Abstract
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.
1
Introduction
Carotid cavernous sinus fistulas (CCFs) are abnormal arteriovenous communications formed in the cavernous sinus and roughly classified into direct and indirect types. Most direct CCFs are formed acutely after serious head traumas tearing the wall of the intracavernous carotid artery and need emergent interventions. Indirect types are gradually formed from nontraumatic origins and cause more subtle signs because of its less shunt flow. In general, indirect CCFs are only treated if symptoms are intractable or intolerable or vision is threatened . Most CCFs are treated by endovascular procedures via transarterial route, transvenous route, or their combinations, with variable embolic materials . Tolosa-Hunt syndrome has clinical symptoms represented by painful ophthalmoplegia caused by pathologies in the cavernous sinus and orbital apex. Magnetic resonance imaging is highly sensitive to detect the causative pathologies for Tolosa-Hunt syndrome . Although dental implants have been popular as daily practices, postprocedural gingivitis and maxillary sinusitis are common complications . In rare instances, paranasal sinusitis has been reported to progress into cavernous sinus thrombosis or caused orbital apex syndrome and Tolosa-Hunt syndrome . In a patient, Tolosa-Hunt syndrome was highly speculated to progress into a CCF . Here, we present a peculiar case of Tolosa-Hunt syndrome probably caused by a CCF formed in the setting of implant-associated chronic infection.
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Case report
A 61-year-old woman presented with painful ophthalmoplegia and was referred to our department. The patient had undergone a placement of dental implant in the posterior maxilla on the right 5 months before the presentation. She had a local maxillary sinusitis 1 month later and was treated by antibiotic agent administration. Her medical history was unremarkable for diabetes mellitus or allergy, and she had not been aware of any preceding head trauma or infection. On examination, the patient was afebrile and well oriented while presented with orbital swelling. The pupil was dilated on the right with sluggish light reflex. The ocular position was abductive on the right with serious restrictions on upward, downward, and innerward gazes, with retro-orbital. Conjunctival chemosis was not noted. Neuroophthalmological examination did not identify visual impairment or increased intraocular pressure. Bruit was not audible. The patient was not aware of the pain in the site of implant or maxilla. Blood examination showed normal findings. Computed tomographic scans revealed intact structure of the maxillary, sphenoid, and ethmoidal sinuses ( Fig. 1 ). Magnetic resonance imaging identified an abnormal structure in the right cavernous sinus with flow void signals ( Fig. 2 ). Catheter angiography revealed a CCF fed by the bilateral meningohypophyseal trunks of the intracavernous internal carotid artery, peripheral branches of the right internal maxillary, anterior and petrosal branches of the right middle meningeal artery, and pharyngeal branches of the left ascending pharyngeal artery ( Fig. 3 ). The venous drainage routes were totally antegrade through to the pterygoid and basilar plexuses via ipsilateral or contralateral cavernous sinuses. Reflux to the cerebral cortical and ophthalmic veins was not found ( Fig. 4 ). The patient underwent coil embolization via the right internal maxillary and middle meningeal arteries and left ascending pharyngeal artery that resulted in a remarkable flow reduction of the CCF. Postoperatively, the patient showed a moderate resolution of the orbital pain, whereas little improvement was noted in the oculomotor nerve paresis.
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