Transcatheter coil embolization of multiple coronary artery-to-left ventricle fistulas: report of a rare case
Coronary artery fistula (CAF) is uncommon but remains the most frequent hemodynamically significant congenital coronary artery anomaly.1 The majority of fistula is single and drains into the right heart, only 3.5% into the left ventricle.2 A large fistula requires closure to prevent complications such as myocardial ischemia resulting from coronary steal, congestive heart failure, endocarditis and potential aneurysmal dilatation and rupture.3-5 Here we presented a rare case of CAF with multiple origins involving left anterior descending artery (LAD), left circumflex branch (LCX) and right coronary artery (RCA), and draining into the left ventricle, which was successfully closed by coil embolization. CASE REPORT A 61-year-old man who complained of chest pain and fatigue on exertion for two years, was admitted to our hospital in March 2007. Physical examination revealed nothing particular and no continuous murmur audible. A chest X-ray showed a cardiothoracic ratio of 45.5%, and there was normal sinus rhythm with no significant ST-T changes on regular electrocardiogram (ECG). On two-dimensional echocardiography, the RCA was dilated. Doppler echocardiography showed a continuous flow into the left ventricle (LV) beneath the posterior leaflet of the mitral valve (Figure 1). A RCA-to-LV fistula was suspected. Selective coronary angiograms demonstrated a common large tortuous fistula arising from the distal LAD, LCX and RCA draining into the LV. The proximal LAD, LCX and RCA were dilated and tortuous (Figure 2). After careful discussion, a staged transcatheter coil embolism regimen was taken.Figure 1.: Doppler echocardiography shows a continuous flow into the left ventricle beneath the posterior leaflet of the mitral valve.Figure 2.: Selective coronary angiograms show a common fistula arising from the distal LCA and RCA running in the LV.The RCA was engaged with a 7F JR 4.0 (Cordis, USA) guiding catheter. Due to the microcatheter (Cook, Europe) not long enough to reach CAF, we had to shorten the length of JR 4.0 guiding catheter by cutting the proximal of 7F JR 4.0 guiding catheter and connecting to 6F JR 4.0 guiding catheter tail-to-tail. A runthrough 0.3556 mm (0.014') guide wire (Terumo, Japan) and a microcatheter were passed through this guiding catheter to the RCA together, after guide wire removed, 2 larger microcoils (Cook) (6 mm × 5 mm) were placed as anchors and then 2 smaller microcoils (3 mm × 3 mm) were released to embolize the CAF. And then the LCX-CAF was successfully closed with similar method. Briefly, 7F JL 4.0was used to engage the left coronary artery (LCA), a microcatheter was positioned in the distal LCX over the guide wire, and 4 microcoils (two 6 mm × 5 mm, two 3 mm × 3 mm) were used to close LCX-CAF successfully. Repeated angiography 15 minutes later showed satisfactory result of the RCA-CAF and LCX-CAF without residual shunting. A month later, the patient was readmitted for another transcatheter closing for LAD-CAF. Satisfied result in LCX and RCA was verified by repeated coronary angiogram. 7F JL 4.0 was used to engage the LCA, 2 microcoils (3 mm × 3 mm) were used to close LAD-CAF through microcatheter successfully. Complete closure of the coronary artery fistula was confirmed by angiography (Figure 3). The patient is on close follow up. At 6 months, patients did not present with any symptoms of chest discomfort and repeated Doppler echocardiography confirmed persistent occlusion of the fistula with no residual shunt.Figure 3.: Post-intervention selective coronary angiograms show complete occlusion of the fistula.DISCUSSION CAF, an uncommon entity of congenital heart disease, first described by Krause in 1865, was characterized by the involved coronary artery having a normal origin from the aorta with a fistulous communication with the atria or ventricles, or with the pulmonary artery (PA).6 During the embryonic development, sinusoid gap of trabeculae of cardiac muscle is connected to coronary artery. Along with the development of myocardium the sinusoid gap was compressed and degraded to capillary. But if sinusoid gap persisted instead of degraded it would turn to be CAF. The majority of fistula drains into the right heart, with 15% into the pulmonary artery and 7% into the coronary sinus. Congenital arteriosystemic fistula rarely drains into the left heart, only 5% draining into the left atrium and 3.5% into the left ventricle.2 And most of the CAF is single. Here we presented a case of CAF with multiple origins involving LAD, LCX and RCA, and draining into the left ventricle. CAF with large shunt can cause heart failure, myocardial ischemia, arrhythmia, infective endocarditis or aneurysm progressive enlargement and rupture.3-5 In this case with significant shunt, blood may preferentially flow to the coronary fistula at the expense of the other coronary arteries, a phenomenon also known as “coronary steal”. Coronary steal is particularly evident during times of stress and can result in myocardial ischemia to territories not supplied by the arterial fistula. So the patient presented with chest pain. Recently, transcatheter intervention was provided as a new therapeutic approach for characteristics of high efficacy and micro-invasive. Coils, detachable balloons, polyvinyl alcohol foam and occlusive devices have been successfully used in the closure of CAF.7-11 In this case with multiple CAF, which might result in longer operation time, greater contrast volume, it needs to be performed by stage for the sake of safety. Moreover, the location of CAF, which is located in the distal coronary artery and extremely tortuous, inspired us to use coil other than Amplatzer duct occluder or else.10 During implementing the RCA coil embolization, due to the obvious tortuous and the microcatheter can not reach the satisfactory position, we connect a 7F JR 4.0 guiding catheter with a 6F JR 4.0 one, which can effectively shorten the length of guiding catheter and easily send the microcatheter to proper position to release coil. This technology is mainly used in the percutaneous transcoronary angioplasty. The application to CAF embolization is rarely to be reported. In conclusion, we successfully treated a complicated, high-flow coronary artery fistula by transcatheter coil embolization. Catheter embolization is an effective and safe treatment for coronary artery fistula in the strictly selected cases.
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