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Case History :

A 3-month-old baby was referred to the cardiologist for evaluation of a murmur. Physical examination revealed an active precordium, bounding pulses, and a 3/6 continuous murmur heard throughout the right precordium. The electrocardiogram was normal. Transthoracic echocardiography showed a markedly dilated proximal right coronary artery with a dilated branch coursing posteriorly through the right atrium. Selective coronary angiography also showed a markedly dilated right coronary artery (8.8 mm in diameter). It was later on diagnosed to be an AV Fistula.

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Investigation Reports :

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Discussion :

Treatment Approach for Coronary AV Fistulae

Although coronary artery fistulae are a relatively rare entity, they may give rise to such complications as cardiac failure, ischemia, thrombosis, arrhythmia, endocarditis, and even rupture. Surgical closure has been reported, either by external ligation of the fistula or by internal patching of the orifice. However, surgery entails the risks attendant upon cardiopulmonary bypass and median sternotomy.

Although successful fistula occlusion has been reported with inflatable balloons, polyvinyl alcohol foam, and umbrellas, the use of implantable coils is currently considered the best method, due to improved control and delivery techniques. The risks of transcatheter coil occlusion are low, and there have been no reports of death in children. One fatality in an adult patient has been reported. In children who are asymptomatic, cardiologists perform catheterization when the femoral artery has grown enough to avoid complications. Developments in catheter and coil technology have made it possible to use 4- or 5-F catheters safely in infants. In addition, the fistula may be entered from the right atrium or ventricle; generally, this requires placing a wire through the fistula from the coronary artery.

Little has been reported about the intermediate- to long-term results following transcatheter closure of coronary artery fistulae in children. Immediately after occlusion, physicians generally perform chest radiography, electrocardiography, echocardiography, and Holter monitoring. It is also recommended to evaluate patients at 2, 6, and 12 months after coil occlusion. Physicians often perform Holter monitoring again at 12 months to evaluate the possibility of vascular compromise to the sinoatrial or atrioventricular node and associated rhythm disturbance. Echocardiography is an excellent method for evaluating ventricular dimensions, ventricular function, coronary artery dimensions, and fistula leakage. Cardiac catheterization and selective coronary angiography may be performed 12 to 24 months after coil occlusion to evaluate coronary artery dimensions and fistula persistence, particularly in those children who have limited echocardiographic windows.

The persistence of fistula leakage after coil occlusion is problematic. Given the risk of bacterial endocarditis and the presence of a foreign body (the coil), many researchers recommend repeat catheterization and coil occlusion after 3 to 6 months. Placement of additional coils appears to be efficacious in abolishing persistent fistula leaks.

Following coronary artery fistula occlusion, several children have demonstrated persistent coronary artery dilatation as late as 4 years, which is the duration of our follow-up. These children pose a therapeutic dilemma. Cardiologists administer low-dose aspirin therapy (3 to 5 mg/kg per day) until coronary normalization occurs. Severe coronary artery dilatation (>10 mm) may warrant anticoagulation with warfarin, especially in patients with sluggish coronary flow, although there is little information available concerning the risk of coronary thrombosis in this group.

Transcatheter coil occlusion of coronary artery fistulae is a safe and effective procedure in children. Cardiologists recommend that children undergo this procedure in centers where experienced anesthesiologists and excellent pediatric and intensive care facilities are available, in order to minimize complications and expedite recovery.