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Ann Thorac Surg 2006;82:1117-1119
© 2006 The Society of Thoracic Surgeons


Case report

Aortoesophageal Fistula Successfully Treated by Endovascular Stent-Graft

Roderik Metz, MDa, A. Nikola Kimmings, MD, PhDa, Hence J.M. Verhagen, MD, PhDb, Inne H. M. Borel Rinkes, MD, PhDa, Richard van Hillegersberg, MD, PhDa,*

a Department of Surgery, University Medical Center Utrecht, Utrecht, the Netherlands
b Department of Vascular Surgery, University Medical Center Utrecht, Utrecht, the Netherlands

Accepted for publication January 24, 2006.

* Address correspondence to Dr van Hillegersberg, University Medical Center Utrecht, Department of Surgery (G04.228), Heidelberglaan 100, Utrecht, 3584 CX the Netherlands (Email: r.vanhillegersberg{at}azu.nl).


    Abstract
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The case of a patient with an aortoesophageal fistula is presented, which was caused by ingestion of a chicken bone and was treated by endovascular stent-graft placement and esophagectomy with early reconstruction. The diagnostic and therapeutic options and challenges encountered in treating an aortoesophageal fistula are discussed.


    Introduction
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An aortoesophageal fistula (AEF) is a rare complication of foreign body ingestion [1, 2]. Most perforations are the result of an aneurysm of the aorta [3], although esophageal malignancy, ulcers, and iatrogenic interventions have also been described. Classic symptoms of an AEF are mid-thoracic pain, a sentinel arterial hemorrhage, followed by exsanguination after a symptom-free period (Chiari's triad). Despite different possibilities for therapeutic interventions for AEF [1–4], this condition is generally fatal.

A previously healthy 31-year-old man presented at our emergency department with severe hematemesis. His complaints had commenced 8 days earlier after swallowing a chicken bone. The complaints consisted of mild thoracic pain, dysphagia, general malaise, and coughing. These subtle clinical symptoms were initially diagnosed as pneumonia for which he received antibiotics from his general practitioner. On admission the patient was hemodynamically stable and an esophago-gastroscopy was done showing a tear in the distal esophagus presumed to be a Mallory-Weiss lesion, which was treated by adrenaline injection.

The next day the patient became hemodynamically unstable and rapidly developed severe swelling of the neck with breathing difficulties. An emergency tracheotomy was performed and computed tomographic angiography revealed a mass with air in the mediastinum and contrast leakage from the aorta 2 cm distal to the left subclavian artery (Fig 1). The diagnosis of AEF was apparent, and an endovascular thoracic aortic stent-graft was immediately placed (TAG 28-10 [W. L. Gore & Assoc, Flagstaff, AZ]) with intentional overstenting of the left subclavian artery. Intraoperative angiography showed complete exclusion of the fistula and no endoleaks.


Figure 1
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Fig 1. Computed tomographic scan showing contrast leakage from the thoracic aorta (arrow).

 
After stent placement, the patient became hemodynamically stable but acute respiratory insufficiency reoccurred the following morning. Emergency surgical decompression of the neck was required. Therefore the neck was explored by a longitudinal right-sided incision along the anterior border of the sternocleidomastoid muscle and a large amount of blood and clot was evacuated. After this, oxygen saturation improved, and the patient was again transferred to the operating room for further exploration of the neck. A second endoscopy in the operating room showed an oval-shaped deep defect in the mid-esophagus. An esophagectomy had to be performed because such a large defect combined with local necrosis was considered unsuitable for primary repair. The esophagus was transected through the neck incision and deviated externally after positioning of a stripper. During manipulation of the mediastinum, massive bleeding occurred for which a second endograft was placed inside the first one (TAG 28-15 [W. L. Gore & Assoc, Flagstaff, AZ]) (Fig 2). Fortunately, an additional 2 cm of proximal seal could be obtained due to the left common carotid artery originating from the brachiocephalic trunk (Bovine variant). The patient was stabilized and a transhiatal esophagectomy was to be finished 2 days later.


Figure 2
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Fig 2. Computed tomographic scan showing the stent-grafts in the aorta.

 
In the following days the patient improved markedly. Control computed tomography illustrated evident reduction of mediastinal infiltration. The decision was made to proceed to early reconstruction, thereby circumventing the inevitable obliteration of the original esophageal bed. A gastric tube was constructed and pulled through the original pre-vertebral route. This was initially inhibited due to edema in the mediastinum, but placing the gastric tube into a laparoscopy camera bag enabled smoother positioning without harming the vascularization.

Subsequently the patient had an uneventful postoperative course. At present, 8 months postoperatively, the patient is doing well.


    Comment
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This case shows several critical points in clinical decision making as follows in more detail.

Bleeding Complication of AEF
The widely accepted treatment for AEF is thoracotomy with aortic graft interposition, followed by reconstruction of the esophagus. This procedure shows extremely high mortality and morbidity rates due to the poor condition of the patients at the time of surgery [3, 4]. Furthermore, there is a significant chance of paraplegia. Endovascular stent placement for AEF has been described with good results [1, 5]. It stops the bleeding and provides the opportunity to stabilize and optimize the patient's condition before further treatment. It is debated however, whether endovascular stenting can be seen as definitive treatment or only as a bridge to further aortic surgery, because graft material is placed in an infected area without any adjunctive procedures like omental plasty [5, 6]. Although short-term results of endovascular treatment of AEF are promising, there is no knowledge about the long-term outcome. In this case, our patient was prescribed oral antibiotics with close surveillance for signs of infection. He stopped taking his prescribed medication, but there are no signs of infection 8 months after stenting. Still, as the patient is such a young age, stenting is considered a temporarily life-saving solution, and the patient will undergo open aortic surgery in the near future.

Management of Esophageal Perforation
With respect to esophageal perforation, primary closure and drainage is advocated if the perforation is diagnosed within 24 hours [3, 7]. In this patient the perforation had existed for more than a week, resulting in severe mediastinitis and esophageal necrosis. Therefore primary repair was deemed unsuitable and the risk of residual leakage of the repair with continuing mediastinitis was unacceptably high. Esophageal resection and drainage of the mediastinum were considered the best option. The timing of the esophagectomy depended on whether or not the patient could be stabilized after such severe hemorrhage.

Early secondary construction using a gastric tube was considered optimal because of sufficient improvement in the patient's general condition within a week and subsidence of the infiltration in the mediastinal space. The gastric tube construction is generally delayed for more than 6 weeks until the patient has fully recovered, but then positioning of the tube through the original esophageal bed is no longer possible.

This case of a young patient, who survived an AEF after ingestion of a chicken bone, presents the considerations encountered in diagnostics, and especially the choice and timing of therapeutic options. The excellent clinical results after short-term follow-up seem to indicate that these choices were prudent. Nonetheless, we wish to emphasize that managing each case of an aortoesophageal perforation demands an individualized approach and an experienced surgical team.


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  1. Assink J, Vierhout BP, Snellen JP, et al. Emergency endovascular repair of an aortoesophageal fistula caused by a foreign body J Endovasc Ther 2005;12:129-133.[Medline]
  2. Sica GS, Djapardy V, Westaby S, Maynard ND. Diagnosis and management of aortoesophageal fistula caused by a foreign body Ann Thorac Surg 2004;77:2217-2218.[Abstract/Free Full Text]
  3. da Silva ES, Tozzi FL, Otochi JP, et al. Aortoesophageal fistula caused by aneurysm of the thoracic aortasuccessful surgical treatment, case report and literature review. J Vasc Surg 1999;30:1150-1157.[Medline]
  4. Flores J, Shiiya N, Kunihara T, Yoshimoto K, Yasuda K. Aortoesophageal fistulaalternatives of treatment case report and literature review. Ann Thorac Cardiovasc Surg 2004;10(4):241-246.[Medline]
  5. Gonzales-Fajardo JA, Gutierrez V, Martin-Pedrosa M, Del Rio L, Carrera S, Vaquero C. Endovascular repair in the presence of aortic infection Ann Vasc Surg 2005;19:94-98.[Medline]
  6. Burks JA, Faries PL, Gravereaux EC, Hollier LH, Marin ML. Endovascular repair of bleeding aortoenteric fistulasa 5-year experience. J Vasc Surg 2001;34:1055-1059.[Medline]
  7. Brinster CJ, Singal S, Lee L, Marshall MB, Kaiser LR, Kucharczuk JC. Evolving options in the management of esophageal perforationreview. Ann Thorac Surg 2004;77:1475-1483.[Abstract/Free Full Text]




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