Combined Endovascular and Surgical Treatment of

Case Reports

Combined Endovascular and Surgical Treatment of Primary Aortoesophageal Fistula

Enrico Maria Marone, MD Giovanni Coppi, MD Andrea Kahlberg, MD Yamume Tshomba, MD Roberto Chiesa, MD

Primary aortoesophageal fistula is a rare cause of massive upper gastrointestinal bleeding. Conservative treatment of aortoesophageal fistula results in a 60% in-hospital mortality rate with no late survival, and conventional surgical treatment has a reported in-hospital mortality rate that approaches 40%. Thoracic endovascular aortic repair is an innovative and less invasive technique for the treatment of aortoesophageal fistula. It enables the rapid control of aortic bleeding and prevents fatal early exsanguination. However, the technique does not repair the esophagus, and there remains a substantial risk of mediastinitis and infection of the stent-graft. Herein, we report the cases of 2 patients in whom we used a combined treatment: thoracic endovascular aortic repair and delayed surgical repair of the esophagus. The esophageal repair involved direct suture of the esophageal wall and reinforcement with an intercostal muscle flap. Early follow-up evaluations suggest that our treatment of both patients was successful. We discuss the advantages and limitations of our technical choices and briefly review the pertinent medical literature. (Tex Heart Inst J 2010;37(6):722-4)

P Key words: Aorta, thoracic/surgery; aortic diseases/ diagnosis/etiology/surgery; esophageal fistula/diagnosis/etiology/therapy; stents; surgical flaps; treatment outcome From: Department of Vascular Surgery, VitaSalute University, Scientific Institute H. San Raffaele, 20132 Milan, Italy Address for reprints: Giovanni Coppi, MD, Department of Vascular Surgery, Vita-Salute University, Scientific Institute H. San Raffaele, via Olgettina 60, 20132 Milan, Italy E-mail: [email protected] © 2010 by the Texas Heart ® Institute, Houston

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rimary aortoesophageal fistula (AEF) is a rare cause of massive upper gastrointestinal bleeding. The chief causes of AEF are erosion of the esophageal wall by a thoracic aortic aneurysm (in up to 54% of cases), and the ingestion of foreign bodies (in 19% of cases).1 Conservative treatment of AEF results in a 60% inhospital mortality rate with no late survival, and conventional surgical treatment has a reported in-hospital mortality rate of nearly 40%.2 Thoracic endovascular aortic repair (TEVAR) has recently gained recognition as a possible technique for the emergent treatment of AEF, despite a considerable risk of infection.3 Herein, we describe the cases of 2 patients who emergently presented with primary AEF. We treated both with use of a combined approach: endovascular exclusion of the aortic ruptures, followed by surgery to correct the esophageal lesions.

Case Reports Two patients presented at our institution with hypotension associated with an episode of hematemesis. Patient 1 was a 57-year-old man without a pertinent medical history. Emergent computed tomographic angiography (CTA) showed primary AEF that was caused by the rupture of a penetrating aortic ulcer into the esophagus. Patient 2 was a 66-year-old man with a history of hypertension, coronary artery disease, cerebrovascular disease, and chronic obstructive pulmonary disease. Emergent CTA led to the diagnosis of primary AEF that was due to a ruptured distal thoracic aortic aneurysm. Both patients were conscious upon arrival. Each underwent fluid replacement and transfusion of concentrated red blood cells (patient 1, 300 cc; and patient 2, 800 cc). Emergent TEVAR was performed to rapidly control bleeding and prevent fatal exsanguination. In patient 1, a 28 × 110-mm Relay® stent-graft (Bolton Medical, Inc.; Sunrise, Fla) was deployed; in patient 2, a 32 × 150-mm Relay stent-graft was used. The endovascular procedures were performed according to our protocol.4 Final angiography showed exclusion of the aortic lesions, with no evident extravasation of contrast media. The patients’ postprocedural courses were uneventful. Surgical repair of the esophageal lesions was scheduled after 4 and 5 days, respectively. Through a right thoracotomy in the 5th intercostal space, the intercostal muscles were mobilized as a pedicled

Combined Treatment of Primary AEF

Volume 37, Number 6, 2010

flap, with use of the posterior intercostal vascular bundle. The distal part of the esophagus was exposed and mobilized with special care, due to the tight adhesion of the esophageal wall to the aorta. In patient 1, the stent-graft was visible through the breached aortic wall (Fig. 1). After tissue débridement, the esophageal lesion was repaired with use of a double-layered absorbable suture. The intercostal muscle flap was then placed in the aorto­ esophageal space and was sutured to the esophageal wall (Fig. 2). Copious washing of the aortic bed was performed to prevent contamination and mediastinitis. Two thoracic drains were placed close to the esophagus—1 cephalad and 1 close to the lower tract. In patient 2, precautionary jejunostomy was performed, because the esophageal lesion was more extensive than expected. Both patients were placed on parenteral nutrition and intravenous antibiotic therapy with fluconazole, piperacillin/tazobactam, metronidazole, and levofloxacin. Subsequent CTAs and esophagograms showed complete exclusion of the aortic lesions, repair of the esophageal breaches, and no signs of infection. The gradual reintroduction of oral food intake started on the 20th and 31st postoperative days, respectively. The patients were discharged from the hospital after 27 and 39 days, respectively. Patient 1 was doing well 24 months postoperatively, with no fever or increase of inflammatory markers. His antibiotic therapy was stopped after 6 months. Patient 2 intermittently continued broad-spectrum antibiotic therapy for 11 out of 18 postoperative months, due to mild leukocytosis and recurrent fever. At 6 months, CTA revealed no sign of infection, mediastinal fluid collections, or periprosthetic air bubbles (Fig. 3).

Fig. 2 Patient 2. Photograph shows complete reconstruction of the esophageal wall with the interposition of a pedicled intercostal muscle flap.

A

B

Fig. 1 Patient 1. Intraoperative photograph shows an aortoesophageal fistula. The previously inserted stent-graft is visible through the breached aortic wall after esophageal mobilization (arrow).

Texas Heart Institute Journal

Fig. 3 Patient 2. Comparative computed tomographic angiography A) preoperatively and B) upon 6-month follow-up. The aortic lesions have been completely excluded, with no signs of endoleak, mediastinal infection, or stent-graft contamination.

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Discussion Several options for AEF repair have been described in the surgical literature. Albeit different in terms of technique, all rely on the same concept: expeditious management of the aortic rupture (with extra-anatomic bypass or in situ reconstruction using antibiotic-soaked grafts or homografts), in association with immediate esophageal repair (direct suture or esophageal resection with esophagogastroplasty or coloplasty) or delayed repair (esophageal diversion with postponed reconstruction).3 However, these procedures result in high intraoperative and perioperative mortality rates. In both of our patients, TEVAR enabled rapid control of bleeding through a less invasive approach than that of traditional surgery. However, using TEVAR alone would have left the esophageal defects untreated, increasing the risks of mediastinitis and death.3 Accordingly, we decided to treat the esophageal lesions when the patients were in stable condition, even though no signs of mediastinitis were present. We believed that the best treatment was direct suture of the esophageal breaches in association with intercostal muscle-flap coverage. This was feasible because the defects were small enough for direct repair, and the surrounding esophageal walls were neither extensively necrotic nor grossly contaminated. The decision to use the intercostal muscle flaps to strengthen and protect the sutured esophageal lesions arose from the recognition that striated muscle is better suited to close contaminated wounds than is omentum or pericardial fat.5 Moreover, using intercostal muscle avoids decubitus of the aortic stent-graft on the esophageal wall, reducing the risk of recurrent AEF. We found that TEVAR is a good emergency alternative for preventing early fatal exsanguination in patients with AEF. However, TEVAR can rarely be used as a stand-alone procedure.6 When the patient’s condition has stabilized, stent-graft removal can be considered and the esophageal lesion given special surgical attention. A nationwide, multicenter survey that we conducted in Italy yielded information on 25 aortoesophageal and aortobronchial fistulae that were treated with TEVAR.7 The data showed that patients who undergo combined treatment (TEVAR + esophageal/bronchial repair) may have lower graft-infection rates and better survival rates than patients who undergo TEVAR alone. We believe that long-term prognosis is strictly affected by mediastinal infection and graft contamination. In order to minimize these events in our patients, we always administer broad-spectrum antibiotic therapy: the specific medications are decided upon in collaboration with infectious-­disease specialists and guided by available culture results. Upon discharge from the hospital, patients take oral antibiotics for at least 6 months, and the therapy ends in the absence of clinical, biochemical, and 724

Combined Treatment of Primary AEF

computed tomographic angiographic signs of infection. All patients must undergo thorough clinical and imaging follow-up, with strictly monitored blood tests and computed tomographic scans at 1, 3, 6, and 12 months, and yearly thereafter. In accordance with the early follow-up evaluations, our 2 patients were treated successfully. Long-term data will be necessary to establish the durability of the proposed combined treatment of AEF.

References 1. Hollander JE, Quick G. Aortoesophageal fistula: a comprehensive review of the literature. Am J Med 1991;91(3):279-87. 2. Kieffer E, Chiche L, Gomes D. Aortoesophageal fistula: value of in situ aortic allograft replacement. Ann Surg 2003;238(2): 283-90. 3. Prokakis C, Koletsis E, Apostolakis E, Dedeilias P, Dougenis D. Aortoesophageal fistulas due to thoracic aorta aneurysm: surgical versus endovascular repair. Is there a role for combined aortic management? Med Sci Monit 2008;14(4):RA48-54. 4. Marone EM, Baccari P, Brioschi C, Tshomba Y, Staudacher C, Chiesa R. Surgical and endovascular treatment of secondary aortoesophageal fistula. J Thorac Cardiovasc Surg 2006; 131(6):1409-10. 5. Bhalla M, Wain JC, Shepard JA, McLoud TC. Surgical flaps in the chest: anatomic considerations, applications, and radiologic appearance. Radiology 1994;192(3):825-30. 6. Topel I, Stehr A, Steinbauer MG, Piso P, Schlitt HJ, Kasprzak PM. Surgical strategy in aortoesophageal fistulae: endovascular stentgrafts and in situ repair of the aorta with cryopreserved homografts. Ann Surg 2007;246(5):853-9. 7. Chiesa R, Melissano G, Marone EM, Kahlberg A, MarroccoTrischitta MM, Tshomba Y. Endovascular treatment of aortoesophageal and aortobronchial fistulae. J Vasc Surg 2010;51 (5):1195-202.

Volume 37, Number 6, 2010