The role of TEVAR in the management of a recurrent aorto-gastric fistula

The role of TEVAR in the management of a recurrent aorto-gastric fistula An aorto-gastric fistula is a catastrophic and rare cause of an upper gastrointestinal bleed. The diagnosis requires a high index of suspicion and expedient management as any delay in each of these component, will be to the detriment of the patient. We report a case of a patient with two episodes of this rare event, with haemodynamic compromise, 15 years after having had a trans-hiatal oesophagectomy for an adenocarcinoma of the oesophagus who presented on both occasions. He had thoracic endo-vascular aortic repair (TEVAR) on both presentations and survived. This case exemplifies the fact that while TEVAR is a good bridging therapy for the management of an aorto-enteric fistula. It however should not be considered as the definitive management for patients who are operable or patients who do not have prohibitive surgical risk. haematemesis and hypovolaemic shock. His medical history was INTRODUCTION remarkable for a trans-hiatal oesophagectomy and gastric pull-up Despite the first case of an aorto-enteric fistula (AEF) being discov- for adenocarcinoma 15 years prior. After initial fluid resuscitated, ered in 1818; its rare incidence results in a low index of clinical oesophago-gastro-duodenoscopy (OGD) was performed which was suspicion. Its usual presentation is that of sudden onset of mas- complicated by cardiac arrest. He was intubated and fluid resusci- sive haematemesis; and this along with it being rare often results tated until haemodynamically stable. Computed tomography aor- in a late or missed diagnosis and hence a high mortality. tography (CTA) demonstrated an aorto-oesophageal fistula (Fig. 1). The definitive role of thoracic endo-vascular aortic repair A rapid decision was made to proceed with a TEVAR limited to (TEVAR) in the management of AEFs is still undefined. It however that segment of aorta using Cook Zenith Alpha 24/105 stent graft. provides a rapid means to achieve haemodynamic stability and is The procedure was successful. The patient was later offered defini- associated with low morbidity and mortality. Despite the advan- tive procedure, which he refused. Follow-up OGD after 4 weeks tages of TEVAR the current standard of care is still open surgery revealed a gastric ulcer, with no abnormal cells on histology, for which involves replacement of the diseased segment of aorta and which he was placed on high dose proton-pump inhibitor. oesophagus and excision of the fistula. The mortality of open He re-presented with hypovolaemic shock and massive repair is usually high and hence a technique which is definitive haematemesis four months later. A diagnosis of AEF was again with a low morbidity and mortality is still being sought after. confirmed on CTA (Fig. 2), just proximal to the previous aortic stent graft. He again had emergency percutaneous TEVAR cov- ering the descending aorta from the level just below the left CASE REPORT subclavian artery to just proximal to the celiac artery. Again, he The purpose of this case report is to describe the clinical course of was offered definitive surgery, but he was still not keen to have a 57-year-old male who presented with acute onset of massive this at that time. Received: December 20, 2017. Accepted: January 29, 2018 Published by Oxford University Press and JSCR Publishing Ltd. All rights reserved. © The Author(s) 2018. This is an Open Access article distributed under the terms of the Creative Commons Attribution License (http://creativecommons.org/licenses/by/4.0/), which permits unrestricted re-use, distribution, and reproduction in any medium, provided the original work is properly cited. Downloaded from https://academic.oup.com/jscr/article-abstract/2018/2/rjy014/4859680 by Ed 'DeepDyve' Gillespie user on 16 March 2018 2 T.-A.T. Russell et al. Figure 1: CT aortogram showing the extravasation of contrast into the stomach. Figure 3: Endoscopic view of aortic stent graft in base of gastric ulcer. Majority (54%) of AEF are due to an aortic aneurysm; foreign body and advanced cancer respectively are the second and third most common causes. In this case the pathology was a gastric ulcer. There are case reports of patients who had AEF early after Sweet’s oesophagectomy [5] but there is no mention Figure 2: CT aortogram showing the extravasation of contrast from the aorta in the literature of such a late presentation, 15 years later post just proximal to the stent graft. oesophagectomy. Theuse of endo-vascularstrategiestotreat a fistula was first report by Chuter et al. [6], who used a stent graft to exclude an Two months later, repeat OGD revealed a persistent gastric aorto-bronchial fistula . Even though there are no guidelines to ulcer with a visible segment of aortic stent graft in its base address the useofTEVAR in themanagement of AEFs its use in (Fig. 3). He, then accepted the option of definitive repair. He other aortic pathologies such as aortic dissections and aneurysms, was immediately transferred to a cardio-thoracic tertiary centre and the benefits of low morbidity and mortality associated with where he underwent a thoracotomy, repair of aorto-gastric fis- these procedures favour its use when available to rapidly salvage tula with primary stomach repair and thoracic and abdominal a patient exsanguinating due to an aorto-oesophageal fistula [5]. aorta replacement with a Dacron graft using left heart bypass. The benefits of TEVAR as a first-choice procedure in these cases He made a good recovery and had no complications. His aortic are; the avoidance of a thoracotomy, aortic cross-clamp and stent-graft culture grew Candida albicans and vancomycin- cardio-pulmonary bypass and their associated complications, sensitive and vancomycin-resistant enterococcus. especially in a haemodynamically compromised patient. TEVAR however has its known complications such as: embolic DISCUSSION events example stroke; paraplegia and visceral ischaemia. Its Even though the first case of a successful repair of an AEF was overall safety profile is nevertheless quite impressive with mortal- performed by Jones [1], the mortality remains high and some ity of 1.9–2.1% compared with 11% mortality with open surgery for series quote as high a mortality as 81% [2]. The principle behind descending thoracic aorta aneurysms [7, 8]. Due to its excellent the management is rapid control of bleeding, reconstruction of results TEVAR is safe, particularly in patients who have high surgi- the aorta and oesophagus and prevention of mediastinal con- cal risks with multiple comorbidities. It is therefore by extrapola- tamination. This operation is predominantly done through a tion a good procedure for extremist patients with AEF. thoracotomy with the use of cardio-pulmonary bypass. There is however on-going debate about the use TEVAR in a Rapid diagnosis is essential as there is typically a narrow contaminated field, and of the risk of mediastinitis and the mor- window before exsanguination occurs. Conservative manage- bidity and mortality associated with an infected stent graft post ment of watchful waiting is not an option as this is associated aortic stenting for AEF. For these reasons, many case reports sug- with 100% fatality [3]. Clinically these patients present with gest the use of TEVAR being only temporary, as a first-choice life- massive haematemesis, however, Chiari described a triad of saving measure, and that a definitive elective surgical procedure chest pain, fever and haematemesis, of which only few patients is to be done once the patient recovers from the ordeal of the mas- present with this constellation of symptoms [4]. sive bleed. This extrapolation however is just from case reports as Downloaded from https://academic.oup.com/jscr/article-abstract/2018/2/rjy014/4859680 by Ed 'DeepDyve' Gillespie user on 16 March 2018 The role of TEVAR in the management 3 there are no randomized study to prove this. There is also a query 3. Chiari H. Injury of the esophagus with perforation of the aor- of whether these patients should receive lifelong broad-spectrum ta produced by a foreign body. Berl Klin Wochenschr 1914;51: antibiotics [9], there is however no consensus on the duration of 7–9. antibiotic therapy post TEVAR. 4. Hollander JE, Quick G. Aortoesophageal fistula: a comprehen- TheroleofTEVAR in themanagementofaorto-enteric fistulae sive review of the literature. Am J Med 1991;91:279–87. is inconclusive. It is nonetheless an excellent bridging therapy and 5. Ren W, He Y, Wang X, Peng Z. Secondary aorto-esophageal a possible definitive therapy for patients with haematemesis who fistula after esophagectomy treated with endovascular treat- are high risk for surgery or have advanced cancer respectively. ment: a case report. Chu. D, ed. Medicine 2017;96:e6555. 6. Chuter TAM, Ivancev K, Lindblad B, Brunkwall J, Arén C. Endovascular stent-graft exclusion of an aortobronchial fis- tula. J Vasc Intervent Radiol 1996;7:357–9. CONFLICT OF INTEREST STATEMENT 7. Matsumura JS, Cambria RP, Dake MD, Moore RD, Svensson LG, None declared. Synder S, et al. International controlled clinical trial of thoracic endovascular aneurysm repair with the Zenith TX2 endovascu- largraft:1-yearresults. JVascSurg 2008;472:247–57. REFERENCES 8. Andrassy J, Weidenhagen R, Meimarakis G, Rentsch M, Jauch 1. Jones JC. Complication of the surgery of patent ductus arter- KW. Endovascular versus open treatment of degenerative iosus. J Thorac Surg 1947;16:305–13. aneurysms of the descending thoracic aorta: a single center 2. Zhang X, Liu J, Li J, Hu J, Yu F, Li S, et al. Diagnosis and treat- experience. Vascular 2011;19:8–14. ment of 32 cases with aortoesophageal fistula due to esopha- 9. Konstantinos X, Ilias D, Michalis S, Konstantinos F. Aortoenteric geal foreign body. Laryngoscope 2011;121:267–72. fistulae: present day management. Int Surg 2011;96:266–73. Downloaded from https://academic.oup.com/jscr/article-abstract/2018/2/rjy014/4859680 by Ed 'DeepDyve' Gillespie user on 16 March 2018 http://www.deepdyve.com/assets/images/DeepDyve-Logo-lg.png Journal of Surgical Case Reports Oxford University Press

The role of TEVAR in the management of a recurrent aorto-gastric fistula

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Abstract

An aorto-gastric fistula is a catastrophic and rare cause of an upper gastrointestinal bleed. The diagnosis requires a high index of suspicion and expedient management as any delay in each of these component, will be to the detriment of the patient. We report a case of a patient with two episodes of this rare event, with haemodynamic compromise, 15 years after having had a trans-hiatal oesophagectomy for an adenocarcinoma of the oesophagus who presented on both occasions. He had thoracic endo-vascular aortic repair (TEVAR) on both presentations and survived. This case exemplifies the fact that while TEVAR is a good bridging therapy for the management of an aorto-enteric fistula. It however should not be considered as the definitive management for patients who are operable or patients who do not have prohibitive surgical risk. haematemesis and hypovolaemic shock. His medical history was INTRODUCTION remarkable for a trans-hiatal oesophagectomy and gastric pull-up Despite the first case of an aorto-enteric fistula (AEF) being discov- for adenocarcinoma 15 years prior. After initial fluid resuscitated, ered in 1818; its rare incidence results in a low index of clinical oesophago-gastro-duodenoscopy (OGD) was performed which was suspicion. Its usual presentation is that of sudden onset of mas- complicated by cardiac arrest. He was intubated and fluid resusci- sive haematemesis; and this along with it being rare often results tated until haemodynamically stable. Computed tomography aor- in a late or missed diagnosis and hence a high mortality. tography (CTA) demonstrated an aorto-oesophageal fistula (Fig. 1). The definitive role of thoracic endo-vascular aortic repair A rapid decision was made to proceed with a TEVAR limited to (TEVAR) in the management of AEFs is still undefined. It however that segment of aorta using Cook Zenith Alpha 24/105 stent graft. provides a rapid means to achieve haemodynamic stability and is The procedure was successful. The patient was later offered defini- associated with low morbidity and mortality. Despite the advan- tive procedure, which he refused. Follow-up OGD after 4 weeks tages of TEVAR the current standard of care is still open surgery revealed a gastric ulcer, with no abnormal cells on histology, for which involves replacement of the diseased segment of aorta and which he was placed on high dose proton-pump inhibitor. oesophagus and excision of the fistula. The mortality of open He re-presented with hypovolaemic shock and massive repair is usually high and hence a technique which is definitive haematemesis four months later. A diagnosis of AEF was again with a low morbidity and mortality is still being sought after. confirmed on CTA (Fig. 2), just proximal to the previous aortic stent graft. He again had emergency percutaneous TEVAR cov- ering the descending aorta from the level just below the left CASE REPORT subclavian artery to just proximal to the celiac artery. Again, he The purpose of this case report is to describe the clinical course of was offered definitive surgery, but he was still not keen to have a 57-year-old male who presented with acute onset of massive this at that time. Received: December 20, 2017. Accepted: January 29, 2018 Published by Oxford University Press and JSCR Publishing Ltd. All rights reserved. © The Author(s) 2018. This is an Open Access article distributed under the terms of the Creative Commons Attribution License (http://creativecommons.org/licenses/by/4.0/), which permits unrestricted re-use, distribution, and reproduction in any medium, provided the original work is properly cited. Downloaded from https://academic.oup.com/jscr/article-abstract/2018/2/rjy014/4859680 by Ed 'DeepDyve' Gillespie user on 16 March 2018 2 T.-A.T. Russell et al. Figure 1: CT aortogram showing the extravasation of contrast into the stomach. Figure 3: Endoscopic view of aortic stent graft in base of gastric ulcer. Majority (54%) of AEF are due to an aortic aneurysm; foreign body and advanced cancer respectively are the second and third most common causes. In this case the pathology was a gastric ulcer. There are case reports of patients who had AEF early after Sweet’s oesophagectomy [5] but there is no mention Figure 2: CT aortogram showing the extravasation of contrast from the aorta in the literature of such a late presentation, 15 years later post just proximal to the stent graft. oesophagectomy. Theuse of endo-vascularstrategiestotreat a fistula was first report by Chuter et al. [6], who used a stent graft to exclude an Two months later, repeat OGD revealed a persistent gastric aorto-bronchial fistula . Even though there are no guidelines to ulcer with a visible segment of aortic stent graft in its base address the useofTEVAR in themanagement of AEFs its use in (Fig. 3). He, then accepted the option of definitive repair. He other aortic pathologies such as aortic dissections and aneurysms, was immediately transferred to a cardio-thoracic tertiary centre and the benefits of low morbidity and mortality associated with where he underwent a thoracotomy, repair of aorto-gastric fis- these procedures favour its use when available to rapidly salvage tula with primary stomach repair and thoracic and abdominal a patient exsanguinating due to an aorto-oesophageal fistula [5]. aorta replacement with a Dacron graft using left heart bypass. The benefits of TEVAR as a first-choice procedure in these cases He made a good recovery and had no complications. His aortic are; the avoidance of a thoracotomy, aortic cross-clamp and stent-graft culture grew Candida albicans and vancomycin- cardio-pulmonary bypass and their associated complications, sensitive and vancomycin-resistant enterococcus. especially in a haemodynamically compromised patient. TEVAR however has its known complications such as: embolic DISCUSSION events example stroke; paraplegia and visceral ischaemia. Its Even though the first case of a successful repair of an AEF was overall safety profile is nevertheless quite impressive with mortal- performed by Jones [1], the mortality remains high and some ity of 1.9–2.1% compared with 11% mortality with open surgery for series quote as high a mortality as 81% [2]. The principle behind descending thoracic aorta aneurysms [7, 8]. Due to its excellent the management is rapid control of bleeding, reconstruction of results TEVAR is safe, particularly in patients who have high surgi- the aorta and oesophagus and prevention of mediastinal con- cal risks with multiple comorbidities. It is therefore by extrapola- tamination. This operation is predominantly done through a tion a good procedure for extremist patients with AEF. thoracotomy with the use of cardio-pulmonary bypass. There is however on-going debate about the use TEVAR in a Rapid diagnosis is essential as there is typically a narrow contaminated field, and of the risk of mediastinitis and the mor- window before exsanguination occurs. Conservative manage- bidity and mortality associated with an infected stent graft post ment of watchful waiting is not an option as this is associated aortic stenting for AEF. For these reasons, many case reports sug- with 100% fatality [3]. Clinically these patients present with gest the use of TEVAR being only temporary, as a first-choice life- massive haematemesis, however, Chiari described a triad of saving measure, and that a definitive elective surgical procedure chest pain, fever and haematemesis, of which only few patients is to be done once the patient recovers from the ordeal of the mas- present with this constellation of symptoms [4]. sive bleed. This extrapolation however is just from case reports as Downloaded from https://academic.oup.com/jscr/article-abstract/2018/2/rjy014/4859680 by Ed 'DeepDyve' Gillespie user on 16 March 2018 The role of TEVAR in the management 3 there are no randomized study to prove this. There is also a query 3. Chiari H. Injury of the esophagus with perforation of the aor- of whether these patients should receive lifelong broad-spectrum ta produced by a foreign body. Berl Klin Wochenschr 1914;51: antibiotics [9], there is however no consensus on the duration of 7–9. antibiotic therapy post TEVAR. 4. Hollander JE, Quick G. Aortoesophageal fistula: a comprehen- TheroleofTEVAR in themanagementofaorto-enteric fistulae sive review of the literature. Am J Med 1991;91:279–87. is inconclusive. It is nonetheless an excellent bridging therapy and 5. Ren W, He Y, Wang X, Peng Z. Secondary aorto-esophageal a possible definitive therapy for patients with haematemesis who fistula after esophagectomy treated with endovascular treat- are high risk for surgery or have advanced cancer respectively. ment: a case report. Chu. D, ed. Medicine 2017;96:e6555. 6. Chuter TAM, Ivancev K, Lindblad B, Brunkwall J, Arén C. Endovascular stent-graft exclusion of an aortobronchial fis- tula. J Vasc Intervent Radiol 1996;7:357–9. CONFLICT OF INTEREST STATEMENT 7. Matsumura JS, Cambria RP, Dake MD, Moore RD, Svensson LG, None declared. Synder S, et al. International controlled clinical trial of thoracic endovascular aneurysm repair with the Zenith TX2 endovascu- largraft:1-yearresults. JVascSurg 2008;472:247–57. REFERENCES 8. Andrassy J, Weidenhagen R, Meimarakis G, Rentsch M, Jauch 1. Jones JC. Complication of the surgery of patent ductus arter- KW. Endovascular versus open treatment of degenerative iosus. J Thorac Surg 1947;16:305–13. aneurysms of the descending thoracic aorta: a single center 2. Zhang X, Liu J, Li J, Hu J, Yu F, Li S, et al. Diagnosis and treat- experience. Vascular 2011;19:8–14. ment of 32 cases with aortoesophageal fistula due to esopha- 9. Konstantinos X, Ilias D, Michalis S, Konstantinos F. Aortoenteric geal foreign body. Laryngoscope 2011;121:267–72. fistulae: present day management. Int Surg 2011;96:266–73. Downloaded from https://academic.oup.com/jscr/article-abstract/2018/2/rjy014/4859680 by Ed 'DeepDyve' Gillespie user on 16 March 2018

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Journal of Surgical Case ReportsOxford University Press

Published: Feb 1, 2018

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