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Tongue necrosis: a rare complication of prolonged oral intubation

Tongue necrosis: a rare complication of prolonged oral intubation This is a case report of necrosis of more than two-third of the dorsal tongue in a 74-year-old male following prolonged oral intubation for vascular surgery. This necrosis progressed from the left tongue to involve much of the dorsal tongue bilaterally. A diagnosis of rhabdomyolysis was made evident by compartment syndrome of the legs with an elevated creatine kinase level of 89 789 u/l (units per litre). The literature also reveals that vasopressin has been linked with reported cases of tongue necrosis. Other possible aetiological factors were discussed in this finding. CASE REPORT INTRODUCTION A 74-year-old male patient was admitted in 2018 by the The tongue is a well-vascularized organ, with a blood supply Vascular Team for a fenestrated cuff repair (type 1a) of an aortic from the lingual artery, a branch of the external carotid artery. endovascular leak. The patient had been admitted in 2009 for a It has a limited secondary contralateral supply from the submu- 7.1 cm abdominal aortic aneurysm of the infra-renal aorta, and cosal plexus. Necrosis of the tongue is not a common finding [1]. an endovascular aneurysm repair had been performed with an Tongue necrosis has most frequently been associated with endurant bifurcated device. At the latest admission, a computer giant cell arthritis [1–5], as seen in temporal arthritis, often angiogram of the aorta revealed a type 1a endovascular leak of diagnosed by a high erythrocyte sedimentation rate [1, 2, 6, 7]. 6.4 cm. It may also occur with cardiogenic shock [1], leading to end- Regular medications included warfarin, bisoprolol, glicalzide, organ hypoperfusion. Vascular compromise may occur with use linagliptin, prednisolone, ramipril and salbutamol. There were of vasoconstrictors [2, 3] such as vasopressin, especially when a significant medical comorbidities including chronic obstructive high dose or continuous infusion is required to maintain blood pressure [2, 5]. pulmonary disease (COPD), high body mass index, atrial fib- Tongue necrosis has also been associated with vascular rillation, diabetes mellitus type 2, hypertension, smoking and diseases such as, Kawasaki disease [7, 8], Wegener granulomato- obstructive sleep apnoea. sis [1, 3, 6], hypercoagulable condition such as disseminated The patient was admitted to the adult intensive care unit intravascular coagulation and rheumatoid hyperviscosity (AICU) at our institute. The vascular surgery was prolonged with syndrome [6]. over 10 hours with oral intubation. Received: July 31, 2019. Revised: August 23, 2019. Accepted: September 9, 2019 Published by Oxford University Press and JSCR Publishing Ltd. All rights reserved. © The Author(s) 2019. This is an Open Access article distributed under the terms of the Creative Commons Attribution Non-Commercial License (http://creativecommons.org/ licenses/by-nc/4.0/), which permits non-commercial re-use, distribution, and reproduction in any medium, provided the original work is properly cited. For commercial re-use, please contact journals.permissions@oup.com 1 Downloaded from https://academic.oup.com/jscr/article-abstract/2019/11/rjz284/5628319 by DeepDyve user on 03 December 2019 2 N.A. Adegbite et al. At day 15, he was discharged to the ward but deteriorated the following day with respiratory failure secondary to hospital- acquired pneumonia and mucus plugging of the left lung. He was reintubated and readmitted to AICU. Inotropic sup- port was required for hypotension and bradycardia. Episodes of melana were noted and had blood transfused. Percutaneous tracheostomy was performed. At Day 34, the patient was taken back to theatre by the maxillofacial team. Much of the anterior and part of the posterior third of the dorsum of the tongue was necrotic (Fig. 2). The necrotic area was removed, and the tongue debrided to healthy tissue. At 12 months review, the dorsum of the tongue had healed with considerable scarring (Fig. 3). Despite the significant restric- tion in tongue movement and speech deficit, the patient how- ever declined further surgical intervention. Figure 1: Necrosis of the tip of tongue. DISCUSSION Ischaemic necrosis of the tongue has been reported in cases of giant cell arteritis (GCA), circulatory shock, Kawasaki disease, Wegener’s granulomatosis, disseminated intravascular coagu- lation and prolonged use of vasopressin. It is probably most commonly associated with GCA. There was no evidence of these alternative causes except for prolonged use of vasopressin. Necrosis of the tongue in this case is unique because it was probably due to the prolonged oral intubation with compression by an anaesthetic tube. The necrosis of the dorsum of the tongue was progressive from the left to the right side to involve the side not compressed by the tube. Using a flexible rather than a rigid tube would probably have minimized the risk of compression. In retrospect, a soft material Figure 2: Bilateral dorsal tongue involvements. (e.g. wet or greased sponge) could have been used as a barrier between the tongue and the oral tube. It is speculated that type 2 respiratory failure, exacerbated by the pre-existing COPD and mucus plugging, together with the compartment syndrome may have resulted in reduced tissue perfusion and rhabdomyolysis. The pre-existing multiple comor- bidities probably contributed to the development of multiple organ failure. The very high creatine kinase is a feature of this case not previously reported elsewhere. ACKNOWLEDGEMENT We are grateful to patient for allowing this report. FUNDING This study was funded by Leicester Royal Infirmary. Figure 3: Extensive scarring of the tongue. REFERENCES On Day 2, a diagnosis of rhabdomyolysis was made with 1. Benjamin RR, Immerman SB, Morris L. Ischemic necrosis of clinical and imaging evidence of compartment syndrome of the the tongue in patients with cardiogenic shock. Laryngoscope legs with an elevated creatine kinase of 90 000 u/l. Bilateral 2010;120:1345–9. fasciotomies was performed of both lower limbs due to a sud- 2. Jinbeom C, Kiyoung S, Dosang L. Ischemic necrosis of the den onset of bilateral compartment syndrome. The patient had tongue in surgical patients with septic shock: a case report. developed acute renal failure, and continuous renal replacement BMC Surgery 2016;16:48. therapy was commenced for metabolic acidosis. 3. Carter LM, Brizman E. Lingual infarction in Wegener’s granu- On Day 3, a black tongue was noted, and referral was made to lomatosis: a case report and review of the literature. Head Face the maxillofacial team (Fig. 1). Blackness of the left tongue dor- Med 2008;4:19. [PubMed: 18718013]. sum was obvious. The patient was very unwell. Chlorhexidine 4. Sofferman RA. Lingual infarction in cranial arteritis. JAMA mouth was prescribed. 1980;243:2422–3. [PubMed: 7373821]. Downloaded from https://academic.oup.com/jscr/article-abstract/2019/11/rjz284/5628319 by DeepDyve user on 03 December 2019 Tongue necrosis 3 5. Zimmermann AT, Brown M. Tongue infarction in giant 7. Paul MB, Alex DA, Wichita K. Tongue necrosis as an cell (temporal) arteritis. Intern Med J 2008:38:376. [PubMed: unusual presentation of carotid artery stenosis. JVascSurg 18402568]. 2011;54:837–9. 6. Brodmann M, Dorr A, Hafner F, Gary T, Pilger E. Tongue 8. Scardina GA, Fuca G, Carini F. Oral necrotizing microvasculitis necrosis as first symptom of giant cell arteritis (GCA). Clin in a patient affected by Kawasaki disease. Med Oral Patol Oral Rheumatol 2009;28:S47–9. [PubMed: 19277817]. Cir Buccal 2007:12:E560–4. [PubMed: 18059239]. http://www.deepdyve.com/assets/images/DeepDyve-Logo-lg.png Journal of Surgical Case Reports Oxford University Press

Tongue necrosis: a rare complication of prolonged oral intubation

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Oxford University Press
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Published by Oxford University Press and JSCR Publishing Ltd. All rights reserved. © The Author(s) 2019.
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2042-8812
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Abstract

This is a case report of necrosis of more than two-third of the dorsal tongue in a 74-year-old male following prolonged oral intubation for vascular surgery. This necrosis progressed from the left tongue to involve much of the dorsal tongue bilaterally. A diagnosis of rhabdomyolysis was made evident by compartment syndrome of the legs with an elevated creatine kinase level of 89 789 u/l (units per litre). The literature also reveals that vasopressin has been linked with reported cases of tongue necrosis. Other possible aetiological factors were discussed in this finding. CASE REPORT INTRODUCTION A 74-year-old male patient was admitted in 2018 by the The tongue is a well-vascularized organ, with a blood supply Vascular Team for a fenestrated cuff repair (type 1a) of an aortic from the lingual artery, a branch of the external carotid artery. endovascular leak. The patient had been admitted in 2009 for a It has a limited secondary contralateral supply from the submu- 7.1 cm abdominal aortic aneurysm of the infra-renal aorta, and cosal plexus. Necrosis of the tongue is not a common finding [1]. an endovascular aneurysm repair had been performed with an Tongue necrosis has most frequently been associated with endurant bifurcated device. At the latest admission, a computer giant cell arthritis [1–5], as seen in temporal arthritis, often angiogram of the aorta revealed a type 1a endovascular leak of diagnosed by a high erythrocyte sedimentation rate [1, 2, 6, 7]. 6.4 cm. It may also occur with cardiogenic shock [1], leading to end- Regular medications included warfarin, bisoprolol, glicalzide, organ hypoperfusion. Vascular compromise may occur with use linagliptin, prednisolone, ramipril and salbutamol. There were of vasoconstrictors [2, 3] such as vasopressin, especially when a significant medical comorbidities including chronic obstructive high dose or continuous infusion is required to maintain blood pressure [2, 5]. pulmonary disease (COPD), high body mass index, atrial fib- Tongue necrosis has also been associated with vascular rillation, diabetes mellitus type 2, hypertension, smoking and diseases such as, Kawasaki disease [7, 8], Wegener granulomato- obstructive sleep apnoea. sis [1, 3, 6], hypercoagulable condition such as disseminated The patient was admitted to the adult intensive care unit intravascular coagulation and rheumatoid hyperviscosity (AICU) at our institute. The vascular surgery was prolonged with syndrome [6]. over 10 hours with oral intubation. Received: July 31, 2019. Revised: August 23, 2019. Accepted: September 9, 2019 Published by Oxford University Press and JSCR Publishing Ltd. All rights reserved. © The Author(s) 2019. This is an Open Access article distributed under the terms of the Creative Commons Attribution Non-Commercial License (http://creativecommons.org/ licenses/by-nc/4.0/), which permits non-commercial re-use, distribution, and reproduction in any medium, provided the original work is properly cited. For commercial re-use, please contact journals.permissions@oup.com 1 Downloaded from https://academic.oup.com/jscr/article-abstract/2019/11/rjz284/5628319 by DeepDyve user on 03 December 2019 2 N.A. Adegbite et al. At day 15, he was discharged to the ward but deteriorated the following day with respiratory failure secondary to hospital- acquired pneumonia and mucus plugging of the left lung. He was reintubated and readmitted to AICU. Inotropic sup- port was required for hypotension and bradycardia. Episodes of melana were noted and had blood transfused. Percutaneous tracheostomy was performed. At Day 34, the patient was taken back to theatre by the maxillofacial team. Much of the anterior and part of the posterior third of the dorsum of the tongue was necrotic (Fig. 2). The necrotic area was removed, and the tongue debrided to healthy tissue. At 12 months review, the dorsum of the tongue had healed with considerable scarring (Fig. 3). Despite the significant restric- tion in tongue movement and speech deficit, the patient how- ever declined further surgical intervention. Figure 1: Necrosis of the tip of tongue. DISCUSSION Ischaemic necrosis of the tongue has been reported in cases of giant cell arteritis (GCA), circulatory shock, Kawasaki disease, Wegener’s granulomatosis, disseminated intravascular coagu- lation and prolonged use of vasopressin. It is probably most commonly associated with GCA. There was no evidence of these alternative causes except for prolonged use of vasopressin. Necrosis of the tongue in this case is unique because it was probably due to the prolonged oral intubation with compression by an anaesthetic tube. The necrosis of the dorsum of the tongue was progressive from the left to the right side to involve the side not compressed by the tube. Using a flexible rather than a rigid tube would probably have minimized the risk of compression. In retrospect, a soft material Figure 2: Bilateral dorsal tongue involvements. (e.g. wet or greased sponge) could have been used as a barrier between the tongue and the oral tube. It is speculated that type 2 respiratory failure, exacerbated by the pre-existing COPD and mucus plugging, together with the compartment syndrome may have resulted in reduced tissue perfusion and rhabdomyolysis. The pre-existing multiple comor- bidities probably contributed to the development of multiple organ failure. The very high creatine kinase is a feature of this case not previously reported elsewhere. ACKNOWLEDGEMENT We are grateful to patient for allowing this report. FUNDING This study was funded by Leicester Royal Infirmary. Figure 3: Extensive scarring of the tongue. REFERENCES On Day 2, a diagnosis of rhabdomyolysis was made with 1. Benjamin RR, Immerman SB, Morris L. Ischemic necrosis of clinical and imaging evidence of compartment syndrome of the the tongue in patients with cardiogenic shock. Laryngoscope legs with an elevated creatine kinase of 90 000 u/l. Bilateral 2010;120:1345–9. fasciotomies was performed of both lower limbs due to a sud- 2. Jinbeom C, Kiyoung S, Dosang L. Ischemic necrosis of the den onset of bilateral compartment syndrome. The patient had tongue in surgical patients with septic shock: a case report. developed acute renal failure, and continuous renal replacement BMC Surgery 2016;16:48. therapy was commenced for metabolic acidosis. 3. Carter LM, Brizman E. Lingual infarction in Wegener’s granu- On Day 3, a black tongue was noted, and referral was made to lomatosis: a case report and review of the literature. Head Face the maxillofacial team (Fig. 1). Blackness of the left tongue dor- Med 2008;4:19. [PubMed: 18718013]. sum was obvious. The patient was very unwell. Chlorhexidine 4. Sofferman RA. Lingual infarction in cranial arteritis. JAMA mouth was prescribed. 1980;243:2422–3. [PubMed: 7373821]. Downloaded from https://academic.oup.com/jscr/article-abstract/2019/11/rjz284/5628319 by DeepDyve user on 03 December 2019 Tongue necrosis 3 5. Zimmermann AT, Brown M. Tongue infarction in giant 7. Paul MB, Alex DA, Wichita K. Tongue necrosis as an cell (temporal) arteritis. Intern Med J 2008:38:376. [PubMed: unusual presentation of carotid artery stenosis. JVascSurg 18402568]. 2011;54:837–9. 6. Brodmann M, Dorr A, Hafner F, Gary T, Pilger E. Tongue 8. Scardina GA, Fuca G, Carini F. Oral necrotizing microvasculitis necrosis as first symptom of giant cell arteritis (GCA). Clin in a patient affected by Kawasaki disease. Med Oral Patol Oral Rheumatol 2009;28:S47–9. [PubMed: 19277817]. Cir Buccal 2007:12:E560–4. [PubMed: 18059239].

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Journal of Surgical Case Reports – Oxford University Press

Published: Nov 1, 2019

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