Endovascular repair of pseudoaneurysms after open surgery for aortic coarctation

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1 Interactive CardioVascular and Thoracic Surgery 22 (2016) doi: /icvts/ivv297 Advance Access publication 26 October 2015 ORIGINAL ARTICLE ADULTCARDIAC Cite this article as: Kotelis D, Bischoff MS, Rengier F, Ruhparwar A, Gorenflo M, Böckler D. Endovascular repair of pseudoaneurysms after open surgery for aortic coarctation. Interact CardioVasc Thorac Surg 2016;22: Endovascular repair of pseudoaneurysms after open surgery for aortic coarctation Drosos Kotelis a, Moritz S. Bischoff a, Fabian Rengier b, Arjang Ruhparwar c, Matthias Gorenflo d and Dittmar Böckler a, * a b c d Department of Vascular and Endovascular Surgery, Heidelberg University Hospital, Heidelberg, Germany Department of Diagnostic and Interventional Radiology, Heidelberg University Hospital, Heidelberg, Germany Department of Cardiac Surgery, Heidelberg University Hospital, Heidelberg, Germany Department of Pediatric Cardiology and Congenital Heart Diseases, Heidelberg University Hospital, Heidelberg, Germany * Corresponding author. Department of Vascular and Endovascular Surgery, Heidelberg University Hospital, Im Neuenheimer Feld 110, Heidelberg, Germany. Tel: ; fax: ; dkotelis@hotmail.com (D. Böckler). Received 24 July 2015; received in revised form 8 September 2015; accepted 21 September 2015 Abstract OBJECTIVES: To analyse early and long-term results of thoracic endovascular aortic repair (TEVAR) in patients with pseudoaneurysms after open aortic coarctation (CoA) repair. METHODS: A total of 11 patients of 418 patients who had been treated with TEVAR during the period from January 1998 to April 2015 (8 males; median age 53 years) were retrospectively analysed. Dacron patch aortoplasty was primarily performed in 9 patients and subclavian flap aortoplasty in 2 patients. Seven of the 11 patients had asymptomatic pseudoaneurysms (median diameter 56 mm, range mm) diagnosed by routine screening. Symptomatic patients presented with haemoptysis, lower limb ischaemia, haemodynamic collapse and back pain and underwent emergency repair (4/11). Adjunctive procedures at the proximal landing zone were required in 7/11 patients. The median number of implanted endoprostheses per patient was 1 (range: 1 5). The median follow-up was 60 months (range months). RESULTS: Technical success was achieved in 91% (10/11; 1 secondary elective open conversion). The 30-day mortality was 0%. The stroke rate was 18% (2 non-disabling strokes). In 2 patients (20%), stent-graft displacement during deployment was observed. The reintervention rate was 33% (Type Ib endoleak, left arm claudication, partial coverage of the left common carotid artery). Clinical success during follow-up was achieved in 10/11 patients. In 9/10 patients, aneurysm sac shrinkage was observed. The Type II endoleak rate was 10% (1/10; intercostal artery). The overall mortality rate was 9% (1 patient died of amyotrophic lateral sclerosis). CONCLUSIONS: Endovascular treatment of post-coarctation pseudoaneurysms is feasible in elective and emergency cases, yielding durable results in the long term. Due to anatomical specifics, implantation may be challenging and requires careful procedural planning. On-site cardiothoracic surgery backup is essential in case open conversion is required. Keywords: Aorta TEVAR Thoracic endovascular aortic repair Pseudoaneurysm Aortic coarctation INTRODUCTION Open surgical repair of coarctation of the aorta (CoA) has been associated with late pseudoaneurysm formation during follow-up with a reported incidence rate between 11 and 24% [1, 2]. Left untreated, these pseudoaneurysms may be prone to fatal rupture [3, 4]. In previous series, thoracic endovascular aortic repair (TEVAR) of post-coa pseudoaneurysms has been reported to be feasible and safe, associated with 100% technical success in all reported cases [4 7]. Endovascular therapy in post-coa patients may be challenging, however. Complicating factors include the proximity of the pathology to the supra-aortic vessels, the often severely angulated aortic arches in relatively young patients and the coincidence sometimes of aneurysmatic and stenotic aortic segments. In previous series, follow-up was relatively short [5 7]. Given the long life expectancy of most post-coa patients, long-term durability of the endovascular therapy is of paramount interest. The aim of this study was to describe potential procedural pitfalls and to analyse the long-term durability of the endovascular repair of post-coa pseudoaneurysms. MATERIALS AND METHODS Study design and patient population This is a retrospective single-centre analysis of 11 patients (8 males), who underwent TEVAR for late pseudoaneurysms after open surgery for aortic coarctation between March 1997 and April The Author Published by Oxford University Press on behalf of the European Association for Cardio-Thoracic Surgery. All rights reserved.

2 D. Kotelis et al. / Interactive CardioVascular and Thoracic Surgery 27 Demographic, procedural and outcome data were extracted from a prospectively institutional TEVAR database with a total of 418 patients treated for various thoracic aortic pathologies. Patient characteristics are given in Table 1. Open CoA repair had been performed by Dacron patch aortoplasty in nine and subclavian flap aortoplasty in 2 patients at a median age of 20 years (range: 2 43 years). Subsequent endovascular repair for postoperative pseudoaneurysm formation was undertaken at a median of 32 years after the CoA repair (range: years). Seven of 11 patients were asymptomatic and diagnosis was made by routine follow-up computed tomographic (CT) screening. The median diameter of the pseudoaneurysms in the asymptomatic patients was 56 mm (range: mm). Out of 4 symptomatic patients, one presented with haemoptysis with CTA showing a contained ruptured pseudoaneurysm with a maximal diameter of 84 mm; a second patient experienced haemodynamic collapse and CT work-up detected contained rupture of a 49-mm-large pseudoaneurysm. One patient presented with bilateral leg ischaemia caused by a severe restenosis, located distally to a pseudoaneurysm measuring 54 mm at the site of the CoA repair. The last of the 4 symptomatic patients had back pain and CT revealed a contained ruptured, 75-mm-large pseudoaneurysm. All 4 symptomatic patients had no radiological follow-up after the open coarcation repair. Seven patients (64%) were on antihypertensive medication at presentation. Endovascular procedure Lengths of proximal (PLZ) and distal landing zones (DLZ) were intended to be at least 20 mm and were preoperatively evaluated using 3D reconstruction software and centreline measurements (intuition, TeraRecon or OsiriX PRO). Oversizing of the endoprosthesis was 15 20%. All TEVAR procedures undertaken before October 2010 were performed in an operation theatre equipped with an Axiom U imaging system (Siemens Healthcare, Erlangen, Germany). Since October 2010, TEVAR is performed in a hybrid operation theatre featuring an Artis Zeego multiaxis imaging system (Siemens Healthcare, Erlangen, Germany). Adenosine-induced cardiac arrest (n = 5), and since March 2009 rapid pacing (n = 4) were used for exact stent-graft placement under general anaesthesia. Patients received single-shot antibiotic prophylaxis and 3000 IU of heparin before sheath insertion. Transfemoral cut-down was performed in 8 patients, iliac conduit due to small access vessels in 2 patients and a percutaneous approach in 1 patient. In 3 patients, right-brachial access was used for endograft deployment over a femoro-brachial wire. Angiography of the aortic arch was routinely performed over a percutaneous transfemoral approach with the exception of the 3 cases with additional right-brachial access. The Gore TAG and, since 2010, the Gore C-TAG stent-graft device were used in 2 and 5 cases, respectively. The Medtronic Valiant, Cook Zenith, LeMaitre Endofit and NuMED covered CP Stent were used each in a patient. Follow-up and definitions Zones of endograft attachment, outcomes, technical and clinical success were defined according to the reporting standards for TEVAR [8]. Technical success was defined as successful exclusion of the aneurysm at completion angiography. Follow-up included history, physical examination, CT angiography or magnetic resonance (MR) angiography, and plain chest radiography before discharge, at 6 months, at 1 year and annually thereafter. Duplex ultrasound was performed to assess carotid subclavian bypass grafts, when necessary. Postoperatively, all patients were put on lifelong aspirin therapy. The median followup was 60 months (range: months). No patient was lost to follow-up. RESULTS Technical outcome Adjunctive procedures at the PLZ were required in 7/11 patients. In 2 patients, proximal fixation was in Zone 1, with prior right ORIGINAL ARTICLE Table 1: Patients characteristics (n = 11) Patient Sex/age at CoA repair in years Type of surgical reconstruction Presentation at TEVAR Size of pseudoaneurysm (mm) Age at TEVAR in years 1 Male/39 Patch aortoplasty Haemoptysis due to aortobronchial fistula Male/23 Patch aortoplasty Routine screening Male/29 Patch aortoplasty Routine screening Female/28 Subclavian flap aortoplasty Routine screening Male/2 12 End-to-end anastomosis Subclavian flap aortoplasty Routine screening also demonstrating recurrent coarctation Male/9 Patch aortoplasty Routine screening Female/18 Patch aortoplasty Routine screening Female/43 Patch aortoplasty Routine screening Male/16 Patch aortoplasty Collapse due to contained aortic rupture Male/19 Patch aortoplasty Lower extremity ischaemia Male/20 Patch aortoplasty Back pain due to contained aortic rupture CoA: coarctation of the aorta; TEVAR: thoracic endovascular aortic repair.

3 28 D. Kotelis et al. / Interactive CardioVascular and Thoracic Surgery carotid to left subclavian bypass grafting with reimplantation of the left common carotid artery (LCCA) in one patient and right to left carotid carotid bypass and vascular plug occlusion of the LSA in the other. In 6 patients, the PLZ was in Zone 2 and primary LSA revascularization was performed in all elective cases (3/6 patients). In one emergency patient, the LSA was occluded with a vascular plug before placement of the thoracic stent graft to prevent a Type II endoleak. In another 2 emergency cases, the LSA was overstented without prior revascularization. In one patient, the stent graft partially covered the ostium of the LCCA; therefore, a right-to-left carotid carotidal bypass was required. Technical success was achieved in 10/11 patients (91%) (Fig. 1). In one patient, despite femoro-brachial wire position, the stent-graft device (LeMaitre Endofit ) could not be delivered due to severe tortuosity of the distal aortic arch (Fig. 2). The patient received secondary elective open surgical repair of the 65-mm-large pseudoaneurysm. The median number of implanted endoprostheses per patient was 1 (range: 1 5). In 2 patients, implantation of multiple devices was required in order to achieve complete sealing due to repeated stent-graft dislocation during deployment (Table 2). The median procedure and fluoroscopy time were 105 min (range: min) and 15 min (range: min), respectively. The median intensive care unit stay was 2 days (range: 1 12 days). The median in-hospital stay was 12 days (range: 8 22 days). Clinical outcome Early results. There was no in-hospital or 30-day mortality. The stroke rate was 18% (2/11). These 2 patients suffered a right-sided cerebral stroke on the first and third postoperative days, respectively. Equally, both developed mild left-sided hemiparesis and fully recovered after neuro-rehabilitation. There was no spinal cord injury. The overall reintervention rate was 33% (3/10). In one patient, postoperative CT angiography revealed a Type Ib endoleak that had not been detected during TEVAR. The patient underwent secondary distal stent-graft extension. In another patient, FU CT angiography detected partial coverage of the LCCA ostium. The patient was asymptomatic. A bare metal balloon-expandable stent (Cordis Genesis ) was implanted at the ostium of the LCCA 1 year postoperatively, in order to prevent LCCA occlusion. One of 4 patients with LSA coverage without prior revascularization developed arm claudication and underwent left subclavian bypass 1 year after the TEVAR procedure. Long-term results. Clinical success after a median follow-up of 60 months was achieved in 10/11 patients, excluding the patient requiring open surgical repair. Pseudoaneurysm sac shrinkage was observed in 9/10 patients after TEVAR, whereas, in the remaining patient, the aneurysm sac remained stable. The Type II endoleak rate was 10% (1/10; intercostal artery). One patient died of amyotrophic lateral sclerosis 15 months after TEVAR, yielding a total mortality rate of 9% (1/11). So far, FU in all other patients is uneventful. DISCUSSION Figure 1: 3D volume-rendered CT angiography (posterior view) shows the successful exclusion of a pseudoaneurysm following TEVAR. TEVAR: thoracic endovascular aortic repair; CT: computed tomography. The herein reported experience with endovascular treatment of post-coa pseudoaneurysms demonstrates that TEVAR is feasible in elective and emergency cases, yielding durable results in the long term. Nevertheless, the procedures can be very challenging and carry potential pitfalls, which are highlighted below. Long-term survival of patients undergoing open surgical repair for CoA is good, approaching 90% after 25 years [1, 2]. Yet, the development of post-coa pseudoaneurysms is a common finding in these patients, especially with patch aortoplasty using synthetic materials such as Dacron (8/11 patients in this series) with an associated risk of aneurysm formation in up to 51% of patients [1, 2]. Another factor holding an increased risk of aneurysm development is advanced age at the time of coarctation repair, with the threshold found to be 13.5 years of age [2]. Thus, the American Heart Association/American College of Cardiology guidelines recommend that all patients after CoA repair should undergo CT or magnetic resonance imaging (MRI) imaging at least every 5 years to assess for pseudoaneurysm formation. As a result, the majority of our patients were diagnosed during routine screening before becoming symptomatic [9]. On the other hand, all patients presenting symptoms had no radiological follow-up after the open coarctation repair. Although maximal aortic diameter remains the strongest predictor of rupture for atherosclerotic aneurysms, the unique morphology of post-coa aneurysms evokes scepticism with regard to the application of such data to this specific patient population [3]. Rupture was observed also at smaller diameters in these patients and so most surgeons advocate treatment in morphologies raising concerns regarding rupture, even if the size

4 D. Kotelis et al. / Interactive CardioVascular and Thoracic Surgery 29 ORIGINAL ARTICLE Figure 2: Anterior (A) and posterior (B) views of a 49-mm large pseudoaneurysm. Note the small access vessels and the gothic tortuous arch. Table 2: Endovascular repair and outcome Patient PLZ Primary LSA revascularization Stent grafts Complications Secondary procedures FU (months) 1 2 No Gore TAG Right cerebral stroke LSA transposition Cook Zenith Gore TAG Type Ib endoleak Distal extension with Gore 114 TAG ITT LSA transposition LeMaitre Endofit (ITT) Open repair 5 3 NuMED Covered CP Stent CSBPG LeMaitre Endofit Partial LCCA coverage Secondary LCCA stent LSA transposition Gore CTAG Type II endoleak via 53 intercostal artery 8 1 CSBPG and left CCA Gore CTAG , reimplantation 9 2 Vascular plug occlusion of the Gore CTAG , , Partial LCCA coverage Intraoperative 46 LSA , , Right cerebral stroke Left arm claudication CCBPG Secondary LSA bypass 10 2 No Gore CTAG CCBPG and vascular plug occlusion of the LSA Medtronic Valiant , Gore CTAG , PLZ: proximal landing zone; LSA: left subclavian artery; CCA: common carotid artery; CSBPG: carotid subclavian bypass; CCBPG: carotid carotidal bypass; LCCA: left common carotid artery; FU: follow-up; ITT: intention to treat.

5 30 D. Kotelis et al. / Interactive CardioVascular and Thoracic Surgery does not exceed 6 cm, as in 2 of our patients [3]. As previously reported, rupture can be fatal in up to 38% of cases and so early treatment of these middle-aged patients is crucial [3, 4]. Endovascular repair of post-coa pseudoaneurysms is an appealing alternative to redo open surgical management, which is associated with marked morbidity and mortality rates of more than 10% [3, 10]. Yet, in contrast to studies reporting 100% technical success [6, 11], this analysis clearly demonstrates that TEVAR can be very challenging in this patient group. Starting with the endograft choice, this must be conformable to the steep aortic arch in this rather young patient population. Owing to the proximity of the pathology to the supra-aortic vessels, a proximal stentgraft anchorage not exceeding Zone 2 is required in the majority of patients. LSA coverage without revascularization may not be as well tolerated in this rather young and active patient population with a higher likelihood of arm claudication in comparison to older patients [3, 12]. Regarding the stent-graft length, the necessitated device length is rather short in these patients with need for 100-mm-long devices in most cases as seen in this series. In cases where longer stent grafts are needed, tapered devices might be of benefit to conform to the calibre mismatch with large proximal and smaller distal aortic diameters in post-coa patients. Furthermore, recurrent coarctation is not rare and may occasionally complicate the endovascular repair procedure. In these cases, ordinary stent grafts may not yield sufficient radial strength to treat the stenosis. This was the reason why a covered balloonexpandable stent was chosen in 1 patient with concomitant recurrent coarctation along with pseudoaneurysm formation. The iliac vessels in these typically younger non-aneurysmatic patients are often small and, given the necessity of 20-F sheaths or larger, the need for prosthetic conduits, as in 2 patients in this series, is above the overall TEVAR average [3]. Furthermore, delivery of the device into the aortic arch can be very demanding due to aortic tortuosity and, as observed in one of our patients, endovascular treatment can fail for this reason. An alternative to the femoro-brachial guidewire that was used in this case to facilitate endograft passage into the aortic arch can be a trans-septal through-and-through guidewire from the right common femoral vein to the left common femoral artery, as reported by Kölbel et al. [13, 14]. Despite its potential complications (e.g. cardiac tamponade, mechanical damage to the valves etc.), the trans-septal approach can be beneficial in order to optimize endograft control in selected patients and so reduce the risk of unintentional coverage of supra-aortic vessels, as was partly the case in 2 of our patients, or of stent-graft dislocation as was the case in 2 of our patients [14]. The risk of stent-graft dislocation is higher when aneurysms coincide with restenosis in some post-coa patients and with stent grafts without active fixation. Rapid pacing is very helpful in the authors experience for exact stent-graft placement in the aortic arch [15]. Furthermore, a right-brachial access for angiographic reasons can be performed as an alternative to the transfemoral access and can be very helpful in such challenging TEVAR procedures. On-site cardiothoracic surgery backup is essential in case of technical failure that cannot be managed by endovascular means, and open conversion might be required immediately. Stroke risk during TEVAR was directly associated with the atheromatous burden of the aortic arch and the PLZ before [16]. In the post-coa patient population with the proximity of the lesions to the supra-aortic vessels, patients are at increased risk of stroke, as seen also in this analysis. Spinal cord injury was observed in this TEVAR population since these patients mostly require a shortsegment aortic coverage of the proximal descending aorta. Close radiological follow-up by CT or MRI is important in order to maintain long-term clinical success in this young patient population. This study is limited by the retrospective design and potential selection bias, the small sample size and the missing open surgical control group. CONCLUSIONS Endovascular repair of post-coa pseudoaneurysms is feasible and meanwhile first-line treatment in many centres. Owing to the specific aortic morphology in this patient population, TEVAR is challenging due to the morphology with the need of LSA management, relevant intraoperative revision rates and unpredicted need of multiple devices. Long-term follow-up shows satisfactory results. On-site cardiothoracic surgery backup is essential in case open conversion is required. ACKNOWLEDGEMENTS The authors thank Elke Klövekorn for her precious help with the revision of the manuscript. Conflict of interest: none declared. REFERENCES [1] Cramer JW, Ginde S, Bartz PJ, Tweddell JS, Litwin SB, Earing MG. Aortic aneurysms remain a significant source of morbidity and mortality after use of Dacron( ) patch aortoplasty to repair coarctation of the aorta: results from a single center. Pediatr Cardiol 2013;34: [2] von Kodolitsch Y, Aydin MA, Koschyk DH, Loose R, Schalwat I, Karck M et al. 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