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Surgical Repair of an Aneurysm of the Innominate Artery with Fistulization into the Trachea
Surgical Repair of an Aneurysm of the Innominate Artery with Fistulization into the Trachea Jean-Philippe Guibaud, MD, Marie Nadine Laborde, MD, Jean Dubrez, MD, Nicolas Elia, MD, Jan Remes, MD, Francesco Rosato, MD, Xavier Roques, MD, and Eugene Baudet, MD, Bordeaux, France
This report describes a 43-year-old patient presenting with an aneurysm located at the junction between the innominate artery and aorta, with spontaneous fistulization into the trachea. Emergency treatment of this unusually located lesion was undertaken to prevent intratracheal rupture. After ligation of the innominate artery, right common carotid artery, and right subclavian artery due to the risk of infection, the tracheal fistula was treated by direct closure and exclusion using an autologous pericardial flap. The procedure was performed under extracorporeal circulation and circulatory arrest with profound hypothermia.
Isolated aneurysms located at the junction between the innominate artery (IA) and aorta are rare.1-3 The main risks are rupture4 and compression of neighboring organs.1,5 Spontaneous fistulization into the trachea has not been reported in the literature. Surgical repair is the only effective therapy. In this case report, we discuss use of extracorporeal circulation (ECC), cerebral protection, vascular reconstruction, and treatment of tracheal fistula.
CASE REPORT A 43-year-old man (weight, 76 kg; height, 1.90 m) was hospitalized for recurrent syncope and tonic-clonic seizures ongoing for 1 week. The patient had no significant medical history and his only risk factor for cardiovascular
Service de Chirurgie Cardiovasculaire and Service de Chirurgie Thoracique, Hoˆpital du Haut-Le´veˆque, CHU de Bordeaux, Bordeaux, France. Correspondence to: J.P. Guibaud, MD, Service de Chirurgie Cardiovasculaire, Hoˆpital du Haut Leveˆque, 5 avenue de Magellan, 33604 Pessac, France. Ann Vasc Surg 2001; 15: 412-414 DOI: 10.1007/s100160010070 © Annals of Vascular Surgery Inc. Published online: April 26, 2001 412
disease was moderate smoking. On physical examination he had a heart rate of 80 beats/min, arterial blood pressure of 110/70, absence of right radial and carotid pulses, and bruits in both subclavian arteries and the left carotid artery. A thoracic computed tomography (CT) scan revealed a large aneurysm at the junction between the IA and the aortic arch with presence of intraaneurysmal thrombus. The aneurysm included the IA and left common carotid artery (LCCA) but the left subclavian artery (LSCA) was intact. Superior vena cava syndrome was present at a subclinical stage. A brain CT scan depicted a round, hypodense area of ischemic origin, located in the anterior aspect of the right internal capsule of the brain. Thoracic aortography (Figs. 1 and 2) demonstrated thrombosis of the IA and compression of the LCCA. Narrowing >50% was observed at the origin of the LSCA. Following respiratory compromise with pneumopathy and fever at 39°C, the patient was intubated and ventilated. Two episodes of hemoptysis occurred during tracheobronchial aspiration. Surgery was undertaken to prevent imminent intratracheal rupture. Under general anesthesia, the left iliac arteries were exposed and ECC with total hemodilution and profound hypothermia was installed. Median sternotomy was performed at 26°C and the ascending aorta was clamped at 23°C with the heart in ventricular fibrillation. Two liters of cold crystalloid cardioplegia was administered in an antegrade manner. The aneurysm involved the entire IA
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Case reports 413
Fig. 1. Angiography showing compression of the left common carotid artery and stenosis of the left subclavian artery. Notice the presence of thrombus in the right common carotid artery and right subclavian artery. Fig. 2. Angiography (late view) showing revascularization of the right subclavian artery by the right vertebral artery.
including the junction with the aortic arch. The diameter of the IA was 35 mm at the origin and 65 mm at the widest point. The aneurysm compressed and displaced the trachea, LCCA, and LSCA. The innominate vein was flattened. After aneurysmotomy under profound hypothermia and circulatory arrest and removal of intraaneurysmal thrombus, an arteriotracheal fistula 5 mm in diameter was located 40 mm above the carina. Exclusion aneurysmorraphy was performed with ligation of the right common carotid artery and right subclavian artery. The aortic arch was reconstructed using a woven polyester patch. Rewarming was then undertaken. Circulatory arrest lasted 26 min. The fistula was closed using four direct sutures. A 30 × 30 mm pericardial pedicle flap was applied to the fistulous area and attached by direct suture. Rewarming, resuscitation, and hemostasis were uneventful. At the end of the procedure the patient was reintubated under endoscopic control to exclude the fistulous tracheal segment and allow placement of an occlusive balloon distal to the fistula. Since standard intubation catheters were not long enough for this purpose, it was necessary to telescope two catheters together. The patient was extubated 44 hr after the procedure. No cardiovascular or neurological complications occurred. Histological examination of the surgical specimen confirmed that the aneurysm was of atherosclerotic origin. The patient was rehospitalized 4 months later for syncope without seizure or micturition. A cervical duplex scan confirmed the diagnosis of right subclavian steal syndrome. Since the patient refused to undergo surgical revascularization, regular surveillance was instituted. One year later the patient was leading a normal life.
DISCUSSION This is the first case report describing spontaneous fistulization of an aneurysm at the junction between the IA and aorta with spontaneous rupture into the trachea. This case illustrates the severity of aneurysm in this unusual location.3,5,6 Surgical repair must be undertaken on an emergency basis. Our strategy consisted of beginning ilioiliac ECC prior to median sternotomy1,2 to avoid intraoperative rupture and allow repair of the aortic arch.5,7,8 In patients with severe tracheal compression, ECC can be initiated before nasotracheal intubation under local anesthesia.9 If reconstruction of the aortic arch is planned, profound hypothermia offers the best means of cerebral protection5,8 since compression of neck vessels7 often prevents use of other methods, i.e., antegrade or retrograde cerebral perfusion. Revascularization of cervical arteries is mandatory to prevent subclavian steal syndrome.1,2,5,8 However, infection can be a limiting factor in this regard. In cases involving low-grade infection of the operative field, an autologous conduit (artery or vein) or arterial homograft can be used for arterial reconstruction.8 In our patient, however, the risk of graft infection was high because of the presence of pneumopathy and the necessity for postoperative mechanical ventilation. In the absence of tracheomalacia,10 tracheal fis-
414
Case reports
tula can be repaired by direct closure using separate sutures. Use of a pericardial flap limits the risk of recurrent fistula.5 Endoscopic inspection of the nasotracheal tube is necessary to ascertain exclusion of the fistulous area. If the fistula is located in the distal trachea, it may be necessary to assemble catheters as described in this case. ECC is mandatory for repair of a large aneurysm involving the junction between the IA and aorta located proximal to the aortic arch. Choice of cerebral protection depends on the patency status of the supraaortic vessels. Reconstruction of the supraaortic arteries is mandatory to avoid subclavian steal, but it may be necessary to postpone this step until resolution of infection. REFERENCES 1. Thomas V. Intrathoracic aneurysms of the innominate and subclavian arteries. J Thorac Cardiovasc Surg 1972;63:461471. 2. Schumacher PD, Wright CB. Management of arteriosclerotic
Annals of Vascular Surgery
3.
4.
5.
6.
7.
8. 9. 10.
aneurysm of the innominate artery. Surgery 1979;85:489495. Brewster DC, Moncure AC, Darling RC, et al. Innominate artery lesions: problems and lessons learned. J Vasc Surg 1985;2:99-112. Stolf NAG, Bittencourt D, Verginelli G, Zerbini EJ. Surgical treatment of ruptured aneurysm of the innominate artery. Ann Thorac Surg 1983;35:394-399. Bower TC, Pairolero PC, Hallett JW Jr, et al. Brachiocephalic aneurysm: the case for early recognition and repair. Ann Vasc Surg 1991;5:125-132. Ketonen P, Meurala H, Harjola PT, et al.. Management of arteriosclerotic aneurysm of the innominate and subclavian arteries. Scand J Thorac Cardiovasc Surg 1983;17:129-133. McFarland JJ, Khan MB, Bellows CF, et al. Superior vena cava syndrome caused by aneurysm of the innominate artery. Ann Thorac Surg 1995;59:227-229. Bower TC. Aneurysm of the great vessels and their branches. Semin Vasc Surg 1996;9:134-146. MacGillivray RG. Tracheal compression caused by aneurysm of the aortic arch. Anaesthesia 1985;40:270-277. Matsushita T, Nakamura A, Kawachi H, et al. A case report of surgical treatment for aneurysm of the brachiocephalic artery associated with tracheal stenosis. J Jpn Assn Thorac Surg 1997;45:754-758.
This report describes a 43-year-old patient presenting with an aneurysm located at the junction between the innominate artery and aorta, with spontaneous fistulization into the trachea. Emergency treatment of this unusually located lesion was undertaken to prevent intratracheal rupture. After ligation of the innominate artery, right common carotid artery, and right subclavian artery due to the risk of infection, the tracheal fistula was treated by direct closure and exclusion using an autologous pericardial flap. The procedure was performed under extracorporeal circulation and circulatory arrest with profound hypothermia.
Isolated aneurysms located at the junction between the innominate artery (IA) and aorta are rare.1-3 The main risks are rupture4 and compression of neighboring organs.1,5 Spontaneous fistulization into the trachea has not been reported in the literature. Surgical repair is the only effective therapy. In this case report, we discuss use of extracorporeal circulation (ECC), cerebral protection, vascular reconstruction, and treatment of tracheal fistula.
CASE REPORT A 43-year-old man (weight, 76 kg; height, 1.90 m) was hospitalized for recurrent syncope and tonic-clonic seizures ongoing for 1 week. The patient had no significant medical history and his only risk factor for cardiovascular
Service de Chirurgie Cardiovasculaire and Service de Chirurgie Thoracique, Hoˆpital du Haut-Le´veˆque, CHU de Bordeaux, Bordeaux, France. Correspondence to: J.P. Guibaud, MD, Service de Chirurgie Cardiovasculaire, Hoˆpital du Haut Leveˆque, 5 avenue de Magellan, 33604 Pessac, France. Ann Vasc Surg 2001; 15: 412-414 DOI: 10.1007/s100160010070 © Annals of Vascular Surgery Inc. Published online: April 26, 2001 412
disease was moderate smoking. On physical examination he had a heart rate of 80 beats/min, arterial blood pressure of 110/70, absence of right radial and carotid pulses, and bruits in both subclavian arteries and the left carotid artery. A thoracic computed tomography (CT) scan revealed a large aneurysm at the junction between the IA and the aortic arch with presence of intraaneurysmal thrombus. The aneurysm included the IA and left common carotid artery (LCCA) but the left subclavian artery (LSCA) was intact. Superior vena cava syndrome was present at a subclinical stage. A brain CT scan depicted a round, hypodense area of ischemic origin, located in the anterior aspect of the right internal capsule of the brain. Thoracic aortography (Figs. 1 and 2) demonstrated thrombosis of the IA and compression of the LCCA. Narrowing >50% was observed at the origin of the LSCA. Following respiratory compromise with pneumopathy and fever at 39°C, the patient was intubated and ventilated. Two episodes of hemoptysis occurred during tracheobronchial aspiration. Surgery was undertaken to prevent imminent intratracheal rupture. Under general anesthesia, the left iliac arteries were exposed and ECC with total hemodilution and profound hypothermia was installed. Median sternotomy was performed at 26°C and the ascending aorta was clamped at 23°C with the heart in ventricular fibrillation. Two liters of cold crystalloid cardioplegia was administered in an antegrade manner. The aneurysm involved the entire IA
Vol. 15, No. 3, 2001
Case reports 413
Fig. 1. Angiography showing compression of the left common carotid artery and stenosis of the left subclavian artery. Notice the presence of thrombus in the right common carotid artery and right subclavian artery. Fig. 2. Angiography (late view) showing revascularization of the right subclavian artery by the right vertebral artery.
including the junction with the aortic arch. The diameter of the IA was 35 mm at the origin and 65 mm at the widest point. The aneurysm compressed and displaced the trachea, LCCA, and LSCA. The innominate vein was flattened. After aneurysmotomy under profound hypothermia and circulatory arrest and removal of intraaneurysmal thrombus, an arteriotracheal fistula 5 mm in diameter was located 40 mm above the carina. Exclusion aneurysmorraphy was performed with ligation of the right common carotid artery and right subclavian artery. The aortic arch was reconstructed using a woven polyester patch. Rewarming was then undertaken. Circulatory arrest lasted 26 min. The fistula was closed using four direct sutures. A 30 × 30 mm pericardial pedicle flap was applied to the fistulous area and attached by direct suture. Rewarming, resuscitation, and hemostasis were uneventful. At the end of the procedure the patient was reintubated under endoscopic control to exclude the fistulous tracheal segment and allow placement of an occlusive balloon distal to the fistula. Since standard intubation catheters were not long enough for this purpose, it was necessary to telescope two catheters together. The patient was extubated 44 hr after the procedure. No cardiovascular or neurological complications occurred. Histological examination of the surgical specimen confirmed that the aneurysm was of atherosclerotic origin. The patient was rehospitalized 4 months later for syncope without seizure or micturition. A cervical duplex scan confirmed the diagnosis of right subclavian steal syndrome. Since the patient refused to undergo surgical revascularization, regular surveillance was instituted. One year later the patient was leading a normal life.
DISCUSSION This is the first case report describing spontaneous fistulization of an aneurysm at the junction between the IA and aorta with spontaneous rupture into the trachea. This case illustrates the severity of aneurysm in this unusual location.3,5,6 Surgical repair must be undertaken on an emergency basis. Our strategy consisted of beginning ilioiliac ECC prior to median sternotomy1,2 to avoid intraoperative rupture and allow repair of the aortic arch.5,7,8 In patients with severe tracheal compression, ECC can be initiated before nasotracheal intubation under local anesthesia.9 If reconstruction of the aortic arch is planned, profound hypothermia offers the best means of cerebral protection5,8 since compression of neck vessels7 often prevents use of other methods, i.e., antegrade or retrograde cerebral perfusion. Revascularization of cervical arteries is mandatory to prevent subclavian steal syndrome.1,2,5,8 However, infection can be a limiting factor in this regard. In cases involving low-grade infection of the operative field, an autologous conduit (artery or vein) or arterial homograft can be used for arterial reconstruction.8 In our patient, however, the risk of graft infection was high because of the presence of pneumopathy and the necessity for postoperative mechanical ventilation. In the absence of tracheomalacia,10 tracheal fis-
414
Case reports
tula can be repaired by direct closure using separate sutures. Use of a pericardial flap limits the risk of recurrent fistula.5 Endoscopic inspection of the nasotracheal tube is necessary to ascertain exclusion of the fistulous area. If the fistula is located in the distal trachea, it may be necessary to assemble catheters as described in this case. ECC is mandatory for repair of a large aneurysm involving the junction between the IA and aorta located proximal to the aortic arch. Choice of cerebral protection depends on the patency status of the supraaortic vessels. Reconstruction of the supraaortic arteries is mandatory to avoid subclavian steal, but it may be necessary to postpone this step until resolution of infection. REFERENCES 1. Thomas V. Intrathoracic aneurysms of the innominate and subclavian arteries. J Thorac Cardiovasc Surg 1972;63:461471. 2. Schumacher PD, Wright CB. Management of arteriosclerotic
Annals of Vascular Surgery
3.
4.
5.
6.
7.
8. 9. 10.
aneurysm of the innominate artery. Surgery 1979;85:489495. Brewster DC, Moncure AC, Darling RC, et al. Innominate artery lesions: problems and lessons learned. J Vasc Surg 1985;2:99-112. Stolf NAG, Bittencourt D, Verginelli G, Zerbini EJ. Surgical treatment of ruptured aneurysm of the innominate artery. Ann Thorac Surg 1983;35:394-399. Bower TC, Pairolero PC, Hallett JW Jr, et al. Brachiocephalic aneurysm: the case for early recognition and repair. Ann Vasc Surg 1991;5:125-132. Ketonen P, Meurala H, Harjola PT, et al.. Management of arteriosclerotic aneurysm of the innominate and subclavian arteries. Scand J Thorac Cardiovasc Surg 1983;17:129-133. McFarland JJ, Khan MB, Bellows CF, et al. Superior vena cava syndrome caused by aneurysm of the innominate artery. Ann Thorac Surg 1995;59:227-229. Bower TC. Aneurysm of the great vessels and their branches. Semin Vasc Surg 1996;9:134-146. MacGillivray RG. Tracheal compression caused by aneurysm of the aortic arch. Anaesthesia 1985;40:270-277. Matsushita T, Nakamura A, Kawachi H, et al. A case report of surgical treatment for aneurysm of the brachiocephalic artery associated with tracheal stenosis. J Jpn Assn Thorac Surg 1997;45:754-758.