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Aortoesophageal fistula secondary to thoracic stent-graft fracture: a case report
(Portuguese PDF version)

Felipe Nasser,¹ Adnan Neser,² Jose Carlos Ingrund,³ Charles Edouard Zurstrassen,⁴ Flavio de Macedo Cavaleiro Ribeiro,⁴ Ricardo Vagner Moreira^,4 Elias Arcenio Neto^,4 Marcelo Calil Burihan^,5 Orlando Costa Barros⁶

1. Assistant, Department of Vascular Surgery, Hospital Santa Marcelina (HSM), São Paulo, SP, Brazil. Head, Department of Vascular and Interventionist Radiology, HSM, São Paulo, SP, Brazil.
2. Head, Vascular Surgery Service, HSM, São Paulo, SP, Brazil.
3. Supervisor, Vascular Surgery Service, HSM, São Paulo, SP, Brazil.
4. Intern vascular surgeons, Department of Vascular and Interventionist Radiology, HSM, São Paulo, SP, Brazil.
5. Assistant physician, Vascular Surgery Service, HSM, São Paulo, SP, Brazil.
6. Assistant physician, Vascular Surgery Service, HSM, São Paulo, SP, Brazil. Head, Department of Vascular Ultrasonography, HSM, São Paulo, SP, Brazil.

Correspondence:
Am J Med. 1991;91:297-87.
Felipe Nasser
Rua Santa Marcelina, 177
CEP 08270-070 - São Paulo, SP, Brazil
Tel.: 55 11 8224.0505
E-mail: nasser.felipe@gmail.com

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ABSTRACT

The endovascular treatment of thoracic aortic aneurysms has undergone considerable development over the past years. However, late complications of this new therapeutic modality have only recently been observed and analyzed. Aortoesophageal fistulas are rare complications of the endovascular treatment of aortic aneurysms, and there are few reports in the literature. We report a case of a patient with thoracic aortic aneurysm treated 4 years ago and with complications due to stent-graft fracture. After a new endovascular intervention, there was extrusion of the old graft through the esophagus with fistula formation. Despite the treatment, the patient died 9 days later due to massive digestive hemorrhage. Technical aspects and review of the literature are discussed.

Keywords: Aneurysm, fistula, graft implantation.

J Vasc Bras. 2006;5(4):317-20

Article submitted July 31, 2006, accepted December 4, 2006.

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INTRODUCTION

The endovascular therapy was introduced in surgical practice as a new therapeutic modality in the treatment of aortic lesions, such as aneurysms,^1-5 dissections,^6,7 penetrating aortic ulcers,^8,9 traumatic aortic rupture,^10,11 among others. However, despite the high technical success rates and improvement in morbidity and mortality rates, its long-term results remain uncertain.¹² To date, most adverse events are associated with the prosthetic material and are especially manifested through endoleaks.¹² Complications such as arterial lesion, upper limb ischemia, distal embolization, stroke and paraplegia have also been reported.¹³

Aortoesophageal and aortobronchic fistulas (AEF) are rare complications of the surgical and endovascular treatment of aortic diseases, but are almost always fatal in the absence of proper treatment, besides presenting considerable morbidity and mortality.^14-18

This study aims at describing the occurrence of extrusion into the esophagus of a previously implanted stent-graft in a patient with thoracic aneurysm of the descending aorta.

CASE REPORT

A 48-year-old male patient, hypertensive, smoker, with history of stroke 2 years ago. Four years ago he was submitted to an endovascular treatment of thoracic aortic aneurysm (TAA) in another service, but the authors are unaware of the material used.

The patient was admitted to our service with history of unproductive cough, associated with dyspnea to medium efforts, both started approximately 3 months ago. He was presenting progressive worsening of clinical status, reporting severe dyspnea and orthopnea during hospitalization, associated with disfagia for solid foods.

On physical examination, skin pallor or cyanosis were not present, and there were auscultatory crackles in pulmonary bases with normal cardiac auscultation. There were no significant changes in vital signs.

Thoracic computed tomography (CT) at admittance showed descending aortic aneurysm and fractured stent-graft in its mid-inferior segment contained inside the aneurysmal sac, besides compression of the left mainstem bronchus. An esophagogram was also performed at hospitalization, which showed marked compression of the esophagus caused by stent-graft and aortic aneurysm.

Figure 1 - Esophagogram showing compression of the esophagus by the fractured graft

Due to the imminent risk of aneurysm rupture, a new repair was performed using the endovascular technique, and a Talent graft (Medtronic) was implanted, culminating in the exclusion of the aortic aneurysm and fractured graft, respectively.

Figure 2 - Aortography before the treatment. Image showing the recanalized thoracic aortic aneurysm and the fractured graft inside it

Figure 3 - Control aortography after endovascular treatment

The patient recovered satisfactorily from surgery, with important improvement in symptoms and hospital discharge on the seventh postoperative day. Ten days after discharge, the patient was admitted again with complaint of strong precordial pain, irradiating to the dorsal region, followed by acute sialorrhea and disfagia for liquids. A new tomography revealed maintenance of the aortic aneurysmal dilatation; however, it did not present signs of rupture or leakage. Nevertheless, there were also images of gaseous content situated between the new stent-graft and the aneurysmal sac. An upper digestive endoscopy showed esophageal ulcerated lesion associated with presence of fistulas in the aorta and left mainstem bronchus. Surprisingly, the former fractured graft was also seen, located in the aneurysmal sac with no signs of bleeding. With the aid of endoscopic clamps, the fractured fragments of the stent-graft were carefully removed. After the capture, a nasoenteral tube was inserted for aspiration and food.

Figure 4 - Endoscopic image of the removal of one of the fragments of the fractured aortic graft

Figure 5 - Group of stents removed from the fistula tract from the aorta to the esophagus

Seven days after the event, a new endoscopy was performed. There was partial healing of the esophageal wall and the patient was in full recovery.

Two days later, however, the patient presented a discrete episode of cough with hemoptoic sputum, culminating in massive hematemesis and death 6 h later.

DISCUSSION

Secondary aortoenteric fistulas (AEF) are well known and described entities, occasionally present after prosthetic repair of the aorta. However, for being rarer, the exact incidence of secondary AEF is still unknown. It is believed to be lower than the incidence of secondary aortoduodenal fistulas, estimated between 0.4-4% of thoracic abdominal reconstructions.¹⁹

As to secondary AEF, there are few available cases in the literature, with high mortality rates, not only due to the frequent presence of infection in the graft material, but also due to the high incidence of aortic pseudoaneurysms and massive bleedings. Secondary AEF was also observed after endovascular repair of TAA. The first case was published in 1998, when Dake reported a series of 103 cases of thoracic descending aneurysms treated with endovascular stent-grafts.¹³ A new case was recently published by Hance, in which the author also reports the occurrence of AEF 15 months after the endovascular repair due to acute traumatic dissection of the thoracic aorta.¹⁵ In this case, the repair was successfully achieved through open surgery of the aorta and esophagus. Eggebrecht, in a retrospective analysis of 60 patients treated with endovascular repair of the thoracic aorta, also observed it in three cases.³ The three patients involved died; the first due to massive hematemesis and the other two, who underwent conservative treatment, due to mediastinitis and sepsis.

In terms of physiopathology, it is believed that AEF secondary to endovascular treatment occurs due to the development of pseudoaneurysms, presence of stent-grafts with endoleaks, stent erosion through the aorta into the esophagus, or perforation of the aorta and esophagus through the stent-graft fixation clamps.¹⁴

The present case surprises because of the occurrence of secondary fistulas between the aorta and the esophagus, and due to the occurrence of extrusion of the fractured aortic stent-graft into the esophagus, which could be captured by endoscopic clamps. Unfortunately, the patient's poor clinical conditions did not allow us to perform a definitive treatment of the AEF, which is the open surgical repair.

In conclusion, we support the need of judicious follow-up in patients treated with endovascular technique. This is the only form of obtaining an early diagnosis of these complications, and may promote proper treatment in earlier stages, which are therefore in better conditions.

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