Interact CardioVasc Thorac Surg 2009;9:124-126. doi:10.1510/icvts.2009.203489
© 2009 European Association of Cardio-Thoracic Surgery
Case report - Cardiac general |
Treatment of high-output coronary artery fistula by off-pump coronary artery bypass grafting and ligation of fistula
Balakrishnan Mahesh*,
Manoraj Navaratnarajah,
Kwabena Mensah and
Mohamed Amrani
Center for Cardiothoracic Surgery and Transplantation, Harefield Hospital, Harefield, UK
Received 1 February 2009;
received in revised form 12 March 2009;
accepted 16 March 2009
*Corresponding author. Department of Thoracic Surgery, Norfolk and Norwich University Hospital, Norwich NR4 7UY, UK. Tel.: +44-1603-286286.
E-mail address: drbmahesh{at}googlemail.com (B. Mahesh).
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Abstract
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Coronary artery fistulas (CAF) are uncommon entities often associated with myocardial ischemia and high output failure. Surgical options include ligation of the fistula, with/without simultaneous coronary artery bypass grafting (CABG). We report a case of left main coronary artery (LMCA) fistula to the coronary sinus (CS), which was associated with high-output bi-ventricular failure, and moderate mitral (MR) and tricuspid regurgitation (TR), related to the volume overload and annular dilatation. This was tackled elegantly by off-pump CABG to protect the territories supplied by the LMCA, followed by ligation of the fistula. This resulted in resolution of the MR and TR. Intraoperative transesophageal echocardiogram (TEE) greatly facilitated the surgical treatment, by identifying the origin and the draining points for the fistula, and aided in the quantification of MR and TR, which had regressed sufficiently at the end of the procedure and did not require surgical correction. This article outlines the importance of multi-disciplinary treatment approach for this complex condition.
Key Words: Coronary artery fistula; Off-pump coronary revascularization; Mitral regurgitation; Coronary revascularization
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1. Introduction
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Coronary artery fistulas (CAF) are uncommon, often presenting with myocardial ischemia and high-output congestive heart failure (CHF) [1]. Surgical options include ligation of the fistula, with/without simultaneous coronary artery bypass grafting (CABG) [1–3]. This may be performed without cardiopulmonary bypass (CPB) [4, 5]. We report a case of left main coronary artery (LMCA) fistula to the coronary sinus (CS), which was associated with high-output CHF, and moderate mitral (MR) and tricuspid regurgitation (TR), related to volume-overload and annular dilatation. This was tackled by CABG without CPB [off-pump-CABG (OPCABG)] to protect the territories supplied by the LMCA, followed by ligation of the fistula. Intraoperative transesophageal echocardiogram (TEE) greatly facilitated surgical treatment, by identifying origin and drainage of the fistula, and by quantifying the MR and TR, which had regressed sufficiently at the end of the procedure and did not require surgical correction.
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2. Case
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A 74-year-old lady presented with CHF. Echocardiogram revealed dilated, volume-overloaded right (RV) and left ventricles (LV), pulmonary hypertension, with moderate MR and TR. Computerized-tomographic angiogram (CTA) revealed a massively enlarged LMCA, with anomalous drainage into CS (Fig. 1a–f), leading to high-output arteriovenous-fistula (AVF). Left anterior descending (LAD) and circumflex arteries arose from this dilated LMCA (Fig. 1a).
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Fig. 1. Computerised tomograms showing (a) reconstruction of the left main coronary fistula with coronaries arising from it (arrowhead); (b, e) tortuous course of the fistula in the posterior atrioventricular groove with calcification (arrowhead), (c, d) fistula arising from the left CS of Valsalva of the aorta (dark arrowhead), running in the posterior AV groove (white arrowhead), and (f) draining into the CS (arrowhead).
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She underwent operative intervention through median-sternotomy. Intra-operative TEE confirmed origin and drainage of the fistula (Fig. 2a, b). Ligating the fistula on CPB with aortic cross-clamping and administration of cardioplegia through the aortic root or coronary ostia would not allow delivery of cardioplegia to myocardium due to the fistula. To obviate this problem, operative strategy involved initially performing OPCABG to revascularize the myocardium supplied by the fistula. Left internal thoracic artery (LITA) and long saphenous vein (LSV) were harvested, and using off-pump stabilizers, the LITA was anastomosed to the mid-LAD artery. Having protected the LAD-territory, the LSV was anastomosed distally to the obtuse-marginal artery using off-pump stabilizers, and proximally to the aorta. Having completely revascularized the myocardial territories supplied by the LMCA fistula, we exposed the fistula in the left atrioventricular groove using off-pump stabilizers; a non-calcified portion of the fistula was identified, mobilised, ligated and excised, the ends were overrun with 4-0 polypropylene (Fig. 2c–d). Intraoperative TEE greatly facilitated entire decision-making process, identifying the origin and drainage of the fistula, and in quantifying MR and TR during surgery. Following closure of the high-output AVF and myocardial revascularization, TEE revealed near-complete resolution of MR and TR. She made an uneventful recovery.
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Fig. 2. Transesophageal echocardiogram showing (a) dilated, aneurysmal LMCA emerging from the left CS of Valsalva of the aorta (arrowhead), and (b) entering into the CS (arrowhead). Intraoperative photographs showing (c) completed saphenous vein graft to the OM (SVG to OM), and ligation of the fistula (arrowhead), and (d) excision of a segment of the fistula, and overrunning of the ends with 4-0 polypropylene suture.
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3. Discussion
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CAFs arise as a result of anomalous connection between coronary arteries and another cardiac chamber or vessel [1], usually on the right side of the circulation [2, 3]. These may be congenital [2], acquired, traumatic or iatrogenic [6], and present with exertional dyspnea, fatigue, myocardial ischemia, and CHF, depending on the shunt [6]. Myocardial blood supply may be adequate at rest, but may be inadequate during exercise, leading to angina [1], akin to steal phenomenon [3]. Patients with large fistulas may present with CHF in infancy. Symptoms and complications of large or multiple CAFs may be apparent in patients diagnosed at later ages [3], and include bacterial endocarditis, thrombosis, embolization, pulmonary hypertension, myocardial ischemia, infarction, dissection and rupture [3, 6]. Investigations used to diagnose include echocardiography, magnetic resonance imaging, CTA and coronary angiography [1, 6].
The majority of small, symptomatic CAFs do not need intervention. CHF may occur in infants with large fistulas and may need intervention. Initial medical management may lead to decrease in symptoms; eventually 50% of fistulas become asymptomatic due to decrease in relative size of the fistula and shunt with growth, or spontaneous regression [3]. Intervention may be required for symptomatic patients or those with complications, which may develop in 50% of patients [3]. Approximately 10–30% of patients with CAFs have another congenital anomaly, such as Tetralogy of Fallot, PDA, and atrial septal defect [3]. Most commonly, CAFs are treated with surgical ligation or closure by a variety of percutaneous methods including coils, microcoils and balloons, double-umbrella devices, and vascular occlusion devices [6].
Abdelmoneim and associates described 30 patients with CAFs; 17 patients were managed conservatively, seven patients underwent surgical closure, and the remaining underwent percutaneous closure by coil embolization [1]. Device closure has  10% leak rate [3]. Surgical ligation of CAF is usually performed on CPB, especially when they are located laterally and drain into the CS. With off-pump techniques, ligation of laterally located fistulas can be performed without CPB [4]. Intraoperative TEE plays an important role in evaluating the efficacy of fistula obliteration [5].
In our case of CAF from the LMCA to CS, there was high-output CHF, with bi-ventricular dilatation, pulmonary hypertension, moderate MR and TR. This fistula needed to be ligated to manage the high-output CHF. Had we cross-clamped the aorta and administered cardioplegia into aortic root, myocardial cardioplegic arrest would be impossible to achieve due to run-off of cardioplegia from the LMCA into CS. In younger patients, where the fistula may not be calcified, temporary occlusion with a soft clamp or vascular sling may allow cardioplegic arrest. But, when the fistula is calcified, as in our case, manipulating it more often than absolutely necessary may embolize calcific material distally in the fistula and into the coronary arteries, with serious consequences. Our strategy was the safest, and involved minimal manipulation of the fistula in the non-calcified portion. OPCABG prior to ligating the CAF obviated the need for cardioplegia, revascularized the myocardium supplied by LAD and circumflex arteries arising from the CAF, and minimized the risk of myocardial infarction on ligating the fistula. Great care was taken while exposing and mobilizing the fistula, not to embolise clot and calcific material distally. Intraoperative TEE played an important role in evaluating efficacy of fistula obliteration [5]. Following ligation of fistula and resolution of high-output state, TEE confirmed that volume overload on RV and LV had abated sufficiently to result in near-complete resolution of secondary MR and TR.
In conclusion, we describe the first case of this nature, using combination of OPCABG to protect the myocardial territories subtended by the fistula, ligation of fistula using off-pump stabilizers, and judicious use of TEE to identify origin and drainage of the fistula, presence of MR and TR related to volume-overload, and near-complete resolution of valvular incompetence following surgical ligation of fistula and correction of high-output state. This reveals the importance of multi-modality approach in managing these complex fistulas.
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References
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Abstract
1. Introduction