Cas clinique

DOI of or1Vascular

Hospital, Turi2Departme

Turin, Italie.

CorrespondSurgery Unit,Turin, Italie, E

Ann Vasc Surhttp://dx.doi.or� Annals of V�Edit�e par ELS

Traitement en urgence d’une fistuleaorto-esophagienne et d’une fistuletrach�eo-esophagienne par endoproth�eseaortique thoracique et endoproth�eseoesophagienne : Un cas mal diagnostiqu�een tant que cancer oesophagien

Emanuele Ferrero,1 Andrea Viazzo,1 Michelangelo Ferri,1 Rodolfo Rocca,2 A. Pecchio,1

Salvatore Piazza,1 Pia Cumbo,1 Giuseppe Berardi,1 Franco Nessi,1 Turin, Italie

Les fistules aorto-esophagiennes sont rares mais mortelles si non trait�ees. La chirurgie thora-cique ouverte est associ�e �a une mortalit�e et �a une morbidit�e op�eratoires �elev�ees. Nous rap-portons le cas d’un homme de 77 ans qui, trait�e par r�eparation aortique endovasculairethoracique (TEVAR) pour an�evrysme thoracique descendant dans un autre centre, apr�es un�episode aigu d’h�emat�em�ese et de moelena avait �et�e transfer�e dans notre centre. Le scannercorps entier montrait la re-perfusion du sac an�evrysmal thoracique descendant (8,8 cm dediam�etre) au niveau de la zone proximale et distale d’ancrage de TEVAR (endofuite de type I)sans signes clairs de fistule avec la lumi�ere oesophagienne. Le malade a eu un nouveau TEVAR�a travers l’implantation pr�ec�edente avec ancrage proximal tr�es proche de l’art�ere sous-clavi�eregauche et ancrage distal juste au-dessus du tronc coeliaque. En raison de la pr�esence d’unefistule trach�eo-esophagienne, une endoproth�ese oesophagienne a �et�e implant�ee peu apr�es, etune j�ejunostomie a �et�e faite. �A 30 jours, le malade �etait en bonne �etat g�en�eral, mais il est mort �a3 mois. Les fistules aorto-oesophagiennes sont rares et habituellement mortelles ; l’identificationpr�ecoce et le traitement par TEVAR empechent l’exsanguination imm�ediate des malades, maisapr�es d�eploiement de l’endoproth�ese, la plupart des malades sont �a risque de complicationsinfectieuses. L’arret du saignement et la restauration de la circulation sont l’urgence primordiale,mais les complications infectieuses et la r�eparation oesophagienne demeurent des probl�emesnon r�esolus.

iginal article: 10.1016/j.avsg.2011.06.009.

and Endovascular Surgery Unit, Mauriziano Umberto In, Italie.

nt of Gastroenterology, Mauriziano Umberto I Hospital,

ance : Emanuele Ferrero, Vascular and EndovascularMauriziano Umberto I Hospital, Largo Turati 62, 10128-mail: emaferrero@libero.it

g 2011; 25: 1142.e1-1142.e5g/10.1016/j.acvfr.2013.02.011ascular Surgery Inc.EVIER MASSON SAS

Aortoesophageal fistula (AEF) is rare but fatal

if untreated; it results from an abnormal communi-

cation between the aorta and the esophagus

and constitutes less than 10% of all aortoenteric

communications; the clinical manifestation is gas-

trointestinal hemorrhage, melena, and hemateme-

sis. Generally, it is caused by thoracic aortic

aneurysms, foreign body ingestion, esophageal mali-

gnancy and traumatic aorticwounds, ruptured pene-

trating aortic ulcers, esophageal or bronchogenic

malignancies, and thoracic surgery.1,2 The thoracic

endovascular aortic repair (TEVAR) has become a

therapeutic alternative for high-risk patients with

1216.e1

Fig. 1. (A) Computed tomography (CT) scan showed a large ATA with proximal and distal type I leak. (B) CT scan

showed sealing of the aneurysm after new thoracic endovascular aortic repair inside previous implantation.

1216.e2 Cas cliniques Annales de chirurgie vasculaire

thoracic aortic disease, but can present AEF as late

complication. In literature, secondary AEFs are

reported as a sequel of TEVAR.3-8 Generally, in these

cases, surgical treatment is to perform an aortic graft

followed by reconstruction of the esophagus, but it

has high rate ofmorbidity andmortality in this group

of patients, so endovascular repair could be a the-

rapeutic option9,10 even if reports of antemortem

diagnosis and successful salvage of patients with

aortoesophageal fistula are rare.11

Fig. 2. Postoperative CT scan showed presence of air

bubbles in the mediastinum.

CASE REPORT

A 77-year-old man was brought to our center because of

multiple episodes of hematemesis and melena. Initially,

in another hospital, the presence of esophageal cancer

was suspected, but the results of biopsy performed were

negative, and after a thoracic and abdominal computed

tomography (CT) scan, a suspicion of an aortoesophageal

fistula was placed. He had a history of atrial fibrillation

under oral anticoagulant therapy, diabetes mellitus, dila-

ted cardiomyopathy, macrocytic anemia and thrombocy-

topenia from myelodysplasia, chronic cerebral vascular

disease and previous ischemic stroke, hyperthyroidism,

mild chronic renal insufficiency, previous exclusion of

thoracic aneurysm by TEVAR (in another center: Valiant

thoracic stentegraft, 32-36 � 150 mm; Medtronic, MN).

The patient underwent new CT scan that showed the pre-

sence of reperfusion of the thoracoabdominal aneurysmal

sac (8.8 cm in diameter) in the proximal and distal TEVAR

landing zone (endoleak type Ie Fig. 1A) without clear

signs of fistulous tract with the esophageal lumen. An

Fig. 3. (A) Barium radiography reported a bronchial-esophageal fistula. (B) Bronchial-esophageal fistula excluded by

esophageal endoprosthesis.

Vol. 25, No. 8, 2011 Cas cliniques 1216.e3

esophagogastroduodenoscopy was performed, and it

showed, at 30 cm from dental arch, a foramen (15 mm in

diameter) with regular margins; the bottom of the lesion

was covered by granulation tissue and blood clots; no

bleedingwas observed, and the lesion appeared suggestive

not of malignancy but of esophageal fistula. Because of

persistent intermittent hematemesis and the need for

blood transfusion, patient underwent new TEVAR treat-

ment (proximally two Relay 28-200 and 28-150 mm:

Bolton Medical, FL; distally Endurant 32-49 mm: Med-

tronic, MN), inside previous implantation, with proximal

landing very close to left subclavian artery and distal

landing just above celiac trunk. The CT scan performed on

fourth postoperative day showed perfect sealing of the

aneurysm (Fig. 1B), but presence of air bubbles in the

mediastinum (Fig. 2), and a bronchial-esophageal fistula

was detected. A barium X-ray examination of the eso-

phagus was performed; it confirmed the presence of the

bronchial-esophageal fistula (Fig. 3A). Due to the poor

general condition of the patient and high operative risk in

executing an esophageal reconstruction, we decided to

exclude the bronchial-esophageal fistula and position

an esophageal endoprosthesis (Fig. 3B) associated with

jejunostomy. The patient underwent enteral nutrition via

a needle catheter jejunostomy; even though the patient

did not present fever or leukocytosis due to air collection,

detected onCT, we started an aggressive antibiotic therapy

(tigecycline, ertapenem, teicoplanin). At 30 days’ follow-

up, the patient was alive in fairly general condition; no

spinal cord and visceral ischemia was present. After 3

months, the patient died suddenly without a specific

cause, probably due to infection.

DISCUSSION AND CONCLUSION

AEF is a rare cause of massive gastrointestinal bleed-

ing and is usually a fatal condition. In the English

literature, a relatively small number of TEVARs for

AEF/ABF are published to date, consisting primarily

of single case reports or small case series with less

than 12 patients,12 and this may account for a

considerable underestimation of the problem. The

most common cause of fistulization is the erosion of

a thoracic aortic aneurysm,13 probably due to the

compression of the esophagus or the airways by the

aneurysm that may result in a local inflammatory

response with formation of stable adhesions and

1216.e4 Cas cliniques Annales de chirurgie vasculaire

tissue necrosis leading to erosion and final fistuli-

zation. While, in ruptured aortic aneurysm, the

extravasation of blood and periaortic hematoma,

with increased local inflammatory response and

compression of surrounding organs, may play a role

in late fistulization.14 Chiesa15 in his report consi-

dered the excessive TEVAR stent-graft oversizing

as possible additional mechanism of AEF fistuliza-

tion: He found that 42% of patients presenting late

AEF/aortobronchial fistulae had a proximal over-

sizing of 20% or more; this probably caused a

deterioration of the arterial wall, the stent-graft

migration, and the aortic aneurysm enlargement.

Generally, common presenting symptoms of AEF

are characteristic Chiari triad features, including

chest pain and sentinel hematemesis of red blood

followed at a variable interval of time by rapidly fatal

massive exsanguinating hematemesis.11 In a few

cases, AEF can be suspected on the basis of isolated

sepsis or septic embolism in a lower extremity16;

other suggestive findings include dysphagia and/or

chest pain and history of surgical treatment

involving the thoracic aorta. The CT scan is the first

imaging study performed in most of the cases, but

rarely detects fistulous tracts. Endoscopy is the most

sensitive and specific method for the diagnosis of

AEF, but it often requires sedation and entails the

risk of dislodging clots during the progress of the

endoscope, which can cause fatal bleeding. Early

recognition, a high index of suspicion, and impro-

ved critical care and emergency services have lead to

an increasing number of cases being recognized

before fatal hemorrhage. Mortality after surgery for

thoracic aortic fistulae reaches 61% in cases of pri-

mary etiology and 78% in cases of secondary fistu-

lae.17,18 Endoluminal repair of thoracic aortic

disease requiring emergent or urgent treatment has

yielded encouraging early results with low morbi-

dity and mortality rates compared with open sur-

gery.19 TEVAR prevents immediate exsanguination

in patients with AEF, but after deployment of the

endograft, most patients are at risk for infectious

complications and death. Cessation of bleeding and

restoration of circulation are of paramount urgency,

whereas esophageal reconstruction could be carried

out later, even if this did not guarantee the survival

of the patient. In the overtly moribund patient,

TEVAR has been proposed as the most appropriate

definitive strategy, as a palliative procedure, whe-

reas in good surgical candidates, coverage of the

aortic lesion, along with an aggressive antibiotic

therapy, may be used to achieve an improvement in

the patient’s general conditions, serving as a

‘‘bridge’’ to open surgical treatment of the aortic

and/or esophageal/ bronchial defect. Following

successful TEVAR, in absence of clear signs of rein-

fection or bleeding, there is no general consensus

concerning the need of staged surgical intervention.

Although clear limitations of the TEVAR strategy

alone include the risk of reinfection and septic

complication, due to the inability to debride or drain

the mediastinum, some success with this approach

has been reported in the literature.20,21 We think

that TEVAR alone does not provide complete and

durable cure for AEF, that these patients need to be

followed closely to evaluate the opportunity and the

appropriate timing for a secondary surgical pro-

cedure according to patient conditions and the grade

of sepsis, but in patients at high risk for complica-

tions with open surgical repair, we consider TEVAR

as a potential definitive treatment. In conclusion,

AEF, although rare and probably underestimated, is

considered a fatal condition. AEF sometimes is a late

sequelae of TEVAR. Surgical and endovascular

treatments are associated with high mortality, but

conservative treatment is not a viable option

because AEF is inevitably fatal if untreated; in these

cases, TEVAR has a predominant role in controlling

the massive hemorrhage associated with AEF, but

infectious diseases and esophageal repair remains an

open problem.

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