A 61-year-old woman presented with painful ophthalmoplegia, Tolosa-Hunt syndrome. The patient had undergone a placement of dental implant 5 months before the presentation and had a local maxillary sinusitis 1 month later. She had not been aware of any preceding head trauma or infection. On examination, the patient showed serious right oculomotor nerve paresis and retro-orbital pain. Blood examination showed normal findings. Magnetic resonance imaging identified abnormal structure in the right cavernous sinus with flow void signals. Angiography revealed a carotid cavernous sinus fistula fed by the intracavernous branches of the internal carotid artery on both sides, right internal maxillary and middle meningeal arteries, and left ascending pharyngeal artery. The patient underwent coil embolization via both external carotid arteries. We assumed that local maxillary sinusitis caused by dental implant might spread hematogenously into the sphenoid and cavernous sinuses and formed a carotid cavernous sinus fistula, which presented with Tolosa-Hunt syndrome. Implant-associated infection has to be managed promptly with adequate manner before it spreads.
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