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The locked-in syndrome is typically caused by basilar artery thrombosis, resulting in ventral pontine infarction. Infectious basilar arteritis is a rare alternative etiology.We present a 24-year-old female with a prodrome of facial pain without fever or meningismus, who developed a locked-in syndrome due to an invasive fungal infection after bone marrow transplantation. The clinical course and neuroimaging demonstrating sinusitis with adjacent pontine infarction but without basilar artery thrombosis are presented. The infectious differential diagnosis and management of invasive fungal infection are discussed.Facial pain without fever or meningismus may be an early symptom of invasive fungal rhinosinusitis, which may involve small basilar pontine perforating arteries, leading to a locked-in syndrome in immunosuppressed patients. Early recognition and treatment may prevent life-threatening neurologic complications.