ARTICLE IN PRESS doi:10.1510/icvts.2009.227900
Interactive CardioVascular and Thoracic Surgery 10 (2010) 820–822 www.icvts.org
Case report - Congenital
Management of a pulsatile mass coming through the sternum. Pseudoaneurysm of ascending aorta 35 years after repair of tetralogy of Fallot Rizwan Attia*, Prem Venugopal, Donald Whitaker, Christopher Young Guy’s and St Thomas’ NHS Trust, Westminster Bridge Road, London, SE1 7EH, UK Received 10 November 2009; received in revised form 5 December 2009; accepted 9 December 2009
Abstract We describe a case of an ascending aortic pseudoaneurysm during long-term follow-up after repair of tetralogy of Fallot (TOF). The patient had a complex cardiac surgical history with multiple operations for the correction of TOF. The aneurysm was located at the presumed site of previous aortic cannulation. It was initially treated percutaneously with an Amplatzer姠 septal occluder device, with limited early success. After 12 months it was found to have migrated into the sac and open surgical repair was undertaken successfully. 䊚 2010 Published by European Association for Cardio-Thoracic Surgery. All rights reserved. Keywords: Tetralogy of Fallot; Pseudoaneurysm; Amplatzer姠 septal occluder device
1. Case A 64-year-old patient presented with dyspnoea, paroxysmal nocturnal dyspnoea, dysphagia and back pain. She had a complex cardiac surgical history starting with an unsuccessful Pott’s shunt for tetralogy of Fallot aged 4. This was followed by a modified Blalock-Taussig (BT) shunt aged 14. A total correction of Fallot (which included a trans annular patch and ligation of right BT shunt) was performed aged 28. Four years later she presented with a pulsating mass in the precordium. The right ventricular outflow tract was aneurysmal. This was excised and repaired. Aged 62 she now presented with a pulsatile mass visible through the sternum. She was diagnosed as having a pseudoaneurysm of the ascending aorta. This was closed with an 18 mm Amplatzer姠 muscular ventricular septal defect (VSD) device by interventional cardiology as the risk of open surgical procedure was considered high. The procedure resulted in a satisfactory seal at the neck of the aneurysm. One year later, however, the device was found to have migrated into the pseudoaneurysm (Fig. 1). Open repair of the ascending aortic pseudoaneurysm was performed under hypothermic circulatory arrest (HCA). Femoro-femoral cardiopulmonary bypass (CPB) was established. A left sub-mammary incision was made to allow venting of the left ventricular apex. HCA was achieved at 15 degrees. A median sternotomy was performed. The pseudoaneurysm contained thrombus. The Amplatzer姠 device was embedded into the wall of the pseudoaneurysm *Corresponding author. Department of Cardiac Surgery, 6th Floor, East Wing, St Thomas’ Hospital, Westminster Bridge Road, London SE1 7EH, UK. Tel.: q44 7525857015; fax: q44 020 7188 1006. E-mail address:
[email protected] (R. Attia). 䊚 2010 Published by European Association for Cardio-Thoracic Surgery
which had eroded into the sternal medullary cavity (Fig. 2). Dense adhesions were present throughout the mediastinum. The neck of the aneurysm was 2.5 cm; it was closed with a Haemashield Patch䊛. Carbondioxide was insufflated throughout circulatory arrest. CPB was re-established and standard de-airing manoeuvres carried out. A Gore-tex䊛 shield was placed over ascending aorta prior to closure, behind the sternum, in case further surgery is required in the future. The heart was weaned off CPB uneventfully without inotropic support (CPB time 156 min and HCA time of 9 min). The postoperative recovery was complicated by a right middle cerebral artery stroke on day 3, from which she recovered well. She remains well at 12 months followup. 2. Discussion The medical and surgical experience with modified BT shunt and one-stage repair of tetralogy are extensive. However, complications still occur. Pseudoaneurysm development is rare but has been described in the literature in this setting w1x. As far as we are aware this is the first documented case of pseudoaneurysm presenting 35 years after initial intervention. Pseudoaneurysms usually form at the site of aortic cannulation, aortotomy or anastomoses. They can be lethal as they are prone to rupture, thrombosis, distal embolisation and fistula formation. Percutaneous approach to closure of large ascending pseudoaneurysms has been described using Amplatzer姠 device w2, 3x. Surgical repair is recommended but may be associated with high morbidity and mortality w4, 5x. Utilising the Amplatzer姠 septal occluder has its own challenges. These include the diameter of ascending aorta
ARTICLE IN PRESS R. Attia et al. / Interactive CardioVascular and Thoracic Surgery 10 (2010) 820–822
821 Editorial New Ideas Work in Progress Report Protocol Institutional Report Proposal for Bailout Procedure Negative Results Follow-up Paper State-of-the-art Best Evidence Topic Nomenclature
Fig. 2. The device has eroded into the posterior table of sternum. Defect in the ascending aorta visible that required patch closure and exclusion of the aneurysm sac.
Historical Pages
In conclusion, long-term follow-up is needed to fully evaluate the effectiveness and safety of percutaneous approach for closure of pseudoaneurysms. Open surgical repair entails higher risk in patients with multiple comorbidities and poses technical challenges but remains the gold standard for treatment.
Brief Case Report Communication
and angle of approach that make device misalignment likely. The device was oversized to compensate for this. Although initially the device achieved an adequate seal, migration to other sites remains a serious hazard w6x. Continued expansion of the neck of the pseudoaneurysm ultimately led to embolisation of the device into the sac. The Amplatzer姠 device provided a temporising measure when the patient initially presented, allowing a more planned operative approach later. The operative management of ascending aortic pseudoaneurysms remains technically challenging. Mortality rates from 29 to 46% are quoted in the literature. This is usually due to fatal haemorrhage resulting from rupture of the pseudoaneurysm during sternal re-entry or surgical repair techniques w4x. As in our case, median sternotomy with femoro-femoral bypass and HCA has been the strategy of choice w5x. The main tenants of operative approach being prevention against cardiac injury and ensuing exsanguination; decompression of left ventricle during cooling and neurological protection. Our strategy for chest re-entry was to decompress the left ventricle with an apical vent. Divide the sternum at low flow with the patient fully cooled to 15 degrees and to fully mobilise the right ventricle before reaching the pseudoaneurysm. There are alternative strategies for reoperation for giant false aneurysm of the thoracic aorta. Bachet et al. w7x describe an alternate useful technique of separate carotid cannulation and selective antegrade cerebral perfusion with cold blood during circulatory arrest at moderate core hypothermia. This technique allows for safe chest re-entry without general deep hypothermia, reduced CPB and HCA times and neurological protection. The mean CPB and HCA time in their series was 159"52 min and 34"9 min, respectively. This is comparable to our CPB time of 156 min and HCA time of 9 min. These techniques have allowed safe reoperations on patients who are considered as high surgical risk.
ESCVS Article
Fig. 1. Computer tomogram demonstrating the Amplatzer姠 muscular VSD device displacement into the pseudoaneurysm. The two disks of the circular device are seen eroded through the pseudoaneurysm into the posterior table of sternum. VSD, ventricular septal defect.
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R. Attia et al. / Interactive CardioVascular and Thoracic Surgery 10 (2010) 820–822
References w1x Sundararaghavan S, Khalid O, Suarez WA, Bove EL. Single-stage repair of tetralogy of Fallot with pseudoaneurysm: a unique approach. Ann Thorac Surg 2004;77:2183–2184. w2x Kanani RS, Neilan TG, Palacios IF, Garasic JM. Novel use of the Amplatzer septal occluder device in the percutaneous closure of ascending aortic pseudoaneurysm: a case series. Cath Cardiovasc Inter 2007;69:146–153. w3x Hussain J, Strumpf R, Wheatley G, Diethrich E. Percutaneous closure of aortic pseudoaneurysm by Amplatzer occluder device – case series of six patients. Cath Cardiovasc Inter 2009;73:521–529. w4x Sullivan KL, Steiner RM, Smullens SN, Griska L, Meister SG. Pesudoaneurysm of the ascending aorta following cardiac surgery. Chest 1998;93: 138–143. w5x Katsumata T, Moorjani N, Vaccari G, Westaby S. Mediastinal false aneurysm after thoracic aortic surgery. Ann Thorac Surg 2000;70:547– 552. w6x DiBardino DJ, McElhinney DB, Kaza AK, Mayer JE Jr. Analysis of the US food and drug administration manufacturer and user facility device experience database for adverse events involving Amplatzer septal occluder devices and comparison with the society of thoracic surgery congenital cardiac surgery database. J Thorac Cardiovasc Surg 2009; 137:1334–1341. w7x Bachet J, Pirotte M, Laborde F, Guilmet D. Re-operation for giant false aneurysm of the thoracic aorta: how to reenter the chest? Ann Thorac Surg 2007;83:1610–1614.
eComment: Pseudoaneurysms of the ascending aorta following cardiac surgery – are they preventable?
Author: Frank Edwin, Walter Sisulu Pediatric Cardiac Center, Sunninghill Hospital, Johannesburg, South Africa doi:10.1510/icvts.2009.227900A The case reported by Attia and colleagues w1x is unusual in the sense that the patient presented with pseudoaneurysm 35 years after the last cardiac operation. Such pseudoaneurysms generally result from a disruption of the aortic wall as a consequence of aortic procedures within the first few months of the cardiac operation. Aortic suture lines, aortic cannulation sites, needle puncture sites (for pressure recording), cross-clamp sites and bypass graft sites have all been
reported as predisposing entities w2x. Perioperative infection, poor intraoperative suture technique, and pre-existing aortic wall disease, alone or in combination may act in concert with surgical trauma to cause an ascending aortic pseudoaneurysm. According to Atik and colleagues w3x, almost 80% of affected patients had the operation performed within two weeks of diagnosis of aortic pseudoaneurysm. This highlights the importance of intraoperative events in the etiology of this disease. In the same study w3x, infection was found to be the predominant cause with almost half of the patients having a history of native or prosthetic valve endocarditis or mediastinitis. In most pseudoaneurysms of the ascending aorta, the site of aortic wall disruption leading to aneurysm formation is anteriorly located, giving rise to pseudoaneurysms that bulge anteriorly. Emaminia and colleagues w4x, however, reported a posteriorly-located aortic pseudoaneurysm resulting from intraoperative injury of the posterior aortic wall caused by the tip of the introduced cardioplegia cannula. These authors w4x emphasized the importance of avoiding aortic wall injury especially in the small aorta during introduction of the cardioplegia cannula. Summarily, intraoperative events and perioperative infection demand careful attention if this devastating complication of cardiac surgery is to be prevented. Appropriate suture technique, careful handling of the aortic wall, strict asepsis and aggressive treatment of perioperative infection are advocated by this author. These measures are even more important in the aorta with pre-existing disease that compromises the tensile strength of the aortic wall. References w1x Attia R, Venugopal P, Whitaker D, Young C. Management of a pulsatile mass coming through the sternum. Pseudoaneurysm of ascending aorta 35 years after repair of tetralogy of Fallot. Interact CardioVasc Thorac Surg 2010;10:820–822. w2x Sullivan KL, Steiner RM, Smullens SN, Griska L, Meister SG. Pesudoaneurysm of the ascending aorta following cardiac surgery. Chest 1998;93:138–143. w3x Atik FA, Navia JL, Svensson LG, Vega PR, Feng J, Brizzio ME, Gillinov AM, Pettersson BG, Blackstone EH, Lytle BW. Surgical treatment of pseudoaneurysm of the thoracic aorta. J Thorac Cardiovasc Surg 2006; 132:379–385. w4x Emaminia A, Amirghofran AA, Shafa M, Moaref A, Javan R. Ascending aortic pseudoaneurysm after aortic valve replacement: watch the tip of the cardioplegia cannula! J Thorac Cardiovasc Surg 2009;137:1285– 1286.
