Ann Thorac Surg 2007;83:291-293
© 2007 The Society of Thoracic Surgeons
Case Reports
Surgical Treatment of Bilateral Aneurysmal Coronary to Pulmonary Artery Fistulas Associated With Severe Atherosclerosis
Salah A.M. Said, MDa,*,
Willem G. de Voogt, MD, PhDb,
Mohamed Soliman Hamad, MDc,
Jacques Schonberger, MD, PhDc
a Departments of Cardiology, Hospital Group Twente, Hengelo, The Netherlands
b St. Lucas Andreas Hospital, Amsterdam, The Netherlands
c Department of Cardiothoracic Surgery, Catharina Hospital, Eindhoven, the Netherlands
Accepted for publication May 10, 2006.
* Address correspondence to Dr Said, Department of Cardiology, Hospital Group Twente, Geerdinksweg DL Hengelo, 141, 7555 the Netherlands (Email: samsaid{at}home.nl).
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Abstract
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A 39-year-old diabetic patient with an old inferior wall infarction presented with disabling angina pectoris, despite medical treatment. Coronary angiography showed severe triple-vessel coronary artery disease, and bilateral coronary to pulmonary fistulas originating from the right coronary artery and the left anterior descending coronary artery. Both coronary artery saphenous vein bypass grafting and ligation of the fistulas was performed.
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Introduction
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The indications for closure of coronary artery fistulas associated with or without coronary artery narrowing are discussed. It is pointed out that coronary fistulas may promote progression of existing atherosclerotic coronary artery disease distal to the fistula.
Congenital solitary coronary artery fistulas may originate from one (unilateral), two (bilateral), or three (multilateral) coronary arteries. They may terminate in any of the cardiac chambers or intrathoracic large vessels with relatively low pressure. Coronary to pulmonary artery fistulas (CPFs) comprise 15% to 20% of the fistulas [1]. Coronary to pulmonary artery fistulas may present asymptomatically. When symptomatic, they can cause angina pectoris, ventricular or supraventricular arrhythmias, congestive heart failure, and infective endocarditis [2, 3].
Small-sized fistulas are conservatively and medically treated and further managed by a careful follow-up. However, large-sized symptomatic fistulas are treated either by percutaneous embolization or by surgical ligation, depending on the anatomical characteristics of the fistula [3].
A 39-year-old man with diabetes who sustained an inferior wall myocardial infarction some years previously, presented with severe angina, which persisted despite extensive medical treatment. On physical examination a continuous cardiac murmur was heard at the second left intercostal space. Further findings were unremarkable.
The electrocardiogram showed an old inferior wall infarction and the exercise stress test was indicative for myocardial ischemia and angina pectoris at 70% of target heart rate.
The coronary arteriogram demonstrated significant three-vessel disease and bilateral coronary to pulmonary fistulas (Fig 1).
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Fig 1. (A) Fistula (see arrow) originating from the proximal part of the right coronary artery (RCA) and ending in the pulmonary artery (PA). (B) Proximal segment of the first diagonal branch shows 80% stenosis. Fistulas (see arrows) originating from the proximal left anterior descending coronary artery (LAD) exits to the PA. Circumflex coronary artery demonstrates severe proximal stenosis. (LCA = left coronary artery; PA = pulmonary artery; RCA = right coronary artery.)
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The fistulas were photographed during surgery (Fig 2). It was noticed that the left anterior descending coronary artery was small and of poor quality. Triple-saphenous vein coronary bypass surgery combined with dual ligation of the fistulas was performed. The proximal ends of the fistulas were ligated using 3-0 nonabsorbable ligature. The distal ends on the pulmonary artery side were electrocauterized or sutured with a nonabsorbable suture. A venous jump graft was used to graft the left anterior descending coronary artery, first obtuse marginal branch of the circumflex artery, and the posterior descending coronary artery of the right coronary artery. The operative and postoperative courses were uneventful.
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Fig 2. Photograph taken during surgery after a median sternotomy and longitudinal opening of the pericardium shows bilateral coronary to pulmonary fistulas with aneurysmal formation (arrows). (PA = pulmonary artery; RAA = right atrial appendage.)
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Comment
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In CPFs, the fistula originates from the coronary arteries and drains with equal frequency into the right or left pulmonary arteries [4].
Baim and colleagues [2] reported a series of 363 cases, including 19 cases (5%) of bilateral fistulas. They showed that bilateral fistulas more often (50%) terminate into the pulmonary artery than unilateral fistulas (17%) [2].
In a recent Dutch Registry of congenital solitary fistulas in 51 adult patients, 16% of the fistulas were bilateral. When unilateral coronary artery fistulas (80%) were present, 33% originated from the right, 18% from the circumflex, and 46% from the left anterior descending coronary arteries. Aneurysmal formation was reported in 35% of the patients. Nearly all fistulas (97%) were tortuous and 27% demonstrated multiplicity of origin. Multiplicity of exits was found in 33% of coronary artery fistulas. Surgical ligation combined during valvular and coronary surgical procedures was performed in 25% of the cases, percutaneous therapeutic embolization in 5%, and 70% were treated by conservative medical management [5].
Whether the fistulas are unilateral, bilateral, or multilateral, in the presence or absence of atherosclerotic coronary artery disease, angina pectoris may be one of the symptoms and although it is rare, myocardial infarction may also develop.
It is suggested that CPFs may potentiate ischemia and angina pectoris, but it remains speculative whether the induction of premature or acceleration of coronary atherosclerosis distal to the fistulas may be attributed to CPFs. Therefore some authors suggest an early closure because of the progressive nature of the lesion, its possible complications, and the low mortality rate of the surgical technique [6].
Successful surgical intervention in a symptomatic bilateral CPF without atherosclerosis [7] and a covered graft stent in a unilateral CPF causing isolated right heart failure [3] have been used for permanent occlusion of the fistulas. Furthermore, surgical procedures combined with bypass grafting for CPF in association with atherosclerosis and aneurysmal formation have been reported [8].
Treatment of asymptomatic patients is controversial. If they are symptomatic, medical, surgical, or percutaneous (ie, balloons, coils, covered graft stent, occlusive devices), management is considered. Fistula anatomy and characteristics determine which strategy or techniques should be chosen, which are tailored to the individual patient. However, an endocarditis prophylactic antibiotic is indicated in both symptomatic and asymptomatic fistulas.
Simple coronary artery fistulas can be ligated without cardiopulmonary bypass. As some coronary artery fistulas may become aneurysmal, repair through the pulmonary artery under cardiopulmonary bypass is indicated [6]. Different surgical techniques have been used (ie, selective external fistula ligation without extracorporeal circulation [the epicardial approach] [8], direct suture of the fistulous ostium from within the involved cardiac chamber, or an intrathoracic vessel [the endocardial approach] [2]). A combination of these techniques was applied in our patient. In the department of Cardiothoracic Surgery of Catharina Hospital, the left internal mammary artery is almost always used to revascularize the left anterior descending coronary artery, especially in young patients. However, in this particular patient, the left anterior descending coronary artery was of small caliber and poor quality. Internal policy is to avoid the use of the left internal mammary artery when the left anterior descending coronary artery is small or of bad quality to avoid postoperative spasm.
In conclusion, in this case report bilateral coronary to pulmonary fistulas associated with severe atherosclerosis are described. The suggested accelerated progression of coronary artery disease distal to the fistula and the hazard of rupture due to progression of the diameter of the aneurysmal formation through the years make surgical closure of the fistulas during concomitant cardiac surgery warranted.
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Acknowledgments
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We wish to thank Dr Mohamed Nasr, cardiac surgeon, National Heart Institute, Cairo, Egypt, and Dr Mohamed Sobhy, cardiologist, University Hospital of Alexandria, Egypt, for referring the patient to the Dutch Cardiac-Surgery Team.
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References
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- Kirklin JW, Barratt-Boyes BG. Congenital anomalies of the coronary arteriesIn: Kirklin JW, Barratt-Boyes BG, editors. Cardiac surgery. New York: Churchill Livingstone; 1993. pp. 1167-1193.
- Baim DS, Kline H, Silverman JF. Bilateral coronary artery-pulmonary artery fistulasReport of five cases and review of the literature. Circulation 1982;65:810-815.[Free Full Text]
- Atmaca Y, Altin T, Özdöl C, Pamir G, Ça
lar N, Oral D. Coronary-pulmonary artery fistula associated with right heart failure: successful closure of fistula with a graft stent Angiology 2002;53:613-616.[Medline] - Komatsu S, Sakata Y, Ueda Y, et al. Estimation of shunt flow in coronary-pulmonary fistula by lung perfusion scintigraphy with technetium-99m macroaggregated albumin Am J Cardiol 1998;82:1158-1161.[Medline]
- Said SAM, van der Werf T. Dutch survey of coronary artery fistulas in adults: congenital solitary fistulas Int J Cardiol 2006;106:323-332.[Medline]
- Hirose H, Takagi M, Miyagawa N, et al. Coronary atherosclerosis with dual coronary artery fistulas Scan Cardiovasc J 1998;32:313-314.
- van Dam DW, Noyez L, Skotnicki SH, Lacquet LK. Multiple fistulas between coronary and pulmonary arteries Eur J Cardio-thoracic Surg 1995;9:707-708.[Abstract]
- Fujimoto N, Onishi K, Tanabe M, et al. Two cases of giant aneurysm in coronary-pulmonary artery fistula associated with atherosclerotic change Int J Cardiol 2004;97:577-578.[Medline]
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Abstract
Introduction
