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Ann Thorac Surg 1997;63:1235-1242
© 1997 The Society of Thoracic Surgeons

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Original Article: Cardiovascular

Coronary Artery Fistulas in Infants and Children: A Surgical Review and Discussion of Coil Embolization

Divisions of Cardiovascular-Thoracic Surgery and Cardiology, Children's Memorial Hospital, and the Departments of Surgery and Pediatrics, Northwestern University Medical School, Chicago, Illinois

	Abstract

Top Footnotes Abstract Introduction Material and Methods Retrospective Cine Angiogram... Results Comment References

 
Background. Coronary artery fistula (CAF) is a rare congenital anomaly that can be complicated by intracardiac shunts, endocarditis, myocardial infarction, or coronary aneurysms. Recent reports have emphasized the efficacy of percutaneous transcatheter techniques. The purpose of this article is to review a 28-year surgical experience with CAF as a standard for comparison and to discuss the emergence and efficacy of transcutaneous catheter coil embolization as an alternative form of therapy.

Methods. From 1968 to 1996, 17 patients (age, 6 weeks to 16.5 years; mean age, 5.5 years) were diagnosed with CAF: 8 of 12 by echocardiography and 17 of 17 by cardiac catheterization. All patients with isolated CAF (n = 13) were asymptomatic despite significant clinical, electrocardiographic, and chest roentgenographic findings in 10. Sixteen had congenital CAF and 1 had acquired CAF after tetralogy of Fallot repair with injury of the anomalous left anterior descending coronary artery. Associated anomalies included tetralogy of Fallot (2), atrial septal defect (1), and patent ductus arteriosus (1). Nine fistulas originated from the right coronary artery and eight from the left. Drainage was to the right ventricle (9), right atrium (4), pulmonary artery (3), and left atrium (1).

Results. All patients had a median sternotomy with epicardial or endocardial ligation. Cardiopulmonary bypass was used in 8; 1 of these (iatrogenic CAF) required distal internal mammary artery bypass graft. There were no operative or late deaths. Follow-up evaluation by physical examination (17), echocardiography (8), and catheterization (2) showed no evidence of recurrent or residual CAF. A retrospective review of the 16 available cine cardioangiograms showed that coil embolization was possible in, at most, 6 patients.

Conclusions. Early surgical management of CAF is a safe and effective treatment resulting in 100% survival and 100% closure rate. Transcatheter embolization is a reasonable alternative to standard surgical closure in only a very small, select group of patients. These surgical results should be considered the standard against which transcatheter techniques are compared.

	Introduction

Top Footnotes Abstract Introduction Material and Methods Retrospective Cine Angiogram... Results Comment References

 
See also page 1242.

A congenital coronary artery fistula is an abnormal direct communication between any coronary artery and any of the cardiac chambers, as well as the superior vena cava, the coronary sinus, the pulmonary artery, and the pulmonary veins. Although the term is not entirely accurate, these communications are commonly referred to as coronary-cameral fistulas^* and are quite rare, occurring in approximately 1 in 50,000 patients with congenital heart disease [1]. In contrast, coronary artery fistulas are also found in association with pulmonary atresia with intact ventricular septum [2] and hypoplastic left heart syndrome [3]. However, the consequences and management of these fistulas and associated cardiac lesions are quite different and are not discussed in this article. Acquired coronary artery fistulas can occur as a result of intracardiac congenital heart operations [4] or transcutaneous catheter techniques used for myocardial biopsy [5] and coronary angioplasty [6]. These fistulas have varied consequences and range in importance from incidental findings at coronary angiography to important lesions of hemodynamic significance [5, 7].

Coronary artery fistulas involving the right heart structures are more common than those involving the left heart structures [8]. The great majority of infants and children with coronary artery fistulas are asymptomatic. Symptoms tend to occur more in the older age groups (more than 20 years of age) who may present with fatigue, dyspnea, angina, and congestive heart failure [9]. Other complications include endocarditis (5%) [10], aneurysmal formation [11], and in rare cases, rupture [12, 13]. The earliest descriptions were by Krause [14] and Brooks [15]. Successful surgical correction was first reported by Biörck and Crafoord in 1947 [16].

Guidelines are needed to determine the comparative therapeutic efficacy of operation and coil occlusion. The purpose of this article is to review a 28-year surgical experience with coronary artery fistulas in infants and children, and to discuss the emergence and efficacy of transcutaneous catheter coil embolization as an alternative form of therapy.

	Material and Methods

Top Footnotes Abstract Introduction Material and Methods Retrospective Cine Angiogram... Results Comment References

 
Retrospective Patient Review
Between 1968 and 1996, 17 patients, aged 6 weeks to 16.5 years (mean age, 5.0 years; median age, 2.6 years) were diagnosed with coronary artery fistula (Table 1). All patients with isolated congenital coronary artery fistula (n = 13) were reportedly asymptomatic despite continuous murmurs in all, electrocardiographic evidence of ventricular hypertrophy in 10, and chest roentgenographic evidence of cardiomegaly or increased vascular markings in 10. Diagnosis was by echocardiography in 8 of 12 patients and by cardiac catheterization in all. Four children had coronary artery fistula in association with other lesions, which included tetralogy of Fallot in 2 (one of these fistulas was an iatrogenically caused communication between an anomalous left anterior descending coronary artery and the right ventricle), atrial septal defect in 1, and patent ductus arteriosus in 1. Eight fistulas originated from the right coronary artery, and ten from the left (1 patient had two fistulas: one originated from the right coronary artery and the other from the left coronary artery). Drainage was to the right ventricle (9), right atrium (4), pulmonary artery (3), and left atrium (1).

View larger version (81K): [in this window] [in a new window]   Fig 1. . Diagrammatic representation of epicardial dissection and instrument isolation of a congenital right coronary artery-to-right ventricular fistula. Careful dissection with continuous electrocardiographic and echocardiographic monitors will ensure safe and total fistula closure.

View larger version (196K): [in this window] [in a new window]   Fig 2. . Transesophageal color-flow Doppler echocardiograms (A) before and (B) after epicardial ligation. Transesophageal echocardiography can also be used to detect wall motion abnormalities that might signal coronary flow problems. Arrow indicates fistulous tract. (RV = right ventricular.)

View larger version (58K): [in this window] [in a new window]   Fig 3. . Diagrammatic representation of a right coronary artery-to-right ventricular fistula by right ventricular exposure through the right atrium and tricuspid valve. Aortic cross-clamping and cardioplegia optimizes exposure and allows fistula identification by controlled cardioplegia administration. Continuous cardiopulmonary bypass without aortic cross-clamping in a beating or fibrillating heart can also be employed.

	Retrospective Cine Angiogram Analysis for Coil Occlusion

Top Footnotes Abstract Introduction Material and Methods Retrospective Cine Angiogram... Results Comment References

	Results

Top Footnotes Abstract Introduction Material and Methods Retrospective Cine Angiogram... Results Comment References

 
All patients survived without clinical or electrocardiographic evidence of myocardial infarction (Table 1). No patient had residual or recurrent coronary artery fistula as noted by disappearance of murmur or by echocardiographic evaluation. The average follow-up time was 7.2 years and was defined by the last documented clinical visit (some patients were lost to long-term follow-up because of relocation and perceived feelings of well being). All patients were asymptomatic and doing well.

The retrospective cine angiogram analysis for efficacy of operation versus coil occlusion revealed similar assessment by the surgeon and pediatric cardiologist agreeing on potential therapy in 15 of the 16 cases available for review (10 for operation and 5 for coil occlusion; in 1 case there was disagreement) (Table 2). The factors against coil occlusion were distal fistula, adjacent vessel at risk, need for concomitant distal coronary bypass, large fistula, and young age (Figs 4, 5). Factors that favored coil occlusion were proximal location, extraanatomic terminal fistulous vessel (away from the normal coronary artery distribution), and older age (Fig 6).

View larger version (132K): [in this window] [in a new window]   Fig 4. . Coronary angiogram demonstrating a left coronary artery-to-left ventricle fistula. The fistulous tract was aneurysmal, had multiple communications, and was adjacent to the left main coronary artery; for these reasons this patient was an unlikely candidate for coil occlusion.

View larger version (145K): [in this window] [in a new window]   Fig 5. . Coronary angiogram demonstrating two fistulous tracts (right coronary artery and left coronary artery) to the right ventricle. Small arteries with distal multiple communications excluded this patient from consideration for coil occlusion.

View larger version (172K): [in this window] [in a new window]   Fig 6. . Coronary angiogram demonstrating a left anterior descending artery-to-right atrium fistula. The fistulous tract is proximal enough, away from adjacent vessels, and large enough to consider catheter cannulation and coil embolization, even with the two communications to the right atrium.

	Comment

Top Footnotes Abstract Introduction Material and Methods Retrospective Cine Angiogram... Results Comment References

Our experience is similar to other reports [8, 17, 18] with regard to frequency distribution of pathologic anatomy, incidence of associated cardiac lesions, judicious use of cardiopulmonary bypass, low operative mortality (0% in this series), low incidence of perioperative complications, and uniform success in fistula ligation (Table 3). Our younger population (mean age, 5.0 years; median age, 2.6 years) and lack of symptoms in those with isolated coronary artery fistula probably reflect the representative patient pool of a children's hospital and the fact that preoperative symptoms are more likely to occur after 20 years of age [9].

The conduct of the operation has changed very little over time [19]. Approximately half of all fistulas are ligated epicardially without cardiopulmonary bypass. Our experience mirrors these reported results (epicardial ligation in 9 of 17 patients). Cardiopulmonary bypass is reserved for inaccessible lesions or for those patients with coexisting cardiac lesions that require repair [17, 18]. Newer myocardial protection techniques favor more accurate intracavitary exploration and definitive ligation. Transesophageal echocardiography has been shown to accurately assess intraoperative repairs [20] and was successfully used to confirm ligation in our last patient (see Fig 2).

Coronary artery fistulas have been closed using Gianturco coils [21, 22], detachable balloons [21, 23, 24], polyvinyl alcohol foam [25], and double-umbrella devices [22]. Although the number of patients who have had transcatheter procedures to treat coronary artery fistulas is small (fewer than 28 reported cases), a number of conclusions can be drawn from these reports. First, transcatheter procedures do not work in all patients. In fact, only 15 of 19 patients reported from the two largest series [21, 22] were able to have the fistula successfully closed. Of the 15 patients in whom closure was successful, only 7 (47%) underwent closure primarily with Gianturco coils; the remainder underwent closure with detachable balloons or double-umbrella devices, which are not presently available in the United States. Thus, from these two studies, only 36% (7/19) of coronary arteriovenous fistulas were able to be closed with Gianturco coils. In addition, there have been complications associated with the transcatheter method of coronary fistula occlusion, which include transient T-wave inversion on the electrocardiogram, which is associated with a small increase in creatine kinase levels [21, 24], embolization of the coil to the pulmonary artery [22], and transient arrhythmias [21–23].

Based on a review of the literature [21–25] and our personal experience, the requirements for satisfactory coil embolization of a coronary arteriovenous fistula include the following: the ability to safely cannulate the branch coronary artery that supplies the fistula, the absence of large branch vessels that can be inadvertently embolized, the presence of a single, narrow restrictive drainage site into the cardiac chamber or vessel, and the absence of multiple fistulous communications. Using these criteria, we reviewed the angiograms of 16 patients who had surgical repair of coronary arteriovenous fistulas. In these 16 patients both surgeon and cardiologist believed that coil embolization was possible in only 5, and the cardiologist-but not the surgeon-believed that coil occlusion was possible in 1 additional patient. The major reasons why coil occlusion was thought not to have been possible included adjacent vessels at risk, multiple communications, patient age limiting safe access to the branch coronary artery, and large fistula size. Thus in our retrospective evaluation of individuals with coronary fistulas that were successfully closed at operation, we believed that coil embolization could have been possible in at most 6 of 16 individuals (37%), a figure similar to that observed in the reports by Reidy and co-workers [21] and Perry and associates [22].

We have shown that asymptomatic infants and children with coronary artery fistula often have significant clinical, electrocardiographic and roentgenographic findings that warrant therapeutic closure. Surgical closure of coronary artery fistulas in children is a safe and effective method of treatment with excellent long-term results. Based on currently available devices, transcatheter embolization is a reasonable alternative to standard surgical closure in only a very small, select group of patients. These surgical results should be considered the standard against which transcatheter techniques are compared.

	Footnotes

Top Footnotes Abstract Introduction Material and Methods Retrospective Cine Angiogram... Results Comment References

 
Presented at the Forty-Third Annual Meeting of the Southern Thoracic Surgical Association, Cancun, Mexico, Nov 7–9, 1996.

Address reprint requests to Dr Mavroudis, Division of Cardiovascular-Thoracic Surgery, M/C 22, Children's Memorial Hospital, 2300 Children's Plaza, Chicago, IL 60614.

^* The "cameral" part of coronary–cameral fistula indicates a cardiac chamber (the atria and ventricles). This description technically excludes the other cardiac drainage sites for coronary artery fistula such as pulmonary artery, superior vena cava, coronary sinus, and pulmonary veins. We prefer to call these lesions "coronary artery fistula" without making reference to the drainage site. Added descriptors such as "congenital" or "acquired" can be used as appropriate.

	References

Top Footnotes Abstract Introduction Material and Methods Retrospective Cine Angiogram... Results Comment References

 

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This Article Abstract Alert me when this article is cited Alert me if a correction is posted Citation Map Services Email this article to a friend Similar articles in this journal Similar articles in PubMed Alert me to new issues of the journal Add to Personal Folders Download to citation manager Author home page(s): Constantine Mavroudis Carl L. Backer Permission Requests Citing Articles Citing Articles via HighWire Citing Articles via Google Scholar Google Scholar Articles by Mavroudis, C. Articles by Gevitz, M. Search for Related Content PubMed PubMed Citation Articles by Mavroudis, C. Articles by Gevitz, M. Related Collections Related Article