Pseudoaneurysm after aortic coarctation repair presenting with an aortobronchial fistula successfully treated with an aortic stent graft

Pseudoaneurysm after aortic coarctation repair presenting with an aortobronchial fistula successfully treated with an aortic stent graft

Clinical Radiology (2005) 61, 104–108 CASE REPORT Pseudoaneurysm after aortic coarctation repair presenting with an aortobronchial fistula successfu...

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Clinical Radiology (2005) 61, 104–108

CASE REPORT

Pseudoaneurysm after aortic coarctation repair presenting with an aortobronchial fistula successfully treated with an aortic stent graft G. Munnekea,*, T. Loosemoreb, J. Smithb, M. Thompsonb, R. Morgana, A.-M. Bellia Departments of aInterventional Radiology, and bSurgery, St George’s Hospital, London, UK

Introduction Aortobronchial fistula (ABF) presents with massive, life-threatening haemoptysis. Without prompt treatment the condition is uniformly fatal. Some patients present with repeated smaller haemoptyses with the fistula apparently thrombosing between episodes. Post-surgical pseudoaneurysm of the descending aorta is the most common cause of ABF accounting for 60% of cases.1 Anastomotic pseudoaneurysm after surgical repair is a recognized complication occurring in 9% of cases, with a mortality of 36% if left untreated.2 Pseudoaneurysm formation complicated by ABF after Dacron patch plasty for aortic coarctation, is well described and may occur in as many as 32% of patients after the procedure.3,4 These patients will have had at least one thoracotomy and therefore represent high reoperative risks. Re-operative surgery is associated with a 13.8% mortality, a 13–36% risk of recurrent laryngeal nerve palsy and a 5–6% risk of phrenic nerve injury.4,5 Endovascular stent grafting is therefore an attractive minimally invasive option with low morbidity and mortality, despite the risk of infection from the bronchial tree. It has produced encouraging results in treating diseases of the descending aorta,6–8 and there are reports in the literature of its safe application in ABF due to a variety of causes.9–14 Recent results from the EUROSTAR and United Kingdom Thoracic Endograft registries8 show a 9.3% 30 day mortality for patients with various thoracic aortic pathologies treated with stent grafts.7 Four hundred and forty-three * Correspondent: G. Munneke, Department of Interventional Radiology, St George’s Hospital, Blackshaw Road, London SW17 0QT, UK. Tel.: C44 208 725 3298; fax: C44 208 725 2936. E-mail address: [email protected] (G. Munneke).

patients were included, 35% of which were treated as emergencies. Paraplegia from spinal cord ischaemia complicated the procedure in 4% of patients with aneurysms and 0.8% of patients with aortic dissection. We present a case of ABF due to a pseudoaneurysm at the site of previous coarctation repair treated successfully with a covered stent graft.

Case report A 53-year-old woman was admitted as an emergency with haemoptysis. She had been seen 2 weeks previously in the Accident and Emergency Department with a cough producing blood-stained sputum. She was given oral antibiotics but the haemoptysis persisted and worsened leading to admission. At the age of 27 she had undergone surgery for aortic coarctation with a Dacron patch plasty. Her past medical history also included a bicuspid aortic valve, severe hypertension requiring three antihypertensive drugs, diabetes and asthma. A contrast-enhanced magnetic resonance angiogram of the aorta performed in 2000 demonstrated a saccular pseudoaneurysm at the site of the Dacron patch repair. At this time the pseudoaneurysm measured 45 mm. The patient declined intervention as she was asymptomatic, despite being informed of the risk of rupture. She remained under yearly clinical and computed tomography (CT) review. Three years later the pseudoaneurysm remained static in size (Fig. 1). On the current admission she was afebrile and cardiovascularly stable. Routine haematology and biochemistry was normal and her chest radiograph demonstrated left upper zone consolidation, as well as her aortic pseudoaneurysm (Fig. 2). A

0009-9260/$ - see front matter Q 2005 The Royal College of Radiologists. Published by Elsevier Ltd. All rights reserved. doi:10.1016/j.crad.2005.07.007

Pseudoaneurysm after aortic coarctation repair

Figure 1 Coronal oblique image from a contrastenhanced CT examination of the thoracic aorta. The post-surgical pseudoaneurysm (white arrow) is seen to arise just distal to the left subclavian artery.

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contrast-enhanced CT examination showed that the aortic pseudoaneurysm measured 48 mm in maximum diameter. In view of the history, an aortobronchial fistula was diagnosed. Bronchoscopy demonstrated fresh bleeding in the left upper lobe bronchus with external compression of the airway. After discussing the available therapeutic options with the patient the decision was made to place a thoracic aortic stent graft to exclude the pseudoaneurysm. Pre-intervention imaging showed that the external iliac arteries were 7 mm and therefore too narrow to accommodate the stent delivery sheath, so the right common iliac artery was surgically exposed to insert the device. The left common femoral artery was punctured percutaneously and a pigtail catheter inserted and positioned in the ascending aorta to enable angiographic guidance during stent deployment. Heparin (5000 units) was administered and a 28 mm diameter, 10 cm long Talent thoracic aortic stent graft (Medtronic, MN, USA) was deployed distal to the left common carotid artery, covering the left subclavian artery origin (Fig. 3(a)). A second device measuring 34 mm in diameter and 10 cm long was deployed distal to this, with overlap. Completion angiography demonstrated complete exclusion of the aneurysm sac (Fig. 3(b)). The left subclavian artery continued to fill via reverse flow through the left vertebral artery. The patient was given intravenous vancomycin perioperatively and for 10 days after stenting, and she will be maintained on daily oral ciprofloxacin for the rest of her life. Her recovery was complicated by an episode of angina and fast atrial fibrillation which settled with medical therapy. She was discharged home 16 days postoperatively. At 12 months follow-up she is well with no further haemoptysis. Her CT examination shows complete thrombosis of the pseudoaneurysm and minimal reduction in its diameter to 45 mm (Fig. 4). Initially she experienced mild left arm claudication but this had resolved on follow-up. She will be followed up with yearly CT examinations.

Discussion

Figure 2 Chest radiograph obtained upon admission. The pseudoaneurysm arising from the descending aorta is marked (arrow). Consolidation in the adjacent lung is suggestive of a fistula.

Aortic coarctation represents between 3 and 5% of congenital cardiac malformations.2 Surgical techniques for treating coarctation include Dacron patch plasty, left subclavian flap aortoplasty and coarctectomy with end-to-end anastomosis if there is sufficient remaining aorta after excision of the coarctation or prosthetic interposition graft. The

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Figure 4 Post-intervention CT examination. The stent graft remains fully patent and there is complete thrombosis of the pseudoaneurysm (arrow).

Figure 3 (a) Left anterior oblique subtraction angiogram performed at the time of stent graft deployment. The pseudoaneurysm is marked (black arrow), notice that the fistula itself is not demonstrated. The stent graft delivery system is in place and the delivery sheath (white arrow) is being withdrawn. (b) Completion angiogram after deployment of two stents. There is no residual filling of the aneurysm.

primary technical success rate is good but pseudoaneurysms occur in 9% of patients after surgery.15 The percentage is much higher in the patients treated with Dacron patch plasty,16 and this has led to the discontinuation of the technique. Angioplasty and stenting have evolved as a treatment for coarctation over the last two decades, with results comparable with those of surgery.17 However, data on long-term follow-up are sparse and patients treated by endovascular techniques may also develop pseudoaneurysms. Knyshov et al.5 reviewed 48 patients with postsurgical coarctation repair pseudoaneurysm and found there was a 100% rupture rate within 15 years if they were managed conservatively. ABF has been reported from 3 weeks post-surgery up to 23 years later.9 ABF should be suspected in any patient with previous thoracic aortic surgery presenting with haemoptysis. Computed tomography examination will readily demonstrate an anastomotic pseudoaneurysm. Consolidation of lung adjacent to the aneurysm is suggestive of a fistula.10 The fistula itself is rarely seen on imaging, but is most reliably demonstrated by trans-oesophageal echocardiography.9 Although bronchoscopy is useful to exclude other causes of haemoptysis, it should be undertaken with great caution in these patients as torrential haemorrhage may be provoked.18 Traditional surgical methods of treating ABF involve a thoracotomy with aortic cross-clamping and aortic repair or bypass grafting.9 In some cases cardiopulmonary bypass and hypothermic circulatory arrest are required for repair.19 When treated electively without evidence of ABF, the perioperative mortality for these aneurysms is 13.8%.4,5

Pseudoaneurysm after aortic coarctation repair

However, if they are treated emergently when fistula occurs this rises to between 24 and 41%.18,20 There are two series in the literature of the use of stent grafts for patients with pseudoaneurysms after coarctation surgery. Reidy2 performed stent grafting in five patients with no peri-operative mortality, and aneurysm exclusion at a mean follow-up of 40 months. Ince et al.15 treated six patients with no procedure-related mortality or morbidity at 30 days and 1 year, respectively. One patient died at 11 months from cancer, the others were all well at follow-up of between 11 and 47 months. Bockler et al.21 performed aortic stent grafting in eight patients with aorto-bronchial fistulae after trauma or surgery. Five patients were still alive at a mean follow-up of 30 months. The only ABF-related death occurred after a technically incomplete procedure. There are numerous case reports of endovascular repair of thoracic aortic pseudo-aneurysms associated with ABF.9–14 These papers report on the treatment of a total of 10 patients. Only two of the patients had previous coarctation repairs. In all cases the pseudoaneurysms were successfully excluded by the stent grafts. In one patient there was rupture of the iliac artery by the 27 F delivery sheath. This required immediate surgical repair but the patient made an otherwise uneventful recovery. One patient died 3 weeks after treatment of vancomycin-resistant staphylococcal pneumonia. A CT examination performed before death demonstrated continued exclusion of the pseudoaneurysm. Of the remaining nine patients, eight were still alive at a mean follow-up of 11 months (range 1–24 months). One patient died of congestive cardiac failure 2 years after stenting. There were no reports of graft infection and no secondary procedures were performed in the follow-up period. There is concern regarding the risk of stent graft infection in ABF due to the connection with the bronchial tree which is colonized with bacteria. However, Semba et al.22 have published results of their experience of treating mycotic aneurysms of the thoracic aorta with stent grafts. They treated three patients, one of whom presented with haempotysis suggesting ABF. All of the patients were maintained on daily antibiotics post-procedure, with no evidence of graft infection or failure at a median follow-up of 24 months. One patient died after a cardiac arrest at 25 months, the other two were alive and well at the time of reporting. Kramer et al.23 used stent grafts to treat four patients with aortic pathology in contaminated surgical fields. One of the four patients had an ABF 10 years after thoracic aortic surgery. Technical success was achieved in all patients and they were

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alive and well at a mean follow-up of 13 months. Endovascular stent grafting has also been successfully employed in infected aneurysms of other arteries for example the abdominal aorta,24 the subclavian artery25 and the superficial femoral artery.26 Most ABFs are anatomically suitable for stent grafting. A proximal and distal landing zone of normal calibre aorta measuring 20 mm in length is required to deploy the stent. The left subclavian artery origin can be covered to provide an adequate landing zone if required.27 Patients rarely develop ischaemic symptoms in the left arm as reversal of flow in the left vertebral artery supplies the distal subclavian. Stenting also avoids the surgical complications of damage to the left recurrent laryngeal and phrenic nerves. Endovascular stent grafting is an attractive therapeutic option for patients with ABF after repair of aortic coarctation. This case report adds to the limited published literature on this subject.10,11 The perioperative morbidity and mortality rates and intermediate term outcomes are encouraging. The risk of stent graft infection may be minimized by maintaining the patient on life-long antibiotics. However, the durability of these devices is still unknown and long-term follow-up is recommended in all patients.

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