Eur J Cardiothorac Surg 2001;20:1255-1257
© 2001 Elsevier Science NL
Surgical management of a gigantic circumflex coronary artery aneurysm with fistulous connection to the coronary sinus
Martin H. Chamberlaina,
Ronald Henryb,
Stacey Brannb,
Gianni D. Angelinia
a Bristol Heart Institute, University of Bristol, Bristol Royal Infirmary, Bristol BS2 8HW, UK
b St Clare Medical Centre, Port of Spain, Trinidad and Tobago
Received 23 April 2001;
received in revised form 3 September 2001;
accepted 3 September 2001.
Corresponding author. Tel.: +44-117-928-3145; fax: +44-117-929-9737
e-mail: g.d.angelini{at}bristol.ac.uk
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Abstract
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We report the successful management of a gigantic circumflex coronary artery aneurysm with fistulisation into the coronary sinus. Transoesophageal echocardiography allowed continuous visualisation of ventricular wall motion during dissection and closure of the aneurysm whilst operating on cardiopulmonary bypass on the beating heart.
Key Words: Coronary artery surgery • Circumflex coronary artery • Aneurysm • Coronary sinus • Fistula • Transoesophageal echocardiography
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1. Case report
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A 58-year-old man presented with a respiratory tract infection. Chest radiography revealed an abnormal cardiac silhouette with a 2.5 cm mass, which appeared confluent with the left heart border Fig. 1a
. Both lung fields were normal. He had no history of angina or myocardial infarction. Ten years previously he had undergone antibiotic therapy for sub-acute bacterial endocarditis; the site of this infection was unclear. The remainder of his past medical history and physical examination were unremarkable. Electrocardiogram (ECG) revealed no abnormality. Echocardiography demonstrated a coronary artery aneurysm and this was confirmed with coronary angiography Fig. 1(b–d). Angiography revealed a normal left anterior descending coronary artery, with a large diagonal branch. The circumflex territory was occupied by the large fistula from which three small obtuse marginal branches originated. The right coronary artery was large and terminated in the posterior descending coronary artery. The patient was commenced on warfarin and referred for surgical correction.
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Fig. 1. Pre-operative chest X-ray (A) demonstrates a 2.5 cm mass (arrow) confluent with the left heart border. Coronary angiography illustrates the complex nature of the circumflex artery aneurysm (B, C contrast injection into aortic root: right anterior oblique views; D guide wire within the aneurysm).
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At operation, median sternotomy was performed revealing a well contracting heart. The circumflex coronary artery was grossly dilated from its origin at the left main stem (LMS) coronary artery. An initial dilatation of 2x3 cm in diameter was followed by a long segment curving back on itself leading into a second cavity of 7x6 cm. This led into a third cavity of 3x4 cm in diameter, which appeared to drain into the coronary sinus. The left anterior descending, the diagonal and right coronary arteries were normal.
The patient was established on cardiopulmonary bypass utilising ascending aortic and bi-caval cannulae (which were snared). A transoesophageal echo (TOE) probe was inserted to visualise myocardial segmental wall motion. The circumflex coronary artery was isolated proximally and occluded with a snare for 5 min, during which the patient was discontinued from cardiopulmonary bypass. This did not produce any ECG changes and TOE demonstrated no alteration in regional wall motion in the circumflex artery territory Fig. 2
. The right atrium was opened and the snare on the circumflex coronary artery was released resulting in a large volume of blood emerging from the coronary sinus. The snare was re-occluded and the aneurysm dissected at its epicardial entrance into the coronary sinus. A 0.5x0.5 cm inlet from the aneurysm into the coronary sinus was identified and closed with 3/0 Prolene. The aneurysm was then opened in a distal to proximal fashion with careful inspection for any large coronary artery branches arising from the aneurysm. Repeated inspection of the TOE and ECG was made (as the aneurysm was progressively opened) to reassure the surgeon that the myocardium was not undergoing ischaemia. Two small marginal coronary arteries were identified which were too small to graft and were therefore ligated. Eventually, the origin of the circumflex coronary artery from the LMS was reached and ligated with two ties. There were no haemodynamic or ECG changes throughout the procedure. TOE demonstrated good contractility of all segments, particularly the lateral and inferior walls. The heart was decannulated, protamine administered and the chest closed. The whole procedure was performed on cardiopulmonary bypass without cross-clamping the aorta on the beating heart.
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Fig. 2. Transoesophageal echocardiography of the left ventricle (LV). (A,B) End-diastolic and end-systolic transverse LV views prior to occlusion of the circumflex coronary artery; (C,D) end-diastolic and end-systolic transverse LV views following 5 min occlusion of the circumflex coronary artery.
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The patient made an uneventful recovery with no ECG changes and was discharged home on day 6 on 300 mg aspirin daily. Histopathology of the aneurysm revealed residual coronary artery muscular wall with severe fibrosis secondary to atherosclerosis. Moderate adventitial inflammation was noted but no arteritis was evident.
At 1 year follow-up the patient is symptom free.
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2. Discussion
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Coronary artery aneurysm is a dilatation of part of a coronary artery of more than 1.5 times the diameter of adjoining normal coronary artery [1]. They are rare and were first recognised at post-mortem studies [2]. Many are found coincidentally at coronary angiography, the suggested incidence being 1–2% [2]. The right coronary artery is involved most frequently, followed by the circumflex and left anterior descending coronary arteries [3].
The most common aetiology is atherosclerotic coronary artery disease [3]. Other causes include coronary ectasia and Kawasaki's disease [4]. All have variable degrees of atrophy and destruction of the musculo-elastic elements of the media. Iatrogenic aneurysms develop as a consequence of balloon dilatation of the coronary arteries [5]. Most coronary aneurysms remain asymptomatic, occasionally manifesting themselves as abnormal findings on chest roentograms [6]. Those that present clinically do so with angina or acute myocardial infarction [7] secondary to thrombosis within the aneurysm leading to local occlusion or distal embolisation [5]. Rupture has been described with haemopericardium and tamponade [6]. Echocardiography [8] and coronary angiography [2] play a major role in diagnosing this condition.
Fistulous connection of a circumflex coronary artery aneurysm to the coronary sinus has previously been reported but is an extremely uncommon pathology [9]. Our case was almost certainly a congenital arteriovenous fistula from the origin of the circumflex coronary artery to the coronary sinus. Aneurysmal dilatation probably resulted from the high-pressure fistula and the small outlet into the coronary sinus creating a stenosis. It is interesting what little bearing the excision of the aneurysm had with regard to the myocardial blood supply. This may have been beneficial to the patient as any potential thrombosis within the aneurysm would have been unlikely to cause any major myocardial infarction. The fistulous connection with the coronary sinus, although small, may also have been protective by reducing stasis within the aneurysm.
We have previously reported the use of TOE for intraoperative localisation of a coronary artery fistula [10]. This technique played a crucial role in the management of this case allowing the whole procedure to be performed on bypass on the beating heart without cross-clamping the aorta. It was possible for the operating surgeon to visualise ventricular wall motion when the aneurysm was progressively dissected and ligated at its origin from the LMS. Repeated examination of the TOE and ECG throughout dissection demonstrated that the aneurysm was not crucial to myocardial perfusion and could be over sewn without the need for additional coronary bypass grafting.
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Abstract
1. Case report
