Ann Thorac Surg 2006;82:81-89
© 2006 The Society of Thoracic Surgeons
Original article: Cardiovascular
Thoracic Aorta False Aneurysm: What Surgical Strategy Should Be Recommended?
Mauricio A. Villavicencio, MD,
Thomas A. Orszulak, MD
*
,
Thoralf M. Sundt, III, MD,
Richard C. Daly, MD,
Joseph A. Dearani, MD,
Christopher G.A. McGregor, MB, FRCS,
Charles J. Mullany, MB, MS,
Francisco J. Puga, MD,
Kenton J. Zehr, MD,
Hartzell V. Schaff, MD
Division of Cardiovascular Surgery, Mayo Clinic, Rochester, Minnesota
Accepted for publication February 27, 2006.
* Address correspondence to Dr Orszulak, Mayo Clinic, 200 First St SW, Rochester, MN 55905 (Email: orszulak.thomas{at}mayo.edu).
Presented at the Fifty-second Annual Meeting of the Southern Thoracic Surgical Association, Orlando, FL, Nov 10–12, 2005.
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Abstract
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BACKGROUND: Thoracic aorta false aneurysms (TAFA) are a surgical challenge. The best technical approach remains uncertain.
METHODS: Between 1981 and 2004, 57 patients underwent operation for TAFA (mean age 57 ± 18 years; 43 [75%] were male). Symptoms included dyspnea 25 (44%), chest pain 22 (39%), and fever 18 (32%). Twelve (21%) were asymptomatic. Thirty-seven (65%) had undergone previous operation with a mean interval between operations of 80 ± 90 months. Fifteen (26%) had a mycotic etiology. The TAFA involved the aortic root in 10 (18%), ascending aorta in 28 (49%), arch in 6 (11%), and descending aorta in 13 (32%). Twenty-one (37%) required femorofemoral cannulation and 28 (49%), circulatory arrest. Surgical techniques included graft replacement in 27 (47%), composite root in 10 (18%), patch repair in 10 (18%), and direct suture in 10 (18%).
RESULTS: Operative mortality was 7% (4 patients). Four of 32 (13%) had massive hemorrhage during redo sternotomy, and all of these had planned extramediastinal cannulation (all survived). Follow-up was 100% for 349 patient-years. Actuarial survival was 77% ± 6%, 63% ± 8%, and freedom from recurrent TAFA was 87% ± 5% and 83% ± 7%, at 5 and 10 years, respectively. Univariate analysis identified TAFA greater than 55 mm, urgent operation, and NewYork Heart Association functional class III or IV as predictors of hemorrhage during redo sternotomy. Obesity and ejection fraction of 35% or less were predictors of operative death.
CONCLUSIONS: Thoracic aorta false aneurysm symptoms may be minimal, and consequently a high degree of suspicion plus serial imaging is warranted. Extramediastinal cannulation, deep hypothermia, and circulatory arrest are required for large mediastinal TAFA. Despite serious risks, TAFA correction is possible with good long-term results.
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Introduction
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Thoracic aorta false aneurysms (TAFA) have been reported as a rare but life-threatening complication of aortic surgery, infection, or trauma [1–3]. Nevertheless, there is controversy in the literature about the incidence of this problem. Clinical follow-up has shown a low incidence of TAFA even in high-risk aortic dissection patients [4, 5]. However, systematic magnetic resonance imaging (MRI) after aortic surgery for aneurysms or dissection has demonstrated approximately 15% risk of TAFA development [6–8]. These data highlight the increasing importance of this pathology in referral-based cardiovascular practice.
The clinical presentation, etiologies, diagnostic methods, and surgical approach have been described only in small series, and despite technical advances, mortality remains high. Furthermore, TAFA recurrence in the presence of infection or abnormal tissues is a generalized concern. Consequently, the best surgical strategy to avoid perioperative death and recurrence remains uncertain.
Therefore, we sought to review the clinical presentation, surgical techniques, and long-term results of TAFA patients operated on at our institution in an effort to determine the best approach and to identify the risk factors of mortality, recurrence, and major complications.
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Patients and Methods
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This research project was approved by the Mayo Clinic Foundation Institutional Review Board on July 6, 2004. After approval, we retrospectively reviewed the cardiovascular surgery division database and the radiology division computed tomography (CT) database at the Mayo Clinic, Rochester, Minnesota, to identify patients coming to operation for false aneurysms of the root, ascending aorta, arch, and descending aorta. Thoracic aorta false aneurysm was defined as a pathologic dilatation of the aorta in which the normal arterial layers were missing partially or totally. Only patients with free blood flow inside the TAFA were considered. Graft wrapping was described as such if the aortic wall was wrapped around the prosthetic graft as in the classical Bentall fashion.
All Mayo Clinic patients are asked by regular mail to give consent for clinical record review for research. Patients who have not authorized are deleted from the databases given to the investigators. No patients in this study refused consent to review clinical records. In a second phase, Institutional Review Board–approved questionnaires and HIPPA (Health Insurance Portability and Accountability Act) forms were sent to the patients included in the study by regular mail for follow-up. A maximum of three mailings were sent in case of no response. Only returned questionnaires with patient-approved HIPPA forms were considered. Follow-up with questionnaires and death certificates was performed between July 2004 and January 2005. Death of unknown cause was included as cardiac death to estimate the survival free of cardiac death. Computed tomography and MRI utilized during the follow-up were not reviewed.
Patients who were thought to have risk of bleeding during redo sternotomy were cannulated through the femoral artery–femoral vein and cooled down to 18°C. Left ventricular venting through a small left thoracotomy before the sternotomy was performed in the case of moderate/severe aortic regurgitation or left ventricular distension in the transesophageal echocardiogram. The rest of the patients had either aorta, subclavian, or femoral arterial cannulation plus right atrial or bicaval drainage. In the descending aorta, the technique was partial bypass with femoral or descending aorta inflow cannulation and left atrium drainage in most cases.
In redo surgery, the previous graft was completely resected only in the presence of infection or multiple leaking sites. With one leaking site and no infection, the graft was partially removed, and the edges were refreshed for a new anastomosis. For redo and primary surgery, nonprosthetic material was intended to use when infection was present. Antibiotic therapy was recommended for 6 weeks when no prosthetic material was left behind, and life-long antibiotic suppression therapy when prosthetic material was required. Omental wrapping was not used.
Relevant data were collected on Microsoft Excel 97, and the analysis was done with JMP 5.0.1.2 (SAS Institute, Cary, NC) Statistical Software. Univariate analysis of dichotomous predictors of operative mortality, hemorrhage during sternotomy, reexploration for bleeding, survival, and freedom from TAFA was performed with 
2, Fisher's exact test, and log-rank when appropriate. Multivariate risk analysis was not used because the small number of cases would have produced unacceptably wide confidence intervals. Possible predictors analyzed are listed in the Appendix. Survival analysis was completed using the Kaplan-Meier method. A p value less than 0.05 was considered significant in all cases. Statistical analysis was performed for the whole sample and divided into significant groups: mediastinal versus descending aorta and primary versus redo surgery.
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Results
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In all, 1,692 patients with thoracic aorta surgery between March 1981 and April 2004 were identified. Of them, 58 had a TAFA repair. One patient with descending aorta TAFA treated percutaneously with an endovascular device was excluded. The preoperative demographics are described in Table 1. Two thirds had previous cardiovascular surgery and one quarter, active infectious endocarditis. Nine had an emergent operation: 7 to contain rupture and 2 for composite-homograft dysfunction.
The mean interval between operations was 80 ± 90 months. At the original operation, 32 had a sternotomy and 5 had a left thoracotomy. Eighteen (49%) were operated on at Mayo and the rest elsewhere. It was common in the original operation to have a composite valve-ascending aortic replacement, the aorta wrapped around the graft, and Bioglue (CryoLife Inc, Kennesaw, GA) utilization starting in 2002 (Table 2). Aneurysm was the primary operative indication in redos. In contrast, the etiology or indication in primary patients was endocarditis in 7 (35%), trauma in 6 (30%), penetrating atherosclerotic ulcer in 3 (15%), aneurysm in 2 (10%), and not known in 1 (5%).
Twelve patients (21%) were asymptomatic. The predominant preoperative symptoms were dyspnea 25 (44%), chest pain 22 (39%), and fever 18 (32%). Initial diagnosis was made by CT (n = 22, 39%), echocardiogram (n = 22, 39%), MRI (n = 3, 5%), catheterization (3, 5%), and chest radiograph (n = 2, 4%). In 5 (9%), the TAFA was detected at the time of operation for another disease. The indication in these cases was aortic valve endocarditis in 2, infected epicardial defibrillator in 1, tricuspid valve regurgitation (status post aortic valve replacement) in 1, and composite graft dysfunction in 1.
As described in Table 3, the most common TAFA location was the ascending aorta, and the most frequent disruption site was the distal anastomosis. The TAFA had fistulized in 1 case each to pulmonary artery (Fig 1), trachea, left atrium, right ventricle, and right atrium. Of 15 with evidence of infection, only 8 had a positive culture: 4 Staphylococcus spp, 1 Salmonella sp, 1 Enterococcus faecalis, 1 Escherichia coli, and 1 Propionibacterium sp. In the presence of active infection, 8 had homograft-autologous pericardium and 7 had synthetic material used for repair.
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Fig 1. Magnetic resonance imaging showing a thoracic aorta false aneurysm coming from the proximal anastomosis of an ascending aortic graft. The aneurysm had fistualized to the pulmonary artery trunk.
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The operation details are described in Table 4. Among patients with a previous tube graft replacement, 9 had partial replacement and 18 a complete replacement. Almost all the patients had extracorporeal circulation, and a half of them had circulatory arrest.
Operative mortality was 7% (Table 5). No patient died of massive hemorrhage during the sternotomy. All of them were presternotomy cannulated. The univariate analysis (Table 6) identified the size of the TAFA and the character of the operation as predictors of massive hemorrhage during the sternotomy. Left ventricular dysfunction and obesity were associated with operative mortality.
A comparison of mediastinal versus descending aorta, and redo versus primary operations (Table 7), showed no difference in all major outcomes, with the exception of stroke. Three patients had stroke in the descending aorta or primary group. In 2, the etiology was a penetrating atherosclerotic ulcer, 2 had retrograde reperfusion from the femoral artery or descending aorta, and 2 had circulatory arrest time of 30 or more minutes.
Fifty-three survivors were followed. Thirty-six patients were alive at the time of follow-up. Three patients did not answer, and 2 refused the HIPPA form. However, all survivors had at least 6 months of follow-up described in their clinical records or questionnaires. Mean follow-up was 73 months, with a total of 349 patient-years. Survival and freedom from TAFA recurrence are presented in Figure 2. The freedom from cardiac death was 79% ± 6% and 68% ± 8%, at 5 and 10 years, respectively. Age greater than 65 years and left ventricle dysfunction was associated with poor survival. There were no factors associated with TAFA recurrence (Table 6). There was 1 case of endocarditis during the follow-up. Seventeen deaths occurred during the follow-up: 9 cardiac related, 4 noncardiac, and 4 of unknown cause.
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Fig 2. Kaplan-Meier actuarial survival (light line) and freedom from thoracic aorta false aneurysm (TAFA) recurrence (heavy line).
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Four of 9 cardiac-related deaths were due to false aneurysm recurrence. Seven patients had TAFA recurrence repaired at the Mayo Clinic. One had a TAFA at the distal anastomosis of an ascending aortic replacement for aortic dissection. The false aneurysm was repaired with felted sutures using hypothermia and low-flow state without inspecting the graft from inside. The TAFA recurred in 1 month, and the patient died of left ventricular failure at the time of the reoperation. Three patients originally had aortic valve replacement for endocarditis. Two of them had a TAFA of the root repair with pericardial patch. However, they required reoperation due to recurrent disruption of the root. One had a false aneurysm repair of the cannulation site but came later for newly developed TAFA of the root. One had a TAFA at the distal anastomosis of an ascending aorta replacement for dissection. No signs of infection were found. It was repaired, but the patient returned 3 months later emergently with root infection and root false aneurysm. A repair with homograft was attempted, but the patient did not come off cardiopulmonary bypass. Two patients had TAFA recurrence after composite replacement several years later, without apparent risk factors.
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Comment
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This study suggests TAFA correction is possible despite the serious risks involved. Massive hemorrhage during redo sternotomy is a significant complication, especially in urgent operations and with TAFA diameter greater than 55 mm. However, it could be successfully managed with extramediastinal cannulation and deep hypothermia. Mortality is higher at advanced age and with left ventricular dysfunction, as in most cardiovascular surgery series. Avoidance of potential pitfalls might be critical to decrease the incidence of recurrence. Symptoms could be minimal, and consequently, routine imaging after aortic surgery is recommended.
There is controversy in the literature about the true incidence of TAFA. There are reports describing a low incidence even among high-risk patients. Sabik and colleagues [4] described 205 patients operated on for aortic dissection with a follow-up of 802 patient-years. They found only 1 reoperation for TAFA [4]. Furthermore, Pugliese and colleagues [5] did the same in 178 patients, completing 690 patient-years without finding any TAFA. Nevertheless, some studies suggest this is an underestimation. Mesana and coworkers [6] meticulously followed patients operated on for dissection or aneurysm with MRI finding an incidence of TAFA close to 15%. They also showed that an important percentage of these patients have periprosthetic hematoma that could regress without causing any clinical problems or, on the other hand, could end up in a TAFA. In addition, 10 of 12 patients reoperated on in this study for TAFA after a primary aortic dissection were asymptomatic, and the 2 others had mild chest pain [6]. In our study, 21% were completely asymptomatic, and 65% were in functional class I or II; and for 9%, it was a intraoperative finding. Furthermore, late causes of death of patients operated on for aortic dissection reported in the literature are sudden death, cardiac tamponade, and aneurysm rupture [4]. It is entirely possible that some of these deaths were caused by TAFA, making the true incidence of this pathology unknown. These findings suggest TAFA incidence has been underestimated and routine imaging screening is warranted whenever risk factors are present.
Graft wrapping and Bioglue might have impacted the incidence of TAFA. Graft wrapping could cause excessive tension on the suture lines predisposing to TAFA, or conceal a small fistula that matures at a later date. Hence, the incidence reported by Mesana and associates [6] after aneurysm surgery, with a high percentage of wrapping at the beginning of the experience, could be an overestimation for current surgical practice. In our experience, wrapping was done in 48% of the previous grafts, but 9 of 12 were performed before 1995, confirming a time decreasing tendency. In the recent years, Bioglue has been used for aortic surgery and a decrease in bleeding complications has been reported [9]. However, there is concern that it could cause tissue necrosis leading to redissection or TAFA. Kazui and coworkers [10] described 2 of 5 patients and Coselli and coworkers [9] described 2 of 17 patients with TAFA after Bioglue application. In our group, 4 of 11 (36%) TAFA patients operated on since 2002 had Bioglue exposure. Nevertheless, we do not know in our study how many patients had Bioglue applied and then had a TAFA develop. Hence, the association is suspected, but we could not draw definitive conclusions.
The wide variety of microorganisms involved should alert the clinician requesting microbiology studies. We found Staphylococcus spp as the most prevalent bacteria at the time of the operation, which is consistent with the findings of Dumont and colleagues [2]. Pseudomonas sp, Corynebacterium sp, Mycobacterium tuberculosis, and Candida albicans have been isolated in previous studies [3, 11–14]. We also found Salmonella sp, Escherichia coli, and Enterococcus faecalis in mycotic TAFA. It is normally accepted that infection is associated with recurrence. However, we did not find infection as predictor of TAFA recurrence. This might be related to the small numbers of cases analyzed, with just one case of newly diagnosed endocarditis in the follow-up.
The redo sternotomy technique has been discussed in previous studies. Obviously, we agree with Dumont and coworkers [2] that surgical intervention must be carefully preoperatively planned because of the inherent risk of entry at sternotomy. In these circumstances, the use of femorofemoral bypass or other kind or extramediastinal cannulation before sternotomy is a well-known strategy. We had only 4 patients with subclavian cannulation in the last years of the study. However, this number is expected to grow because of the concern of thromboembolic complications with femoral or retrograde perfusion.
The likelihood of TAFA rupture during sternal reentry has been decided so far by surgeon's criteria looking to the CT scan or to the MRI. Univariate analysis showed significant risk during urgent operations and with diameter above 55 mm. Accordingly, precautions to manage this complication need to be stressed in these cases. Cardiopulmonary bypass and cooling before sternal reentry is important to have time and exposure to dissect the adhesions with circulatory arrest or low-flow state if the TAFA is open. If the patient has moderate or severe aortic regurgitation, cooling will result in left ventricle distention, arrhythmias, failure, and death. In this case, left ventricle venting through a limited left thoracotomy could be a life-saving technique before sternal reentry. Mohammadi and associates [1] advocate the use of bilateral carotid artery cannulation to avoid death and complications of deep hypothermia circulatory arrest. However, our operative mortality rate (7%) does not support this approach if compared with historical series, and univariate analysis failed to identify the need of circulatory arrest as predictor worse outcome. Furthermore, no mediastinal patient had stroke, suggesting that bilateral carotid cannulation is not required for this reason.
What procedure should be done once the leakage site is identified? Optimal exposure with a bloodless surgical field might be critical to avoid recurrence. Upon finding out where is leakage site (only 3% had multiple origins), the tissue edges are refreshed if they are weak or abnormal, and the repair is accomplished. A simple graft, composite replacement, patch, or simple suture could be needed depending upon the localization and the size of the neck. When previous grafts without active infection had a single leakage site, we did not think that complete replacement was necessary as long as the edges were well healed, as has been stated before [1]. This strategy is supported by the 10-year freedom from TAFA recurrence of 83% obtained by our group. If infection is present, allograft material and autologous pericardium should be used whenever possible. LeMaire and colleagues [7] found increased success following this premise in proximal aortic reoperations after composite grafts. Whenever the infected composite graft was replaced by an homograft, the patient survived and vice versa [7]. Nevertheless, the numbers are still small, and judicious use of synthetic material might be appropriate to shorten the operation time in a critically ill patient.
Potential pitfalls are important to analyze to prevent recurrence. One of our patients had early recurrence (1 month). We think that was due to incomplete inspection of the sutures of a previously ascending aorta graft for dissection. Therefore, bloodless visualization might help decreasing recurrence. Three patients had recurrence from an aortic root disrupted by endocarditis. In these cases, an alternative is the use of homograft instead of an aortic prosthesis. Finally, another patient had a early recurrence (3 months) because of infection that was not evident at the time of the first operation. Hence, high suspicion of infection (even if there is no pus or active inflammation) and aggressive antibiotic and surgical treatment are recommended.
On descending aorta false aneurysms, we prefer left partial bypass with a Biomedicus pump (Medtronic, Minneapolis, MN) whenever possible. This approach decreases the dose of heparin needed and the amount of coagulopathy produced when compared with full conventional cardiopulmonary bypass. On the other spectrum of the technique, we prefer it over clamp and sewing because this permits a nonhurried repair and provides distal perfusion during clamping [15].
Is there any strategy in this subgroup that could decrease the incidence of stroke? Avoidance of aortic cross-clamping, long circulatory arrest times, and retrograde arterial flow might improve neurologic outcomes, particularly in the setting of atherosclerotic penetrating ulcers. Left subclavian cannulation or graft side cannulation could help in reducing this devastating complication.
Emerging technology such as endovascular stenting, coil embolization, port-access techniques have appealing applications in the treatment of TAFA. There are scattered but promising reports of TAFA treatment in the literature [16–18]. We think there are selected patients suitable for endovascular stenting. Patients with no evidence of infection and with involvement of the descending aorta appear as a logical indication. Furthermore, for those with penetrating atherosclerotic ulcers, it might be even better because of the high risk of stroke we have seen. We expect an increase number of patients treated with endovascular techniques at our division on the near future.
Study limitations include studying a heterogeneous group of patients. Redo results could be different when compared with primary cases in larger series. The retrospective nature of the study may also affect the certainty of the results. Several surgeons performed the operations, and the outcomes can vary among them. Moreover, numbers are small, and therefore, the statistical power of the study is low.
In conclusion, hypertension, infection, previous aortic operation, and graft wrapping are associated with TAFA. Symptoms may be minimal or absent, and consequently a high degree of suspicion is warranted. Routine image screening is recommended whenever risk factors are present. Extramediastinal cannulation, deep hypothermia, and circulatory arrest are required for large mediastinal TAFA. Despite serious risks, TAFA elimination is possible with a low recurrence rate and good long-term survival.
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Appendix
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Preoperative Factors Tested in Univariate Analysis
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Discussion
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DR CLIFF K. CHOONG (Cambridge, UK): Congratulations on the very good results and the detailed analysis. In your presentation, you had shown a slide that contained a lot of different factors that you had evaluated assessing for risk factors for mortality. You had evaluated date of surgery from 1990 onward as one of those factors. Did you however look at earlier date of surgery such as those performed between 1984 to 1990 as a potential a risk factor, because your series is a very long series, dating from from 1984 to the current period?
DR VILLAVICENCIO: We analyzed that in terms of surgery after 1990, 1995, or 2000 as a dichotomous variable, and we didn't find any differences.
DR ANTHONY L. ESTRERA (Houston, TX): I congratulate you on your talk and a nice presentation. There is a point that is confusing to me, however. In your conclusion, you report a recommended surgical approach to thoracic aortic false aneurysms but you also seem to mix both ascending false aneurysms with thoracoabdominal aortic false aneurysms. I think that it is very important to make this distinction, because your conclusion about your approach will depend on the location of your false aneurysm and whether it is with or without infection. In our experience, the location and etiology of the false aneurysm determines our approach. Could you comment?
DR VILLAVICENCIO: Yes, sir, I understand the point and we thought about it, because one of the limitations of the study is that this is a very heterogeneous group of patients. It was difficult to decide if we were going to do just the mediastinal versus descending, or do just postsurgery, post–graft replacement (of some part of the aorta) versus those ones who didn't have a graft before, or even in the ascending aorta, if we would've had to separate the patients who had a compromise of the root, which I think might have a higher risk than the ones who had just compromise of the ascending aorta.
Hence, we focused on what was the risk on reentry. We analyzed that particular group as a separate one, these are those patients with previous sternotomy, and then we did a separate analysis, and then we found that there was more risk when the aneurysm had a diameter than 55 or the patient had an urgent operation. But I agree that is a limitation of the study. It is too heterogeneous, and the results might vary for each part. However, we tried also to do a univariate analysis to see if that had any influence or not, and we separated mediastinal or descending false aneurysm for the major outcomes, and we didn't find any differences.
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Acknowledgments
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Special thanks to Judy Lenoch for her help in data recollection.
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Top
Abstract
Introduction
