malignancy, or organ transplantation. The most common presenting symptoms include facial pain and swelling, fever, and rhinorrhea. Mortality rates reach nearly 50%. Those with previous organ transplants typically present with sinus symptoms a few weeks to months after transplantation. A 75-year-old man presented with headache. His history was significant for bilateral lung transplantation in 2017. On his fourth presentation, he was admitted to the hospital for work up. Imaging showed small fluid levels within the right maxillary and sphenoid sinuses. Infectious work-up revealed no meningitis. On day three, the patient complained of diplopia on the right. His examination was significant for right lateral rectus palsy. Repeat imaging was performed and showed increasing fluid levels of the right maxillary and sphenoid sinuses, and the otolaryngology consultation service was consulted. Nasal endoscopy was significant for pink, vascularised mucosa with no obvious regions of pallor or necrosis. Endoscopic sinus surgery was performed. There were no findings suggestive of fungus but purulence of the right sphenoid grew Rhizopus. The patient’s nerve palsy did not resolve and progressed to the contralateral orbit. Repeat nasal endoscopy continued to show healthy mucosa while MRI showed enhancement worrisome for meningitis. To our knowledge, this is the only reported case of fungal sphenoid sinusitis resulting in meningitis and death six years after transplant. Current literature documents fungal infection in post-transplant patients up to four years after surgery, with most occurring within the first year. Suspicion for fungal infection should remain high in this patient population. Keywords: mucormycosis, lung transplant, sinusitis, lateral rectus palsy
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