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Innovations: Technology & Techniques in Cardiothoracic & Vascular Surgery:
doi: 10.1097/IMI.0000000000000013
Case Reports

Staged Hybrid Treatment for Contained Rupture of a Large Right Coronary Artery Aneurysm

Khoshbin, Espeed MD; Sogliani, Franco MD; Tang, Augustine MD

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From the Department of Cardiothoracic Surgery, Lancashire Cardiac Centre, Victoria Hospital, Blackpool, UK.

Accepted for publication September 18, 2013.

A video clip is available for this article. Direct URL citations appear in the printed text and are provided in the HTML and PDF versions of this article on the journal’s Web site ( www.innovjournal.com). Please use Firefox when accessing this file.

Disclosure: The authors declare no conflicts of interest.

Address correspondence and reprint requests to Espeed Khoshbin, MD, University Hospital of Central Manchester, Oxford Rd, Manchester, M13 9WL UK. E-mail: khoshbinuk@yahoo.co.uk.

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Abstract

We describe a staged approach to the management of a rare acute condition—contained rupture of a large right coronary artery aneurysm. A covered stent was deployed percutaneously to isolate the aneurysm at presentation followed by planned coronary bypass grafting. Treatment interval was complicated by new-onset pulmonary tuberculosis and subacute thrombosis of the covered stent leading to nonfatal inferior myocardial infarction. Coronary surgery was performed after complete antitubercular treatment and resolution of the acute pericarditis/thrombosis as a consequence of the contained rupture. The advantages of this staged approach included the following: (a) The covered stent prevented both acute myocardial infarction and progressive pseudoaneurysm expansion in the acute phase. (b) Deferred surgery was rendered technically less hazardous while avoiding the undesirable option of having to exclude an extremely calcified dominant right coronary artery. The patient made an excellent postoperative recovery with complete resolution of her symptoms at 6 weeks’ follow-up.

A coronary artery aneurysm (CAA) is defined as vessel dilatation of greater than 1.5 times the diameter of the largest native coronary vessel.1 Rupture of a coronary aneurysm may be catastrophic, leading to cardiac tamponade. Presentation with controlled rupture is, however, rare and may masquerade as a myocardial mass.2 Both surgical3–5 and conservative management4,6 of this condition have already been reported. Here, we describe a staged approach to its management involving primary coronary stenting and interval bypass grafting.

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CASE REPORT

A 76-year-old patient was acutely hospitalized with severe retrosternal pain. A nongated computed tomography showed a pelvic teratoma and presence of a mass on the right side of the heart. A transesophageal echocardiogram showed a large cavitating lesion (approximately 30 mm) overlying the right atrioventricular groove encroaching on a normally functioning tricuspid valve. Doppler signal detected blood flow within this lesion. Oncological assessment supported by further imaging excluded the likelihood of secondary cardiac metastasis from a seemingly benign pelvic tumor. Coronary computed tomographic angiogram confirmed the diagnosis of contained rupture of a large aneurysm arising from the right coronary artery. Invasive coronary angiogram showed a large atheromatous right coronary artery with two separate fistulae communicating with a contained ruptured aneurysm (Figure 1 and Video 1, available at http://links.lww.com/INNOV/A31). There were multiple stenoses before and after these fistulae. During multidisciplinary discussion, a surgical strategy was proposed: the approach would involve excluding the segment of right coronary artery feeding this aneurysm and then grafting the posterior descending artery. In the interim, she developed pneumonia. In light of this deterioration, a less traumatic hybrid approach was agreed. Two covered stents were successfully deployed across the mouth of these two fistulae virtually excluding the communication with the aneurysm (Figure 2 and Video 2, available at http://links.lww.com/INNOV/A32). The surgery was postponed, and she was discharged home on clopidogrel and aspirin. Shortly after discharge, she became constitutionally unwell with significant weight loss and respiratory symptoms. Tuberculosis (TB) was diagnosed on sputum culture. Antitubercular therapy was commenced, with rapid symptom resolution. She remained clinically well until she was acutely rehospitalized with an inferior myocardial infarction (MI) 4 months after stent deployment. Repeat angiogram demonstrated a complete thrombosis of the covered stent. There were no signs of any residual leak into the contained aneurysm. After this event, she developed significant angina but remained clinically stable on medical treatment and had completed a full course of antituberculous treatment. Urgent surgery was scheduled. She underwent coronary artery bypass surgery. Pericardiotomy revealed dense adhesions, most of these over the anterior surface of the heart including some extremely dense adhesions over the atrioventricular groove leading up to the right atrial free wall corresponding to the troubled area. At this stage, the patient was placed on cardiopulmonary bypass, and the rest of the heart was freed. With the heart arrested, attention was turned to the inferior wall. The posterior descending artery was located and grafted using a reversed saphenous vein, and the proximal anastomosis was constructed to the right side of the ascending aorta. The heart was then weaned off cardiopulmonary bypass without difficulty. The patient had an excellent uneventful recovery. She was reviewed 6 weeks after surgery and was symptom-free.

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DISCUSSION

The objective for this staged approach in the acute setting of a contained rupture was to allow adequate time for patient stabilization. This was achieved by using a covered stent, which may temporarily preserve the integrity of the proximal and the mid right coronary artery while permitting gradual reduction in local inflammation, thus improving tissue integrity and the ease of tissue handling during subsequent coronary surgery. The incidence of stent thrombosis and angiographic restenosis has been reported in various clinical settings after the use of the covered stents (5.7% and 31.6%, respectively). Therefore, the incidence of subacute stent occlusion is relatively high.7 Hence, a multidisciplinary decision was made to use the covered stent as a bridge to enable safer surgery at a later date.

The proximal and mid sections of the right coronary artery are the most common sites for coronary aneurysms.8 There have been reports of successful surgical ligation of the coronary artery in which the aneurysm was distal to a small right coronary vessel3 and surgical isolation of the aneurysm with concomitant bypass grafting to the distal vessel.5 The latter, although technically feasible in our case, would necessarily kill all the branches arising from the proximal and the mid right coronary artery leading to dysfunction of the right side of the heart and a likely conduction deficit. Deployment of covered stents during the first phase of our approach was intended to preserve this blood supply and prevent both acute MI and progressive pseudoaneurysm expansion when the patient was clinically most compromised. The deferred surgery was rendered technically less hazardous while avoiding the undesirable option of having to exclude an extremely calcified dominant right coronary artery. Fortunately, the staged treatment also allowed time for resolution of pneumonia and completion of the antituberculous chemotherapy.

The etiology of CAA in adults is predominantly atherosclerotic. Other causes include congenital, autoimmune, iatrogenic/accidental trauma, and infectious.8 Tuberculous coronaritis is a very rare condition resulting from inflammatory vasculitis in coronary vessels.8 It may be a consequence of hematogenous spread or direct contact from an adjacent lesion. There have been reports of a possible link between active pulmonary TB and acute MI9 in addition to evidence linking CAA to Bacillus Calmette Guerin vaccination.10 However, a direct association between active pulmonary TB and development of coronary aneurysm remains unproven. The presence of active TB in this case is probably not causally linked to the coronary aneurysm or subsequent stent thrombosis. The latter, although well recognized, was an unwelcome interval complication during the staged approach.

The occurrence of subacute stent thrombosis in this instance despite uninterrupted dual antiplatelet therapy may well be related to pharmacokinetic idiosyncrasy because platelet function testing immediately before surgery suggested that the patient was clearly a nonresponder to aspirin (10% inhibition of 160 × 109/L platelets [platelet function analyzer, PFA-100; Dade Behring, Inc, Deerfield, IL USA]). Consequently, clopidogrel was recommenced immediately after surgery for secondary coronary and graft protection. Perhaps, baseline platelet function testing may help to refine future patient selection for the staged approach in this rare condition.

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REFERENCES

1. Syed M, Lesch M. Coronary artery aneurysm: a review. Prog Cardiovasc Dis. 1997; 40: 77–84.

2. Berrizbeitia LD, Samuels LE. Ruptured right coronary artery aneurysm presenting as a myocardial mass. Ann Thorac Surg. 2002; 73: 971–973.

3. Wan S, LeClerc JL, Vachiery JL, Vincent JL. Cardiac tamponade due to spontaneous rupture of right coronary artery aneurysm. Ann Thorac Surg. 1996; 62: 575–576.

4. Chia HM, Tan KH, Jackson G. Non-atherosclerotic coronary artery aneurysms: two case reports. Heart. 1997; 78: 613–616.

5. Jha NK, Ouda HZ, Khan JA, Eising GP, Augustin N. Giant right coronary artery aneurysm- case report and literature review. J Cardiothorac Surg. 2009; 4: 18

6. Hayat SA, Ghani S, More RS. Treatment of ruptured coronary aneurysm with a novel covered stent. Catheter Cardiovasc Interv. 2009; 74: 367–370.

7. Gercken U, Lansky AJ, Buellesfeld L, et al. Results of the Jostent coronary stent graft implantation in various clinical settings: procedural and follow-up results. Catheter Cardiovasc Interv. 2002; 56: 353–360.

8. Desai PK, Ro JH, Pucillo A, Weiss MB, Herman MV. Left main coronary artery aneurysm following percutaneous transluminal angioplasty: a report of a case and review of the literature. Cathet Cardiovasc Diagn. 1992; 27: 113–116.

9. Bakalli A, Osmani B, Kamberi L, Pllana E. Acute myocardial infarction and pulmonary tuberculosis in a young female patient: a case report. Cases J. 2008; 1: 246

10. Ueda Ikuyo SK, Sugimoto T. An infant with fever, skin tuberculosis and coronary artery aneurysms after Bacillus Calmette Guerin (BCG) vaccination. J Jpn Pediatr Soc. 2003; 107: 1628–1630.

Keywords:

Coronary artery bypass surgery; Coronary artery aneurysm; Hybrid approach; Percutaneous coronary intervention

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