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Candida guilliermondii is an opportunistic pathogen that rarely causes invasive candidiasis even in immunocompromised humans. We report a case presentation of invasive C. guilliermondii rhinosinusitis causing an orbital and intracranial extension (frontal lobe abscess). An aggressive multidisciplinary team management is a key approach in invasive fungal sinusitis and avoided mortality in this case. When orbital apex syndrome secondary to sinusitis is encountered in an immunocompromised patient, the treating physician should consider fungal infection as a causative agent.