Aorto-gastric ﬁstula (AGF) is an uncommon and life threatening complication of oesophagectomy. Usually occurring in the immediate weeks following the procedure at anastomosis lines, this case describes a rare development of an AGF 15 years after an oesophagectomy due to the presence of a benign ulcer. Initially successful endovascular stenting of the thoracic aor- ta was followed by re-bleed, further stenting but eventually open surgery was required. haematemesis, melaena and 4-day history of back pain. Fifteen INTRODUCTION years previously he was treated for a poorly differentiated adeno- A ﬁstula between the thoracic aorta and a pulled-up stomach carcinoma (pT3 pN1 MX) close to the gastro-oesophageal junction (neo-oesophagus) after oesophagectomy is an uncommon and with chemotherapy and trans-hiatal oesophagectomy. He received often fatal complication causing massive haemorrhage. no radiotherapy. Apart from being a life-long smoker he had no Most previous cases documented the development of ﬁstulae other positive social risk factors or medical history. He underwent at suture or anastomosis lines penetrating or perforating into the immediate oesophago-gastro-duodenoscopy (OGD). This showed aorta [1–3]. This is usually seen within weeks of the procedure fresh and clotted blood in the gastric remnant but no bleeding and likely as a complication of acute infection or leak [1, 3, 4]. source was identiﬁed. He had a pulseless arrest during the OGD We describe the management of a patient with AGF that but underwent successful cardiopulmonary resuscitation. Following developed due to benign ulceration 10 years after oesophagect- this a CT angiogram was performed. This demonstrated a ﬁstula omy for cancer. Initially we carried out thoracic aortic endovas- between the neo-oesphagus and the thoracic aorta (Fig. 1). cular stenting, then due to a re-bleed repeated stenting was Following discussion between an upper gastro-intestinal surgeon, followed by open surgery. a vascular surgeon and an interventional radiologist the patient (The case and images have been consented for use in publi- was taken to the operating theatre. Under general anaesthesia an cation by the patient.) initial thoracic aortogram was performed from the right groin to conﬁrm the presence and location of the AGF (Fig. 2). Following this, a covered thoracic stent graft (Cook Medical ZTA-24-105/ CASE REPORT diameter of 24 mm and length of 105 mm), was inserted via the A 57-year-old male presented haemodynamically unstable with a femoral artery under image guidance. The stent graft, usually blood pressure of 85/50 mmHg and a heart rate of 136 bpm with used in aneurysmal repair, was placed in the thoracic aorta across Received: December 11, 2017. Accepted: February 1, 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 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 firstname.lastname@example.org Downloaded from https://academic.oup.com/jscr/article-abstract/2018/2/rjy019/4877012 by Ed 'DeepDyve' Gillespie user on 16 March 2018 2 H.S. Chotai et al. Figure 1: CT angiogram showing aorta-enteric ﬁstula demonstrated by arrow (axial and sagittal view, respectively). Figure 3: Angiogram post primary aortic stenting. Figure 4: OGD following primary stent insertion showing enteric ulcer. Figure 2: Angiogram prior to primary aortic stenting; leak via ﬁstula demon- strated by arrow. stent was relined with further stents extending both proximally and distally beyond the previous stent, with care taken not to occlude the celiac trunk and arteries below this (Cook Medical the ﬁstula and occluded it. Subsequent angiogram showed no leak (Fig. 3). The patient became haemodynamically stable and had an ZTA-26-105 and Cook Medical ZTA-28-109). Again, the patient had a quick recovery. However, repeat endoscopy showed that uneventful recovery. A week later, he underwent a further OGD. This showed a the aortic stent was visible through a defect in the wall of the neo-oesophagus (Fig. 6). benign ulcer (Fig. 4), which was later conﬁrmed histologically. Helicobacter pyloriwas negative. He was discharged home 8 At this point the patient was referred to the Liverpool days post stent insertion with a further 2-week course of co- Thoracic Aneurysm Service where he underwent a left thora- amoxiclav (Amoxicillin/clavulanic acid) and 1 week of ﬂucon- cotomy. The thoracic aorta was clamped proximal and distal to the aortic stents and a left heart bypass was established for dis- azole, as recommended by a consultant microbiologist, and life-long omeprazole. He remained well for the next months. tal perfusion. The aorta was opened, the stents excised and However, 4 and a half months later he again became replaced with a 22 mm Dacron tube graft. After mobilizing the haemodynamically unstable with a further episode of haema- neo-oesophagus from the aorta, the edges of the defect, which appeared clean and healthy, was repaired directly. A week later temesis. A further CT angiogram showed contrast entering the neo-oesophagus from the thoracic aorta (Fig. 5). The patient contrast swallow showed satisfactory repair of the neo- oesophagus with no anastomotic leak. was again taken to the emergency theatre and the previous Downloaded from https://academic.oup.com/jscr/article-abstract/2018/2/rjy019/4877012 by Ed 'DeepDyve' Gillespie user on 16 March 2018 Successful management of an aorto-gastric ﬁstula occurring 15 years after oesophagectomy 3 . Our patient had no sign of malignancy for over a decade. Radiotherapy, though not a factor in this case, can also contribute to the breakdown of soft tissue planes in the irradiated ﬁeld . In the absence of other predisposing factors and in the pres- ence of a previous truncal vagotomy that was part of the ori- ginal oesophagectomy, smoking may have contributed to the development of a benign ulcer in the pulled-up gastric rem- nant, adhering to and then eroding into the aorta. The treatment for AGF is difﬁcult. Conservative treatment commonly fails with inevitable death . In some patients, with recurrent malignancy, palliation may have a role. Formal open repair through thoracotomy has been considered deﬁni- tive treatment but carries a high risk of mortality even in spe- cialized institutions in the acutely unwell patient. Our patient was treated initially by endovascular means using a covered thoracic aortic stent graft, normally used in aneurysmal repair. Infection and/or mechanical pressure from Figure 5: Second admission: CT angiogram showing aorta-enteric ﬁstula demon- this resulted in the failure of this treatment and open repair strated by arrow (axial and sagittal view, respectively). became necessary. However, the initial stenting meant that this major operation could be carried out in a stable patient who was systemically well. This is likely to have contributed to a satisfactory outcome. Ongoing concern remains regarding recurrent graft infection. Life-long surveillance, prolonged anti- biotic treatment and abolishing likely septic sources are neces- sary to reduce this risk. Our experience points towards the temporizing role of aortic stent-grafts in patients with AVG but, due to the lack of trials, treatment for these difﬁcult patients has to be tai- lored individually. CONFLICT OF INTEREST STATEMENT None declared. REFERENCES 1. Brookes V, Stafford J. Peptic ulceration and perforation of the stomach after oesophagectomy. Thorax 1952;7:167–9. Figure 6: Second admission: OGD post second stent insertion demonstrating 2. Strong S, Higgs S, Streets C, Titcomb D, Barham P, Blazeby J, visibility of aortic stent via defect in wall of neo-oesphagus. et al. Aorto-conduit ﬁstula developing four years after eso- phagectomy. J Surg Case Rep 2012;2012:8–8. The removed aortic stents grew candida albicans sensitive 3. Uittenbogaart M, Sosef M, van Bastelaar J. Sentinel bleeding to Fluconazole. The patient continues on antibiotics, including as a sign of gastroaortic ﬁstula formation after oesophageal antifungals. He remains well and is due to undergo treatment surgery. Case Rep Surg 2014;2014:1–4. for his extensively carious teeth. 4. Sato O, Miyata T, Matsubara T, Shigematsu H, Yasuhara H, Ishimaru S. Successful surgical treatment of aortogastric ﬁs- DISCUSSION tula after an esophagectomy and subsequent endovascular AGFisanuncommonand oftenlethalcomplicationofprevious graft placement: report of a case. Surg Today 1999;29:431–4. oesophagectomy. It may present within weeks after surgery  5. Hollander J, Quick G. Aortoesophageal ﬁstula: a comprehen- due to anastomotic problems or later, due to recurrent malignancy sive review of the literature. Am J Med 1991;91:279–87. Downloaded from https://academic.oup.com/jscr/article-abstract/2018/2/rjy019/4877012 by Ed 'DeepDyve' Gillespie user on 16 March 2018
Journal of Surgical Case Reports – Oxford University Press
Published: Feb 1, 2018
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