Demonstration of Coronary-Pulmonary Artery Fistula with Saccular Aneurysm in Multidetector Computed Tomography

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1 J Radiol Sci 2011; 36: Demonstration of Coronary-Pulmonary Artery Fistula with Saccular Aneurysm in Multidetector Computed Tomography Chiung-Ying Liao 1 Ching-Pei Chen 2 Ching Hsueh 1 Albert D. Yang 1 Shang-Yun Ho 1 Kwo-Whei Lee 1 Department of Radiology 1, Division of Cardiovascular Medicine 2, Changhua Christian Hospital, Changhua, Taiwan Abstract Coronary-pulmonary artery fistula is an uncommon cardiac anomaly, usually congenital. Most coronary-pulmonary artery fistulas are clinically and hemodynamically insignificant and are usually found incidentally. This report describes 15 case of complex coronary-pulmonary artery fistula with 5 originated from the left anterior descending coronary artery (33%), 2 originated from the right coronary artery (13%) and 8 originated from the bilateral coronary arteries. Variable size of dilated aneurismal structure was found in 13 of patients. Coronary-pulmonary artery fistula is supplied by either single or both coronary arteries and mostly drains to the left side of the pulmonary trunk. It is typically located in the anterolateral aspect of the pulmonary trunk. The complex anatomy of the fistula was shown in detail by multidetector computed tomography (MDCT) using multiplanar reconstruction and 3D volume rendering techniques. Coronary-pulmonary artery fistula (CPAF) is first described by Krause in 1865 [1]. Coronary artery anomalies are rare, with an incidence of 0.2% to 1.2% [2], among which CPAF is usually detected in 0.1% to 0.2% of coronary angiograms [3-4]. Although not all coronary-pulmonary artery fistulas are clinically or hemodynamically significant, some can result in serious consequences including myocardial ischemia, myocardial infarction, or sudden death [5]. Because of the complex structural anatomy and the probability of the multiple fistulas arising from different segments of the coronary arteries and coronary sinuses, conventional coronary angiography may not be sufficient. An ideal investigation technique should be noninvasive, safe and provide adequate anatomic description of the fistula. Here we present fifteen cases of coronary arteries, and terminating in the pulmonary artery with saccular aneurysm formation, clearly demonstrated by using the multidetector computed tomography. Materials and Method A total of 2,267 consecutive patients who underwent contrast-enhanced coronary CT angiography (CCTA) from Oct 2006 to Dec 2010 in our institution were included. We retrospectively reviewed cardiac CT reports using an electronic database, and 15 cases of Coronary-pulmonary artery fistula were collected for assessing the MDCT coronary angiography findings. These Patients were aged from 37 to 70 years (mean, 51 years), including 9 men and 6 women. Nine patients were asymptomatic (60%). The other 6 patients had various symptoms including dyspnea, chest tightness arrhythmia, and palpitations. Heart murmurs were audible in 3 patients and two were systolic murmur and the other one was diastolic murmur. Abnormal electrocardiographic findings were observed in 8 patients (53%), including sinus bradycardia in four patient, prolong QTC, nonspecific ST/T wave abnormality, Q wave in Lead III/ Correspondence Author to: Shang-Yun Ho Department of Radiology, Changhua Christian Hospital, Changhua, Taiwan No. 135, Nan-Hsiao Street, Changhua 500, Taiwan 153

2 avf and evidence of ventricular hypertrophy in one. The characteristics of these patients were summarized in Table l. Contrast-enhanced CT of the heart and great vessels, including coronary arteries, was performed in an attempt to demonstrate the course of the coronary-pulmonary artery fistula. CT coronary angiography was performed using a 64-slice MDCT scanner (Brilliance 64, Philips) with a retrospective electrocardiogram gated protocol. Imaging parameters of 120 kv, 1000 mas, and mm slice collimation were preset for the scan. In this case, gantry rotation speed at 0.4 sec per revolution and a helical pitch of 0.2 were applied. The examination was performed using a single breath-hold technique to cover 120 mm from the cardiac outflow tract to the apex of heart within a total scanning time of about 10 sec. A total of 100 ml nonionic water-soluble iodinated contrast medium at 320 mg I/mL concentration followed by 30 ml of normal saline IV was administered at a rate of 5 ml/ sec. A predefined temporal offset at 75% R-R wave interval (at diastole) of each cardiac cycle was reconstructed to demonstrate the anomaly, using multiplanar reconstruction and 3D volume rendering methods. The anatomy of the coronary artery-pulmonary artery fistula was complex but was well demonstrated after detailed analysis on the 64-slice MDCT coronary angiography. The plexus of tortuous dilated feeding vessels arise from coronary artery and usually coalesces to become dilated aneurismal structure draining into the main pulmonary artery. Results A single fistula presented in all of patients, 5 originated from the left anterior descending coronary artery (33%), 2 originated from the right coronary artery (13%) and 8 originated from the bilateral coronary arteries. Variable size of dilated aneurismal structure was found in 13 patients. Only one patient received percutaneous coils embolization in the dilated feeding vessels due to hemodynamic change and shortness of breath. And the size of the dilated aneurismal structure was measured about 2cm in diameter, which is the largest one. The 64-slice MDCT coronary angiography was showed the plexus of tortuous dilated feeding vessels arise from bilateral coronary arteries and coalesces to become dilated aneurismal structure draining into the main pulmonary artery (Fig. 1a, 1b). Table 1. The clinical symptom and characteristics of these patient Patient No Age, Sex Murmur Significant findings ECG Symptom Associated cardiac lesion 1 46, M Sinus bradycardia LVH Arrhythmia/ palpitation 2 47, F Sinus bradycardia 3 49, M Normal 4 58, M Gr. I-II systolic murmur at apex Sinus bradycardia Chest tightness Hypertension Mitral malve disorder 5 45, F Diastolic murmur Normal Shortness of breath 6 67, F Normal 7 58, M Normal Intermittent chest pain 8 70, M Prolonged QTC 9 40, F Nonspecific ST & T wave abnormality 10 46, F Normal 11 37, M Normal 12 48, M Right ward axis Chest tightness 13 40, F Sinus bradycardia 14 56, M systolic murmur at apex Intermittent chest tightness 15 62, M Q wave in lead III, avf 154

3 In most cases (13/15, 86.6%), the drainage site was the left lateral side of the pulmonary trunk, in one patient (6.7%) it was the right lateral side of the pulmonary trunk, and in one case (6.7%) it was the anteromedial side. Particularly in patients with an aneurysmal sac or in whom the diameter of the fistula vessel was big, the high-density flow jet was shown clearly on CT, and thus the drainage site could be assessed easily (Fig. 1b). The dilated aneurismal structure of other patients were equal or below 1cm in diameter. These patients were treated medically and followed up without interventional therapies such as transcatheter embolization or surgical closure due to normal myocardial perfusion and no significant left-to-right shunt. The distribution of site, origin and size of aneurismal structure is shown in Table 2. Discussion Congenital anomalies of the coronary arteries are found not infrequently, affecting about 0.2% to 1.2% of the general population [2]. CPAF is rare, detected in 0.1% to 0.2% of coronary angiograms [3-4]. Most of them originate from the RCA and almost always drain into low-pressure chambers of the heart (right ventricle 42.5%, right atrium 34%, and pulmonary artery 15%) [3]. Most CPAF is clinically and hemodynamically insignificant and is usually found incidentally. The factors influencing the clinical presentation and prognosis of the CPAF are the size of the communication, the amount of blood drained through it, the resistance of the recipient chamber, and development of myocardial ischemia or infarction (presumably resulting from the coronary steal phenomenon). Frank congestive heart failure is a frequent complication, especially in patients above 40 years of age; however other complications such as arrhythmias, infective endocarditis, rupture of an aneurismal fistula, and sudden death have been described [5]. Until recently, conventional coronary angiography was the diagnostic method of choice for detecting coronary anomalies. This is limited by invasiveness, its planar imaging nature, restricted angle of angiographic projections and concern for the contrast load [6]. So the precise course, complex configuration of the anomalous vessel and anatomical relationship with adjacent structures may be obscured on two-dimensional fluoroscopic imaging [7]. Cardiac MDCT is a new imaging technique in the evaluation of Figure 1 1a 1b Figure 1. a. The 3D volume rendering shows the surface of the complex coronary-pulmonary artery fistula and its relationship with adjacent structure. There are plexus of tortuous vessels arising from the proximal left anterior descending artery (LAD) and right coronary artery (RCA) form a network encircling the main pulmonary artery (MPA) and eventually coalesce to become a dilated aneurismal structure (*), running across the anterior aspect of the main pulmonary artery. b. The MPR at the Right anterolateral projection of the heart shows the aneurismal structure (*) just at the anterior aspect of the main pulmonary artery (MPA), presenting contrast jet phenomenon (arrow head) into the main pulmonary artery, indicating left to right shunt. 155

4 Table 2. The distribution by site of origin and size of aneurismal structure Patient No Involved Size of aneurismal coronary artery structure (mm) drainage site (which side of PA) 1 RCA, LAD 6.8 Lt lateral side 2 LAD 4.7 Lt lateral side 3 LAD 7.4 Lt lateral side 4 RCA 6.3 Lt lateral side 5 RCA, LAD 20 Lt lateral side 6 RCA, LAD 7.6 anteromedial side 7 RCA, LAD 7.5 Lt lateral side 8 LAD 0 Lt lateral side 9 RCA, LAD 10 Lt lateral side 10 RCA, LAD 9 Lt lateral side 11 RCA, LAD 3.7 Rt lateral side 12 LAD 0 Lt lateral side 13 LAD 5.7 Lt lateral side 14 RCA 7.7 Lt lateral side 15 RCA, LAD 10 Lt lateral side cardiovascular system. Although MDCT is inferior in temporal resolution to conventional coronary angiography (40 ms), it has some advantages over conventional angiography, such as short investigation time, noninvasiveness of the procedure, simple preparation, and minimal aftercare. MDCT coronary angiography may be used to identify anomalous origin and course of the coronary arteries like a fistula at its best projection, using multiplanar reconstruction and 3D volume rendering methods, without additional exposures of radiation and contrast medium. In recent reports, CPAF can be correctly identified by MDCT coronary angiography [7-12]. Treatment of asymptomatic patients with insignificant shunting is still a matter of debate [13]. Patients treated conservatively should be followed up closely for appearance of symptom. Most of the adult patients who are asymptomatic remain free of symptoms for long periods [14]. There has been a general agreement that all symptomatic patients with coronary fistulae should undergo closure of the fistulae as soon as diagnosis is made. Ischemic symptoms or positive findings at stress testing in the dependent territory (related to steal of myocardial blood-flow), aneurismal dilatation, with or without mural thrombus of the feeding coronary artery, cardiac cavity overload due to substantial blood shunting are usual indications for intervention to close a coronary fistula [15]. Transcatheter closure is an alternative therapeutic approach using different methods including detachable balloons, stainless steel, and platinum coils, the Amplatz occluder and alcohol or foam injection and covered stents. In asymptomatic patients the indication for CPAF closure is still debated. The prognosis in asymptomatic patients is good and a conservative clinical follow up is recommended because of the low incidence of an adverse outcome [16, 17]. In conclusion, MDCT coronary angiography is a noninvasive 3D imaging technique that provides an excellent overview of the cardiac and vascular anatomy that could be helpful for planning future cardiovascular therapeutic approach, interventional or surgical. This method is valuable particularly in complex vascular malformations, such as coronary pulmonary artery fistula, obviating the need for invasive procedures. References 1. Krause W. Über den Ursprung einer akzessorischen A. coronaria cordis aus der A. pulmonalis. Z Ratl Med 1865; 24: Dodge-Khatami A, Mavroudis C, Backer CL, et al. Congenital Heart Surgery Nomenclature and Database Project: anomalies of the coronary arteries. Ann Thorac Surg 2000; 69: S270-S Fernandes ED, Kadivar H, Hallman GL, et al. Congenital malformations of the coronary arteries: the Texas Heart Institute experience. Ann Thorac Surg 1992; 54: Angelini P. Normal and anomalous coronary arteries: definitions and classification. Am Heart J 1989; 117: Levin DC, Fellows KE, Abrams HL. Hemodynamically significant primary anomalies of the coronary arteries: angiographic aspects. Circulation 1978; 58: Chan MSM, Chan IYF, Fung KH, et al. Demonstration of complex coronary pulmonary artery fistula by MDCT and correlation with coronary angiography. AJR Am J Roentgenol 2005; 184: S28-S32 7. Tomasian A, Lell M, Currier J, Rahman J, Krishnam MS. Coronary artery to pulmonary artery fistulae with multiple aneurysms: radiological features on dualsource 64-slice CT angiography. Br J Radiol 2008; 81: e218-e Ozaki N, Wakita N, Inoue K, Yamada A. Surgical repair of coronary artery to pulmonary artery fistula with Aneurysms. Eur J Cardio-Thoracic 2009; 35: A-R Zeina, J Blinder, U Rosenschein, E Barmeir. Coronary-pulmonary artery fistula diagnosed by multidetector computed tomography. Postgrad Med J 2006; 82:

5 10. Yiginer O, Bas S, Feray H. Demonstration of coronaryto-pulmonary fistula with MDCT and conventional angiography. Int J Cardiol 2008; 134: e126-e Chan SM, Chan YF, Fung KH, et al. Demonstration of Complex Coronary-Pulmonary Artery Fistula by MDCT and Correlation with Coronary Angiography. AJR Am J Roentgenol 2005; 184: S28-S Choi CU, Kim JW, Yong HS, et al. Bilateral coronary artery fistula and communication between two fistulas identified on multidetector computed tomography in patient with coronary artery disease. Int J Cardiol 2008; 127: e118-e Hong GJ, Lin CY, Lee CY, et al. Congenital coronary artery fistulas: clinical considerations and surgical treatment. ANZ J Surg 2004; 74: Luo L, Kebede S, Wu S, Stouffer GA. Coronary artery fistulae. Am J Med Sci 2006; 332: Angelini P. Are all fistulae worth closing? Editorial comment. Tex Heart Inst J 2005; 32: Megan CS, Sol R, Stevev DC, et al. Prognostic significance of clinically silent coronaty artery fistulas. Am J Cardiol 1999; 83: Goldberg SL, Makkar R, Duckwiler G. New strategies in the percutaneous management of coronary artery fistulas: a case report. Catheter Cardiovasc Interv 2004; 61:

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