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Simultaneous Hybrid Treatment of Aneurysmal Aberrant Right Subclavian Artery Nicola Troisi, M.D., Emiliano Chisci, M.D., Leonardo Ercolini, M.D., Clara Pigozzi, M.D., and Stefano Michelagnoli, M.D. Department of Surgery, Vascular and Endovascular Surgery Unit, San Giovanni di Dio Hospital, Florence, Italy ABSTRACT We review the literature and report our experience on the simultaneous hybrid treatment (open and endovascular) of two symptomatic aneurysms of an aberrant right subclavian artery (ARSA). At followup (four years and one year, respectively) both patients were alive and free of symptoms. Hybrid treatment of an aneurysmal ARSA is a safe and effective procedure. However, a larger series of patients with long-term follow-up is necessary to determine the role of this technique. doi: 10.1111/jocs.12467 (J Card Surg

2015;30:80–84) Aberrant right subclavian artery (ARSA), also referred to as lusoria artery, is one of the most common congenital anomalies of the aortic arch with an incidence of 0.5% to 2%.1 Over time ARSA tends to become aneurysmal. In most cases the ARSA is asymptomatic and the diagnosis is incidental.2 Only 5% of patients experience symptoms, including chest pain, dyspnea, cough, and compression dysphagia (‘‘dysphagia lusoria’’).3 Since the first report by Gross in 1946,4 many surgical techniques were adopted; all these techniques include a left thoracotomy or a median sternotomy. The postoperative mortality ranges from 7.7% to 25%.5 Recently, hybrid approaches have emerged, including extra-anatomical surgical bypasses of the supraaortic vessels, and the deployment of a thoracic stentgraft.6 This mini-invasive hybrid approach seems to reduce the rate of postoperative complications.7 We report our experience with this technique and review the literature. PATIENT PROFILES Patient 1 A 75-year-old male was admitted with dysphagia. His medical history was notable for smoking, arterial hypertension, and mild chronic renal failure. Conflict of interest: The authors acknowledge no conflict of interest in the submission. Address for correspondence: Nicola Troisi, M.D., Department of Surgery, Vascular and Endovascular Surgery Unit, San Giovanni di Dio Hospital, Via di Torregalli 3, 50143 Florence, Italy. Fax þ390556932268; e-mail: [email protected]

The patient had recent-onset dysphagia associated with weight loss of 7 kg in three months. A computed tomography angiography (CTA) showed a 62-mm ARSA with a saccular aneurysm of the thoracic aorta (41 mm) (Fig. 1). The patient underwent a simultaneous hybrid procedure. The intervention was carried out under general anesthesia in a hybrid operating room; somatosensory evoked potentials (SEPs) and electroencephalography (EEG) were used to monitor evoked potentials during surgical intervention. The first step was the exposure of both the common carotid and subclavian arteries with a bilateral supraclavicular incision. Both subclavian arteries were ligated and then subsequently reconstructed with a subclavian-to-carotid transposition on the right and a carotid-subclavian prosthetic e-polytetrafluoroethylene (e-PTFE; W. L. Gore and Associates, Inc., Flagstaff, AZ, USA) bypass graft on the left. The second step was the exposure of the right common femoral artery and the deployment of an aortic endograft (TAG, TGE454515; W. L. Gore and Associates, Inc.) via a 0.035-in, 260-cm-long super-stiff wire (Lunderquist; Cook Medical, Inc., Bloomington, IN, USA). The proximal landing zone was just distal to the left common carotid artery. The final angiogram showed exclusion of the aneurysmal sac from the blood flow and the patency of the supra-aortic vessels. The postoperative period was free of complications. The patient was discharged on day 6 in good condition. The dysphagia lasted for about three months. At fouryear follow-up the patient was alive and free of symptoms; a four-year CT-scan documented the

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Figure 1. Patient 1: preoperative CT-scan (a) and its graphic representation (b).

exclusion of the aneurysmal ARSA and the patency of the supra-aortic vessels (Fig. 2). IRB (Institutional Review Board) approval was obtained to report this case. Patient 2 A 85-year-old female was admitted with dyspnea and chest pain. She had a previous history of smoking, arterial hypertension, hypercholesterolemia, type 2 diabetes, coronary artery disease, and severe valvular aortic stenosis. An electrocardiogram and cardiac enzymes were unremarkable. An urgent CT showed a 110-mm aneurysmal ARSA involving the distal aortic arch; furthermore, the patient

had a truncus bicaroticus with a common trunk for both common carotid arteries and the right subclavian artery (Fig. 3). The patient underwent an urgent hybrid simultaneous procedure, performed in a hybrid operating room under general anesthesia with neurological monitoring (SEPs and EEG). The first step was the exposure of both the common carotid and subclavian arteries with a bilateral supraclavicular incision. The right subclavian artery was ligated. The first part of the left subclavian artery was not anatomically accessible from the supraclavicular incision; so, the left subclavian artery was occluded with the deployment of multiple coils through the direct puncture of the artery. Then, the two subclavian arteries

Figure 2. Patient 1: four-year CT-scan (a) and its graphic representation (b).

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Figure 3. Patient 2: preoperative CT-scan (a) and its graphic representation (b).

were reconstructed with a subclavian-to-carotid transposition on the right and a carotid-subclavian prosthetic e-PTFE (W. L. Gore and Associates, Inc.) bypass on the left. The second step was the exposure of the right common femoral artery and the deployment of an aortic endograft (TAG, TGE343420; W. L. Gore and Associates, Inc.) via a 0.035-in, 260-cm-long super-stiff wire (Lunderquist; Cook Medical, Inc.). The proximal landing zone was just distal to the truncus bicaroticus. The final angiogram showed exclusion of the aneurysmal ARSA and the patency of the supra-aortic vessels. The postoperative period was complicated by an acute myocardial infarction. The patient was discharged

on day 12 in a rehabilitative center. At one-year followup the patient was alive and free of symptoms; the severe valvular aortic stenosis remained untreated. A one-year CT-scan documented the exclusion of the aneurysmal ARSA (maximum diameter about 6 cm) and the patency of the supra-aortic vessels (Fig. 4). IRB approval was obtained to report this case. REVIEW OF LITERATURE 8

Kopp et al. published a series on six patients treated with the hybrid approach, reporting encouraging midterm results; the first treated patient survived for 82 months after the repair.

Figure 4. Patient 2: one-year CT-scan (a) and its graphic representation (b).

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A recent metanalysis reviewed the results obtained in 39 patients with ARSA and Kommerell’s diverticulum undergoing hybrid treatment; the overall 30-day mortality rate was 7.7% and the mortality rate was significantly higher in patients when an aneurysmal ARSA was present.7 In this review, the 30-day morbidity rate was substantially high (17.9%). During the follow-up of these 39 patients (mean duration about 16 months) eight endoleaks occurred (20.5%); of these, three endoleaks were type I. The authors of this metanalysis concluded that hybrid exclusion using thoracic stent-graft and distal ligation with reconstruction should be one of the techniques to treat aneurysmal changes of an ARSA. However, the potential for endoleak, dysphagia, and ischemic symptoms caused by nonreconstruction of the ARSA mandate a lifelong surveillance. A more recent systematic review published in 2014 identified 31 reported cases of patients with ARSA who underwent endovascular repair (10 patients without an associated Kommerell’s diverticulum and 21 patients with a Kommerell’s diverticulum).9 The authors reported that 7/31 patients (22.6%) had postoperative complications and three patients died with an overall 30-day mortality of 9.7%. Furthermore, during the follow-up 4/31 patients (12.9%) had an endoleak (one type I, two type II, and one type IV). Table 1 summarizes the results reported in these two reviews. DISCUSSION Treatment of an aneurysmal ARSA is mandatory to relief the symptoms and/or to avoid rupture. In our experience, both patients had a large aneurysm and were symptomatic. In the more recent review published in 2014 only 3/31 patients (9.7%) were asymptomatic.9 Open surgical treatment is associated with a high rate of postoperative morbidity and mortality.5 In particular, the all-cause 30-day mortality rate ranges from 7.7% to 25%.5,7 We choose the hybrid mini-invasive approach for our two elderly and fragile patients. In the literature there are contradictory reports about the timing of the surgery (open and endovascular). Shennib and Diethrich6 reported the results obtained in two cases, one simultaneous and one staged. Our patients were symptomatic and we adopted a simultaneous approach, in order to avoid delay in the treatment and immediately obtain the relief of symptoms.

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We decided to use a less rigid graft, in order to obtain a better seal into the aortic arch. In both cases we used a Gore TAG, a next-generation device engineered for flexibility and conformability in tortuous and angulated arch anatomy. Another key point in this hybrid approach is the importance of ligation of the aberrant subclavian arteries when covered with stent grafts. At the distal end of the subclavian arteries, surgical ligation, or endovascular occlusion were most commonly performed to prevent retrograde blood flow. Endovascular occlusion devices can be placed in antegrade or retrograde fashion. Ligation/occlusion of the subclavian arteries is associated with a low risk of postoperative type II endoleak. In fact, in the review of 2014 only 2/31 type II endoleaks were reported; both patients did not undergo ligation/occlusion of the subclavian arteries.9 Some authors do not ligate the subclavian arteries, but in our patients we preferred to occlude the subclavian arteries in order to avoid retrograde blood flow into the aneurysmal sac and we did not record type II endoleaks during the follow-up. Finally, revascularization of the subclavian arteries is widely debated in the literature. Revascularization is important to prevent ischemic symptoms in the arm and to reduce the risk of posterior cerebral ischemia. In the review of 2012 the aberrant subclavian artery was completely occluded in 31 of 39 cases, but reconstruction was performed only in 27 patients; no symptoms of upper limb or cerebral ischemia were reported in the four patients whose aberrant subclavian artery was not reconstructed.7 Furthermore, in the review of 2014 the authors reported that intentional coverage of left subclavian artery without revascularization was performed in only three patients, while the remaining 28 patients underwent surgical revascularization. The same authors reported that three patients had postoperative right arm ischemia (one critical ischemia and two claudication), but they did not report if the patients affected were those not revascularized.9 In both the reviews the authors strongly recommended the revascularization of the subclavian arteries, especially when overstenting of the left subclavian artery is intended. In our experience, in all the subclavian arteries we performed a revascularization with a prosthetic bypass or a transposition and we did not record any cerebral or upper limb ischemic symptoms.

TABLE 1 Results of Hybrid Treatment of Aneurysmal Aberrant Right Subclavian Artery Author (Year) Yang et al. (2012)7 Vucemilo et al. (2014)9

Number of Patients

Symptomatic

Subclavian Occlusion

Subclavian Revascularization

30-Day Major Morbidity

30-Day Mortality

39 31

27 (69.2%) 28 (90.3%)

31 (79.5%) N/A

27 (69.2%) 28 (90.3%)

7 (17.9%) 7 (22.6%)

3 (7.7%) 3 (9.7%)

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In conclusion, hybrid treatment of an aneurysmal ARSA could be considered a feasible, safe, and effective procedure. However, long-term results and larger multicenter experiences are needed. Acknowledgment: The authors wish to thank Mrs. Cristina Cantini for the support in the iconography.

REFERENCES 1. Freed K, Low VH: The aberrant subclavian artery. AJR Am J Roentgenol 1997;168:481–484. 2. Turkenburg JL, Versteegh MI, Shaw PC: Case report: Aneurysm of an aberrant right subclavian artery diagnosed with MR imaging. Clin Radiol 1994;49:837–839. 3. Asherson N, Bayford D: His syndrome and sign of dysphagia lusoria. Ann R Coll Surg Engl 1979;61:63–67.

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4. Gross RE: Surgical treatment for dysphagia lusoria. Ann Surg 1946;124:532–534. 5. Kieffer E, Bahnini A, Koskas F: Aberrant subclavian artery: Surgical treatment in thirty-three adult patients. J Vasc Surg 1994;19: 100–109; discussion 110–1. 6. Shennib H, Diethrich EB: Novel approaches for the treatment of the aberrant right subclavian artery and its aneurysms. J Vasc Surg 2008;47:1066–1070. 7. Yang C, Shu C, Li M, et al: Aberrant subclavian artery pathologies and Kommerell’s diverticulum: A review and analysis of published endovascular/hybrid treatment options. J Endovasc Ther 2012;19:373–382. 8. Kopp R, Wizgall I, Kreuzer E, et al: Surgical and endovascular treatment of symptomatic aberrant right subclavian artery (arteria lusoria). Vascular 2007;15:84–91. 9. Vucemilo I, Harlock JA, Qadura M, et al: Hybrid repair of symptomatic aberrant right subclavian artery and Kommerell’s diverticulum. Ann Vasc Surg 2014;28:411–420.

Simultaneous hybrid treatment of aneurysmal aberrant right subclavian artery.

We review the literature and report our experience on the simultaneous hybrid treatment (open and endovascular) of two symptomatic aneurysms of an abe...
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