Ann Thorac Surg 2001;71:2026-2027
© 2001 The Society of Thoracic Surgeons
Case report
Rupture of an aneurysm of a coronary arteriovenous fistula
Takahiko Misumi, MDa,
Kuni Nishikawa, MDa,
Mikito Yasudo, MDa,
Takaaki Suzuki, MDa,
Hiroya Kumamaru, MDa
a Division of Cardiovascular Surgery, Ashikaga Red Cross Hospital, Ashikaga, Japan
Accepted for publication April 24, 2000.
Address reprint requests to Dr Misumi, Division of Cardiovascular Surgery, Ashikaga Red Cross Hospital, 3-2100 Honjo, Ashikaga-shi, Tochigi 326-0808, Japan
e-mail: cdm88010{at}par.odn.ne.jp
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Abstract
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Coronary arteriovenous fistula is a relatively rare congenital malformation. The course of this malformation is generally benign, and rupture of the fistula is thought to be very rare. We report a case of rupture of an aneurysm of a coronary arteriovenous fistula that originated from the distal left circumflex coronary artery and drained into the right atrium; this rupture was successfully treated with an operation.
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Introduction
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Coronary arteriovenous fistula is a relatively rare congenital malformation. The course of this malformation is generally benign, and rupture of the fistula is thought to be very rare [1]. This report describes our experience with a case of rupture of the aneurysm of the coronary arteriovenous fistula and a successful operation to correct it.
A 63-year-old woman was admitted to our hospital in emergency because of syncope. She was born and grew up without any incidents, and she had no history of cardiac symptoms. On physical examination, her consciousness was clear, but her blood pressure was 60/40 mm Hg. Echocardiography demonstrated moderate volume of pericardial effusion and an unidentified cystic mass behind the aortic root. The pericardiocentesis was performed and 120 mL of blood was aspirated, and then the patient’s hemodynamic condition rapidly improved. The left coronary angiography showed a dilated and tortuous fistulous vessel that originated from the distal portion of the left circumflex coronary artery and drained into the right atrium (Fig 1). Chest computed tomographic scan showed two aneurysmal formations of the fistula 30 to 40 mm in diameter running beneath the main pulmonary trunk and the aortic root (Fig 2). The diagnosis was rupture of the aneurysm of the coronary arteriovenous fistula, and the patient was referred to us for urgent surgical treatment.
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Fig 1. The left coronary angiography showed a dilated and tortuous fistulous vessel that originated from the distal portion of the left circumflex coronary artery and drained into the right atrium. (LAD = left anterior descending artery.)
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Fig 2. Chest computed tomographic scan showed two aneurysmal formations of the fistula running beneath the main pulmonary trunk and the aortic root. Arrow indicates the fistula. (Ao = aorta; PA = pulmonary artery; AN = aneurysm.)
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The median sternotomy and the exposure of the right femoral vessels were performed simultaneously. By cannulating one venous cannula into the right atrium from the right femoral vein and one arterial cannula into the right femoral artery, cardiopulmonary bypass was instituted. After the cardiopulmonary bypass was established, the pericardium was opened, and about 100 mL of serosanguinous pericardial effusion was drained. The fistula turned out to originate from the distal portion of the left circumflex coronary artery, running along the posterior atrioventricular groove and beneath the main pulmonary trunk and the aortic root. It formed two aneurysms in its pathway and drained into the junction of the right atrium and the superior vena cava. The clot formation was observed around the aneurysms. After the removal of the clot, the rupture site was identified at the proximal aneurysm. The ascending aorta was cross-clamped, and cardioplegic solution was administrated into the aortic root, clamping the origin of the fistula digitally to avoid the cardioplegic solution’s being stolen. After the cardiac arrest, the right atrium was opened. The draining site of the fistula was found at the junction of the right atrium and superior vena cava; it was about 5 mm in diameter. We directly closed the site with a pledgeted mattress suture. Then we ligated the fistula at the origin from the left circumflex coronary artery with three metal clips. We removed the aortic clamp and opened the aneurysm at the rupture site in order to ensure that there was no blood flow in the fistula.
The postoperative course was uneventful, and the follow-up coronary angiography showed no residual blood flow to the fistula (Fig 3). The histopathologic examination of the resected aneurysmal wall revealed no sign of infectious process.
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Fig 3. The postoperative coronary angiography showed no residual blood flow to the fistula. Right anterior oblique, 30 degrees.
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Comment
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Coronary arteriovenous fistula is a relatively rare congenital malformation. Sometimes we can find small coronary arteriovenous fistulae in the ordinary coronary angiography. The course of this malformation is generally benign, but in some cases, significant complications may occur: congestive heart failure, bacterial endocarditis, myocardial ischemia, ventricular arrhythmia, or sudden death [2]. Usually, the fistulous dilatation is uniform throughout, and although it may become aneurysmal (19%; [3]), the rupture of the aneurysm is thought to be very rare [1].
The standard concept of the surgical treatment is to close the fistula at the origin and the terminal. Therefore, median sternotomy and cardiopulmonary bypass are sometimes indicated, either to prevent catastrophic hemorrhage during closure of the fistula or to access it, such as when it is in the posterior site of the heart [2]. When the fistula opens into the right atrium, as in our case, the right atrium should be opened and the fistula closed from inside. Recently, there has been an increasing number of reports about transcatheter closure of fistulas [4]. Further data accumulation is expected to evaluate the effectiveness of this technique.
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References
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Habermann J.H., Howard M.L., Johnson E.S. Rupture of the coronary sinus with hemopericardium. Circulation 1963;28:1143-1144.[Abstract/Free Full Text]
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Kirklin J.W., Barratt-Boyes B.G. Congenital anomalies of the coronary arteries. In: Kirklin J.W., Barratt-Boyes B.G., eds. Cardiac surgery, 2nd ed. New York: Churchill Livingstone, 1993:1167-1193.
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Urrutia-S C.O., Falaschi G., Ott D.A., Cooley D.A. Surgical management of 56 patients with congenital coronary artery fistulas. Ann Thorac Surg 1983;35:300-307.[Abstract]
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Perry S.B., Rome J., Keane J.F., Baim D.S., Lock J.E. Transcatheter closure of coronary artery fistulas. J Am Coll Cardiol 1992;20:205-209.[Abstract]
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Abstract
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