A Rare Case of Triple Coronary Artery Fistulae Originating from Left Main and Right Coronary Arteries - PDF Document

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  1. CORONARY ARTERY FISTULAE | CASE REPORT A Rare Case of Triple Coronary Artery Fistulae Originating from Left Main and Right Coronary Arteries Mehmet Ali Elbey1, Akif Vatankulu2, Osman Sonmez2, Ahmet Bacaksiz2 Date received: 27/9/12, Reviewed: 22/10/12, Accepted: 26/11/12 Key words: coronary artery fistulae, coronary steal, angina pectoris DOI: 10.5083/ejcm.20424884.86 ABSTRACT 1. Dicle University School of Medicine Department of Cardiology Diyarbakır, Turkey. 2. Bezmialem Vakif University School of Medicine Department of Cardiology, Istanbul, Turkey. Dual coronary artery fistulae (CAF) involving both right and left coronary trees are uncommon; accounting for only 5% of all CAFs. A 48-year-old male patient was admitted to our institution for evaluation of chest pain. The coronary angiography revealed fistulae from left main coronary artery (LMCA) and right coronary artery (RCA) to the pulmonary artery. We concluded that angina pectoris was caused by a steal phenomenon and the patient was recommended surgical intervention. To our knowledge, this is the first paper reporting three fistulae associated with multiple aneurysms originating from proximal coronary arteries, which were connected to the pulmonary artery. CORRESPONDENCE Mehmet Ali Elbey, Dicle University, School of Medicine, Department of Cardiology, Diyarbakır, Turkey. PC: 21280, INTRODUCTION CASE REPORT Coronary artery fistulae (CAF) are rare con- genital or acquired coronary artery anomalies characterised by vascular connections from a coronary artery to a cardiac chamber or major central blood vessel. More than 90% of fistu- lae drain into the systemic venous side of the circulation (1, 2). CAF often arise from the right coronary artery system, and clinical presenta- tions are dependent on factors such as the type of fistulae, shunt volume, site of the shunt, and presence of other cardiac conditions (3). CAF is an anomaly resulting in the steal phenomenon of coronary blood flow (4). A 48-year-old male patient was admitted to the hospital with complaint of exertional chest pain. There was no history of significant chest wall trauma or any invasive cardiac procedure. He had previously been diagnosed with hyper- cholesterolemia. At physical examination, a mild continuous murmur (grade 1/6) could be heard, at the level of the second intercostal space of the left parasternal area. Electrocardiography demonstrated inferolateral ST depression. Tel: +90 0412 248 80 01 Fax: +90 0412 248 85 23 Email: elbeymali@hotmail.com Chest x-ray showed mild cardiomegaly and slightly increased pulmonary vascularity. With the clinical suspicion of ischemic coronary ar- tery disease, selective coronary angiography was performed and a fistula that originated from the ostial LMCA (Figure1 A-B), and another two from originated from the ostial RCA were detected (Figure1 C-D). We report an adult patient with three fistulae originating from the proximal LMCA and right coronary sinus to the main pulmonary artery demonstrated noninvasively with MDCT and in- vasively with coronary angiography. Left and right fistulae were connected and drained into the pulmonary artery. Multidedec- tor computed tomography (MCT) was per- formed in order to further evaluate the fistulae associated with aneurysm, which showed that the fistulae comprised three source vessels Figure 2 A-B). There were separate ostiums in the left main coronary artery and right coro- nary arteries. The same tract was also confirmed by MCT. We thought that angina pectoris was caused by a steal phenomenon and the patient was recommended surgical intervention. ISSN 2042-4884 136 EUROPEAN JOURNAL OF CARDIOVASCULAR MEDICINE VOL II ISSUE II

  2. A RARE CASE OF TRIPLE CORONARY ARTERY FISTULAE Figure 1: First fistula originating from the left main coronary artery with drainage into the aneurysmal tract. (A, B, arrow head). Second and third coronary artery fistulae originating from the ostial right coronary artery with drainage into aneurysmal tract (C, D, arrows). Left and right fistulae tract connect and drainage into the pulmonary artery (LMCA: left main coronary artery, L: left descending artery, C: circumflex artery, F: fistula, A: aneurysm). Figure 2: Multi-detector computed tomography showed that the fistulae were composed of three source vessels. Three fistulae, one branching from left main coronary artery (A, arrow head) and another two from the right coronary artery (B), have large aneurysm and were connected to the pulmonary artery (A and B). (L: left descending artery, C: circumflex artery, F: fistula, A: aneurysm). DISCUSSION Dual CAF involving both right and left coronary trees are uncom- mon accounting for only 5% of all CAFs (5). The most common prox- imal communication arises from right coronary artery followed by left anterior descending artery and the distal communication most commonly occurred in the pulmonary artery (1). Haemodynamic consequences of the coronary artery fistula depend on the size of the fistula and the communicating chamber. It causes a shunt from the high-pressure coronary artery to a lower-pressure cardiac chamber or vein in 90% of cases (6). The most frequent complaints of patients with CAF are angina pectoris (due to the coronary steal phenomenon), dyspnoea, palpitations and lethargy (7). Angina pec- toris can be caused by a steal phenomenon and by the manifesta- tion of coronary atherosclerosis in the fistula itself (8). The path-physiologic mechanisms of the symptoms are volume overload as a result of the shunt, coronary steal that causes de- creased myocardial oxygen supply and lack of capillary formation (10). Dual coronary artery fistulae more frequently present phenom- ena of secondary coronary ischemia than of significant left-right shunt with phenomena of coronary steal (11). CAF may cause an- gina pectoris resulting in the steal phenomenon of coronary flow (10) which was also seen in our case. Small coronary artery fistulae are usually silent but large fistulas can be associated with myocardial infarction, arrhythmias, congestive heart failure or endocarditis (6, 9). 137 EUROPEAN JOURNAL OF CARDIOVASCULAR MEDICINE VOL II ISSUE II

  3. HEALTHCARE BULLETIN | CORONARY ARTERY FISTULAE Small-sized fistulae are medically treated and further managed by a careful follow-up. However, large-sized symptomatic fistulas are treated either by surgical ligation or by percutaneous embolisation, depending on the anatomical characteristics of the fistulae (12). But controversies exist regarding the management of asymptomatic patients (6). In our case, because of the characteristics of the fistulae (especially left side fistula) surgical treatment was recommended for the patient. Cases of dual coronary artery fistulae have been reported previous- ly (1, 5, 10); however, to our knowledge triple coronary artery fistulae has not been reported before. This is the first report of three fistu- lae associated with multiple aneurysms originating from proximal LMCA and proximal RCA, which were connected to the pulmonary artery. REFERENCES 1 Vavuranakis M, Bush CA, Boudoulas H. Coronary artery fistulas in adults: incidence, angiographic characteristics, natural history. Cathet Cardiovasc Diagn 1995;35(2):116-20. 2 Yamanaka O, Hobbs RE. Coronary artery anomalies in 126,595 patients undergoing coronary arteriography. Cathet Cardiovasc Diagn 1990;21:28-40. 3 Gowda RM, Vasavada BC, Khan IA. Coronary artery fistulas: clinical and therapeutic considerations. Int J Cardiol 2006;107:7-10. 4 Schmid M, Achenbach S, Ludwig J, et al. Visualization of coronary artery anomalies by contrast-enhanced multi-detector row spiral computed tomography. Int J Cardiol 2006;111(3):430–435. 5 Hirose H, Takagi M, Miyagawa N, et al. Coronary atherosclerosis with dual coronary artery fistulas. Scand Cardiovasc J 1998;32:313-4. 6 Oncel D, Oncel G. Right coronary artery to left ventricle fistula-effective diagnosis with 64-MDCT. Int J Cardiovasc Imaging 2007;23(2):287-291. 7 Tirilomis T, Aleksic I, Busch T, Zenker D, Ruschewski W, Dalichau H. Congenital coronary artery fistulas in adults: surgical treatment and outcome. Int J Cardiol 2005;98:57-9. 8 Urrutia-S CO, Falaschi G, Ott DA, Cooley DE. Surgical management of 56 patients with congenital coronay artery fistulas. Ann Thorac Surg 1983;35:300-7. 9 Latson LA (2007) Coronary artery fistulas: how to manage them. Catheter Cardiovasc Interv 2007 70:110–116. 10 Kim MJ, Kwon HY, Hwang CS, et al. Dual Fistulas of Ascending Aorta and Coronary Artery to Pulmonary Artery. Korean Circ J 2011; 41:213-216. 11 Kidawa M, Peruga JZ, Forys J, Krzeminska-Paula M, Kasprzak JD. Acute coronary Syndrome or steal phenomenon -a case of right coronary to right ventricle fistula. Kardiol Pol 2009;67:287-90. 12 Said SA, Voogt WG, Hamad MS and Schonberger J. Surgical Treatment of Bilateral Aneurysmal Coronary to Pulmonary Artery Fistulas Associated With Severe Atherosclerosis. Ann Thorac Surg 2007;83:291–3. 138 EUROPEAN JOURNAL OF CARDIOVASCULAR MEDICINE VOL II ISSUE II