Interact CardioVasc Thorac Surg 2007;6:413-414. doi:10.1510/icvts.2006.149849
© 2007 European Association of Cardio-Thoracic Surgery
Case report - Cardiac general |
Surgical exclusion of a symptomatic circumflex coronary to right atrium fistula
Chakib Benlafqiha,b,
Bertrand Léobona,*,
Valérie Chabbertc and
Yves Glocka
a Department of Cardiovascular Surgery B (Pr. Fournial), CHU Rangueil, Toulouse, France
b Department of Cardiovascular Surgery A, CHU Ibn Sina, Rabat, Morocco
c Department of Radiology, CHU Rangueil, Toulouse, France
Received 5 December 2006;
received in revised form 12 February 2007;
accepted 13 February 2007
*Corresponding author. Service de chirurgie cardiovasculaire B, CHU de Rangueil, 1 avenue jean Poulhès, 31059 Toulouse Cedex, France. Tel.: +33-561-322-651; fax: +33-561-322-959.
E-mail address: bertrand.leobon{at}libertysurf.fr (B. Léobon).
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Abstract
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Coronary artery fistulas are rare and half of them are symptomatic. Diagnosis is confirmed by echocardiography and coronarography and can be precisely located by multislice CT-scan. We report the case of a 56-year-old female patient with congestive heart failure caused by a coronaro-cardiac fistula established between the proximal circumflex coronary artery and the right atrium. Surgical exclusion of the fistula was achieved by ligation of both extremities and a running suture on the aneurysmal vessel. Follow-up at 6 months was satisfactory with an asymptomatic patient and absence of recurrence of the fistula on echocardiography.
Key Words: Cardiac anatomy; Computed tomography; Coronary artery pathology
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1. Introduction
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We report the case of a 56-year-old woman with a symptomatic coronary artery fistula, from circumflex artery to the right atrium, responsible for heart failure, treated successfully by conventional surgery.
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2. Case report
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This 56-year-old female patient was admitted for congestive heart failure. She had a history of diabetes, treated hypertension, moderate renal dysfunction and chronic atrial fibrillation (AF). There was no antecedent of angina pectoris or myocardial infarction. Clinical examination found signs of congestive heart failure and a continuous murmur in the left interscapulovertbral area. ECG confirmed AF. Trans-thoracic and transesophageal echocardiography revealed an important left to right shunt with pulmonary hypertension (plumonary systolic pressure=70 mmHg). This shunt was due to a coronaro-cardiac fistula localized in the left atrioventricular sulcus with drainage into the right atrium, right chambers were dilated. There was no left ventricular dysfunction and a mitral regurgitation grade I–II. These results were confirmed by coronarography and CT-scan showing a large tortuous vessel originating from the end of the left main coronary artery and following the left atrioventricular sulcus with drainage into the right atrium in the area of the coronary sinus (Fig. 1a–c). After medical treatment and regression of the congestive signs, surgical treatment of this coronary to right atrium fistula was decided.
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Fig. 1. (a, b) Pre-operative multislice CT-scan showing the origin of the fistula on the proximal circumflex coronary artery, its route (arrows), and its distal drainage in the right atrium (LM: Left Main coronary artery, RA: Right Atrium). (c) Image from the coronarography showing the fistula during left main injection (arrows). (d, e) Intraoperative views of the fistula (arrows) before (c) and after (d) its exclusion.
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Under cardiopulmonary bypass, heart luxation revealed an aneurysmal circumflex artery immediately downstream the first left marginal artery. After several convolutions in the left atrioventricular sulcus, the dystrophic vessel joined the right atrium (Fig. 1d). Through a right atriotomy the opening of the fistula could be identified 15 mm next to the coronary sinus ostium. Temporary clamping of the proximal segment of the aneurysmal circumflex artery resulted in an important reduction of the outflow in the right atrium and had no consequence on ECG and left ventricular wall motion on intraoperative transesophageal echocardiography. Thus, we performed a ligation and section of the proximal aneurysmal artery and the closing of the distal ostium in the right atrium. As a complement, an external running suture suppressed the whole aneurysmal vessel (Fig. 1e). AF was treated by epicardial bipolar radiofrequency ablation. Immediate postoperative course was simple. There was no sign of myocardial ischemia on biology, ECG or echocardiography. We noted an early recurrence of AF. The continuous murmur had disappeared and the patient's rehabilitation was brief. Echocardiography before discharge found a normal left ventricular function without wall motion dysfunction. There was no more patent fistula or shunt. At 6 months, clinical and echocardiographic follow-up was satisfactory: the patient was asymptomatic, still with AF but without recurrence of murmur or congestive heart failure.
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3. Discussion
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The first description of coronary artery fistulas is given by Krause in 1865, and the first surgical treatment was reported in 1958 by Fell. They are rare and represent 0.2–0.4% of congenital heart diseases [1] and some can disappear during childhood [1, 2]. Coronary fistulas are due to persistence of intramyocardial primitive sinusoids or to a lack in the development of the collateral branches from the involved artery [3]. Their diagnosis is often made in children where they are rarely symptomatic. They are more often symptomatic in adults, according to the localization and size of the fistula. They can be revealed by dyspnea or angina and can cause sudden death, myocardial infarction, heart failure, endocarditis, embolism or rupture with hemopericardium. Forty to 55% of the patients are asymptomatic and symptoms or risk of lethal complications increase with age [2, 4].
Diagnosis is evoked by a continuous systolodiastolic murmur located according to the fistula position. Echocardiography can show the aneurysmal tortuous vessel and dilatation of the receiving chamber or pulmonary hypertension. Coronarography confirms the diagnosis and gives a description of the involved coronary artery, of its branches and of its drainage. Our patient also benefited from a multislice CT-scan giving anatomical images of this lesion (Fig. 1a,b).
Although some spontaneous closure of coronary fistulas have been reported [1, 2], it seems clear for most authors that symptomatic fistulas should be treated [3, 4]. For asymptomatic patients, many authors also recommend a treatment if the shunt is significant [3, 4]. The necessity of a treatment for asymptomatic fistulas without shunt is more controversial, even if good results and low morbidity and mortality rates are in favor [3–6].
Surgery is the gold standard for the treatment of coronaro-cardiac fistulas. In some cases, percutaneous coronary intervention can be preferred. This procedure requires several conditions: fistula has to be unique, with a distal narrowing to avoid embolism through the draining chamber [7, 8]. For the majority of the authors, surgical procedure consists in the closing of the coronary inflow and of the distal ostium through a cardiotomy of the draining chamber [3]. To avoid recurrence of the fistula, the opening of the whole fistula and ligation of branches has been proposed. We preferred not to open the fistula and to suppress it by ligation of the whole aneurysmal vessel with an external running suture, reducing the risk of postoperative bleeding (Fig. 1e). In a few cases this procedure can induce myocardial ischemia requiring revascularization by aorto-coronary bypass, justifying an intraoperative monitoring of ECG and transesophageal echocardiography.
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4. Conclusion
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Coronary fistulas are rare but more frequently discovered, thanks to imaging improvements. They can cause severe complications justifying a treatment in most cases. The surgical treatment remains the gold standard with a weak morbi-mortality even if some cases can benefit from percutaneous closing.
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References
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- Griffiths SP, Ellis K, Hordof AJ, Martin E, Levine OR, Gersony WM. Spontaneous complete closure of a congenital coronary artery fistula. J Am Coll Cardiol 1983; 2:1169–1173.[Abstract]
- Tkebuchava T, Von Segesser LK, Vogt PR, Jenni R, Arbenz U, Turina M. Congenital coronary fistulas in children and adults: diagnosis, surgical technique and results. J Cardiovasc Surg (Torino) 1996; 37:29–34.[Medline]
- Cheung DL, Au WK, Cheung HH, Chiu CS, Lee WT. Coronary artery fistulas: long-term results of surgical correction. Ann Thorac Surg 2001; 71:190–195.[Abstract/Free Full Text]
- Rittenhouse EA, Doty DB, Ehrenhaft JL. Congenital coronary artery- cardiac chamber fistula. review of operative management. Ann Thorac Surg 1975; 20:468–485.[Abstract]
- John S, Perianayagam WJ, Muralidharan S, Nandakumar V, Mansfield R, Krishnaswamy S, Sukumar IP, Cherian G. Surgical treatment of congenital coronary artery fistula. Thorax 1981; 36:350–354.[Abstract]
- Aggoun Y, Bonhoeffer P, Sidi D, Bonnet D, Acar P, Kachaner J. Congenital coronary-cardiac fistula in children. Effects of surgical occlusion and percutaneous embolization. Arch Mal Coeur Vaiss 1997; 90:605–609.[Medline]
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Abstract
1. Introduction