Half A Purple Painful Tongue

Abstract

To the Editor: A 73-year-old man abruptly developed left tongue and left facial pain, peaking in intensity 2 hours after onset. After 24 hours of sustained discomfort, the gentleman presented to the emergency room. He had difficulty describing the pain but characterized it as “hard.” It was most severe on the left side of his tongue. A less severe pain was also present in his left cheek, radiating to his ear. He stated that within a few hours of onset of the pain, the left side of his tongue had turned a purplish-black color, “It looked like someone drew a line with a pen right down the middle.” The discoloration had resolved about 6 hours before his presentation, but family members confirmed his observations. He denied any recent trauma, facial numbness, swelling, or weakness. He had sustained a left inferior cerebellar stroke 10 years earlier, with complete resolution of symptoms. He had a patent foramen ovale with right-to-left flow with cough or Valsalva and hypertension, for which he was on an outpatient regimen of a calcium channel blocker and angiotensin-converting enzyme inhibitor (his only medicines). Initial examination showed a blood pressure of 240/130 in both arms, a hyperemic tongue on the left side with flattening of the papilla and two shallow excoriations, tenderness to left tongue palpation but no left facial or scalp tenderness and no palpable temporal arteries, normal upstroke and no bruits of the carotid arteries, and normal neurologic examination, including no papilledema or retinal hemorrhages. The remainder of the physical examination was unremarkable. He had an elevated creatine kinase of 550 IU/L, with a normal myocardial band fraction. All other values were normal, including sedimentation rate. Our primary differential diagnosis included vascular occlusion; arteritis; neurologic pain, including varicella zoster and tic douloureux; and a focal anatomic abnormality, such as parotitis or salivary duct stone. Carotid duplex ultrasound studies were performed because of the patient's history of cerebrovascular disease. The ultrasound revealed complete occlusion of the left external carotid artery (Figure 1), with minimal plaque involvement elsewhere, right or left side. We attributed this man's tongue pain and discoloration to acute occlusion of the left external carotid artery. The tongue is a highly vascular muscle and receives its blood supply from bilateral lingual arteries (the second branching of the external carotid arteries). It is unclear why this man had tongue ischemia, given the bilateral vascular supply to the tongue. In fact, bilateral ligation of the external carotid arteries is a surgical treatment of severe epistaxis1 and is usually well tolerated, although cases of claudication on mastication after this procedure have been reported.2 A review of the literature detailed two similar cases of acute unilateral facial and tongue pain;3,4 evidence of external carotid artery occlusion was diagnosed with angiography in both cases. It should be noted that tongue necrosis has been reported in patients with temporal arteritis.5 This man's pain resolved within 72 hours of presentation, and he sustained no motor deficits and no tissue necrosis. He was discharged on aspirin (given his past history of cerebrovascular disease) and a revised antihypertensive regimen. In the future, our differential diagnoses for unilateral facial or tongue pain will include occlusion of the external carotid artery. Power flow Doppler ultrasound reveals no flow through the external carotid artery .