Table of Contents  
CASE REPORT
Year : 2021  |  Volume : 8  |  Issue : 3  |  Page : 253-256

Thoracic endovascular repair for catastrophic aorto-enteric fistula secondary to esophageal stent: Case report and literature review


1 Department of Vascular Surgery, Royal North Shore Hospital, Sydney; Department of Medicine Faculty, Sydney University, Camperdown, NSW, Australia
2 Department of Vascular Surgery, Royal North Shore Hospital; Department of Medicine Faculty, University of New South Wales, Sydney, NSW, Australia
3 Department of Vascular Surgery, Royal North Shore Hospital, Sydney, NSW, Australia

Date of Submission08-Jan-2021
Date of Acceptance19-Jan-2021
Date of Web Publication6-Jul-2021

Correspondence Address:
Animesh A Singla
Department of Vascular Surgery, Royal North Shore Hospital, Camperdown; Department of Medicine Faculty, University of New South Wales, Sydney, NSW
Australia
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Source of Support: None, Conflict of Interest: None


DOI: 10.4103/ijves.ijves_7_21

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  Abstract 


Secondary aorto-enteric fistula (SAEF) following esophageal stenting is a rare but recognized pathology. These are multifactorial in etiology and can lead to life-threatening catastrophic hemorrhage. With the advent of thoracic endovascular covered stent technology, a minimally invasive means of addressing is problem has emerged. We aim to discuss the pathophysiology and underlying principles in managing this difficult problem. Prompt early recognition and multidisciplinary care are critical to success.

Keywords: Aortoenteric fistula, endovascular, thoracic endovascular repair


How to cite this article:
Kotecha K, Singla AA, Nguyen D. Thoracic endovascular repair for catastrophic aorto-enteric fistula secondary to esophageal stent: Case report and literature review. Indian J Vasc Endovasc Surg 2021;8:253-6

How to cite this URL:
Kotecha K, Singla AA, Nguyen D. Thoracic endovascular repair for catastrophic aorto-enteric fistula secondary to esophageal stent: Case report and literature review. Indian J Vasc Endovasc Surg [serial online] 2021 [cited 2021 Jul 25];8:253-6. Available from: https://www.indjvascsurg.org/text.asp?2021/8/3/253/320625




  Introduction Top


Secondary aorto-enteric fistula (SAEF) following esophageal stenting is a rare but recognized pathology. Causes are often multifactorial involving patients with underlying malignancy, prior radiation-induced tissue damage, and poor nutritional state. The placement of a rigid esophageal stent further predisposes to an abnormal communication leading to an aortoenteric fistula. This case report highlights the successful management of a SAEF using thoracic endovascular stent-graft repair. The inherent anatomical pathophysiology and principles in managing such complex cases are discussed.


  Case Report Top


A 64-year-old male was admitted with new-onset hematemesis. He had a past medical history of esophageal squamous cell carcinoma (SCC) which had been treated with curative intent by neoadjuvant chemoradiotherapy. He had bilateral pulmonary emboli but otherwise no disease. He had recent staging of his disease which identified new metastasis with pulmonary and hepatic involvement. As he was deemed a nonsurgical candidate, an esophageal stent was placed for relief of dysphagia symptoms.

On current admission, his white cell count was 6.2, and C-reactive protein was 28. His blood cultures grew Enterococcus faecium and Streptococcus oralis, treated with intravenous vancomycin and amoxicillin-clavulanate. Urgent endoscopy showed a patent stent with granulation tissue and likely ulceration at the proximal end of the stent (25 cm from the incisors). A large amount of clot was seen at this spot and suctioned exposing underlying likely cancer recurrence. The remainder of the esophageal stent was unaffected and no biopsy was taken to due to the risk of erosion/catastrophic hemorrhage [Figure 1].
Figure 1: Endoscopy. (a) Highlighting proximal esophagus with squamous cell carcinoma recurrence. (b) Proximal stent with evidence of recurrence and fresh bleeding. (c) Patent distal esophageal stent

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The patient underwent further investigation with contrast-enhanced computed tomography scanning [Figure 2]. This identified an aorto-enteric fistula between the esophagus and thoracic aorta located at the proximal end of the stent, with an associated aortic pseudoaneurysm. The images were reconstructed using multiplanar reformatting and three-dimensional volumetric rendering to identify proximal and distal landing zones [Figure 3]. His overall disease process carried a median 12-month life expectancy and was thus deemed reasonable for intervention. He was urgently transferred to our tertiary referral center. On admission to intensive care, the patient had a catastrophic bleed and went into hypovolemic shock. He was transferred directly to the hybrid operating theatre for emergency endovascular repair, while the massive transfusion protocol was activated, and he was resuscitated with 13 units of packed red blood cells, eight units of fresh frozen plasma, ten units of cryoprecipitate, and a pool of platelets. Whilst both groins were surgically prepped, the patient had rapid sequence induction followed by bilateral femoral access under ultrasound guidance. As the patient remained stable following induction, percutaneous closure devices were inserted bilaterally (Perclose Proglide, Abbott Laboratories, Illinois, USA).
Figure 2: Sagittal and axial planes highlighting esophageal stent with communicating aortic pseudoaneurysm

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Figure 3: Three-dimensional volumetric reconstruction highlighting thoracic aortic pseudoaneurysm creating a fistula with esophageal stent

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Following an aortic arch diagnostic angiogram, the area of focal pseudoaneurysm was seen at the site of the aortoenteric fistula. The landing zones were mapped, and the patient underwent insertion of a single covered thoracic sent (28 cm × 180 cm CoverSeal – Medtronic, Dublin Ireland). The proximal landing zone was distal to the left subclavian artery (Ishimaru landing zone 3). The stent had adequate wall apposition and the final angiographic run showed seal over the location of the likely fistula [Figure 4]. Due to significant coagulopathy, heparin was not given before stent deployment. Postdeployment dilatation was not performed due to concern of a fragile aorta.
Figure 4: Pre- and postthoracic covered stent deployment highlight location of pseudoaneurysm and adequate seal post stent deployment

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Post-operatively the patient stabilized and was observed in ICU for further 24 h. He received infectious disease input and placed on lifelong suppressive amoxicillin-clavulanate post discharge.


  Discussion Top


Definition and incidence

Aortoenteric fistula can be categorized as primary or secondary, and in relation to anatomical location. Primary aortoenteric fistula is defined as a spontaneous communication between the native aorta and any portion of the GI tract.[1] A secondary aortoenteric fistula occurs when there is a false communication between an enteric structure and a previous aortic graft, for which there are predisposing factors.

Esophageal malignancy is a known risk factor in the development of secondary aortoenteric fistula. Self-expanding metallic stents are now widely used in the management of obstructive disease of the esophagus. Various stents exist, including bare metal and covered stents. Following the initial introduction in 1983, they have become a mainstay in the palliative management of the malignant esophageal disease.[2] Adverse events relating to esophageal stent placement have been recognized, including esophageal injury, tear, rupture, and aorto-esophageal fistula formation.[2] Previous retrospective series looking into adverse events reported 153 patients affected. Of these, 43 died, 14 of massive hemorrhage post stent implantation.[3] Zhan et al. reviewed the literature for cases of aorto-esophageal fistula following esophageal stent implantation and found that nine of fourteen patients died of hemorrhage postimplantation.[4] Other series utilizing esophageal stents also report 10% incidence of AEF formation.[5]

Pathophysiology of aorto-enteric fistula development

In our patient, there are several risk factors which may have led to higher propensity for aorto-esophageal fistula formation. In particular, the history of prior radiotherapy for esophageal SCC would likely have led to a natural loss of tissue integrity in the mediastinum, with weakened esophageal mural strength and aortic adventitial strength. This combined with the underlying malignant process (and likely recurrence), will also have contributed to the development of the aortoenteric fistula.

There are also several anatomical and physiological processes which occur during AEF formation, which may explain the relatively high frequency of this condition in this patient population. Within the superior mediastinum,[6] 22 cm from the incisors, the esophagus is crossed by the aortic arch on its left side. Above the esophageal opening in the diaphragm, firm connective tissue connects the posterior surface of the esophagus to the aorta. Combined with the mechanical erosion from continuous aortic pulsations at this location, this area may be predisposed to communication. Finally, the esophageal stent itself plays a potentially critical role in AEF formation. The typical placement of these stents often results in proximal struts finishing at this potential natural area of communication, which may cause localized ischemia and necrosis with ulceration. A combination of all of these predisposed our patient to AEF formation. The endoscopy showed a large area of clot at this location, which was likely providing temporary tamponade from massive exsanguinating hemorrhage.[4]

Principles of thoracic endovascular repair in aorto-enteric fistula

On presentation, management of an AEF is similar to that of a ruptured mycotic aneurysm: principles include permissive hypotension (aiming for central nervous system perfusion rather than a systolic target), similar to the management of ruptured abdominal aortic aneurysms.[7] AEF is essentially an infection in which the bacteria-free aorta becomes contaminated by the gastrointestinal tract, and therefore antibiotic coverage is an essential part of therapy. Historically, open thoracic repair of aortic and esophageal defect with extensive debridement of the infected tissue (and replacement of infected aorta via allograft) was considered the gold standard. True lateral positioning with a posterolateral thoracotomy can provide access and repair of the entire thoracic aorta. This can be combined with a midline sternotomy or a trapdoor incision to enhance exposure.[8] Following open aortic repair, the esophageal repair must be considered. Approaches can include primary repair with or without reinforcement, exclusion diversion, and single-stage esophageal resection with or without primary reconstruction, but there is no consensus or gold standard for open esophageal surgery.[9]

In the endovascular era, international consensus guidelines have shifted towards thoracic endovascular repair (TEVAR) in the treatment of unstable patients. Although the long-term outcomes remain unclear,[10] it provides a temporizing means to seal massive exsanguination with minimal hemodynamic insult. An important, but the understated overarching principle in the treatment of endovascular hemorrhage control is effective multidisciplinary care. In the acute setting, these patients should be managed in centralized, tertiary referral centers (with activation of code-crimson protocols to allow expedited transfer to hybrid suites). These have a clear correlation of improved patient outcomes,[11] relating to the overall volume. Critical to the successful management of this case was the experienced anesthetist, who was able to manage hemodynamics under local anesthetic but poised for rapid induction. The presence of experienced endovascular scrub nurses and assistants allowed for the timely and life-saving arrest of the hemorrhage. Pooled analyses of EVAR/TEVAR for AEF suggest that there is an early survival benefit compared to early in situ repair, implying that TEVAR with planned subsequent explanation and definitive in situ repair may provide improved overall survival benefit relative to in situ reconstructions without endovascular temporization.[12] However, recurrent hemorrhage after endograft exclusion of the AEF can occur as early as 1 month after endograft placement.[12] Takeno et al., who reviewed the literature for 150 cases of AEF, found that of 59 patients who underwent some surgical therapy for AEF and whose cause of death was documented, 26 died of sepsis after surgery (compared to 33 from hemorrhage). This finding corroborates what is already intuitive to most surgeons, namely that TEVAR neither removes the infectious nidus nor corrects the enteric defect. There may be rare occasions where small enteric defects with minimal contamination can be managed with antibiotics and endovascular exclusion. For patients who are otherwise reasonable surgical candidates, open surgical repair of AEF should be offered, followed by esophagectomy for complete cure. However, given this patient's burden of metastatic disease, temporizing TEVAR was the surgical ceiling of care. Finally, the risk of spinal cord ischemia,[13] a recognized grave complication post TEVAR, can be minimized with several specialized techniques exist to minimize the risk of spinal cord ischemia including epidural cooling, retrograde perfusion, or complete cardiopulmonary bypass. This is deemed highly morbid, and in an unstable patient such as ours, would almost certainly be lethal due to the hemodynamic insult. Therefore, TEVAR is appropriate as a damage-control surgery for hemostasis as a bridge to stent replacement and esophageal repair.


  Conclusion Top


This case report highlighted successful management of a complex, life-threatening surgical emergency. Principles of permissive hypotension, multidisciplinary care, and use of specialized code-crimson pathways were critical to successful treatment. There appears to be a clear danger area of potential anatomical weakness in these patients. When combined with rigid esophageal stent strut at this location, the likelihood of AEF development is likely high in this patient population. Early utilization of CT angiography with prompt diagnosis, will lead to the highest chance of survival.

Declaration of patient consent

The authors certify that they have obtained all appropriate patient consent forms. In the form the patient (s) has/have given his/her/their consent for his/her/their images and other clinical information to be reported in the journal. The patients understand that their names and initials will not be published and due efforts will be made to conceal their identity, but anonymity cannot be guaranteed.

Financial support and sponsorship

Nil.

Conflicts of interest

There are no conflicts of interest.



 
  References Top

1.
Chung J. Management of Aortoenteric Fistula. Adv Surg 2018;52:155-77.  Back to cited text no. 1
    
2.
Spaander MC, Baron TH, Siersema PD, Fuccio L, Schumacher B, Escorsell Às, et al. Esophageal stenting for benign and malignant disease: European Society of Gastrointestinal Endoscopy (ESGE) Clinical Guideline. Endoscopy 2016;48:939-48.  Back to cited text no. 2
    
3.
Li Y, Wang X, Bian R, Sun J, Zhao M. Medical Device Adverse Events Primary Analysis on Esophageal Stents. Zhongguo Yi Liao Qi Xie Za Zhi 2017;41:48-50.  Back to cited text no. 3
    
4.
Zhan Y, Xu Z. Massive hemorrhage from an aortoesophageal fistula caused by esophageal stent implantation: A case report and literature review. Medicine (Baltimore) 2019;98:e18303.  Back to cited text no. 4
    
5.
Aryaie AH, Singer JL, Fayezizadeh M, Lash J, Marks JM. Efficacy of endoscopic management of leak after foregut surgery with endoscopic covered self-expanding metal stents (SEMS). Surg Endosc 2017;31:612-7.  Back to cited text no. 5
    
6.
McMinn RH. In: Last's Anatomy. 9th ed. Philadelphia, USA: Elsevier; 2019. p. 382-400.  Back to cited text no. 6
    
7.
Sörelius K, Budtz-Lilly J, Mani K, Wanhainen A. Systematic Review of the Management of Mycotic Aortic Aneurysms. Eur J Vasc Endovasc Surg 2019;58:426-35.  Back to cited text no. 7
    
8.
Valentine RJ, Wind G. In: Anatomic Exposures in Vascular Surgery. 4th ed. Philadelphia, USA: Wolters Kluwer; 2020. 180-200.  Back to cited text no. 8
    
9.
Kaman L, Iqbal J, Kundil B, Kochhar R. Management of Esophageal Perforation in Adults. Gastroenterology Res 2010;3:235-44.  Back to cited text no. 9
    
10.
Miyahara S, Nomura Y, Shirasaka T, Taketoshi H, Yamanaka K, Omura A, et al. Early and midterm outcomes of open surgical correction after thoracic endovascular aortic repair. Ann Thorac Surg 2013;95:1584-90.  Back to cited text no. 10
    
11.
Brescia AA, Patel HJ, Likosky DS, Watt TM, Wu X, Strobel RJ, et al. Volume-Outcome Relationships in Surgical and Endovascular Repair of Aortic Dissection. Ann Thorac Surg 2019;108:1299-306.  Back to cited text no. 11
    
12.
Kakkos SK, Bicknell CD, Tsolakis IA, Bergqvist D, Hellenic Co-operative Group on Aortic Surgery. Choice-management of secondary aorto-enteric and other abdominal arterio-enteric fistulas: A review and pooled data analysis. Eur J Vasc Endovasc Surg 2016;56:770-86.  Back to cited text no. 12
    
13.
Xue L, Luo S, Ding H, Zhu Y, Liu Y, Huang W, et al. Risk of spinal cord ischemia after thoracic endovascular aortic repair. J Thorac Dis 2018;10:6088-96.  Back to cited text no. 13
    


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  [Figure 1], [Figure 2], [Figure 3], [Figure 4]



 

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