Case reports
Lingual Infarction: A Review of the Literature Enrique Ginzburg, MD*, William E. Evans, MD, Wynn Smith, MD,
Miami, Florida and Columbus, Ohio
Lingual infarction, although a very rare entity has been reported in 16 cases since 1961. The goal of this article is to alert the physician to the symptoms of tongue claudication to prevent the morbid effects of tongue infarction and blindness due to cranial arteritis. A discussion of the disease and the differential diagnosis is included. (Ann Vasc Surg 1992;6:000-000). KEY WORDS:
Lingual infarction; giant cell arteritis; tongue claudicatiom
The o c c u r r e n c e of lingual ischemia or infarction, although rare, has been seen due to trauma [1], arteritis [2-6] and carotid artery disease [7]. The importance of this finding and early diagnosis is paramount since early treatment will limit tissue loss and progression of disease.
CASE REPORT A 79-year-old white woman presented to her physician with acute right ocular blindness and a three month history of right facial and jaw pain. The patient also experienced increasing pain on swallowing and difficulty articulating words. One month prior to this admission she complained of fatigue, lethargy, and swelling of the tongue with inability to eat. She was hospitalized and during her admission was found to have a left lower lobe pneumonia. She completed medical treatment of her pneumonia and had a percutaneous endoscopic gastrostomy placed to supplement her nutrition. The patient was
From the Department of Surgery, University of Miami/ Jackson Memorial Medical Center*, Miami, Florida and the Saint Anthony Medical Center, Columbus, Ohio. Reprint requests: Enrique Ginzburg, MD, University of Miami~Jackson Memorial Medical Center, Department of Surgery (D40), 1611 N.W. 12 Avenue, Miami, Florida 33136.
seen again one month later with a foul smelling centrally infarcted segment of her tongue. On physical examination, this thin yet very alert woman, had a mobile tongue with a central area of dark necrotic tissue extending to the base of the tongue posterior to the circumvallate papillae. This can be clearly seen in Figure 1 as the dark area of the tongue posterior to health tissue bordered by sloughed mucosa. She had a right carotid bruit and only Doppler signals of her temporal arteries were evident. The patient underwent noninvasive vascular studies which showed stenosis of 20-30% of her left common carotid artery, 40-50% of the left internal carotid artery, and an open left external carotid artery. The right side studies showed 20-30% stenosis of her common carotid artery, 80-99% stenosis of her internal carotid artery, and a very stenotic external carotid artery. The patient had a negative systemic lupus antibody test and an elevated erythrocyte sedimentation rate (ESR). She underwent debridement with biopsies which came back negative for neoplasm. Arteriogram was performed which showed greater than 90% stenosis of the right internal carotid artery with an occluded right lingual artery stump (see Fig. 2). In conjunction with right lingual artery occlusion there is also left sided lingual artery occlusion as seen in Figure 3, which is an extremely important factor contributing to lingual infarction. The patient was offered a temporal artery biopsy for diagnostic purposes but refused under recommendation of her neurologist. His clinical impression was that the
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Fig. 1. Original presentation of this patient's infarcted tongue. After debridement patient was left with a sole central ischemic area of necrosis surrounded by healthy tissue.
patient had giant cell arteritis with an elevated ESR of 120. He promptly placed her on a steroid regimen. During her current hospitalization the patient underwent further debridement of the necrotic lingual tissue with marked improvement in oral intake and improved phonation. The patient will be scheduled for elective carotid surgery if no improvement results in her carotid disease after steroid protocol.
451
Fig. 3. Arteriogram of left carotid artery showing total occlusion of the lingual artery (arrow) consistent with Missen's autopsy results.
Since that first report, lingual necrosis has been commonly associated with giant cell temporal arteritis. This arteritis is a general vasculitis predominantly affecting the large and medium-sized arteries in people over 50 years of age [9]. Cranial arteritis has the common manifestations of headache and visual loss, presenting in nearly 50% of patients with polymyalgia rheumatica [I0]. Although otolaryngologic manifestations of craDISCUSSION nial arteritis are said to be infrequent, approxiThe rarity of lingual infarction is evident from a mately 25% of these patients have complaints or 25 year literature search on the subject revealing symptoms of masticatory claudication, tongue pain, fewer than 20 cases reported in the world literature, and frank lingual infarction prior to a diagnosis of the first in the United States in 1967, by Freedman, arteritis [11]. These symptoms of intermittent an oral surgeon [8], preceded by Horton in 1966 tongue claudication and paroxysmal blanching are [12]. important warnings of impending glossal infarction, just as visual scotoma are to permanent visual loss [10]. The most dreaded consequence of giant cell arteritis is visual loss which is usually irreversible. ~t One study revealed a 46% rate of total or partial blindness in 358 cases of temporal arteritis [13]. It also has other clinical manifestations such as lip and scalp necrosis, carpal tunnel syndrome, limb claudication, stroke, angina pectoris and pulmonary disease [9]. Giant cell arteritis mainly affects the superficial temporal, vertebral and ophthalmic arteries [14]. However, large arteries may be involved in temporal arteritis, and more than 50% of patients will have concomitant bruits of the carotid, subclavian, and brachial arteries, which disappear under steroid treatment [11]. Lab analysis of sedimentation rates Fig. 2. Arteriogram of carotid artery showing high rade stenosis of active arteritis and occluded stump are usually elevated with a mean of 100 ram/hr. Normochromic anemia refractory to iron therapy is rrow) of the lingual artery.
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also a common finding in these patients [11]. Midtongue carcinoma, abscess of floor of the mouth, and arterial embolization and atrial fibrillation have also been responsible for characteristic tongue infarcts. Moniliasis and Vincent's angina are also common early considerations [11]. The low incidence of tongue infarction with giant cell arteritis has been attributed to the abundant vascular supply to the tongue, consisting mainly of the lingual artery and both the ascending pharyngeal and external fascial artery [15]. However, lingual artery involvement has been suggested to be very common in giant cell arteritis. Missen demonstrated inflammatory changes, postmortem, in 9 out of I0 arteries in five cases of giant cell arteritis [16]. This was also demonstrated in our patient with bilateral occlusions of the lingual artery, as seen in Figures 2 and 3. Tongue pain can be demonstrated by requiring repetitive rapid protrusion with resulting symptoms of pain, burning, or fatigue with tongue blanching [I0]. Prompt treatment with corticosteroids in large doses followed by a lower maintenance dose is important to limit and present further vascular complications once arteritis is diagnosed.
REFERENCES 1. 2. 3. 4. 5. 6. 7. 8. 9. 10. 11. 12. 13.
ACKNOWLEDGMENT
14.
The authors wish to thank Kathy Boston, RN, Cuba Reinhardt, RN, RVT, Sherry Kistler, RN, RVT, Margaret Kuhn, RN, RVT, and Kim Oren for their assistance. Imm
15. 16.
SIEGEL RL, SPEISER AM, BIKOFSKY VM. Tongue ischemia from a soft drink can: a case report. J Am Dental Assoc 1990;121(5):607-608. ROSEMAN BB, GRANITE E. Massive tongue necrosis secondary to temporal arteritis. J Oral Maxillofacial Surg 1984 ;42(10):682-684. PEDERSON AT, JEPSEN FL. Lingual infarction in giant cell arteritis: a case report. J Laryngol Otolaryngol 1983; 97(5):479--483. DARE B, BYRNE E, ROBERTSON A. Acute lingual ischemia complicating temporal arteries, Med J Aust 1981; 1(10):534. JENKINS PR. Lingual infarction in cranial arteritis. Dental Update 1985;12(5):303-305. ARNUNG K, NIELSEN IL. Temporal arteritis and gangrene of the tongue, ACTA Med Scand 1979;206(3):239-240. GOODMAN JM, ZINK WL, COOPER DF. Hemilingual paralysis caused by spontaneous carotid artery dissection. Arch Neurol 1983;40(10):653-654. FREEDMAN GL, HOOLEY JR. Ischemic necrosis of the tongue: a case report, Oral Surg 1967;24(6):821-824. SHERARD RK, COLERDGE ST. Giant cell arteritis. J Emerg Med 1986;4(4):293-299. SOFFERMAN RA. Cranial arteritis in otolaryngology. Ann Otolaryngology 1980;89:215-219. SOFFERMAN RA. Lingual infarction in cranial aneritis. JAMA 1980;243(23):2422-2423. HORTON BT. Complications of temporal arteritis. Br Med J 1966;3:105-106. BIRKHEAD NC, WAGENER HP, WEIHMANN J. Treatment of temporal arteritis with adrenal corticosteroids. JAMA 1957;163:821-827. WILKINSON MD, RUSSEL RWR. Arteries of head and neck in GCA. Arch Neurol 1972;27:378-391. REED C, INGLIS MJ. Acute massive gangrene of the tongue. Br Med J 1965;2:575-576. MISSEN GAK. Gangrene of the tongue. Br Med J 1961 ;l: 1343-1344.
Lingual Infarction: A Review of the Literature Enrique Ginzburg, MD*, William E. Evans, MD, Wynn Smith, MD,
Miami, Florida and Columbus, Ohio
Lingual infarction, although a very rare entity has been reported in 16 cases since 1961. The goal of this article is to alert the physician to the symptoms of tongue claudication to prevent the morbid effects of tongue infarction and blindness due to cranial arteritis. A discussion of the disease and the differential diagnosis is included. (Ann Vasc Surg 1992;6:000-000). KEY WORDS:
Lingual infarction; giant cell arteritis; tongue claudicatiom
The o c c u r r e n c e of lingual ischemia or infarction, although rare, has been seen due to trauma [1], arteritis [2-6] and carotid artery disease [7]. The importance of this finding and early diagnosis is paramount since early treatment will limit tissue loss and progression of disease.
CASE REPORT A 79-year-old white woman presented to her physician with acute right ocular blindness and a three month history of right facial and jaw pain. The patient also experienced increasing pain on swallowing and difficulty articulating words. One month prior to this admission she complained of fatigue, lethargy, and swelling of the tongue with inability to eat. She was hospitalized and during her admission was found to have a left lower lobe pneumonia. She completed medical treatment of her pneumonia and had a percutaneous endoscopic gastrostomy placed to supplement her nutrition. The patient was
From the Department of Surgery, University of Miami/ Jackson Memorial Medical Center*, Miami, Florida and the Saint Anthony Medical Center, Columbus, Ohio. Reprint requests: Enrique Ginzburg, MD, University of Miami~Jackson Memorial Medical Center, Department of Surgery (D40), 1611 N.W. 12 Avenue, Miami, Florida 33136.
seen again one month later with a foul smelling centrally infarcted segment of her tongue. On physical examination, this thin yet very alert woman, had a mobile tongue with a central area of dark necrotic tissue extending to the base of the tongue posterior to the circumvallate papillae. This can be clearly seen in Figure 1 as the dark area of the tongue posterior to health tissue bordered by sloughed mucosa. She had a right carotid bruit and only Doppler signals of her temporal arteries were evident. The patient underwent noninvasive vascular studies which showed stenosis of 20-30% of her left common carotid artery, 40-50% of the left internal carotid artery, and an open left external carotid artery. The right side studies showed 20-30% stenosis of her common carotid artery, 80-99% stenosis of her internal carotid artery, and a very stenotic external carotid artery. The patient had a negative systemic lupus antibody test and an elevated erythrocyte sedimentation rate (ESR). She underwent debridement with biopsies which came back negative for neoplasm. Arteriogram was performed which showed greater than 90% stenosis of the right internal carotid artery with an occluded right lingual artery stump (see Fig. 2). In conjunction with right lingual artery occlusion there is also left sided lingual artery occlusion as seen in Figure 3, which is an extremely important factor contributing to lingual infarction. The patient was offered a temporal artery biopsy for diagnostic purposes but refused under recommendation of her neurologist. His clinical impression was that the
450
VOLUME 6 N o 5 - 1992
LINGUAL INFARCTION
Fig. 1. Original presentation of this patient's infarcted tongue. After debridement patient was left with a sole central ischemic area of necrosis surrounded by healthy tissue.
patient had giant cell arteritis with an elevated ESR of 120. He promptly placed her on a steroid regimen. During her current hospitalization the patient underwent further debridement of the necrotic lingual tissue with marked improvement in oral intake and improved phonation. The patient will be scheduled for elective carotid surgery if no improvement results in her carotid disease after steroid protocol.
451
Fig. 3. Arteriogram of left carotid artery showing total occlusion of the lingual artery (arrow) consistent with Missen's autopsy results.
Since that first report, lingual necrosis has been commonly associated with giant cell temporal arteritis. This arteritis is a general vasculitis predominantly affecting the large and medium-sized arteries in people over 50 years of age [9]. Cranial arteritis has the common manifestations of headache and visual loss, presenting in nearly 50% of patients with polymyalgia rheumatica [I0]. Although otolaryngologic manifestations of craDISCUSSION nial arteritis are said to be infrequent, approxiThe rarity of lingual infarction is evident from a mately 25% of these patients have complaints or 25 year literature search on the subject revealing symptoms of masticatory claudication, tongue pain, fewer than 20 cases reported in the world literature, and frank lingual infarction prior to a diagnosis of the first in the United States in 1967, by Freedman, arteritis [11]. These symptoms of intermittent an oral surgeon [8], preceded by Horton in 1966 tongue claudication and paroxysmal blanching are [12]. important warnings of impending glossal infarction, just as visual scotoma are to permanent visual loss [10]. The most dreaded consequence of giant cell arteritis is visual loss which is usually irreversible. ~t One study revealed a 46% rate of total or partial blindness in 358 cases of temporal arteritis [13]. It also has other clinical manifestations such as lip and scalp necrosis, carpal tunnel syndrome, limb claudication, stroke, angina pectoris and pulmonary disease [9]. Giant cell arteritis mainly affects the superficial temporal, vertebral and ophthalmic arteries [14]. However, large arteries may be involved in temporal arteritis, and more than 50% of patients will have concomitant bruits of the carotid, subclavian, and brachial arteries, which disappear under steroid treatment [11]. Lab analysis of sedimentation rates Fig. 2. Arteriogram of carotid artery showing high rade stenosis of active arteritis and occluded stump are usually elevated with a mean of 100 ram/hr. Normochromic anemia refractory to iron therapy is rrow) of the lingual artery.
ANNALS OF VASCULAR SURGERY
LINGUAL INFARCI10N
452
also a common finding in these patients [11]. Midtongue carcinoma, abscess of floor of the mouth, and arterial embolization and atrial fibrillation have also been responsible for characteristic tongue infarcts. Moniliasis and Vincent's angina are also common early considerations [11]. The low incidence of tongue infarction with giant cell arteritis has been attributed to the abundant vascular supply to the tongue, consisting mainly of the lingual artery and both the ascending pharyngeal and external fascial artery [15]. However, lingual artery involvement has been suggested to be very common in giant cell arteritis. Missen demonstrated inflammatory changes, postmortem, in 9 out of I0 arteries in five cases of giant cell arteritis [16]. This was also demonstrated in our patient with bilateral occlusions of the lingual artery, as seen in Figures 2 and 3. Tongue pain can be demonstrated by requiring repetitive rapid protrusion with resulting symptoms of pain, burning, or fatigue with tongue blanching [I0]. Prompt treatment with corticosteroids in large doses followed by a lower maintenance dose is important to limit and present further vascular complications once arteritis is diagnosed.
REFERENCES 1. 2. 3. 4. 5. 6. 7. 8. 9. 10. 11. 12. 13.
ACKNOWLEDGMENT
14.
The authors wish to thank Kathy Boston, RN, Cuba Reinhardt, RN, RVT, Sherry Kistler, RN, RVT, Margaret Kuhn, RN, RVT, and Kim Oren for their assistance. Imm
15. 16.
SIEGEL RL, SPEISER AM, BIKOFSKY VM. Tongue ischemia from a soft drink can: a case report. J Am Dental Assoc 1990;121(5):607-608. ROSEMAN BB, GRANITE E. Massive tongue necrosis secondary to temporal arteritis. J Oral Maxillofacial Surg 1984 ;42(10):682-684. PEDERSON AT, JEPSEN FL. Lingual infarction in giant cell arteritis: a case report. J Laryngol Otolaryngol 1983; 97(5):479--483. DARE B, BYRNE E, ROBERTSON A. Acute lingual ischemia complicating temporal arteries, Med J Aust 1981; 1(10):534. JENKINS PR. Lingual infarction in cranial arteritis. Dental Update 1985;12(5):303-305. ARNUNG K, NIELSEN IL. Temporal arteritis and gangrene of the tongue, ACTA Med Scand 1979;206(3):239-240. GOODMAN JM, ZINK WL, COOPER DF. Hemilingual paralysis caused by spontaneous carotid artery dissection. Arch Neurol 1983;40(10):653-654. FREEDMAN GL, HOOLEY JR. Ischemic necrosis of the tongue: a case report, Oral Surg 1967;24(6):821-824. SHERARD RK, COLERDGE ST. Giant cell arteritis. J Emerg Med 1986;4(4):293-299. SOFFERMAN RA. Cranial arteritis in otolaryngology. Ann Otolaryngology 1980;89:215-219. SOFFERMAN RA. Lingual infarction in cranial aneritis. JAMA 1980;243(23):2422-2423. HORTON BT. Complications of temporal arteritis. Br Med J 1966;3:105-106. BIRKHEAD NC, WAGENER HP, WEIHMANN J. Treatment of temporal arteritis with adrenal corticosteroids. JAMA 1957;163:821-827. WILKINSON MD, RUSSEL RWR. Arteries of head and neck in GCA. Arch Neurol 1972;27:378-391. REED C, INGLIS MJ. Acute massive gangrene of the tongue. Br Med J 1965;2:575-576. MISSEN GAK. Gangrene of the tongue. Br Med J 1961 ;l: 1343-1344.
