J Thorac Cardiovasc Surg 2007;133:1277-1285
© 2007 The American Association for Thoracic Surgery
Have we gone too far? Endovascular stent-graft repair of aortobronchial fistulas
Grayson H. Wheatley, III, MDa,1,*,
Anthony Nunez, MDb,
Ourania Preventza, MDa,
Venkatesh G. Ramaiah, MDa,
Julio A. Rodriguez-Lopez, MDa,
James Williams, MDa,
Dawn Olsen, PA-Ca,
Edward B. Diethrich, MDa
a Department of Cardiovascular and Endovascular Surgery, Arizona Heart Institute, Phoenix, Ariz
b HeartCare Midwest, S.C., Peoria, Ill.
Presented at the Thirty-second Annual Meeting of the Western Thoracic Surgical Association, Sun Valley Resort, Sun Valley, Idaho, June 21–24, 2006.
Received for publication July 3, 2006; revisions received October 27, 2006; accepted for publication November 6, 2006.
* Address for reprints: Grayson H. Wheatley III, MD, Arizona Heart Institute, 2632 N. 20th Street, Phoenix, AZ 85006. (Email: GWheatley{at}azheart.com).
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Abstract
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Objective: Although endovascular repair of the descending thoracic aorta has emerged as a viable treatment option, little is known about its potential to treat patients diagnosed with aortobronchial fistulas. We reviewed our comprehensive thoracic endografting experience with regard to the endovascular management and subsequent outcome of patients with aortobronchial fistulas to assess whether endoluminal graft repair is a realistic option.
Methods: Between February 2000 and November 2005, 255 patients were successfully treated with an endoluminal graft to the descending thoracic aorta. Indications for intervention included: atherosclerotic aneurysms (109/255, 42.7%), acute and chronic dissections (75/255, 29.4%), miscellaneous (34/255, 13.3%), penetrating aortic ulcers (30/255, 11.8%), and aortobronchial fistulas (7/255, 2.7%).
Results: Average patient age was 73.4 ± 10.1 years, with 4 male patients (4/7, 57.1%) and 3 female patients (3/7, 42.9%). All patients presented with hemoptysis, with 1 patient (1/7, 14.3%) requiring preoperative blood transfusion. Three patients (3/7, 42.9%) were diagnosed with atherosclerotic aneurysms, 3 patients (3/7, 42.9%) had pseudoaneurysms associated with prior open surgical repair, and 1 patient (1/7, 14.3%) had a prior endoluminal graft placed for a traumatic aortic transection. No standard postoperative antibiotic regimen was followed. There were no endoleaks, no incidences of paraplegia, and no endoluminal graft infections. Survival was 100% (7/7) at both 30 days and 1 year, and all patients are currently alive. Follow-up computed tomography was available for all 7 patients, with an average follow-up of 42.6 ± 28.5 months.
Conclusions: Endovascular management of aortobronchial fistulas appears to be safe and well tolerated, even in surgically high-risk patients, with minimal risk of prosthesis infection. Long-term surveillance and continued investigation are warranted.
Abbreviations and Acronyms ABF = aortobronchial fistula; CFA = common femoral artery; CT = computed tomography; DTA = descending thoracic aorta; ELG = endoluminal graft; IDE = investigator device exemption; TEE = transesophageal echocardiogram
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Introduction
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Dr Wheatley
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Aortobronchial fistulas (ABFs) can be associated with a number of pathologic conditions of the descending thoracic aorta (DTA), including atherosclerotic aneurysms, para-anastomotic pseudoaneurysms secondary to previous open surgical repair, and mycotic aneurysms.1-3
In addition, ABFs have been associated with invasive aspergillus infection of the lung, unilateral lung transplantation, and pulmonary tuberculosis.4-6 Usually presenting as massive and/or intermittent hemoptysis, a high index of suspicion and early diagnosis are critical. Although rare, this life-threatening disorder requires urgent intervention and treatment. The operative mortality for traditional open surgical repair of ABF using the clamp-and-sew technique reaches 20% in the modern era.7
As endovascular repair of the DTA is emerging as a viable treatment option for uncomplicated atherosclerotic aneurysms, little is known about its potential to treat patients diagnosed with ABF. Currently, only 1 thoracic endoprosthesis has gained approval by the United States Food and Drug Administration for endovascular treatment of the DTA. Use of this device is limited to atherosclerotic aneurysms; however, several case reports have been published describing endovascular stent-graft repair of ABF using several different types of thoracic endoprostheses.8-10 The results have been, in general, favorable. We reviewed our comprehensive thoracic endografting experience with regards to the endovascular management and subsequent outcome of patients with ABF to assess whether endovascular stent-graft repair is a realistic option.
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Materials and Methods
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Between February 2000 and November 2005, 255 consecutive patients with diverse pathologies of the DTA were prospectively enrolled in an Arizona Heart Hospital Institutional Review Board–approved single-site investigational device exemption clinical study. All patients were treated with TAG endoprostheses (W. L. Gore & Associates, Flagstaff, Ariz) under the investigational protocol and were fully informed and consented prior to implantation of the endoluminal graft (ELG). Enrollment in the study was limited to patients who were deemed to be at "high surgical risk," meaning that they had comorbidities and/or thoracic aortic pathologies that placed them at prohibitive risk for open repair, and, without intervention, an adverse event could be anticipated within days or weeks. During this study period, no patients were treated nonoperatively or with open surgery.
Seven patients with ABF were treated (Table 1). Mean age was 73.4 ± 10.1 years, with 4 male patients (4/7, 57.1%) and 3 female patients (4/7, 42.9%). All patients with ABF presented with hemoptysis (n = 7). Patients with ABF were diagnosed preoperatively based upon clinical findings of hemoptysis and diagnostic tests, which included chest radiography (n = 7), contrast-enhanced computed tomography (CT) angiography (n = 7), bronchoscopy (n = 5), angiography (n = 3), and transesophageal echocardiography (n = 3). All patients were hemodynamically stable at the time of their intervention.
Following discharge, patients were seen within 2 weeks to assess the incision sites and to evaluate the patient’s overall health status. CT scans, plain radiographs, and physical examinations were obtained at 1, 6, and 12 months and yearly thereafter. All significant medical events that resulted in either an unplanned increase in the level of care, permanent sequela, or death were recorded.
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Results
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The TAG endoprosthesis was successfully delivered and deployed in all 7 patients (7/7, 100%), with no open thoracic conversions. Adequate ELG placement and exclusion of the ABF was confirmed by completion angiography. Ten ELGs were deployed in 7 patients. Table 2
lists the sizes of the ELGs deployed along with the method of vascular access for sheath delivery. In 2 patients, the external iliac artery was too small to accommodate the delivery sheath; a retroperitoneal incision was performed, and a 10-mm Dacron conduit was anastomosed to the proximal common iliac artery. The conduit was then used to deliver both the sheath and the device. In 2 patients, the conduit was ligated, and in 1 patient the conduit was tunneled to the ipsilateral common femoral artery (CFA), and an iliac-CFA bypass was performed. In the other 4 patients, vascular access and sheath delivery were safely performed through the CFA. There were no iliac artery ruptures secondary to sheath delivery or removal and no complications associated with vascular access.
To better define the most proximal extent of the landing point for the ELG, the thoracic aorta has been divided into several zones. Zone 1 is the ascending aorta up to and including the origin of the innominate artery; zone 2 includes the origin of the left common carotid artery; zone 3 includes the origin of the left subclavian artery and proximal DTA; and zone 4 is the mid to distal DTA. The ELG was landed in zone 2 in 1 patient with intentional coverage of the left subclavian artery, zone 3 in 5 patients, and zone 4 in 1 patient. In the patient who required coverage of the left subclavian artery to achieve adequate proximal fixation, the subclavian artery was not coil embolized, and no carotid-subclavian bypass was performed. This patient had no signs of arm or hand ischemia and was completely asymptomatic. Neurologically, there were no cerebral vascular accidents or transient ischemic attacks associated with the procedures. There were no incidences of paraparesis or paraplegia. We elected not to utilize spinal drains in any of the patients and maintained increased spinal perfusion by maintaining a systolic blood pressure greater than 140 mm Hg for 48 hours postoperatively. We did not monitor spinal cord somatosensory evoked potentials.
Postoperatively, there were no significant episodes of arrhythmias or ST segment changes noted on telemetry monitoring. There were no episodes of chest pain or myocardial infarctions. One patient developed midback and rib pain on postoperative day 1, which resolved spontaneously by postoperative day 4. Two patients required blood transfusions with each patient receiving 1 U of packed red blood cells. The average amount of nonionic contrast used for the procedures was 336.4 ± 206.7 mL (range 120–640 mL). There were no episodes of acute renal insufficiency secondary to contrast nephropathy, and all patients were aggressively hydrated intravenously with normal saline solution. One patient had a baseline creatinine level of 2.0 mg/dL and was discharged with a creatinine level of 1.9 mg/dL. This patient received a fenoldopam intravenous drip pre- and postoperatively to assist in minimizing contrast nephropathy.
All patients received a CT angiogram of the chest, abdomen, and pelvis prior to discharge. There were no endoleaks or ELG migrations detected. All ABFs appeared to have sealed by the time of the CT angiogram, and there was stability of the aortic diameter over time (Figure 1). The hemoptysis resolved in all patients prior to discharge. All patients received intravenous doses of cefazolin perioperatively. No standard antibiotic regimen was implemented. Five patients were discharged home on oral antibiotics, and 2 patients were not discharged on any antibiotics (Table 3). There were no clinically documented pneumonias and no signs of infection in any of the patients. The average length of stay in the hospital was 4 ± 1.1 days (range 3–6 days). There were no deaths and no reinterventions at 30 days postoperatively. Average follow-up was 42.6 ± 28.5 months (range 10–75 months). All patients are currently alive (7/7, 100%), and no patient has complained of hemoptysis. There have been no episodes, observed either clinically or by CT scan, of ELG infection. There have been no device fractures or migrations and no endoleaks. There have been no reinterventions.
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Figure 1. Aortic diameter (cm) in patients with ABFs treated with an ELG preoperatively and at regularly scheduled postoperative follow-up intervals. ABF, Aortobronchial fistula; ELG, endoluminal graft.
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Only 1 patient required an additional vascular intervention. This patient underwent successful placement of an infrarenal abdominal aortic ELG for an expanding 5-cm abdominal aortic aneurysm 4 years after treatment of the ABF. Three patients have been diagnosed with new medical problems. One patient has developed bladder cancer 5 years after treatment of his ABF, and the bladder cancer is currently controlled with chemotherapy bladder irrigations. Another patient required insertion of a dual-chamber pacemaker for new sick sinus syndrome 2 years following ELG insertion and has also experienced a recent cerebral vascular accident. A third patient has developed a new 2-cm pulmonary nodule, which was detected during routine chest CT imaging for ELG surveillance, and has also developed mild renal insufficiency 3 years following ELG insertion. Three patients have had no new medical problems and are otherwise healthy.
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Discussion
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ABFs represent a challenging therapeutic and diagnostic dilemma. Presenting as either intermittent or massive hemoptysis, mortality is 100% if intervention is not pursued aggressively.11 The fistulous connection results from compression necrosis of the posterior aspect of the left tracheobronchial tree from an associated aneurysm or pseudoaneurysm of the DTA. The size of the fistulous connection determines the magnitude of the hemoptysis. If left untreated, the fistula can continue to enlarge until massive hemoptysis and possibly exsanguination occur. In addition to hemoptysis, additional signs and symptoms of ABF are dyspnea and cough, chest or back pain, pulmonary rales, or hypoxia.12 This disorder can often be confused with pulmonary embolism, pneumonia, or lung abscess, and therefore a high index of suspicion is important.
Several imaging modalities are useful in diagnosing ABF and in differentiating it from other sources of hemoptysis. Chest radiograph, chest CT with intravenous contrast, magnetic resonance imaging, and contrast aortography have all been shown to be successful in demonstrating ABF (Figures 2, 3).
2,11 In addition, bronchoscopy, intravascular ultrasound, and transesophageal echocardiogram (TEE) are useful modalities for making the diagnosis. Bronchoscopy is particularly useful in ruling out other pulmonary sources of the hemoptysis but should be pursued with caution because of the risk of dislodging any clot and potentially reactivating bleeding. In all of our patients, bronchoscopy and chest CT, radiograph, and aortogram were used to confirm the diagnosis. In addition to assisting with making the diagnosis, both intravascular ultrasound and TEE can assist with intraoperative visualization and localization of the ABF and help with identifying the appropriate location for stent-graft deployment. We believe that these two imaging modalities are critical for identifying suitable proximal and distal landing zones for the ELG and ensuring that the ABF is excluded.
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Figure 2. Chest CT demonstrating an ABF (white arrow) from the DTA to the left main stem bronchus. CT, Computed tomographic scan; ABF, aortobronchial fistula.
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Figure 3. Diagnostic angiogram demonstrating an ABF (white arrow) from the DTA to the left main stem bronchus at the site of a pseudoaneurysm. ABF, Aortobronchial fistula; DTA, descending thoracic aorta.
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Most patients with ABF have complex aortic pathologic conditions, such as para-anastomotic pseudoaneurysms or sizeable atherosclerotic aneurysms of the DTA, which make open surgical repair a less than straightforward procedure. In addition to the technical challenges of performing a redo left thoracotomy or dealing with a complex DTA aneurysm, the potential exists for prosthetic graft infection. A recent 19-year review of cases of ABF treated with open surgical repair revealed an operative mortality of 20% with no associated embolic strokes or paraplegias.7 In addition, follow-up with a median of 2.5 years showed no deaths.7 Despite improved results with open surgical repair, an opportunity exists to potentially incorporate less invasive technologies, such as endovascular stent-graft repair, into the treatment paradigm for ABF (Figure 4).
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Figure 4. Chest CT demonstrating successful exclusion of an ABF. CT, Computed tomographic scan; ABF, aortobronchial fistula.
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A review of the literature shows a total of 36 cases of ABF treated with an ELG (Table 4
1,2,4,9,13-28). A meta-analysis demonstrates a cumulative 30-day mortality of 8.3%, with most of the cases reporting successful 1-year survival. In our cohort of 7 patients, we were able to successfully treat the ABF with an ELG in all patients. Although all patients presented with hemoptysis, only 2 patients required a blood transfusion for a hemoglobin value <10 g/dL. The relatively low transfusion rate in our patients is attributed to early recognition and intervention, which allowed us to repair the ABF prior to catastrophic hemorrhage and prevent additional blood loss. Survival was 100% at both 30 days and 1 year. All patients are currently alive up to 75 months postprocedure and have required no reinterventions. Only 1 patient has required a subsequent vascular intervention. This patient required ELG repair for an expanding abdominal aortic aneurysm.
We did not utilize spinal cord protection with a spinal drain in any of the patients in this study. In addition, we do not utilize somatosensory evoked potential monitoring for our thoracic endografting procedures. There were no perioperative or postoperative neurologic complications, such as paraplegia or paraparesis. We use a spinal drain selectively for neurologically high-risk patients, and we place the drain preoperatively in patients with a prior abdominal aortic aneurysm repair (open or endovascular) and/or manipulation of the DTA below the level of T9. In addition, if any patient develops postoperative paraplegia or paraparesis, we will immediately place a spinal drain. Following ELG repair, we help prevent spinal cord ischemia by actively increasing the systolic blood pressure to between 140 and 160 mm Hg for 48 hours postoperatively. The subclavian artery is also an important contributor to spinal cord perfusion and collaterals; however, it can frequently be covered without neurologic or vascular compromise.28 We intentionally covered the left subclavian artery in 1 patient in this study to achieve adequate 2-cm proximal fixation of the ELG. We did not perform a preoperative carotid-subclavian bypass in this patient, and there were no neurologic sequelae. In addition, there was no retrograde type II endoleak from the left subclavian artery, and the patient had no signs of vascular insufficiency of the left arm. We have found that it is safe to manage these patients expectantly, and if left arm claudication develops, then we perform an elective carotid-subclavian bypass.
Vascular access is an important consideration for thoracic endografting procedures. Introduction and removal of large-bore delivery sheaths in small, calcified or tortuous access vessels can be associated with rupture of the iliac artery and subsequent patient mortality. It is critical to assess the quality and size of both external and external iliac arteries in patients who are candidates for endovascular repair to see if the arteries will accommodate the large-bore delivery sheath before the patient can be offered endovascular repair. If the access vessels will not accept the large-bore delivery sheath, then a 10-mm retroperitoneal conduit must be sewn to the distal aorta or proximal common iliac artery to make the vascular access portion of the procedure safe. In this study, 3 patients (3/7, 42.9%) required a retroperitoneal conduit to safely deliver the sheath. This rate is significantly higher than most series for thoracic endografting. Commonly, 15% of patients who are treated with a thoracic ELG require a retroperitoneal conduit.29 It is unclear why our series has a higher rate of retroperitoneal conduits, but 6 of the 7 patients had aggressive atherosclerotic-related disease as their primary aortic pathology (either prior open surgical repair of an aneurysm or primary disease) and thus had associated iliofemoral disease. The absence of mortalities in this study is directly attributed, in part, to the thoroughness of the preoperative planning for vascular access and appropriate use of retroperitoneal conduits.
Prosthetic graft infection is a significant concern following repair of ABF. In open surgical repair, it is recommended that the prosthetic graft and fresh suture line be protected from the repaired bronchial fistula by either wrapping the graft with aneurysmal wall or interposing viable tissue such as pleura, intercostal muscle pedicle flap, omentum, or pericardial fat pad.7,11 Antibiotic irrigation is recommended intraoperatively, and the prosthetic graft can even be soaked in antibiotic solution prior to implantation.7 ELG repair of ABF does not allow for similar maneuvers to protect against endoprosthesis infection. In this study, there were no endoprosthesis infections. It is unclear as to why the infection rate for endovascular repair is minimal, but the ELG remains in the center of the aneurysm sac well away from the actual fistula and source of contamination. Perhaps because there is minimal tissue trauma associated with the deployment of the ELG, as opposed to open surgical repair, the excluded aneurysmal or pseudoaneurysmal cavity is much less likely to become contamination or infected. Additionally, the excluded aneurysmal wall and cavity may still be vascularized and remain viable, to some degree, after endovascular repair from fine collaterals branches that are not disrupted with deployment of the ELG. All patients received 3 intravenous doses of perioperative cephalosporin antibiotics. Subsequently, no routine postoperative antibiotic regimen was followed. In fact, 2 patients received no postoperative antibiotics and were discharged home without any oral antibiotics. In this limited cohort, there does not appear to be an association between postoperative antibiotic administration and freedom from graft infection. Additional studies are needed before definitive recommendations for antibiotic administration can be generated. In the meantime, we feel it is appropriate to discharge the patients with 1 week of outpatient oral antibiotics.
This study has several limitations. First, it is a small series and has associated limitations for making broader generalizations. However, the results are promising for an endovascular approach to ABF when taken in the context of the additional 36 patients reported in the world’s literature. Second, this is a retrospective review and a subset of patients taken from a larger series of thoracic endografting patients from a single center. To better understand the risks and benefits of an endovascular approach, a prospective, randomized study is needed. However, it is unlikely that such a study can be conducted due to the rarity of ABF and the limited number of cases. Most likely, any conclusions that will be drawn will be based upon a meta-analysis of the literature and from large-scale multicenter national research trials for endovascular devices in general. Finally, although the follow-up for patients with ABF treated with an ELG extends up to 6.25 years in this study, the mean follow-up is 42.6 ± 28.5 months. Currently, all patients are alive, and continued surveillance of these patients is critical. There is potential for recurrence of the fistula, as 1 of the patients developed an ABF 6 years following placement of a thoracic ELG. It will be important to assess any late graft infections, device failures, or migrations or adverse patient outcomes.
In conclusion, this study demonstrates that it is possible to treat patients with ABF using an endovascular approach and that the results compare favorably with open surgical repair. Patients tolerated the procedure well, and hemoptysis resolved in all 7 patients. There were no deaths, no episodes of paraplegia, and no endoprosthesis infections. All patients are still alive and there have been no reinterventions. Careful preoperative planning is essential for optimal results when patients are being considered for endovascular repair, especially with regards to vascular access considerations. In our cohort, duration and type of postoperative antibiotics does not appear to correlate with freedom from graft infection. Although endovascular technologies are emerging for the treatment of many types of aortic pathologies, significant questions relating to this therapeutic modality are yet to be resolved including long-term device durability, improved long-term patient outcomes, and economic concerns of increased device cost and expenses related to long-term graft surveillance with CT. Additional investigation and continued graft surveillance are critical to help determine the future role of endovascular technologies.
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Footnotes
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1 Grayson H. Wheatley III reports consulting fees from W.L. Gore and associates. 
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References
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Abstract
Introduction
