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 Table of Contents  
ORIGINAL ARTICLE
Year : 2018  |  Volume : 51  |  Issue : 2  |  Page : 50-57

Occult aortic fistulation affects late outcome of ruptured descending thoracic aortic aneurysms after emergency thoracic endovascular aortic repair in patients with initial hematemesis/hemoptysis


Division of Cardiovascular Surgery, Department of Surgery, National Cheng Kung University Hospital, College of Medicine, National Cheng Kung University, Tainan, Taiwan

Date of Submission14-May-2017
Date of Decision09-Aug-2017
Date of Acceptance25-Oct-2017
Date of Web Publication24-Apr-2018

Correspondence Address:
Dr. Chung-Dann Kan
Department of Surgery, Division of Cardiovascular Surgery, National Cheng Kung University Hospital, College of Medicine, National Cheng Kung University, Tainan
Taiwan
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Source of Support: None, Conflict of Interest: None


DOI: 10.4103/fjs.fjs_80_17

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  Abstract 

Background: Although thoracic endovascular aneurysm repair (TEVAR) has been widely used as the first choice of emergency surgical procedure for ruptured descending thoracic aortic aneurysms (rDTAAs), the risk factors of adverse outcome have less been investigated.
Purpose: To investigate the outcomes of patients undergoing TEVAR for rDTAA and to identified risk factors of worse prognoses.
Materials and Methods: The surgical outcome of TEVAR for rDTAA in National Cheng Kung University Hospital was retrospectively analyzed. From February 2008 to December 2016, 27 patients were included, after excluding patients with traumatic aortic injury, infected aneurysm, esophageal malignancy-related aortoesophageal fistula or those in association with aortic dissection.
Results: There were 5 (18.5%) 30-day mortalities, including 3 (11.1%) intraoperative deaths. Seven additional patients died during follow-up and the estimated survival rate at 1 year and 3 years was 61.3 ± 9.7% and 50.5 ± 10.6%, respectively. Among these patients with late mortality, five patients presented with hematemesis or hemoptysis preoperatively. Aortoesophageal fistula was confirmed in three patients by esophagogastroduodenoscopy presenting with hematemesis. These patients underwent subsequent open debridement along with esophagectomy after TEVAR and remained alive during follow-up. On the other hand, those with possible occult aortic fistulations that were not detected by endoscopic examinations and not surgically managed had worse late outcomes (P = 0.058).
Conclusions: For patients with rDTAA having hematemesis or hemoptysis as part of the initial presentations, careful survey for possible aorta-related fistulation is important. Although definite diagnosis of fistulation might be difficult, surgical exploration for hematoma evacuation, adequate debridement, and repair of intraoperative identified fistulation should be advocated.

Keywords: Aortobronchial fistula, aortoesophageal fistula, aortopulmonary fistula, ruptured descending thoracic aortic aneurysm, thoracic endovascular aortic repair


How to cite this article:
Lin TW, Kan CD. Occult aortic fistulation affects late outcome of ruptured descending thoracic aortic aneurysms after emergency thoracic endovascular aortic repair in patients with initial hematemesis/hemoptysis. Formos J Surg 2018;51:50-7

How to cite this URL:
Lin TW, Kan CD. Occult aortic fistulation affects late outcome of ruptured descending thoracic aortic aneurysms after emergency thoracic endovascular aortic repair in patients with initial hematemesis/hemoptysis. Formos J Surg [serial online] 2018 [cited 2018 Oct 20];51:50-7. Available from: http://www.e-fjs.org/text.asp?2018/51/2/50/231145


  Introduction Top


Ruptured descending thoracic aortic aneurysm (rDTAA) is a highly lethal condition and remains a challenging surgical emergency. The traditional surgical treatment via left thoracotomy for an rDTAA is associated with high mortality, with a 30-day mortality rate of more than 30% even in an experienced center. Furthermore, more than half of the survivors would develop significant and even permanent neurological, respiratory, and renal complications.[1] Thoracic endovascular aortic repair (TEVAR) has been widely used in elective situations for patients with an anatomically suitable descending thoracic aneurysm. In the emergency setting of managing an rDTAA, TEVAR theoretically provides a less invasive approach and results in quicker exclusion of the aortic aneurysm. In comparisons between TEVAR and the traditional open repair, some studies demonstrated that TEVAR may have trends toward improved short-term and mid-term survival.[1],[2],[3],[4],[5],[6] Nevertheless, considerable postoperative morbidities still developed in patients with rDTAA treated with TEVAR, including endoleak, which occurred in nearly one-fifth of the patients.[1],[2],[4],[5],[6] Furthermore, multiple factors might affect both the short-term and long-term outcomes of rDTAA treated with TEVAR, including anatomical and morphological features of the aortic aneurysm, as well as the clinical presentations of patients. In this study, we retrospectively evaluated the outcomes of patients who underwent TEVAR for rDTAA in a single tertiary medical center and also identified risk factors of adverse early and late outcomes.


  Materials and Methods Top


The study was approved by the Institutional Review Board (IRB) in National Cheng Kung University Hospital (IRB No.: ER-100–146). The IRB waived the requirement of patient informed consent.

Study population, definitions, and follow-up protocol

From February 2008, when the first case of rDTAA was treated with endovascular procedure in the National Cheng Kung University Hospital, to December 2016, 50 patients have undergone emergency TEVAR for descending thoracic aorta catastrophes with various etiologies. Eleven patients with infected aortic aneurysms were excluded, including those with fever or a positive perioperative blood culture and suggestive imaging finding of an infectious process on computed tomography (CT), such as presence of periaortic soft-tissue mass and fluid or air accumulation. Three patients with secondary aortoesophageal fistula caused by invasion of advanced esophageal malignancy and the other nine patients with traumatic aortic injuries, ruptured dissecting aneurysms, or acute type B aortic dissections were also excluded from this study. Finally, 27 patients who had undergone TEVAR for rDTAA were included and retrospectively analyzed. The diagnosis of ruptured aortic aneurysm was established by using CT angiography (CTA). Ruptured aortic aneurysm was defined as the presence of contrast medium extravasation outside the aorta, disruption of the contour of the aortic aneurysm with periaortic hematoma, pleural effusion, or pseudoaneurysm formation. If only localized periaortic or mediastinal hematoma was present, the aortic aneurysm was defined as a contained rupture. The anatomic location of the aneurysm was between the left subclavian artery and the celiac trunk. Patients with aortic arch aneurysms, that is, those with aneurysmal change of the aorta proximal to the orifice of left subclavian artery, were excluded from this study.

Hemodynamic instability was defined as the presence of shock (systolic arterial blood pressure <90 mm Hg) or the need for cardiopulmonary cerebral resuscitation (CPCR). Perioperative blood transfusion was defined as the amount of blood product transfused intraoperatively and within 24 h postoperatively. Renal failure was defined as a 3-fold increase in serum creatinine level, a decrease in glomerular filtration rate by 75%, a urine output of <0.3 mL/kg/h for 24 h, or anuria for 12 h based on the risk, injury, failure, loss of kidney function, end-stage kidney disease criteria, or need for dialysis. Respiratory failure was defined as prolonged mechanical ventilation for more than 7 days after operation or the need for tracheostomy.

A CTA would be arranged before hospital discharge and annually thereafter for the survivors. All patients presenting with hematemesis/hemoptysis preoperatively and successfully salvaged from emergency TEVAR underwent at least once esophagogastroduodenoscopy and bronchoscopy during hospitalization. Patients with “occult aortic fistulation” are defined as those presented with hematemesis/hemoptysis at the time of diagnosis of rDTAA, but without subsequent definite diagnosis of aortic-related fistula based on any perioperative diagnostic modality.

Surgical techniques

All endovascular procedures were performed under general anesthesia in a hybrid operating room equipped with a fluoroscopic unit. Prophylactic lumbar spinal drains were only inserted in 2 hemodynamically stable patients before the surgical procedure (7.4%). The brands of the stent graft devices used included Cook Zenith TX 2 (William Cook Europe Aps, Bjaeverskov, Denmark; n = 19), Medtronic Valiant (Medtronic Ireland, Galway, Ireland; n = 7), and Gore TAG (W. L. Gore and Associates, Flagstaff, Arizona, USA; n = 1). The diameter of the stent graft was oversized by 10%–20% the diameter of the aortic landing zone. The endovascular devices were routinely advanced through the common femoral or external iliac arteries using the open technique and deployed under fluoroscopic guidance. Balloon angioplasty or even stent implantation to achieve an adequate vascular route is necessary in two patients (7.4%) with stenotic iliac arteries. None of the patients underwent fenestrated stent graft implantation or the chimney technique. The left subclavian artery was not routinely embolized at its origin or revascularized even if covered by the stent graft, unless there was type II endoleak from the left subclavian artery or evident left arm malperfusion. When the left common carotid artery coverage was necessary for an adequate landing zone (zone 1), a bypass procedure was performed. If landing zone at ascending aorta (zone 0) was necessary, innominate artery bypass would be performed through a median sternotomy. A final angiography was routinely performed to assess the position of the endograft and the presence of any type of endoleak.

Statistical analysis

Among predictive variables of early outcomes, the categorical variables were analyzed using the Fisher's exact test and the continuous variables were analyzed using the 2-tailed Student t-test or Mann–Whitney U-test. The Kaplan–Meier survival curve and log-rank test were used to evaluate the estimate survival during follow-up. P < 0.05 was considered statistically significant. Statistical analysis was performed using the Statistical Package for the Social Sciences version 18.0 for Windows (IBM SPSS, Chicago, IL, USA).


  Results Top


Patient and procedural characteristics

The mean age of the 27 patients was 72.07 ± 13.79 years, and 20 (74.1%) of them were male. Hemodynamic instability was present in 9 patients (33.3%), while preoperative CPCR was needed in three patients (11.1%). Fourteen patients (51.9%) were considered as having a contained ruptured aortic aneurysm based on CTA findings, as defined previously. Ten patients (37.0%) had hematemesis or hemoptysis as part of their initial presentations. The aneurysm was fusiform in 17 patients (63.0%) and saccular in 10 (37.0%). The proximal landing zone at zone 0 was necessary in 3 patients (11.1%), and both of proximal landing zones at zone I and zone II were necessary in 6 patients (22.2%), and the rest 12 patients (44.4%) had the proximal landing zone at zone III. Transcarotid artery bypass was performed in five patients before deployment of the stent graft with coverage of the left carotid artery. In one hemodynamic unstable patient who required zone I landing, the TEVAR procedure was performed first and bypass from a right subclavian artery to a left carotid artery was subsequently performed. Of the 12 patients with a stent graft covering the left subclavian artery, 6 (50.0%) had either embolization or direct ligation of the left subclavian artery near the orifice. The patient demographics, clinical presentations, and operative characteristics are shown in [Table 1].
Table 1: Patient demographics, clinical presentations and operative characteristics

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Early outcomes

Three intraoperative mortalities (11.1%) occurred, of which 2 died from failure exclusion of the ruptured aneurysm by the endovascular prostheses and uncontrolled bleeding. Meanwhile, the other patient had an intraoperative cardiac arrest despite successful stent graft deployment, which was likely to be caused by preoperative profound shock. The 30-day mortality rates were 18.5% (n = 5). Both of the additional two patients died after initial successful TEVAR because of the development of various postoperative complications and multiorgan failure. Major morbidities developed in 45.8% (n = 11) of the 24 patients who were alive when discharged from the operating theatre. Renal failure (37.5%, n = 9), respiratory failure (33.3%, n = 8) and systemic infectious complication (25.0%, n = 6) were the most common postoperative complications. The predictors of 30-day mortality are shown in [Table 2].
Table 2: Predictors of 30-day mortality

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Endoleak was detected on the final angiography after completion of the procedure in the operating theatre or on CTA before hospital discharge in five patients (20.8%). One (4.2%) of them had type I endoleak, and the remaining four patients (16.7%) had type II endoleak. There was no type II endoleak arising from not embolized left subclavian artery. Among the patients with endoleak, one patient underwent reintervention by means of transarterial embolization (TAE) for type II endoleak. The other 4 patients with primary endoleak did not undergo any reintervention such as proximal stent graft extension for type I endoleak or TAE for type II endoleak. The main reason why these endoleaks were not addressed was that none of these patients had persistent exsanguination and that the usual postoperative comorbidities made the families favor conservative management. Two of the 4 patients died within 30 days after TEVAR; as mentioned above, both of the additional two mortalities after initial successful TEVAR were caused by multiorgan failure, which was not directly related to the presence of primary endoleak.

Late outcomes

The estimated 1-year and 3-year survival was 61.3 ± 9.7% and 50.5 ± 10.6%, respectively [Figure 1]. Seven patients died beyond 30 days after the initial TEVAR procedure; among them, five of them had hematemesis or hemoptysis as the initial presentation. The postoperative survival of these 5 patients ranged from 113 days to 702 days. The cause of death included recurrent hemoptysis in 1 (20.0%), and paraaortic infection-related sepsis and multiorgan failure in 4 (80.0%). The outcomes of the patients with endovascularly managed rDTAAs and those initially presenting with hematemesis/hemoptysis are summarized in [Figure 2].
Figure 1: The Kaplan-Meier survival curve for patients underwent TEVAR for rDTAA. The estimated 1-year and 3-year survival was 61.3 ± 9.7% and 50.5 ± 10.6%, respectively

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Figure 2: (a) The enrollment, allocation and outcomes of patients undergoing emergency TEVAR in National Cheng Kung University Hospital from February 2008 to December 2016. (b) The outcome of the 10 patients presenting with hematemesis/hemoptysis initially. TEVAR, Thoracic endovascular aortic repair; rDTAA, ruptured descending thoracic aortic aneurysm; intra-OP, intraoperative; MOF, multiorgan failure; AB fistula, aortobronchial fistula; AE fistula, aortoesophageal fistula; EGD, Esophagogastroduodenoscopy

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Ten patients (37.0%) had hematemesis/hemoptysis as a part of presentations when they were diagnosed with rDTAAs. Except for one patient who died intraoperatively due to failure of aneurysm exclusion by endovascular devices and uncontrolled bleeding, we routinely arranged both esophagogastroduodenoscopy and bronchoscopy for all the other patients for possible aorta-related fistulation. Nevertheless, definite evidence of aortoesophageal fistula was confirmed on intraoperative aortography and esophagogastroduodenoscopy only in 3 patients. All of these patients underwent subsequent esophagectomy and survived without evidence of persistent infection or other aorta-related complications. For the rest of the 6 patients, aorta-related fistulation could not be confirmed by CTA or intraoperative aortography and neither esophagogastroduodenoscopy nor bronchoscopy could identify an aortoesophageal or an aortobronchial fistula. Despite prolonged systemic antibiotics were prescribed for all of the patients, five of them eventually died because of recurrent hemoptysis or paraaortic infection as mentioned above during follow-up. A worse prognosis, although not statistically significantly, was observed in these rDTAA patients associated with possible occult aortic fistulations, i.e. that presented with hematemesis/hemoptysis but without definite identification and proper surgical management [Figure 3], P = 0.058].
Figure 3: The Kaplan–Meier survival curve comparing patients undergoing thoracic endovascular aneurysm repair for ruptured descending thoracic aortic aneurysm presenting with and without hematemesis or hemoptysis initially. Those with hematemesis or hemoptysis as the initial presentation but without a definite diagnosis and surgical management of aortic fistulation (i.e., those with “occult aortic fistulation”) had nonstatistically significant worse late outcomes (P = 0.058)

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Three patients with endoleaks detected on postprocedural angiography or CTA survived to discharge; however, all of them died of late aorta-related complications during follow-up. Unresolved aortic aneurysm-related aortobronchial fistula developed in one patient with type I endoleak half year after initial TEVAR and the patient died because of massive hemoptysis. The other two patients also had hematemesis or hemoptysis as the initial presentation and both of them died because of paraaortic infection and sepsis. In addition to possible occult aortoesophageal or aortobronchial fistula, endoleak-related persistent accumulation of hematoma might also contribute the development of paraaortic infection in these two patients.


  Discussion Top


Since the 2000s, TEVAR has been increasingly used as the primary method of management for acute aortic emergencies of the descending thoracic aorta, including rDTAAs.[7],[8],[9],[10],[11],[12],[13],[14],[15] The early results of endovascular repair for rDTAAs varied, although most reports showed a 30-day mortality rate <20%. Considerable postoperative morbidities were still reported, including paraplegia, stroke, cardiac complications, pulmonary complications, acute renal failure, and bleeding.[1],[2],[3],[4],[5],[6] Endoleak also developed in nearly 20% of the patients.[1],[2],[6] In the meta-analysis in 2010 that compared open and endovascular repair for rDTAA,[3] Jonker et al. concluded that TEVAR for rDTAA had a lower 30-day mortality rate (18.9%) than open repair (33.3%). Despite the possible short-term survival benefits of TEVAR in comparison to the traditional open repair in the emergency setting, a notable aneurysm-related complication and death still occurred during follow-up in the TEVAR group. Among the late adverse outcomes, aorta-related fistulations are rare but difficultly managed and lethal problems.[16],[17]

In the presented series, the mortality and morbidities of the patients who underwent TEVAR for rDTAA in the emergency were comparable to the published literature. Patients with hematemesis or hemoptysis as part of the initial presentations had worse late outcomes, but not early outcomes. The recently published studies, including two systemic reviews regarding the outcome of TEVAR for aortoesophageal and aortobronchial fistulas, respectively, support that endovascular repair could serve as an initial life-saving procedure for those with aortoesophageal or aortobronchial fistulas.[18],[19],[20],[21],[22],[23] Nevertheless, subsequent debridement and resection of the involved structures are still advocated.[19],[20],[21],[22],[23] However, for most of the patients who presented with hematemesis or hemoptysis in this series, the diagnostic modalities, including CT, intraoperative aortography, panendoscopy, and bronchoscopy, failed to confirm the diagnosis of aortoesophageal or aortobronchial fistula. There are several explanations. First, the bleeding might not be enough to be shown with contrast medium extravasation on CTA or aortography. Second, the rupture site could be difficult to identify on endoscopic examination after TEVAR because the bleeding has already ceased. Third, the symptoms of hematemesis or hemoptysis might not be caused by the true connection between the aorta and the gastrointestinal tract or the airway, but simply caused by aneurysmal erosion to these structures. If the diagnosis of aorta-related fistulation is not established in these patients, the further therapeutic decision could be difficult. Because of lack of definite diagnosis, a conservative management for patients with rDTAA salvaged by emergency TEVAR with initial presentation of hematemesis/hemoptysis was the usual treatment strategy. Broad-spectrum antibiotic therapies were routinely prescribed for an extended period. Despite this, we still observed poor late outcomes in these patients.

Czerny et al. introduced the concept of aortopulmonary fistula which is defined as a connection of aorta to pulmonary parenchyma, in comparison to the usual aortobronchial fistula where the connection is at the central airway.[17] In a broad sense, an aortopulmonary fistula could also refer to pulmonary parenchyma erosion caused by rDTAAs. Prognosis was found not to be different in patients with aortobronchial or aortopulmonary fistula and the authors suggested that an aggressive and radical surgical management should be the most durable treatment for both of these complications.[17],[24] Nevertheless, in our experience, the diagnosis of fistulation between aorta and pulmonary parenchyma was still difficult. A bronchoscopy definitely could not identify those lesions. Identification of contrast medium extravasation on CTA or angiography might be the only diagnostic finding of these aortopulmonary fistulas. After carefully reviewing the CTA of patients presenting with hematemesis/hemoptysis when an rDTAA was diagnosis in these series, we still could not identify any previously undiagnosed aorta-related fistulations, despite a loss of the fat plane between the aorta and airway or a downstream aspirated pneumonitis might be presented. Gallium scintigraphy or positron emission tomography (PET) is helpful to provide hints of possible infection related to the occult aortic fistulations; nevertheless, they still could not identify definite evidence of the presence of fistulations.

Patients with rDTAAs presenting with hematemesis or hemoptysis could associate with primary-infected aortic aneurysms. In this study, we have excluded all patients with suspicious or definite infected aortic aneurysms according the exclusion criteria described above. We also routinely performed series blood cultures for rDTAA patients with hematemesis or hemoptysis before TEVAR, and those with positive cultures were not included in this series. Thus, all of our study groups at least have no active infected aortic aneurysm at the time of operation, based on clinical, laboratory, and image results. The late paraaortic infections and recurrent aortic fistulations in our cases were considered to be subsequent to unmanaged occult fistulas. In the current endovascular era, together with aggressive adjunctive procedures to eliminate the infectious environment and long-term antibiotics suppression, endovascular aortic repair might serve as a reliable definite therapy for infected aortic aneurysms.[25],[26] Applying this strategy of treating infected aortic aneurysms in the current endovascular era, we emphasized again that any occult aortic fistulations should be identified and aggressively managed in patients with rDTAAs.

In addition to the existed occult aortic fistulations, persistent mediastinal hematoma after rDTAA and presence of endoleak were also considered to be the mechanisms of late fistulation formation.[24] Although endoleaks did not result in persistent bleeding after initially successful TEVAR and were not directly related to early mortalities, we found an adverse late outcome in patients with endoleaks. Two of the three patients with endoleak also presented with preoperative hematemesis/hemoptysis, and it would be difficult to define which of these risk factors actually contributed to the late aorta-related death. Nevertheless, a staged operation still should not only be recommended for patients who present with hematemesis or hemoptysis, but also be considered for those with significant mediastinal hematoma and endoleaks. Debridement of the aneurysm sac, evacuation of the hematoma, and ligation of the branches which result in type II endoleaks could be done. Definite surgical procedures, such as resection or repair of the esophagus, bronchus, or pulmonary parenchyma, should be performed if any evidence of aortic fistulation is noted during the second operation. Even stent-graft extirpation and simultaneous aortic graft reconstructions should also be considered.

Limitations

The retrospective observational design and limited number of cases were the main limitations of the present study. During the study, we rarely performed open surgical repair for rDTAAs; thus, we did not provide institutional experience of the results of the comparison between open and endovascular repair for rDTAA. The intraoperative procedural and postoperative management strategy was also not unanimous for each case, which was highly dependent on surgeon preference. Finally, Gallium scintigraphy or PET was rarely arranged in our patients, and some information of possible paraaortic infectious processes might be missed in those presenting with hematemesis/hemoptysis.


  Conclusions Top


In this study, hematemesis or hemoptysis was not an uncommon presentation in patients with rDTAAs. Furthermore, all of the late deaths were aorta related in this study, which is highly related to the possible communication between the aorta and the esophagus or airway. Although the definite diagnosis of aorta-related fistulation was seldom established, a secondary exploratory operation should be still considered for possible occult fistulations. In these patients, subsequent open surgical management, including aggressive debridement and repair of the affected adjunctive structure to the aortic aneurysm, might improve the late outcome.

Financial support and sponsorship

This work is supported by funding from Medical Science and Technology Research Grant, National Cheng Kung University Hospital (NCKUH-10506021).

Conflicts of interest

There are no conflicts of interest.

 
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