View metadata, citation and similar papers at core.ac.uk brought to you by CORE provided by Elsevier - Publisher Connector CASE REPORTS From the Midwestern Vascular Surgical Society Tongue necrosis as an unusual presentation of carotid artery stenosis Paul M. Bjordahl, MD, and Alex D. Ammar, MD, Wichita, Kan A 57-year-old man with premature coronary artery disease presented to the emergency department with left facial pain, numbness, and tongue swelling. The patient was found to have significant tongue necrosis, and subsequent arteriography demonstrated carotid bifurcation stenosis with embolization to the left lingual artery. The patient was successfully treated with debridement of his tongue and left carotid endarterectomy. (J Vasc Surg 2011;54:837-9.) The principal vascular supply to the tongue is through examination, the patient was found to have a pale, ischemic appear- the lingual artery via the external carotid artery. The tongue ing area (2 cm ϫ 3 cm) of the left side of his tongue with evidence is thought to have limited collateral blood supply, largely of mucosal loss. He had no neurological deficits. due to the fibrous septum in its center. The ascending Computed tomographic scan of the head and neck with pharyngeal, facial, and maxillary arteries do not collateralize intravenous contrast identified significant carotid artery stenosis. the tongue. Contralateral flow via a submucosal plexus Subsequent carotid duplex scan revealed 70% stenosis of the left provides collateral supply with some addition from the internal carotid artery and severe stenosis of bilateral external ipsilateral internal carotid artery. Lingual infarction is a rare carotid arteries. The patient underwent bedside debridement of event, and has been reported less than 50 times in the the insensate portion of the necrotic area of the front of his tongue literature. Almost all cases have been attributed to cranial by an oral surgeon. The patient had full thickness necrosis of his arteritis, often with the addition of ergot alkaloids for tongue. Because of his unusual presentation, history of coronary migraine treatment.1 A more unusual cause, with four artery disease, and suspected atypical angina pectoris by the cardi- reported cases, is occlusive atherosclerotic disease of the ologist, arteriography was performed for cardiac assessment and external carotid artery.2-5 Herein, we report the case of a further workup of his carotid occlusive disease. It demonstrated previously asymptomatic male who presented with tongue 80% stenosis of the vein graft to the left anterior descending necrosis due to lingual artery embolic occlusion distal to coronary artery, which was successfully stented. In addition, arte- severe external carotid stenosis. riography confirmed Ͼ90% stenosis of both external carotid arter- ies with embolization to the left lingual artery (Figs 1 and 2). The CASE REPORT patient subsequently underwent left carotid endarterectomy. A 57-year-old man with hypertension, premature coronary The patient had an uneventful postoperative course with artery disease, and prior coronary artery bypass presented to the preservation of his lingual motor function and return of normal emergency department complaining of facial pain and numbness in appearing tongue mucosa. Recovery was lasting with over 2 years his tongue with the sensation that it had been bitten. The pain of follow-up. radiated into his left anterior neck. The patient denied history of amaurosis fugax, hemiparesis, aphasia, or prior facial pain. There DISCUSSION was no history of prior neck surgery or irradiation. On physical Tongue necrosis can be a rare presentation of carotid atherosclerotic disease. To our knowledge, this is the fifth From the Department of Surgery, University of Kansas School of Medicine- reported case of facial pain and tongue necrosis due to carotid Wichita. Competition of interest: none. occlusive disease, and the first to our knowledge, without Presented at the Thirty-fourth Annual Meeting of the Midwestern Vascular evidence of prior transient ischemic attack. Surgical Society, Midwestern Vascular 2010, Indianapolis, Ind, Septem- Because of its much higher frequency, vasculitis must ber 9-11, 2010. be considered as a differential diagnosis in any patient with Reprint requests: Alex D. Ammar, MD, Department of Surgery, Room 1 3082, University of Kansas School of Medicine-Wichita, 929 N. St. tongue ischemia and facial pain. An erythrocyte sedimen- Francis St., Wichita, KS 67214 (e-mail: [email protected]). tation rate would be helpful if vasculitis is suspected clini- The editors and reviewers of this article have no relevant financial relationships cally. Ischemia secondary to a vascular process will almost to disclose per the JVS policy that requires reviewers to decline review of any always be unilateral and confined to the tip or front half of manuscript for which they may have a competition of interest. the tongue. Other causes of tongue ischemia include hy- 0741-5214/$36.00 Copyright © 2011 by the Society for Vascular Surgery. perviscosity syndrome, radiation effects, carotid dissection, doi:10.1016/j.jvs.2011.01.057 and disseminated intravascular coagulation.6-9 837 JOURNAL OF VASCULAR SURGERY 838 Bjordahl and Ammar September 2011 Fig 1. Left carotid arteriogram demonstrating approximately 90% stenosis and in situ thrombus or embolus of the lingual artery with distal occlusion. The thin arrows outline lingual artery thrombus or embolus. The thicker arrow shows abrupt occlusion of the lingual artery presumably due to embolization. Fig 2. Early phase arteriogram demonstrating severe stenosis at the origin of the right external carotid artery. JOURNAL OF VASCULAR SURGERY Volume 54, Number 3 Bjordahl and Ammar 839 Although symptoms such as jaw pain with chewing may 2. Reed C, Inlis MJ. Acute massive gangrene of the tongue. Br Med J become manifest with ligation of one or both external 1965;2:575-6. carotid arteries, circulation to the face is well preserved 3. Bergan JJ. Ischemic necrosis of the tongue; a case report. Q Bull Northwest Univ Med Sch 1959;33:38-40. because of abundant collateral flow. In our case, however, 4. Amen F, Amen AA. Tongue ischaemia in a patient with external carotid severe stenosis in the contralateral external carotid artery artery stenosis. J Laryngol Otol 2002;116:1053-4. and embolization to the ipsilateral lingual artery abruptly 5. Orita Y, Ogawara T, Yorizane S, Nannba Y, Akagi H, Nishizaki K. compromised the limited collateral flow to the tongue, Necrosis of the tongue after transient ischemic attack. Oral Surg Oral resulting in tissue necrosis. Treatment of this condition Med Oral Pathol Oral Radiol Endod 2000;89:316-8. 6. Kamatani T, Yamashita K, Okabayashi T, Maeda H, Toi M, Yamamoto acutely may include antiplatelet therapy. Definitive treat- T. Bilateral ischemic necrosis of the tongue due to disseminated intravas- ment is endarterectomy. Debridement may be necessary for cular coagulation. Int J Oral Maxillofac Surg 2008;37:777-9. local tongue necrosis. 7. Zelch MG, Geisinger MA, Risius B. Tongue necrosis after intraarterial Carotid artery occlusive disease, particularly in the ab- vasopressin therapy. AJR 1985;144:1283-4. sence of transient ischemic attack, is a rare cause of tongue 8. Pfeiffer J, Ridder GJ. [Spontaneous tongue necrosis consecutive to necrosis. 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