In an immunocompromised state, it is very rare for an indolent fungal ball to transform into an invasive fungal rhinosinusitis. However, it is rarely reported that sphenoid fungal ball invades the infratemporal fossa and trigeminal nerve. A 47-year-old female patient undergoing chemotherapy for malignant thymoma with pleural dissemination presents with right facial hypoesthesia. Magnetic resonance image and computed tomography showed bony defects in the sphenoid lateral recess. These lesions extended from the right sphenoid sinus to the infratemporal fossa and mandibular nerve. Endoscopic debridement was performed, and fungal ball was diagnosed in sphenoid lateral recess and infratemporal fossa. Tissue invasion of Aspergillus species was confirmed in pterygoid body and muscles. Systemic antifungal treatment with voriconazole was used, and the disease has been stable without improvement of mandibular nerve palsy. Physicians must know the possible pathophysiology of invasive fungal sinusitis from sphenoid lateral recess to infratemporal fossa and mandibular nerve, which can be treated by endoscopic debridement with minimal morbidity.
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