Case Report - (2021)Volume 12, Issue 6
Aorto Esophageal Fistula (AEF) is a rare but catastrophic complication in thoracic aortic aneurysms with no consensus about the best treatment option. We present a case of AEF as a late complication of TEVAR implantation in a Stanford type-B chronic dissection treated with hybrid aortic surgical approach and percutaneous esophageal stent.
Computed tomography; Aorta; Dissection; Vascular disease
AEF: Aorto Esophageal Fistula; CT: Computed Tomography; ECG: Electrocardiogram; TEVAR: Thoracic Endovascular Aortic Repair
•To recognize cardiovascular complications in chronic aortic diseases.
•To learn the role of multimodality imaging in challenging aortic disease cases.
•To evaluate and discuss percutaneous options in major complications in aortic diseases.
History of presentation
A 64-year-old male presented to the emergency department for the onset of nausea and hypotension. He also reported episodes of melena in the last 3 days. Blood pressure was 95/60 mmHg, heart rate 60 bpm, body temperature 36°C, and respiration rate 24 bpm. Cardiac and thoracic auscultations were unremarkable, and all peripheral pulses were present.
Past medical history
His past medical history included arterial hypertension, dyslipidemia, and a Type-B Stanford aortic dissection in 2000. Due to the progressive aortic dilation, in 2013, a TEVAR was implanted in zone 1 of the aortic arch associated to total debranching of supra-aortic trunks and ligature of the left subclavian artery. Subsequently, due to progressive aortic dilatation and the suspicion of a type 2 endo leak, the left subclavian artery was embolized through the false lumen in 2017. Since then, the patient underwent annual follow-up at our aortic disease unit with stabilization of the proximal thoracic aortic aneurysm diameter, however, there was aortic arch dilation in the proximalsealing zone with proximalTEVAR migrationwithoutevidence of a type I endoleak (Figure 1). The patient was waiting for an elective open thoracoabdominal aneurysm repair.
Figure 1: ECG-gated CT angiography, last follow-up study before hospital admission. (A): oblique sagittal plane showing the aortic arch and descending thoracic aorta with the implanted TEVAR and exclusion of the residual false lumen (asterisk). Double-sided arrow shows the aortic arch dilation and signals the displacement point of the TEVAR. (B): Transverse plane of the aorta showing the maximum diameter of the descending aorta aneurysm (87 mm). (C): Volume rendering cinematic reconstruction showing aortic arch dilation proximal to the TEVAR andsubclavian artery embolization coils (arrows).
Given the patient's clinical presentation with gastrointestinal bleeding, the differential diagnosis included primary digestive bleeding secondary to peptic ulcer disease, cancer, reflux esophagitis, or bleeding esophageal varices. However, because of the patient’s history of chronic aortic dissection, the presence of an aortic-esophageal fistula or aortic rupture was also considered in the differential diagnosis.
The laboratory workup revealed anemia (Hemoglobin of 10.7 g/dL, hematocrit of 33%), D-dimer elevation (2408 ng/mL), leukocyte elevation (12.57 × 109/L) with neutrophilia (82.8%), C-reactive protein 1.89 mg/dL, creatinine 1.18 mg/dL; remaining parameters were unremarkable. The Electrocardiogram (ECG) showed no repolarization abnormalities. A transthoracic 2D echocardiogram (TTE) showed a non-dilated left ventricle with normal systolic function and mild dilation of the ascending aorta.
The patient underwent an ECG-gated CT study showing a significant detachment of the proximal portion of the TEVAR (Type I endoleak), proximal descending aorta aneurysm growth with a maximum diameter of 87 mm (including the false lumen peri- TEVAR), and the presence of small air bubbles in the mediastinum between the false lumen and the esophagus (Figure 2).
Figure 2: ECG-gated CT angiography. Oblique coronal plane of the TEVAR before (A) and after the current admission (B) showing caudal displacement of the proximal anchorage (dotted lines and dotted arrow). Arrowheads signal the subclavian embolization coils. (C): Increased dilation of the descending thoracic aorta (from 87 mm to 110 mm); intra-aneurysm air bubble (arrowhead), and adjacent esophagus (asterisk). (D): Volume rendering cinematic reconstruction of the thoracic aorta.
The esophageal-gastro-duodenoscopy confirmed the suspicion of Aorto Esophageal Fistula (AEF) for the presence of small clots in the esophageal wall and a clear wall continuity solution.
The patient was hospitalized in the Acute Cardiac Care Unit and owing to recurrent hematemesis, progressive fall in hematocrit, possible mediastinal infection (fistulization with the esophagus), and the increased mortality risk associated with combined aortic surgery (open aortic arch and descending thoracic aortic repair) and esophagectomy, the aortic team decided on a hybrid surgical repair. High-dose antisecretory therapy, parenteral nutrition, and antibiotic prophylaxis with amoxicillin-clavulanic acid were started. Firstly, the patient underwent esophageal stent implantation to seal the fistula. Three days later, an aortic repair with a Thoraflex hybrid prosthesis implantation was performed with brachiocephalic trunk implantation on the distal ascending aorta. In the same procedure, a new TEVAR was implanted from the distal part of the Thoraflex to the distal thoracic descending aorta (Figure 3). Thus, the aortic arch and the thoracic descending aorta were repaired and isolated from the esophageal fistula.
Figure 3: CT angiography volume rendering cinematic reconstruction, a posterior view showing the relationship between the TEVAR and the esophageal stent with the nasogastric tube within it.
After surgery, the patient was followed by serial Gated-ECG CT studies with no reported endoleaks or other complications related to the aortic surgical procedure. Twenty days after surgery, the patient presented a new episode of non-hemodynamically significant hematemesis. A proximal detachment of the esophageal stent was shown by CT with no new aortic abnormalities. The endoscopic study again revealed the presence of an esophageal fistula (Figure 4) being treated with endoscopic relocation of the esophageal stent with no further complications. The patient was discharged in stable clinical conditions after 90 days of hospitalization and under treatment with broad-spectrum antibiotic therapy. However, 20 days later, the patient died due to the development of fever and posterior mediastinitis.
Figure 1: CT angiography volume rendering cinematic reconstruction, a posterior view showing the relationship between the TEVAR and the esophageal stent with the nasogastric tube within it.
AEF is a life-threatening condition with a high rate of morbidity and mortality. It is usually related to an aortic or esophageal disease such as esophageal cancer or aortic aneurysms [1,2]. It has been described as a rare and fatal complication after TEVAR with an incidence of<2% [3,4] associated with TEVAR infection in the early postoperative period [5,6] or direct erosion of the stent-graft through the aorta into the esophagus [3-7].
The AEF treatment of choice is under debate since these are high-risk patients. Classically, conservative management has been performed which was associated with a 100% mortality. Also, an esophageal stent (17% post-operatively survival rate) or isolated esophagectomy (19% survival rate) have been reported. The highest survival has been described in cases with aortic replacement/ repair plus surgical esophagectomy with a 1-year survival rate of 46% [8,9]. We reported the first successful case in which non- invasive management of the esophageal pathology (stent) and a hybrid aortic approach (aortic arch surgery and TEVAR) has been described to initially stabilize the hemodynamic situation of the patient and, also, to reduce the morbidity and mortality of a combined esophageal and complete open aortic surgery. Although the procedure was technically successful and the patient could be discharged, the fatal final few weeks later suggests that this approach must be carefully individualized and highlights the importance of esophageal repair after the initial stabilization given the high risk of infection and mediastinitis.
The present case shows that complications after TEVAR implantation are diverse and they can occur at any time, so close follow-up with different imaging techniques (mainly CT) must be performed at least annually .
There is no clear consensus about the treatment of choice in AEF which is associated with a high mortality rate. The best treatment approach is not known, and a bedside multidisciplinary decision by an aortic team seems to be the best option. In this case, a hybrid surgical aortic approach with a non-invasive esophageal intervention (stent) was chosen with a satisfactory in-hospital clinical stabilization of the patient. However, it underlines the relevance of complete esophageal repair and infection control to improve the negative prognosis of these patients.
The authors declare that there is no conflict of interest.
Drs. Lucia La Mura has been supported by a research grant provided by the Cardiopath Ph.D. program.
The authors have reported that they have no conflicts of interest to disclose.
Citation: Mura LL, Valente FX, Servato ML, Barrabes JA, Rodriguez-Lecoq R, Constenla I, et al. (2021) Hematemsis as a Late Complication after Tevar in a Stanford Type-B Chronic Aortic Dissection. J Clin Exp Cardiolog.12: 1000682.
Received: 17-May-2021 Accepted: 03-Jun-2021 Published: 10-Jun-2021 , DOI: 10.35248/2155-98126.96.36.1992
Copyright: 2021 Mura LL, et al. This is an open-access article distributed under the terms of the Creative Commons Attribution License, which permits unrestricted use, distribution, and reproduction in any medium, provided the original author and source are credited.