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Ann Thorac Surg 2008;85:649-651. doi:10.1016/j.athoracsur.2007.08.037
© 2008 The Society of Thoracic Surgeons
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Ibrahim Arif Tarhan
Tamer Kehlibar
Yucesin Arslan
Mehmet Yilmaz
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Case Reports

Resection of a Right Coronary Artery Aneurysm With Fistula to the Coronary Sinus

Azmi Ozler, MD, Ibrahim Arif Tarhan, MD*, Tamer Kehlibar, MD, Yucesin Arslan, MD, Mehmet Yilmaz, MD, Mert Dumantepe, MD, Cansin Pancaroglu, MD

Department of Cardiovascular Surgery, Siyami Ersek Thoracic and Cardiovascular Surgery Training and Research Hospital, Istanbul, Turkey

Accepted for publication August 14, 2007.

* Address correspondence to Dr Tarhan, Hizirbey Caddesi Nesrin Sokak No. 1 D: 11, Göztepe, Istanbul, 34732, Turkey (Email: atarhan{at}gmail.com).


   Abstract
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 Abstract
Introduction
 Comment
 References
 
We are reporting the successful surgical management of a challenging right coronary artery aneurysm with a giant fistula into the coronary sinus. We performed fistula division, coronary sinus size reduction, and complete resection of the right coronary artery aneurysm with bypass to the posterior descending artery.


   Introduction
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 Abstract
 Introduction
Comment
 References
 
Varieties of right coronary artery (RCA) anomalies ranging from anomalous origin from wrong sinus to RCA with high take off [1] have been published to date. In the literature, the most common anomaly of the RCA is the artery arising from the wrong sinus. Another rare form of coronary anomalies is congenital aneurysm associated with a fistula. Because of the complications related to them, they are normally diagnosed in relatively young ages. Generally the draining site may be the right ventricle, the right atrium, and pulmonary veins [2], whereas drainage in the coronary sinus occurs the least frequently [3].

We are here reporting a patient with RCA aneurysm with a giant fistula into the coronary sinus treated successfully by complete resection of the RCA aneurysm with bypass to the posterior descending artery and fistula ligation.

A 65-year-old woman was referred to our cardiovascular surgery clinic with dyspnea, extreme weakness, fatigue, and lowered effort capacity complaints. She was obese (105 kg) and had a history of dyspnea and swelling of the lower legs for the last 5 years.

During her first physical examination, the pulse was arrhythmic (82 beats per minute), the blood pressure was 140/90 mm Hg, and she had a pre-tibial soft edema. Discovery of a continuing murmur over the left sternal border at the fourth intercostal space prompted a more detailed cardiologic investigation. She had a remarkable hepatomegaly. Electrocardiogram revealed atrial fibrillation, biventricular hypertrophy pattern, and additional right axis deviation.

A chest x-ray film revealed a slight enlargement of the right border of the heart. Echocardiography suggested the presence of a large RCA aneurysm and dilated cardiac chambers. Left ventricular ejection fraction was 30%. Selective coronary angiography revealed a giant aneurysm arising from the initial segment of the RCA (Fig 1). Calculated ratio of pulmonary blood flow to systemic blood flow was 1.85.


Figure 1
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  		Fig 1. Computed tomographic angiogram shows large circumvolved right coronary artery (RCA). Small arrow shows RCA ostium, and large arrow shows right coronary artery–coronary sinus fistula. (Ao = aorta; CS = coronary sinus.)

 
For a more detailed anatomic definition, a computed tomographic scan was performed. A computed tomographic angiography revealed a giant circumvolved aneurysm arising from the ostium of the right coronary artery. The aneurysm had many loops over its route and had an hourglass shape at the fistula site into the coronary sinus (Fig 2).


Figure 2
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  		Fig 2. Operational view. (A) Aneurysmal right coronary artery is seen before cardiopulmonary bypass. (B) Arrow shows coronary sinus fistula site. (RCA = right coronary artery; Ao = aorta.)

 
Surgery was performed through a sternotomy with cardiopulmonary bypass. After the pericardial cavity was exposed, a giant and circumvolved RCA was seen. Even on the inferior surface, a RCA aneurysm extended until the posterior descending artery rose. Acute marginal branches were small in size, but the terminal branch posterior descending coronary artery was reasonable, despite the fact that the main coronary artery was abnormally expanded.

After the aorta was clamped, antegrade cardioplegic cardiac arrest was provided. During the cardioplegic infusion, a coronary sinus fistula was manually compressed. After cardioplegia, two small vertical incisions were applied on the coronary artery near the fistula site and on the enlarged coronary sinus. The fistula was identified as a 15-mm hole at the roof of the coronary sinus ostium. The fistula was verified from the right atrium by right angled forceps. The fistula hole was closed with sutures. An aneurysmal segment of the RCA was thoroughly removed up to the fistula, which was located next to the posterior descending branch. The posterior descending coronary artery was bypassed with a saphenous vein graft in an end-to-end fashion. Epicardial microwave ablation was added to the procedure. The patient recovered without complication.


   Comment
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References
 
One of the most serious complications associated with congenital aneurysm associated with a fistula is the coronary sinus rupture. Other complications are endocarditis, mural thrombus embolization with subsequent myocardial infarction [4], and coronary steal syndrome with a reversible thallium perfusion defect [5].

The left-to-right shunt with right ventricular overload can produce congestive heart failure similar to that in our patient. To treat congestive heart failure due to left-to-right shunt and avoid other life-threatening complications, such as aneurysm rupture, we believe that urgent surgery is a necessity.

In such delayed left-to-right shunt cases, myocardial preservation is particularly important due to hypertrophied ventricular muscle. Due to the possibility of a coronary steal phenomenon [6] and embolization risk during cardioplegia, the retrograde route is proposed. However, we avoided inserting retrograde cardioplegia cannulas before the cardiopulmonary bypass due to the fragility of the dilated, thin coronary sinus. We applied antegrade cardioplegic perfusion together with digital closure of the fistula as previously described [7]. During cardioplegic infusion, in case of a fistula from the coronary arterial system to the coronary venous system, pressure inside the system would be too high and would perforate the thin coronary system.

Surgical treatment should aim at closing the shunt and preserving the natural myocardial perfusion. Wauthy and colleagues [8] reported the successful ligation of a smaller fistula in a younger patient. Leaving the large segments of coronary vessels intact may induce thromboembolism. This is why we resected the whole aneurysmal segment and completed it with the bypassing of the posterior descending branch in our case. Although the right coronary main body expanded too much, there were not any remarkable branches worth bypassing except the posterior descending coronary artery.

When we opened the aneurysmal RCA entirely, during selective left coronary antegrade cardioplegic infusion, all small acute marginal branches excessively bleeded retrograde. After dividing all marginal branches, we resected the body of the right coronary artery prior to the posterior descending coronary artery. The right coronary artery ostium on the aorta was repaired directly with a 3/0 nylon suture. After ligation of the coronary fistula hole over the coronary sinus, the exceedingly (2.5 cm) large coronary sinus was reduced and repaired with a continuous suture to avoid coronary sinus thrombosis. Resection of the aneurysmal RCA should prevent further thrombosis in the dilated segment and embolus to the acute marginal branches.

Complete resection of the right coronary aneurysm and distal division of a fistula at the coronary sinus emergence is the basis of this procedure, unlike the previous publications.

We conclude that in coronary artery aneurysm with coronary sinus fistula, the surgical strategy should be carefully planned. A fistula must be manually compressed while infusing cardioplegic solution. To identify and securely control the coronary origin of the fistula, the possible presence of "en cascade" aneurysms should be excluded. Complete aneurysmal coronary artery resection can be applied to avoid relapses and further embolic potential. Revascularization of the remarkable branches is essential for an enlarged myocardium.


   References
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 Abstract
 Introduction
 Comment
 References
 

  1. Tarhan A, Kehlibar T, Yilmaz M, et al. Right coronary artery with high takeoff Ann Thorac Surg 2007;83:1867-1869.[Abstract/Free Full Text]
  2. Lowe JE, Oldham Jr HN, Sabiston Jr DC. Surgical management of congenital coronary artery fistulas Ann Surg 1981;194:373-380.[Medline]
  3. Ieva R, Correale M, De Luca G, Di Biase M. A coronary artery fistula with aneurysm draining into the coronary sinus: role of transesophageal echocardiography Am J Geriatr Cardiol 2007;16:112-114.[Medline]
  4. Straumann E, Niederhäuser U, Meili C, Christen S, Bertel O. Recurrent myocardial infarction caused by a giant coronary aneurysm Int J Cardiol 1998;63:305-307.[Medline]
  5. Glynn Jr TP, Fleming RG, Haist JL, Hunteman RK. Coronary arteriovenous fistula as a cause for reversible thallium-201 perfusion defect J Nucl Med 1994;35:1808-1810.[Abstract/Free Full Text]
  6. IL KH, Koshiji T, Okamoto M, Arai Y, Masumoto H. Surgical repair of coronary arteriovenous fistula: a simple and useful approach to identify the fistulous communication Eur J Cardiothorac Surg 2001;20:850-852.[Abstract/Free Full Text]
  7. Misumi T, Nishikawa K, Yasudo M, Suzuki T, Kumamaru H. Rupture of an aneurysm of a coronary arteriovenous fistula Ann Thorac Surg 2001;71:2026-2027.[Abstract/Free Full Text]
  8. Wauthy P, Demanet H, Deuvaert FE. Surgical treatment of coronary artery fistula with aneurysm Acta Chir Belg 2003;103:532-533.[Medline]



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[Abstract] [Full Text] [PDF]

This Article
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Ibrahim Arif Tarhan
Tamer Kehlibar
Yucesin Arslan
Mehmet Yilmaz
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