Accepted Manuscript Aortobronchial Fistula after Thoracic Endovascular Aortic Repair (TEVAR) for Descending Thoracic Aortic Aneurysm John Nicholas Melvan, Jacob DeLaRosa, Julio C. Vasquez PII:
S0890-5096(17)30332-1
DOI:
10.1016/j.avsg.2016.10.040
Reference:
AVSG 3178
To appear in:
Annals of Vascular Surgery
Received Date: 5 May 2016 Revised Date:
7 September 2016
Accepted Date: 13 October 2016
Please cite this article as: Melvan JN, DeLaRosa J, Vasquez JC, Aortobronchial Fistula after Thoracic Endovascular Aortic Repair (TEVAR) for Descending Thoracic Aortic Aneurysm, Annals of Vascular Surgery (2017), doi: 10.1016/j.avsg.2016.10.040. This is a PDF file of an unedited manuscript that has been accepted for publication. As a service to our customers we are providing this early version of the manuscript. The manuscript will undergo copyediting, typesetting, and review of the resulting proof before it is published in its final form. Please note that during the production process errors may be discovered which could affect the content, and all legal disclaimers that apply to the journal pertain.
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Aortobronchial Fistula after Thoracic Endovascular Aortic Repair (TEVAR) for Descending Thoracic
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Aortic Aneurysm
3 John Nicholas Melvan1, Jacob DeLaRosa1, 2 and Julio C. Vasquez2
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Division of Cardiothoracic Surgery, Emory University, Atlanta, GA 1, Portneuf Medical Center,
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Pocatello, ID2
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Conflicts of Interest: No funding sources or conflicts of interests to disclose.
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Acknowledgements: None.
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Journal: Annals of Vascular Surgery.
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Consent: Consent has been obtained for publication.
Correspondence:
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Julio C. Vasquez, M.D.
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777 Hospital Way, Ste G-11
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Pocatello, ID 83201
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Phone: (208) 239-2580
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Fax: (208) 239-2589
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Email:
[email protected]
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ABSTRACT Continued enlargement of the aneurysm sac after thoracic endovascular aortic repair (TEVAR) is a known risk after endovascular treatment of thoracic aortic aneurysms. For this reason, periodic
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outpatient follow up is required to identify situations that require repair. Here we describe an
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aortobronchial fistula (ABF) in a patient lost to follow up, that presented 3 years after an elective TEVAR
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done for a primary, descending thoracic aortic aneurysm (dTAA). Our patient arrived in extremis and
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suffered massive hemoptysis leading to her demise. CT angiogram near the time of her death
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demonstrated a bleeding ABF immediately distal to her previous TEVAR repair. Aortic aneurysmal
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disease remains life threatening even after repair. Improved endovascular techniques and devices have
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resulted in decreased need for reintervention. However, this case demonstrates the risk of thoracic aortic
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disease progression and highlights the importance of establishing consistent, long term follow up after
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TEVAR.
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CASE REPORT An 84 year old, hypertensive woman with a history of breast cancer, spine fracture, and chronic back pain was initially evaluated in our office for a primary, 4.7 cm descending thoracic aortic aneurysm
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(dTAA) and treated conservatively. During the following year, she was hospitalized twice for acute on
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chronic back pain that prompted axial imaging to rule out thoracic aortic dissection or rupture. Imaging
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studies demonstrated more than a 1 cm diameter increase in the size of her aneurysm over 12 months.
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Due to the rapid growth of the aneurysm and complicated clinical picture, we recommended she undergo
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an elective, percutaneous thoracic endovascular aortic repair (TEVAR) of her dTAA. We placed
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overlapping Medtronic Talent Captiva endografts covering 18 cm of her descending thoracic aorta. The
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proximal endograft (34x34x160 mm) overlapped with the distal endograft (38x34x112 mm) for
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approximately 9 cm in length (Figure 1A). The proximal landing zone of the native aorta had a diameter
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of 30 mm (40 mm long) and a distal landing zone diameter of 30 mm (37 mm long). The left subclavian
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artery was not covered but several bronchial and intercostal vessels were covered during repair.
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Following an uncomplicated postoperative course, the patient was discharged home and followed
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up in our clinic 2 weeks after her procedure. Our standard TEVAR follow up includes a 2 week post
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procedure office visit, followed by CT angiogram of the thoracic and abdominal aorta at 1 month, 6
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months, and 12 months post procedure. Imaging is performed during yearly follow up visits thereafter.
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After the first postoperative visit the patient did not return phone calls and letters indicating the need to
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return to clinic. The main barrier to her care was poor compliance. Unfortunately, after being lost to
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follow up, she presented 3 years later with massive hemoptysis and profound hypotension. She was
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immediately intubated in our emergency department with prompt return of 500cc bright red blood through
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her endotracheal tube. CT angiogram near the time of her death demonstrated a bleeding aortobronchial
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fistula (ABF) originating distal to her TEVAR repair (Figure 1B and 1C). The patient expired soon after
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presentation as she had an established DNR order and her family requested that aggressive resuscitation
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efforts be withheld. No information is available concerning the interval growth of her dTAA after repair.
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At the time of rupture, the seal of her distal landing zone had been lost and its diameter had grown from
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30 mm at the time of TEVAR repair to 44 mm at the time of her death. This represented a type 1b
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endoleak from the distal edge of her endograft that resulted from interval growth of her aneurysmal
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disease, as the graft was properly sized at the initial intervention. There was no evidence of graft infection
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at the time of her death. Her caretakers did not recall any exacerbation of her chronic back pain and she
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had no previous episodes of hemoptysis. Unfortunately, she had not been evaluated by any other
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physicians over the interval period. Had the patient been clinically stable for repair, we would have
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attempted another endograft stent placement to cover the area of active bleeding, which had resulted in an
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ABF.
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ABF can result from thoracic aneurysm rupture directly into the bronchial tree due to adhesions
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that form between the aorta and airways. Hemoptysis is often the initial presentation of ABF and is
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associated with extremely high mortality. Endograft placement for dTAA results in 96% freedom from
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aneurysm-related death, but up to 14% of patients may need repeat intervention at 5 years for various
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reasons, including progression of aneurysm dilatation beyond the stented aorta [1]. Unfortunately, our
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patient developed aneurysmal progression after TEVAR and arrived in extremis with no time to
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intervene. This case emphasizes the importance of close patient follow up and repeat imaging after
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TEVAR.
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DISCUSSION Repair of thoracic aortic aneurysm is advocated when the diameter of the aneurysm is equal to or greater than 5.5 cm [2]. An endovascular approach is recommended over open surgery when feasible [2,
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3]. Endovascular techniques can result in complications including stroke, paraplegia, visceral ischemia,
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access complications, device migration, endoleaks, or post implantation syndrome [4]. Multiple
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commercially available endografts for TEVAR repair are available including Zenith (Cook Inc.,
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Bloomington, IN), Valiant (Medtronic Corp., Santa Rosa, CA), Conformable GORE TAG (W.L. Gore
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and Associates Inc., Flagstaff, AZ), and Relay Endografts (Bolton Medical Inc., Sunrise, FL) [5].
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Following the use of these endografts in the treatment of thoracic aortic aneurysms, one European registry
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review estimated an annual reintervention rate of 4.6% or 14% risk at 4 years for management of
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endoleaks and progression of aneurysmal disease [5].
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Several series of TEVAR patients have been published that include 5 year outcomes and
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prevalence of reintervention [6-8]. Geisbush et al published their single center experience of 264 TEVAR
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patients, which showed an overall reintervention rate of 22%. Indications for reintervention were led by
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endoleaks (44%) and progression of aortic disease (29%). Risk factors included chronic expanding aortic
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dissections (OR 2.35), hybrid aortic procedures (OR 2.11), and connective tissue disease (OR 7.54) [7].
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This same group reported 5 year mortality of 50% and freedom from complication of 52%, 47.2%, and
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47.2% at 1, 3, and 5 years respectively [6]. A meta-analysis of 17 studies including 567 patients, reported
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considerable variability (range 0-60%) in rates of reintervention after TEVAR for type B aortic
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dissection. Causes for reintervention included endoleaks (8.1%) and progression of aneurysmal disease
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(7.8%). Rare complications included retrograde type A dissection (0.67%), aortoesophageal fistula
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(0.22%), paraplegia (0.45%), and stroke (1.5%) [8]. Based on our imaging studies, we have concluded
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that this patient developed a type 1b endoleak after progression of her aneurysmal disease that lead to
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fistulization into the airway distal to her previous TEVAR repair.
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Different rates of reintervention are reported depending on the specific type of endoleak.
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Preventza et al reported on 249 patients undergoing dTAA repair with TEVAR, and found that endoleaks 5
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occurred in 38 patients: type 1a (n = 13, 34.2%), type 1b (n = 15, 39.5%), type 2 (n = 8, 21.1%), and type
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3 (n = 2, 5.3%) respectively. Of these, 12 patients required reintervention; type 1a (n = 8, 66.7%), type 1b
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(n = 2, 16.7%), combined type 1 and 3 (n = 1, 8.3%), and type 3 (n = 1, 8.3%) [9]. Szeto et al reviewed
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680 patients undergoing TEVAR at the University of Pennsylvania and found that endoleaks (45/80)
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accounted for the greatest majority of their cases requiring reintervention (11.7%, 80/680); type 1 (24/45),
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type 2 (5/45), type 3 (9/45), and multiple/unclear (7/45). Other lesser causes of endograft failure in their
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series included proximal aortic events (14%), distal aortic events (18%), multiple failure modes (5%),
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endograft infection (4%), and carotid occlusion/stent collapse (3%). The median time for reintervention
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was 210 days [10]. Boufi et al have found that short proximal aortic neck length may be the only
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independent risk factor for the development of a secondary type I endoleak, not clinical urgency or aortic
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length covered. Using receiver operating characteristics, Boufi and colleagues have advocated for ≥ 24
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mm landing zone [11]. The proximal landing zone in our case was 30 mm.
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Our patient originally underwent TEVAR for a primary dTAA using a Talent (Medtronic) thoracic stent graft with Captiva delivery system. This device was first launched in 2005 and received
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FDA approval in 2008. Outcomes from the use of these endografts are available for at least 7 separate
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clinical trials [12]. Following the development of second generation Valiant endografts, Talent and
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Valiant endografts were compared head-to-head. Effectiveness end points only differed in that
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reinterventions due to endoleak after > 30 days were significantly greater for Talent versus Valiant
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endografts (6.5% v 0%). Loss of graft patency, stent graft migration at 1 month, and endoleak at 12
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months were not different between devices [13]. Valiant endografts were not available for our patient at
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the time of her procedure.
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ABF results from the erosion of an aneurysm into the bronchial tree or pulmonary parenchyma
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that is adjacent to the aneurysm. Previous placement of a prosthetic graft and severe atherosclerosis have
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been associated with this condition [14]. Little evidence however is available concerning the incidence of
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ABF after TEVAR [15-18]. Piceche et al studied all available case reports and reviews for postoperative
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aortic fistulas into the airways after cardiothoracic surgery. Uniformly fatal if untreated, surgical mortality 6
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rates were 15% for open repair and 6.8% for endovascular repair [17]. Hemoptysis was the first, and often
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the only, clinical finding of ABF. These authors found that aortic fistulas into the airways were more
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common after dTAA procedures compared to other fistula after cardiac and thoracic surgery [17]. Using a
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nationwide survey of 17 centers in Italy, Chiesa et al reported a 1.7% (19/1113) incidence of
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aortoesophageal and aortobronchial fistula after TEVAR. Higher rates of fistula formation in this survey
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were associated with aortic pseudoaneurysms, complex operations, and emergent procedures. Overall
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survival was a dismal 16% [15]. Similarly, Czerny and colleagues reviewed the European Registry of
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Endovascular Aortic Repair Complications, comprised of 4680 TEVAR patients at 14 European centers,
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and found the incidence of aortobronchial or aortopulmonary fisulta after TEVAR was 0.56%. Leading
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mechanisms of fistula formation were external compression of the bronchial tree, endoleak formation, and
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further aortic ischemia [16]. Therefore, ABF after TEVAR are rare, but carry a high mortality rate.
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In conclusion, endovascular treatment of aortic aneurysm is a reliable therapy for the management of this condition. However, like any other therapeutic intervention, it can result in
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complications. Aneurysmal progression and aortic fistulization can occur and result in fatal outcomes.
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TEVAR patients must be educated that aortic aneurysm is a dynamically evolving disease process that
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requires close observation and periodic imaging. Common intervals for follow up are 1 month, 6 months,
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and then yearly. With aggressive outpatient management, the catastrophic outcome experienced by our
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patient can hopefully be avoided.
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FIGURE LEGEND
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Figure 1: CT angiogram taken (A) intraoperatively and (B, C) 3 years postoperatively during rupture of
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the aortrobronchial fistula. Aortic aneurysm distal to the TEVAR stent and filling of the aortobronchial
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fistula are shown.
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