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ISSN: 2835-1568; CODEN: USA

DOI: http://dx.doi.org/10.51521/WJCRCI.2023.220117

CASE REPORT

Aortic-Esophageal Fistula (AEF)- A Case Report

Dr.Gigi Bregadze, Dr.Mariam Grdzelidze & Mr. Davit Tetvadze

Department of Vascular Surgery, New Hospitals, Tbilisi State Medical University,

Tbilisi, Georgia
Received Date:
25/07/2023
Revised Date:
31/07/2023 Abstract
Accepted Date: Aortoesophageal fistula is one of the rarest causes of bleeding in the upper
02/08/2023 gastrointestinal tract. Aneurysm of the thoracic aorta is the leading causing factor
Published Date in such cases. The main challenge for doctors is to diagnose timely, because the
15/08/2023 disease can be accompanied by massive bleeding and rapid worsening of the
patient's condition. A characteristic symptom is Chiari's triad (chest pain,
hematemesis, symptom-free period after hematemesis). However, the triad is
expressed only in 1/3 of patients. The leading diagnostic tools are esophago-
gastro-duodenoscopy (detection accuracy ≈ 25%) and contrast CT angiography,
Corresponding Author:
Mr. Davit Tetvadze, Department of
which in most cases reveals the disease. After the diagnosis, the patient's aorta and
Vascular Surgery, New Hospitals, esophagus are urgently reconstructed by the endovascular method. Massive
Tbilisi State Medical University, Tbilisi, bleeding from the esophagus, diagnostic difficulties, and the insufficient amount of
Georgia, Email: medical equipment in the regions of Georgia make it difficult, and sometimes the
[email protected] possibility of diagnosing the above-mentioned disease is missed altogether, and
Citation: thus complicates the solution. We present a case of aorto-esophageal fistula, the
Dr.Gigi Bregadze, Dr.Mariam patient was brought to our clinic from one of the regions of Georgia, where he was
Grdzelidze & Mr. Davit Tetvadze (2023) being treated with the following diagnose: bleeding from an esophageal ulcer. In
Aortic-Esophageal Fistula (AEF)- A our hospital the patient was immediately diagnosed by computed tomography
Case Report. World J Case Rep Clin
Imag. 2023 July-August; 2(2)1-5. (CT).

Copyrights
© 2023, Mr. Davit Tetvadze, et al., This Keywords:
article is licensed under the Creative Aortic-Esophageal Fistula
Commons Attribution-Non Commercial-
Computed Tomography
4.0-International-License-(CCBY-NC)
(https://worldjournalofcasereports.org/b Vascular Surgery
logpage/copyright-policy). Usage and Minimally Invasive Operations
distribution for commercial purposes Aortic Aneurysm
require written permission. Esophagus, etc.,

Introduction aortic aneurysm, foreign body ingestion, esophageal malignancy, or

postoperative complications. The diagnosis can be made on the basis
An aortoesophageal fistula is a life-threatening cause of of clinical findings alone. (Heckstall & Hollander, 1998). Diagnosis
gastrointestinal bleeding where an abnormal communication of AEF is rarely made before massive hematemesis. However, most
between the esophagus and the aorta may result from a thoracic cases are associated with characteristic Chiari’s triad features of

Davit Tetvadze, et al., 2023

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ISSN: 2835-1568; CODEN: USA

DOI: http://dx.doi.org/10.51521/WJCRCI.2023.220117

aortoesophageal syndrome, including chest pain and sentinel

hematemesis of red blood followed at a variable interval of time by
rapidly fatal massive exsanguinating hematemesis. In the present
series these symptoms were observed in 4 of the 6 patients with true
AEF. (Kieffer, Laurent, & Dominique, 2003). CT angiography is the
mainstay for diagnosis owing to the quick acquisition, widespread
availability, and superior spatiotemporal resolution (Vu, Menias, &
Bhalla, RadioGraphics). Extravasation of contrast material from the
aorta into the esophagus, or vice versa, can be diagnosed with high
confidence by CT angiography.

Pathogenesis
Atherosclerotic disease leading to intimomedial fat deposition is the
most common cause of loss of integrity of an otherwise intact vessel
wall. With the high pulsatile intravascular pressures, this
phenomenon gradually leads to aneurysmal growth. The weakened
aortic wall allows abnormal radial transmission of pulsatile pressure
to the surrounding soft tissues. This abnormal force causes pressure
necrosis and adhesive granulation tissue between the aorta and a
periaortic hollow or solid organ, resulting in a fistula (Picichè, R, &
A, 2003). A false aneurysm, or pseudoaneurysm, does not contain
wall layers and is more prone to sudden rupture instead of controlled
fistula development. A true aneurysm occurs due to weakening of
the wall of an otherwise intact blood vessel, in which case all three Figure 1: X-ray, Coronal plane, A – Blakemore tube esophageal
walls of the artery are thinned but intact. If AeF (Aortic-esophageal balloon, B – Gastric Baloon. The compression of esophageal balloon
Fistula) occurs during aortic intervention and/or aortic graft can’t be seen because of the plane
placement, it is termed a secondary fistula. The pathophysiology of
secondary fistulas is multifactorial and related to foreign body
reaction to the graft, graft cramping, infection, leakage coil
placement, etc.

Case Presentation – A 55-year-old male patient was transferred from

one of the regional clinics of Georgia, where he was being treated
with the diagnosis - esophageal ulcer, thoracic aorta aneurysm
without rupture. Bleeding relapses were noted twice before
transferring, with vomiting of bright red blood for which a
Blakemore tube was inserted. Hemotransfusion took place. Due to
the pain in the chest, a computed tomography of the chest cavity
with I.V contrast was performed, where a 1.6/2.3 cm contrast-filled
mass was noted on the right wall of the aorta, after which the
penetration of the aorta into the esophagus was suspected.

Upon admission, he complains of general weakness, dizziness,

shortness of breath, chest pain, the patient is drowsy, opens his eyes
when called, and is able to perform simple tasks. 5 years ago, left
inguinal hernia plastic surgery was performed, there is no allergy. Figure 2: CT angiography scan, Axial plane, Th.6 vertebral level. A –
Due to the general condition, the patient was intubated. The Inserted and compressed Blakemore tube. B – Aneurysm sac,
extravasation from the aorta. Compressed bronchi can be seen as
condition is stable, a preliminary diagnosis was made:
well on both of sides.

 Thoracic aortic aneurysm, ruptured – I71.1

 Somnolence, stupor and coma – R40
Preoperational Condition
 Other shock – R57.8
The blood shows an anemic picture, post-hemorrhagic anemia,
 Acute respiratory failure – J96.0 which is why blood transfusion became necessary. In the
 essential hypertension – I10 background of high CRP, the blood is sterile. During the ultrasound
examination of the upper part of the abdominal cavity, a large
amount of mixed echogenic mass - blood - is visualized in the
stomach with a Blakemore tube. It was decided to perform an
integrated operative intervention, to insert stent grafts of the aorta
and esophagus. In order to avoid the infection of the aortic stent
graft, the esophageal stent graft should be inserted first, and then the

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ISSN: 2835-1568; CODEN: USA

DOI: http://dx.doi.org/10.51521/WJCRCI.2023.220117

interventions on the aorta must be performed. Recent studies showed It was necessary to mark the exact location. For this, endoscopists
that stent grafting is a promising technique for treatment of infiltrated the distal site of the esophagus with contrast to make the
esophageal perforation. However, the evidence of its benefits is still perforation area more visible (Figure 4).
scarce. Stent grafting seems to be an effective less invasive
technique for the treatment of esophageal perforation. It allows the
preservation of the esophagus in most of patients. (Biancari,
Tauriainen, & Tatu, 2017).

Research Conclusion
Fifty-three consecutive patients underwent stent grafting for
esophageal perforation at Oulu University Hospital, Finland. The
primary endpoint of this study was early and intermediate mortality.
Secondary outcome endpoints were the need for esophagectomy and
additional surgical procedures on the esophagus and
extraesophageal structures. Patients' mean age was 64.6 ± 13.4
years. The mean delay to primary treatment was 23 ± 27 h. The most
frequent cause of perforation was Boerhaave's syndrome (46.5%).
The thoraco-abdominal segment of the esophagus was affected in
58.1% of cases. Minor primary procedures were performed in 25
patients (58.1%) and repeat surgical procedures in 23 patients
(53.5%). repeat stent graftings were performed in 22 patients (50%).
Two patients (4.7%) underwent esophagectomy, one for unrelenting
preprocedural stricture of the esophagus and another for persistent
leakage of a perforated esophageal carcinoma. The mean length of
stay in the intensive care unit was 6.0 ± 7.5 days and the in-hospital
stay was 24.3 ± 19.6 days. In-hospital mortality was 4.6%. Three-
Figure 4: X-ray, Coronal plane. A – The contrast infiltrated area for
year survival was 67.2% (Biancari, Tauriainen, & Tatu, 2017). the better measurement of the perforation.

Stent Graft Implantation – Before esophageal stenting is started. The endoscopist was then free to perform esophageal stent-grafting
Under general anesthesia We isolated the femoral artery and passed (Figures 5,6).
the extra-stiff (Lunderquist) wires into the thoracic aorta to be ready
in case of bleeding. After this using endoscope esophagus lumen is
free. Large blood clots and fresh blood in the lumen. At 28
centimeters from the mouth, there is an ulcerated area of up to 1 cm.
Esophageal stenting was performed under X-ray control by inserting
a 24-Fr esophageal stent of Cook. The penetrated area is closed
without complications (Figure 3).

Figure 3: X-ray, Coronal plane. A – Extra-stiff wire (Lunderquist). B – Figure 5. X-ray, Coronal plane. A – 24fr esophageal
Endoscope to clarify and observe the exact localisation of the
perforation.

Davit Tetvadze, et al., 2023

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DOI: http://dx.doi.org/10.51521/WJCRCI.2023.220117

control, a catheter is inserted from the left brachial artery and the
‘’Pigtail’’ is brought to the exit of the left subclavian artery.
Angiography shows a cone-shaped pathological dilatation of the left
subclavian artery (Figure 8). The graft was placed in the distal
descending direction of the left subclavian artery. The stent-graft is
deployed. The rupture site was covered. Aneurysm is not contrasted
by control angiography. Extravasation does not occur (Figure 9).

Figure 6. X-ray, Coronal plane. A – Graft site fully stent of

Cook is aimed at precise localization covers the perforation site. B,
C – fixating sites.

Part of Vascular Surgery

Now is the time for vascular surgeons to act. The left common
femoral artery was isolated and clamped on tourniquets. The
common femoral and left brachial arteries were cannulated using the Figure 8 A: X-ray, Coronal plane. A – Cone-shaped anomaly of the
Seldinger technique. A hydrophilic wire 0.35" and an angiographic left subclavian artery.
catheter "Pigtail" were transferred from the thigh in the direction of
the aortic arch. Aortography was performed. A saccular aneurysm
of the thoracic aorta can be seen (Figure 7).

Figure 9: X-ray, Indirect plane for a better vision. A – Deployed stent-

graft Fully covering the rupture site. B – esophageal stent. C – Extra-
Figure 7: X-ray, Coronal plane. A Saccular aneurysm of the stiff wire. D – ‘Pigtail’ inserted through the left Subclavian artery
descending thoracic aorta
Conclusion
From the femoral approach, an extra-stiff wire was passed in the
direction of the aortic arch and brought to the ascending aorta. A The patient was discharged from the hospital on 31/12/2022, with
stent-graft (40x170 mm) was transferred to the same wire. For the following prescription: double anticoagulation, low dose

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DOI: http://dx.doi.org/10.51521/WJCRCI.2023.220117

rivaroxaban + long-term aspirin, symptomatic treatment and Conflict of Interest: None

angiologist supervision. Endoleak is not visible on repeated control
computed tomography. The patient spent 5 days in the ICU and 11 Ethical Considerations: None
days in the surgical hospital. In a nutshell We can clearly say that
this case is an obvious example of the benefits of endovascular
surgery as a minimally invasive technique. In the future, similar References
procedures will become more and more available and will replace
open surgical interventions. It is worth noting that similar and more Biancari, F., Tauriainen, T., & Tatu, Y. (2017). Outcome of stent
useful tools are being created today, which helps to achieve better grafting for esophageal perforations: single-center experience.
results. However, it should be noted that 2 weeks after discharge, the Heckstall, R.L., & Hollander, J.E. (1998). Aortoesophageal
patient returned to the clinic by ambulance with signs of fistula: recognition and diagnosis in the emergency department.
mediastinitis, which is why he was handed over to thoracic surgeons Kieffer, E. K., Laurent, C., & Dominique, G. (2003).
and is still being treated. Aortoesophageal Fistula. Value of In Situ Aortic Allograft
Replacement.
Picichè, M., R,P.D., & A,F.A. (2003). Postoperative aortic
Acknowledgements: fistulas into the airways: etiology, pathogenesis, presentation,
diagnosis, and management. Ann Thorac Surg.
This case was acknowledged by GAAVS (Georgian Association of Vu, Q., Menias, C., & Bhalla, B. S. (RadioGraphics).
Angiologist and Vascular Surgeons). We presented this case on 19th Aortoenteric fistulas: CT features and potential mimics.
annual meeting of GAAVS. RadioGraphics, 197-209.

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