Successful rescue of a patient with aortoesophageal fistula secondary to anastomotic leak after esophagectomy

Abstract

Background

Aortoesophageal fistula (AEF) is a rare but invariably life-threatening conditio. The optimal treatment for AEF resulting from postoperative anastomotic leakage following esophageal cancer surgery remains a significant clinical challenge. Here, we report a detailed case of AEF caused by anastomotic leakage from a thoracic esophageal anastomosis after esophageal cancer resection, which was successfully managed with emergent thoracic endovascular aortic repair (TEVAR) for hemorrhage control, followed by elective surgical intervention comprising esophagogastric re-anastomosis and aortic fistula repair with a bovine pericardial patch. The patient achieved long-term survival.

Case presentation

A 68-year-old male underwent minimally invasive McKeown esophagectomy with intrathoracic esophagogastric anastomosis after neoadjuvant chemotherapy and immunotherapy for advanced esophageal squamous cell carcinoma (SCC). Postoperatively, the patient experienced fever followed by hematochezia and hematemesis. Emergency endoscopy was unsuccessful in controlling the active arterial bleeding at the inflamed anastomotic site. Enhanced CT angiography (CTA) did not reveal a definitive aortoesophageal fistula, and subsequent transarterial embolization of the right gastric artery (confirmed by procedural records) failed to control the condition. The patient developed hemorrhagic shock manifested by loss of consciousness and hypotension. A repeat enhanced CT scan revealed active contrast extravasation from the descending aorta into the gastric conduit. An emergency TEVAR was performed to stabilize the hemodynamics. On postoperative day 10, we performed surgical exploration, which confirmed resection of the esophagogastric anastomotic leak, direct repair of the aortic wall defect, and reconstruction via esophagogastric anastomosis with the remnant stomach. Intraoperatively, a stapler clip from previous endoscopic hemostasis was identified as the potential culprit for the fistula formation. Ten months after the onset of AEF, the patient continued chemotherapy and led a normal daily life.

Conclusion

This case underscores that TEVAR is an effective and life-saving hemostatic method for managing life-threatening hemorrhage from AEF. However, TEVAR alone cannot ensure long-term survival due to its inability to control the underlying infection and address the gastrointestinal defect. Long-term survival requires subsequent definitive surgical repair of both the esophageal and aortic components.

Background

Aortoesophageal fistula (AEF) is a rare but potentially life-threatening condition characterized by an abnormal connection between the aorta and the esophagus [1–3]. The clinical management of AEF, particularly when it results from complications of esophageal cancer surgery, remains a challenging issue. This research focuses on the therapeutic strategies for AEF, especially in cases arising from postoperative anastomotic leaks.

Esophageal cancer, a prevalent malignancy with a high mortality rate, often necessitates extensive surgical interventions including esophagectomy and gastroesophageal anastomosis [4, 5]. Despite advancements in surgical techniques and adjuvant therapies, complications such as anastomotic leaks pose significant risks to patient survival. The development of AEF following esophageal cancer surgery is a particularly severe complication, as it can lead to massive and often catastrophic hemorrhage and rapid deterioration of the patient’s condition [6, 7].

The optimal treatment approach for AEF is still under debate. While traditional open surgical methods of managing AEF often involve direct repair of the fistula and the affected aortic segment, these interventions are associated with high morbidity and mortality and may not always be successful. Recent advancements in endovascular techniques, such as thoracic endovascular aortic repair (TEVAR), have introduced new options for managing such critical situations by providing a less invasive means of securing the aortic integrity.

In this study, we present a case of AEF induced by an esophageal anastomotic leak post-esophageal cancer resection, which was successfully managed through an emergency TEVAR procedure as a bridge to definitive surgery. This was followed by a secondary surgical intervention for comprehensive repair of the aortic fistula and re-establishment of esophageal continuity. The patient’s successful recovery and long-term survival underscore the effectiveness of a staged approach, initiating with TEVAR in stabilizing hemorrhagic events, though it also highlights the necessity for subsequent surgical correction to address the septic focus and the gastrointestinal conduit disruption.

Our findings contribute to the understanding of AEF management by illustrating the potential of combining endovascular and surgical approaches to achieve favorable outcomes. This case emphasizes the importance of a multidisciplinary approach involving thoracic surgery, cardiac surgery, and interventional radiology in the management of complex post-surgical complications and offers insights into improving survival rates in patients with this devastating complication.

Case presentation

The patient, a 68-year-old male with a 3-month history of dysphagia, was diagnosed with esophageal SCC. Endoscopic examination revealed wall rigidity and thickening at 33–36 cm from the incisors, impeding gastric passage of the endoscope. Biopsy confirmed SCC, and chest-abdomen CT showed significant wall thickening of the lower esophagus. According to the Japanese Classification of Esophageal Cancer, 11th Edition, the clinical stage was determined to be T3N2M0. The patient received neoadjuvant chemotherapy and immunotherapy consisting of paclitaxel-albumin, nedaplatin, and sintilimab. The patient also had a history of hypertension with poorly controlled blood pressure, despite treatment with nifedipine extended-release and metoprolol. The patient underwent a minimally invasive McKeown esophagectomy with an intrathoracic linear-stapled esophagogastric anastomosis. During surgery, significant aortic tortuosity was noted.

Postoperative complications included fever on postoperative day 9, with chest CT revealing pleural effusion and pulmonary inflammation. By postoperative day 10, fresh blood was noted in the gastric tube drainage, necessitating transfusions of 2 units of red blood cells and 200 ml of plasma. Chest CT-guided drainage was performed, extracting 150 ml of bloody effusion. On postoperative day 11, the patient experienced hematemesis and melena, with significant bleeding observed at the anastomotic site. Emergency endoscopy identified severe anastomotic bleeding, suspected to be arterial in nature. Due to significant inflammation and edema at the anastomotic site, endoscopic hemostasis was unsuccessful, leading to intervention with selective esophageal artery embolization. Two days after the embolization, the patient experienced recurrent upper gastrointestinal bleeding. Resuscitation measures including blood transfusions were undertaken, and CTA showed no active contrast extravasation in the gastric lumen or obvious fistula at the anastomotic site. However, aortic ulceration and a peri-aortic hematoma were observed without a clear aortic fistula.

After selective arterial embolization failed to provide long-lasting hemostasis, it was decided on postoperative day 25 to move on with TEVAR. Since the original CTA did not show a conclusive fistula, this period illustrates the diagnostic difficulty presented by AEF. Initially, conservative and interventional methods were given priority. Only until repeat CTA revealed active aortic contrast extravasation that coincided with the beginning of hemorrhagic shock did the definite indication for TEVAR become apparent, confirming the diagnosis and the necessity of prompt aortic surgery. The decision was made to proceed with an aortic stent graft placement on postoperative day 25 (which was 8 days after the embolization). Percutaneous access to the right common femoral artery was used to do the emergency TEVAR. Intravascular ultrasonography (IVUS) was used to determine the landing zones precisely and evaluate the aorta architecture following systemic heparinization. To cover the extravasation location, a thoracic stent-graft was placed in the distal aortic arch. Completion angiography verified that the fistula had been successfully excluded and that there was no sign of a type I or III endoleak. After the surgery, the patient’s hemodynamics quickly normalized. The patient was transferred to the intensive care unit (ICU) and showed good recovery.

Despite the aortic stent graft placement, a discussion among the medical team concluded that anastomotic leakage likely persisted due to unresolved local infection. As a result, a second surgical intervention was planned. On postoperative day 35, the patient underwent reoperation involving take-down of the previous anastomosis, reanastomosis of the residual stomach and esophagus, with bovine pericardium used for reinforcement of the aortic fistula repair. During the reoperation, extensive thoracic adhesions and significant aortic tortuosity were observed, with dense adhesion between the descending aorta and surrounding structures. The anastomotic site was found to be densely adhered to the lower aortic arch. The gastric wall was incised to reveal a stapler clip from the previous endoscopic hemostasis, which was strongly suspected as the nidus for the fistula formation and persistent leak. Extensive and drastic debridement of all necrotic and inflammatory peri-aortic and mediastinal tissues was carried out during the reoperation following the removal of the anastomosis. In order to establish healthy, bleeding tissue margins, the region surrounding the fistula site—which displayed indications of chronic infection—was carefully debrided. Next, the borders of the aortic wall defect were cleaned. This area was circularly excised, and a prosthetic patch was used for aortic wall repair. Due to extensive dissection of the residual stomach, the decision was made to perform a hand-sewn re-anastomosis of the stomach and esophagus at a higher level under the aortic arch. To preserve aortic integrity, the previously implanted aortic stent graft was maintained in place. A bovine pericardial vascular patch was mainly used to seal the aortic wall defect at the fistula site. This patch reinforced the closure and added another line of defense against any local infection. The intraoperative situation is shown in Fig. 1. The surgery was challenging but ultimately deemed satisfactory. Postoperatively, the patient recovered smoothly and was discharged 18 days after the final surgical intervention.

Fig. 1.

Photographs of the surgical procedure

Discussion

Aortoesophageal fistula (AEF) arising as a complication of esophageal cancer surgery presents a formidable clinical challenge, often characterized by complex management and high mortality. Our case vividly illustrates this scenario, where an anastomotic leak led to the erosion of a stapler clip into the adjacent aorta, culminating in a life-threatening AEF. The combination of anastomotic leak leading to AEF necessitates an intricate and staged approach to treatment. This discussion delves into the critical role of thoracic endovascular aortic repair (TEVAR) as a preliminary life-saving intervention and examines the subsequent necessity for definitive surgery involving esophageal-gastric re-anastomosis and aortic fistula repair. The danger of upfront definitive open surgery was prohibitive in this critically ill patient who presented with hemorrhagic shock. TEVAR was an essential “bridge” or “damage-control” process. Resuscitation, hemodynamic stabilization, and acute inflammatory insult mitigation were all made possible by its prompt, life-saving bleeding management. This made it possible to optimize the patient’s health, give targeted antibiotics, and schedule the ensuing elective, definitive surgical repair under more controlled conditions—all of which are linked to better results.

TEVAR has emerged as a critical intervention in the acute management of AEF, primarily serving as a bridge to more definitive treatment. Its primary advantage lies in its minimally invasive nature, which facilitates rapid stabilization of patients in critical hemorrhagic shock, as seen in our patient who developed hypotension and altered consciousness. Literature indicates a high technical success rate for TEVAR, approximately 87.3%, with a lower 30-day mortality rate (19.4%) compared to open surgical repair (50%) [1, 8]. This demonstrates TEVAR’s efficacy in controlling life-threatening hemorrhage and stabilizing patients who may otherwise face immediate mortality, thereby creating a crucial window of opportunity for further planning and intervention.

However, the use of TEVAR does not address the underlying gastrointestinal defect or remove infected tissue, which can continue to pose significant risks [8]. In our case, despite successful hemostasis with TEVAR, the persistent anastomotic leak and the presence of the eroding stapler clip mandated a second operation. Furthermore, the persistent presence of the stent graft in a potentially septic environment raises concerns about long-term infection and graft-related complications such as endoleaks or graft disintegration. This limitation underscores the absolute necessity for subsequent surgical interventions to address the root cause of the AEF and to achieve source control. After considerable thought, it was decided to conduct a primary re-anastomosis within the chest, even though it was in an area that had already been infected. Achieving what was deemed to be a radical debridement with control of local infection was necessary for this. The goal of the viable bovine pericardium interposition was to separate the suture line. Additionally, the increased morbidity of a cervical esophagostomy and delayed reconstruction were compared with the degree of stomach mobilization and the patient’s general health. This emphasizes how crucial customized surgical judgment is in challenging situations.

The role of esophageal-gastric re-anastomosis and aortic fistula repair post-TEVAR is pivotal. While TEVAR provides immediate hemostatic control, it is inadequate for the long-term management of the gastrointestinal fistula. The persistent gastrointestinal defect demands definitive closure to prevent continuous mediastinal leakage and associated complications like persistent sepsis and potential re-bleeding from other vessels. Our surgical strategy involved not only the resection of the leaking anastomosis and re-anastomosis but also the critical step of addressing the aortic wall defect and interposing a biological barrier (bovine pericardium) between the new anastomosis and the aorta. This multi-component repair is essential to prevent recurrence.

Several studies support the necessity of subsequent surgical intervention. Akashi et al. demonstrated that combined approaches, including esophageal resection and aortic replacement, significantly improve survival rates in AEF cases, compared to TEVAR alone [9]. Similarly, Kawamoto et al. advocated for the early combination of TEVAR with subtotal esophagectomy as an effective treatment for AEF [10]. These findings highlight the importance of addressing both the aortic and gastrointestinal components of AEF. The timing of this definitive surgery, as in our case performed 10 days after TEVAR, is crucial; it must balance the patient’s recovery from the initial insult with the urgency to control ongoing sepsis.

We acknowledge the risk of stent-graft infection and the potential for the graft itself to become a nidus for recurrent or new AEF, particularly at its ends in areas of persistent inflammation or adhesion. This inherent risk underscores why TEVAR is considered a temporary bridge rather than definitive therapy in infected fields. It reinforces the necessity of timely and radical surgical debridement and repair to control the septic focus and mitigate long-term graft-related complications. In practice, early definitive esophageal resection/re-anastomosis and aortic fistula repair after TEVAR can mitigate the risk of ongoing infection and provide a more comprehensive solution. Reports indicate that delaying these procedures can lead to increased morbidity and mortality, particularly due to recurrent bleeding or persistent infection [11, 12]. Therefore, integrating TEVAR with definitive surgical repair is essential for optimal patient outcomes. The successful long-term survival of our patient, leading a normal life 10 months post-operatively, stands as a testament to the efficacy of this combined, multidisciplinary approach.

Conclusion

In summary, TEVAR plays a crucial role in the initial management of AEF by providing critical stabilization and controlling life-threatening hemorrhage. However, its limitations in addressing gastrointestinal defects and ongoing infection highlight the necessity for subsequent definitive surgical interventions, including esophageal-gastric re-anastomosis and aortic fistula repair with tissue interposition. A combined, staged approach ensures both immediate stabilization and long-term resolution of AEF, improving patient outcomes and reducing the risk of complications. Future research should focus on refining these treatment strategies, optimizing the timing between endovascular and surgical phases, and evaluating long-term outcomes to further enhance the management of this complex condition.

Authors’ contributions

Mengchao Xue: Conceptualization, Data curation, Formal analysis, Investigation, Writing - original draft, Writing - review & editing.Mingsheng Wu: Conceptualization, Resources, Supervision, Validation, Project administration.Ming Lu: Conceptualization, Methodology, Supervision, Validation, Visualization, Writing - review & editing, and acted as the corresponding author responsible for all correspondence and revisions. All authors read and approved the final manuscript.

Funding

Project supported by the National Natural Science Foundation of Shandong Province (No.ZR2020QH212).

Data availability

The datasets generated and/or analyzed during the current study are available from the corresponding author on reasonable request.

Declarations

Ethics approval and consent to participate

The Ethics Committee of Shandong University’s Qilu Hospital gave its approval to this study. We attest to the fact that this study complied with the 2013 revision of the Declaration of Helsinki.

Consent for publication

Patients in this study provided written informed consent for their personal clinical details and any identifying images published in this study.

Competing interests

The authors declare no competing interests.