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Thoracic aortic aneurysm causing aorto-esophageal fistula—our experience with a rare disease

Thoracic aortic aneurysm causing aorto-esophageal fistula—our experience with a rare disease Aorto-esophageal fistula is a life-threatening condition, accounting for a small number of cases of upper gastrointestinal bleeding where patients present with one or more features of Chiari’s triad. We present the case of a 43-year-old woman, referred to us with symptoms of central chest pain, sudden onset dysphagia followed by massive hemoptysis. She was diagnosed with an aorto-esophageal fistula due to a ruptured thoracic aortic aneurysm and rushed for an emergency endovascular thoracic aortic stent and feeding jejunostomy with intravenous antibiotics and supportive care. After 6 weeks of surgery, the patient was re-evaluated to plan for an esophageal stent if required. The purpose of this presentation is to make the surgical fraternity aware of the gravity of this disease and novel techniques to manage it. INTRODUCTION CASE REPORT Thoracic aortic aneurysms account for about two-thirds of all A 43-year old women was rushed to the nearest hospital with aorto-esophageal fistulas (AEFs). Early and aggressive treatment presenting complaint of mild epigastric pain since two days is required, as there is risk of massive bleeding, infection, sepsis followed by sudden onset moderate to severe central chest and ultimately death. Conservative medical management results pain, dysphagia and progressive difficulty in breathing. She then in no late survival [1]. developed two episodes of hematemesis with melena. She is an Several types of treatment have been described, including active smoker with no known co-morbidities. The patient was open surgery, temporary control measures such as percutaneous then rapidly resuscitated with multiple blood transfusions and embolization and the use of a Sengstaken–Blakemore tube supportive care to stabilize her vital parameters and referred to and, more recently, endovascular repair. Infected AEF with our center for further management. poor clinical condition renders them at high risk for open On presentation to our center, the patient was pale looking surgery. Endovascular surgery provides palliative treatment or with stable vitals. Chest and abdomen examination was unre- a temporary alternative until patients are healthy enough to markable. Coagulation profile and cardiac enzymes were within tolerate open surgery. normal limits. However, there was a fall in serial hemoglobin. We present a case report of a 43-year-old woman who pre- Ultrasound of abdomen and pelvis was normal. Upper gas- sented at our institute with a thoracic AEF and underwent tho- trointestinal tract (UGI) endoscopy was done immediately, which racic endovascular aortic repair (TEVAR) as a life-saving proce- revealed ulceration with a blood clot at 25 cm of the esophagus dure. (Fig. 1). Received: May 4, 2020. Revised: June 14, 2020. Accepted: June 22, 2020 Published by Oxford University Press and JSCR Publishing Ltd. All rights reserved. © The Author(s) 2020. This is an Open Access article distributed under the terms of the Creative Commons Attribution Non-Commercial License (http://creativecommons.org/li censes/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/2020/7/rjaa242/5875150 by DeepDyve user on 27 August 2020 2 A. Yadav et al. Figure 1: UGI endoscopy, clot with underlying ulceration in the esophagus. Figure 3: CE-CT image, transverse section. Figure 2: CE-CT image showing saccular aneurysm arising from the proximal Figure 4: Fluoroscopy image of endovascular stent before deploying. descending thoracic aorta. Repeat chest X-ray showed developing left-sided pleural effu- immediately taken for emergency TEVAR where 34 × 34 × 167- sion. mm stent graft (Medtronic Inc. Valiant Thoracic Stent Graft) was Contrast-enhanced chest computed tomography (CE-CT) deployed using a guidewire beyond the left subclavian artery with thoracic and abdominal aortogram showed a saccular till above the diaphragm via an incision over the left common aneurysm arising from the right lateral wall of the proximal femoral artery. Check angiogram showed no leak (Figs 4 and 5). descending thoracic aorta (10 × 8 mm, neck 8 mm) with Due to existing mediastinitis, feeding jejunostomy (FJ) was pre- peri-aneurysmal hematoma tracking along the left posterior ferred over esophageal stenting. FJ was performed in the same mediastinum, causing anterior displacement of the esophagus setting after the endovascular procedure. and a left-sided hemothorax suggestive of a ruptured aneurysm Post-operatively, she was continued on broad-spectrum (Figs 2 and 3). antibiotics for mediastinitis with a gradual increase in FJ feed, With the above findings, a diagnosis of ruptured descending which she tolerated well. She had an uneventful recovery and thoracic aortic aneurysm with AEF was made. The patient was was discharged within a week following the procedure. Downloaded from https://academic.oup.com/jscr/article/2020/7/rjaa242/5875150 by DeepDyve user on 27 August 2020 Our experience with a rare disease 3 in 1818. Incidence of AEF is reported to be 5–23% among all the causes of critical gastrointestinal bleeding by a meta-analysis [2]. Chiari [3] reported the classic clinical triad of mid-thoracic chest pain, sentinel arterial hemorrhage and fatal hemorrhage as ‘aorto-esophageal syndrome’. Causes of AEF are ruptured or non-ruptured aortic aneurysm, foreign body ingestion and advanced esophageal malignancy. Some cases of AEFs occur as a complication after surgical pros- thetic repair of an aneurysm or ‘following’ TEVAR [1]. Snyder and Crawford [4] reported the first survival following surgical repair in 1983, but such cases are few. The treatment process involves control or prevention of fatal bleeding followed by definitive aortic treatment, accompanied by methods to stop continuous contamination through the fistula to the aortic prosthesis and treating fistulous esophageal com- munication. At present, treatment options include either open surgery or endovascular repair. It is advisable that patients with clinical signs of infection and extensive contamination of the surround- ing tissues demonstrated by imaging studies, as air bubbles or pleural effusion, or patients with AEF due to foreign body inges- tion, should undergo open surgery immediately. The procedure should include debridement of any devitalized tissue, including the compromised segment of the esophagus, and replacement of the diseased aortic segment [5]. In 1994, thoracic endovascular aortic repair (TEVAR) was Figure 5: Endovascular stent after deployment. introduced as an alternative to open surgical repair for treatment of descending thoracic aortic aneurysm and is routinely used in aortic emergencies or high-risk elderly patients. It is the mainstay for providing provisional or palliative treatment, until the patient becomes fit for a definitive procedure. It offers a minimal access procedure saving major operating time and reducing perioperative complications associated with open repair [6]. Recently, there are concerns regarding an increased number of AEF cases after TEVAR. Reports have suggested that TEVAR fol- lowed by early open repair (bridging) resulted in better outcomes [7], optimal timing being <1 week; otherwise, the stent-graft is being deeply involved in the infection process. Optimal timing for esophageal reconstruction is at least several months [1]. AEF, being rare with a small number of cases, requires fur- ther studies and trials to come up with the best management plan. However, esophageal stenting or reconstruction was not required in our case as esophageal perforation had sponta- neously resolved. CONFLICT OF INTEREST None. Figure 6: Gastrograffin swallow showing no esophageal leak. REFERENCES 1. Yamazato T, Nakamura T, Abe N, Yokawa K, Ikeno Y, Koda Y, et al. Surgical strategy for the treatment of aortoesophageal After 6 weeks, when mediastinitis subsided, gastrograffin fistula. J Thorac Cardiovasc Surg 2018;155:32–40. swallow was done, which showed no esophageal leak suggesting 2. Malik MU, Ucbilek E, Sherwal AS. Critical gastrointestinal spontaneous healing of perforation (Fig. 6). Oral feed was com- bleed due to secondary aortoenteric fistula. J Community Hosp menced with liquid and, subsequently, a normal diet. Intern Med Perspect 2015;5:10. 3. Chiari H. Ueber Premdkorperverletzung des oesophagus mit aortenperforation. Ber Klin Wochenschr 1914;51:7–9. DISCUSSION 4. Snyder DM, Crawford ES. Successful treatment of primary AEF remains a life-threatening condition with a high rate of aorta-esophageal fistula resulting from aortic aneurysm. morbidity and mortality, described for the first time by Dubreuil J Thorac Cardiovasc Surg 1983;85:457–63. Downloaded from https://academic.oup.com/jscr/article/2020/7/rjaa242/5875150 by DeepDyve user on 27 August 2020 4 A. Yadav et al. 5. Flores J, Shiiya N, Kunihara T, Yoshimoto K, Yasuda K. Aortoe- 7. Vallabhajosyula P, Komlo C, Wallen T, Szeto WY. Two-stage sophageal fistula: alternatives of treatment case report and surgical strategy for aortoesophageal fistula: emergent tho- literature review. Ann Thorac Cardiovasc Surg 2004;10:241–6. racic endovascular aortic repair followed by definitive open 6. Chiu P, Goldstone AB, Schaffer JM, Lingala B, Miller D, Mitchell aortic and esophageal reconstruction. J Thorac Cardiovasc Surg R, et al. Endovascular versus open repair of intact descending 2012;144:1266–8. thoracic aortic aneurysms. J Am Coll Cardiol 2019;73:643–51. http://www.deepdyve.com/assets/images/DeepDyve-Logo-lg.png Journal of Surgical Case Reports Oxford University Press

Thoracic aortic aneurysm causing aorto-esophageal fistula—our experience with a rare disease

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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) 2020.
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2042-8812
DOI
10.1093/jscr/rjaa242
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Abstract

Aorto-esophageal fistula is a life-threatening condition, accounting for a small number of cases of upper gastrointestinal bleeding where patients present with one or more features of Chiari’s triad. We present the case of a 43-year-old woman, referred to us with symptoms of central chest pain, sudden onset dysphagia followed by massive hemoptysis. She was diagnosed with an aorto-esophageal fistula due to a ruptured thoracic aortic aneurysm and rushed for an emergency endovascular thoracic aortic stent and feeding jejunostomy with intravenous antibiotics and supportive care. After 6 weeks of surgery, the patient was re-evaluated to plan for an esophageal stent if required. The purpose of this presentation is to make the surgical fraternity aware of the gravity of this disease and novel techniques to manage it. INTRODUCTION CASE REPORT Thoracic aortic aneurysms account for about two-thirds of all A 43-year old women was rushed to the nearest hospital with aorto-esophageal fistulas (AEFs). Early and aggressive treatment presenting complaint of mild epigastric pain since two days is required, as there is risk of massive bleeding, infection, sepsis followed by sudden onset moderate to severe central chest and ultimately death. Conservative medical management results pain, dysphagia and progressive difficulty in breathing. She then in no late survival [1]. developed two episodes of hematemesis with melena. She is an Several types of treatment have been described, including active smoker with no known co-morbidities. The patient was open surgery, temporary control measures such as percutaneous then rapidly resuscitated with multiple blood transfusions and embolization and the use of a Sengstaken–Blakemore tube supportive care to stabilize her vital parameters and referred to and, more recently, endovascular repair. Infected AEF with our center for further management. poor clinical condition renders them at high risk for open On presentation to our center, the patient was pale looking surgery. Endovascular surgery provides palliative treatment or with stable vitals. Chest and abdomen examination was unre- a temporary alternative until patients are healthy enough to markable. Coagulation profile and cardiac enzymes were within tolerate open surgery. normal limits. However, there was a fall in serial hemoglobin. We present a case report of a 43-year-old woman who pre- Ultrasound of abdomen and pelvis was normal. Upper gas- sented at our institute with a thoracic AEF and underwent tho- trointestinal tract (UGI) endoscopy was done immediately, which racic endovascular aortic repair (TEVAR) as a life-saving proce- revealed ulceration with a blood clot at 25 cm of the esophagus dure. (Fig. 1). Received: May 4, 2020. Revised: June 14, 2020. Accepted: June 22, 2020 Published by Oxford University Press and JSCR Publishing Ltd. All rights reserved. © The Author(s) 2020. This is an Open Access article distributed under the terms of the Creative Commons Attribution Non-Commercial License (http://creativecommons.org/li censes/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/2020/7/rjaa242/5875150 by DeepDyve user on 27 August 2020 2 A. Yadav et al. Figure 1: UGI endoscopy, clot with underlying ulceration in the esophagus. Figure 3: CE-CT image, transverse section. Figure 2: CE-CT image showing saccular aneurysm arising from the proximal Figure 4: Fluoroscopy image of endovascular stent before deploying. descending thoracic aorta. Repeat chest X-ray showed developing left-sided pleural effu- immediately taken for emergency TEVAR where 34 × 34 × 167- sion. mm stent graft (Medtronic Inc. Valiant Thoracic Stent Graft) was Contrast-enhanced chest computed tomography (CE-CT) deployed using a guidewire beyond the left subclavian artery with thoracic and abdominal aortogram showed a saccular till above the diaphragm via an incision over the left common aneurysm arising from the right lateral wall of the proximal femoral artery. Check angiogram showed no leak (Figs 4 and 5). descending thoracic aorta (10 × 8 mm, neck 8 mm) with Due to existing mediastinitis, feeding jejunostomy (FJ) was pre- peri-aneurysmal hematoma tracking along the left posterior ferred over esophageal stenting. FJ was performed in the same mediastinum, causing anterior displacement of the esophagus setting after the endovascular procedure. and a left-sided hemothorax suggestive of a ruptured aneurysm Post-operatively, she was continued on broad-spectrum (Figs 2 and 3). antibiotics for mediastinitis with a gradual increase in FJ feed, With the above findings, a diagnosis of ruptured descending which she tolerated well. She had an uneventful recovery and thoracic aortic aneurysm with AEF was made. The patient was was discharged within a week following the procedure. Downloaded from https://academic.oup.com/jscr/article/2020/7/rjaa242/5875150 by DeepDyve user on 27 August 2020 Our experience with a rare disease 3 in 1818. Incidence of AEF is reported to be 5–23% among all the causes of critical gastrointestinal bleeding by a meta-analysis [2]. Chiari [3] reported the classic clinical triad of mid-thoracic chest pain, sentinel arterial hemorrhage and fatal hemorrhage as ‘aorto-esophageal syndrome’. Causes of AEF are ruptured or non-ruptured aortic aneurysm, foreign body ingestion and advanced esophageal malignancy. Some cases of AEFs occur as a complication after surgical pros- thetic repair of an aneurysm or ‘following’ TEVAR [1]. Snyder and Crawford [4] reported the first survival following surgical repair in 1983, but such cases are few. The treatment process involves control or prevention of fatal bleeding followed by definitive aortic treatment, accompanied by methods to stop continuous contamination through the fistula to the aortic prosthesis and treating fistulous esophageal com- munication. At present, treatment options include either open surgery or endovascular repair. It is advisable that patients with clinical signs of infection and extensive contamination of the surround- ing tissues demonstrated by imaging studies, as air bubbles or pleural effusion, or patients with AEF due to foreign body inges- tion, should undergo open surgery immediately. The procedure should include debridement of any devitalized tissue, including the compromised segment of the esophagus, and replacement of the diseased aortic segment [5]. In 1994, thoracic endovascular aortic repair (TEVAR) was Figure 5: Endovascular stent after deployment. introduced as an alternative to open surgical repair for treatment of descending thoracic aortic aneurysm and is routinely used in aortic emergencies or high-risk elderly patients. It is the mainstay for providing provisional or palliative treatment, until the patient becomes fit for a definitive procedure. It offers a minimal access procedure saving major operating time and reducing perioperative complications associated with open repair [6]. Recently, there are concerns regarding an increased number of AEF cases after TEVAR. Reports have suggested that TEVAR fol- lowed by early open repair (bridging) resulted in better outcomes [7], optimal timing being <1 week; otherwise, the stent-graft is being deeply involved in the infection process. Optimal timing for esophageal reconstruction is at least several months [1]. AEF, being rare with a small number of cases, requires fur- ther studies and trials to come up with the best management plan. However, esophageal stenting or reconstruction was not required in our case as esophageal perforation had sponta- neously resolved. CONFLICT OF INTEREST None. Figure 6: Gastrograffin swallow showing no esophageal leak. REFERENCES 1. Yamazato T, Nakamura T, Abe N, Yokawa K, Ikeno Y, Koda Y, et al. Surgical strategy for the treatment of aortoesophageal After 6 weeks, when mediastinitis subsided, gastrograffin fistula. J Thorac Cardiovasc Surg 2018;155:32–40. swallow was done, which showed no esophageal leak suggesting 2. Malik MU, Ucbilek E, Sherwal AS. Critical gastrointestinal spontaneous healing of perforation (Fig. 6). Oral feed was com- bleed due to secondary aortoenteric fistula. J Community Hosp menced with liquid and, subsequently, a normal diet. Intern Med Perspect 2015;5:10. 3. Chiari H. Ueber Premdkorperverletzung des oesophagus mit aortenperforation. Ber Klin Wochenschr 1914;51:7–9. DISCUSSION 4. Snyder DM, Crawford ES. Successful treatment of primary AEF remains a life-threatening condition with a high rate of aorta-esophageal fistula resulting from aortic aneurysm. morbidity and mortality, described for the first time by Dubreuil J Thorac Cardiovasc Surg 1983;85:457–63. Downloaded from https://academic.oup.com/jscr/article/2020/7/rjaa242/5875150 by DeepDyve user on 27 August 2020 4 A. Yadav et al. 5. Flores J, Shiiya N, Kunihara T, Yoshimoto K, Yasuda K. Aortoe- 7. Vallabhajosyula P, Komlo C, Wallen T, Szeto WY. Two-stage sophageal fistula: alternatives of treatment case report and surgical strategy for aortoesophageal fistula: emergent tho- literature review. Ann Thorac Cardiovasc Surg 2004;10:241–6. racic endovascular aortic repair followed by definitive open 6. Chiu P, Goldstone AB, Schaffer JM, Lingala B, Miller D, Mitchell aortic and esophageal reconstruction. J Thorac Cardiovasc Surg R, et al. Endovascular versus open repair of intact descending 2012;144:1266–8. thoracic aortic aneurysms. J Am Coll Cardiol 2019;73:643–51.

Journal

Journal of Surgical Case ReportsOxford University Press

Published: Jul 1, 2020

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