Asian Cardiovasc Thorac Ann 2006;14:e4-e5
© 2006 Asia Publishing EXchange Ltd
A Surgical Case of Symptomatic Coronary Artery-Pulmonary Artery Fistula
Aya Saito, MD,
Minoru Ono, MD,
Noboru Motomura, MD,
Yasutaka Hirata, MD,
Tetsuro Morota, MD,
Shinichi Takamoto, MD
Department of Cardiothoracic Surgery, University of Tokyo, Tokyo, Japan
For reprint information contact: Aya Saito, MD Tel: 81 3 5800 8654 Fax: 81 3 5684 3989 Email: ayasaitou-ths{at}umin.ac.jp, 7-3-1 Hongo, Bunkyo-ku, Tokyo 113-8655, Japan.
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ABSTRACT
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We describe a 55-year-old female patient with multiple plexiform coronary artery-pulmonary artery fistulas, who presented with ischemic episodes accompanying an inversion of T wave on an electrocardiogram even though it was a small shunt size. The surgical procedure involved closure of the openings of the fistulas from inside the pulmonary trunk using cardiopulmonary bypass with additional ligations. Postoperative coronary angiogram revealed complete interruption of the coronary artery-pulmonary trunk fistulas resulting in complete resolution of the patient’s chest symptoms.
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INTRODUCTION
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With increasing use of coronary angiography, coronary artery fistula (CAF) is more frequently encountered. Since clinical presentation varies from asymptomatic to life-threatening conditions, we usually consider several factors (e.g. clinical symptoms, shunt size, other co-existing cardiac lesions) before deciding on surgical treatment. However, there seems no definite criteria for treatment, especially for those with no or nonspecific clinical findings other than the existence of CAF. We report a case presenting with chest pain and dilatation of the fistulous vessels. This CAF was perfectly interrupted by surgery under intraoperative guidance of transoesophageal echocardiography.
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CASE REPORT
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A 55-year-old woman was admitted to our hospital suffering from dyspnea and repetitive chest pain. One year previously, she experienced the first episode of effort angina with an ischemic change in the electrocardiogram (EKG) revealing a terminal negative T wave from V2 through V5. A coronary angiogram revealed CAFs, originating from the left anterior descending artery (LAD) and draining into the pulmonary trunk (PT). The shunt size was small. Angina due to coronary steal was suspected, and medical treatment was initiated. Ten months later, she experienced nitroglycerin-ineffective severe chest pain again. A second coronary angiogram revealed mild growth of the fistulous vessels. She was referred for surgical treatment.
She had been taking medication for hyperlipidemia and brain arteriovenous malformation. Her family history included brain infarction, ischemic heart disease and diabetes mellitus. Her general condition was good. There was no heart murmur. Electrocardiogram at rest on the latest admission showed no evidence of ischemia. Echocardiography revealed normal cardiac function without left ventricular asynergy. Coronary angiogram demonstrated plexiform vessels arising from the proximal LAD and draining into the PT, forming an angiomatous network (Figure 1
). There was no detectable O2 step-up at the PT.
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Figure 1. Coronary angiography demonstrates the most recent preoperative finding, showing the angiomatous network of the fistula. (*) The PT which was enhanced through CAFs. White arrows: two major fistulas arising from the proximal left coronary artery.
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At the operation, the fistulous vessels were quickly found on the surface of the PT, but the origin of the fistulas could not be identified. On a beating heart under cardiopulmonary bypass (CPB), the PT was inspected inside, and a 2-mm opening, where arterial blood was leaking, was found above the annulus of the pulmonary valve. The opening was closed using a 6-0 purse string suture. Transesophageal echocardiogram (TEE) performed after closure of the pulmonary arteriotomy showed another small but obvious shunt, so the PT was re-opened and closed the remaining opening. After additional ligations to the fistulous vessels, repeated TEE confirmed no residual shunt with normal ventricular performance.
The patient was free from chest pain after the surgery, and postoperative coronary angiogram revealed complete interruption of the CAF without any lesion on the native coronary arteries.
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DISCUSSION
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CAF is a relatively rare congenital anomaly, found in about 0.2% of all the adult cases undergoing coronary angiography.1 With an increasing number of catheterization studies, the incidence of asymptomatic cases is on the rise, representing up to 30% – 50% of all CAF cases. The fistulous connection may enter any part of the heart and in about 15% to 20% of cases, connect to the pulmonary artery.2 Clinical features usually include effort dyspnea, heart failure, chest pain and heart murmur. However, none of these is specific for this disease category. Angina, postulated to be due to coronary artery steal, is uncommon, and thus ischemic EKG changes are not frequently seen. The aneurysmal change is rare and expansion of fistulous vessels has been reported to be slow. Operative indications are often dependent on the presence of related symptoms and shunt size.2 Surgical therapy for an asymptomatic patient is still controversial.3
In this case, the patient presented with angina accompanying EKG changes, which were strongly suggestive of coronary steal or vasospastic angina. However, a repeated catheterization study failed to estimate the shunt rate through the fistula. A probable explanation is that the multiple angiomatous CAF resulted in coronary-to-coronary circulation, and thus the estimated arteriovenous shunt rate was negligible. The shunt size is often considered an important factor in determining whether to operate, but it may not always be applicable in a certain group of patients such as the patient illustrated in this case.
The choice of surgical procedures varies on the type of fistulous communication. The principle of surgical treatment is complete interruption of the fistulous vessel by using the least invasive technique. Cardiopulmonary bypass is often necessary. However, aortic clamping is not always required in a patient whose fistula drains into the right-sided heart or the pulmonary artery. The operative result for the repair of CAF is almost perfect. The complicated angiomatous structure in this case prevented us from directly confirming the origin of the fistulas, so an ideal approach was to close the openings from within the PT under CPB without cardiac arrest. The beating heart condition helped to find openings by detecting blood flow from CAFs. To achieve complete interruption, intraoperative TEE was very useful in detecting residual small shunts, as has been recommended in several previous reports.4,5 Recent improvements in echocardiography provide far clearer and more reliable images. Intraoperative screening by TEE is a powerful tool in the armamentarium for the surgical treatment of this type of disease.
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REFERENCES
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- Wilde O, Watt I. Congenital coronary artery fistulae: six new cases with a collective review. Clin Radiol. 1980;31:301–11[Medline]
- Kouchoukos NK, Blackstone EH, Doty DB, Hanely FL, Karp RB. Chapter 32 ‘Congenital anomalies of the coronary arteries’. Kirklin / Barratt-Boyes Cardiac surgery, 3rd edition. Philadelphia, Churchill Livingstone, 2003, 1241–7.
- Cheung DL, Au WK, Cheung HH, Chiu CS, Lee WT. Coronary artery fistulas: long-term results of surgical correction. Ann Thorac Surg 2001;71:190–5.[Abstract/Free Full Text]
- Chamberlain MH, Henry R, Brann S, Angelini GD. Surgical management of a gigantic circumflex coronary artery aneurysm with fistulous connection to the coronary sinus. Eur J Cardiothorac Surg 2001;20:1255–7.[Abstract/Free Full Text]
- Kadir I, Ascione R, Linter S, Bryan AJ. Intraoperative localisation and management of coronary artery fistula using transesophageal echocardiography. Eur J Cardiothorac Surg 1999;16:364–6.[Abstract/Free Full Text]
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ABSTRACT
INTRODUCTION
