Interact CardioVasc Thorac Surg 2009;8:290-291. doi:10.1510/icvts.2008.190678
© 2009 European Association of Cardio-Thoracic Surgery
Case report - Cardiac general |
Surgical repair of coronary sinus atrial septal defect and supraventricular tachycardia
Nobuchika Ozaki*,
Noboru Wakita,
Kyozo Inoue and
Akitoshi Yamada
Department of Cardiovascular Surgery, Kobe Rosai Hospital, 4-1-23 Kagoike-dori, Chuo-ku, Kobe 651-0053, Hyogo, Japan
Received 12 August 2008;
received in revised form 6 October 2008;
accepted 9 October 2008
*Corresponding author. Tel.: +81-78-231-5901; fax: +81-78-242-5316.
E-mail address: nobozaki{at}kobeh.rofuku.ne.jp (N. Ozaki).
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Abstract
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A 67-year-old female had suffered from fatigue and palpitation. Cardiac examination revealed coronary sinus atrial septal defect, moderate mitral and tricuspid regurgitation, coronary artery disease, and supraventricular tachycardia with paroxysmal atrial fibrillation. Surgical repair of the anomaly, regurgitant valves, and arrhythmia associated with coronary revascularization was successfully performed and the patient has been doing well in normal sinus rhythm.
Key Words: Cardiac surgery; Coronary sinus; Congenital heart defect
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1. Introduction
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Coronary sinus atrial septal defect (ASD) is a rare entity. This anomaly was a part of unroofed coronary sinus syndrome, consisting of completely unroofed coronary sinus with or without persistent left superior vena cava (PLSVC), partially unroofed mid or terminal portion of coronary sinus, resulting in coronary sinus ASD [1]. We reported a case of an adult patient with atrioventricular valves regurgitation, coronary artery disease (CAD), supraventricular tachycardia, and coronary sinus ASD, who was successfully treated.
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2. Case report
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A 67-year-old female was referred to our hospital because of fatigue and palpitation. She has had antiarrhythmic drugs for years with a history of electrical defibrillation therapy for unknown supraventricular tachycardia eight years ago. Then ASD was diagnosed as the cause of tachycardiac arrhythmia. On admission, she was in New York Heart Association (NYHA) functional class III and had atrial flutter rhythm of 110–120/min at rest with paroxysmal atrial fibrillation (AF) of 140–170/min on mild effort (Fig. 1a). Transthoracic echocardiogram showed dilated right atrium and ventricle, moderate tricuspid and mitral regurgitation, and coronary sinus ASD without PLSVC (Fig. 2). Cardiac catheterization revealed pulmonary artery systolic pressure of 48 mmHg, Qp/Qs of 2.1, and left to right shunt ratio of 52.4%. Coronary angiography showed a severe stenosis of the proximal left anterior descending artery (LAD). No preoperative examination demonstrated the PLSVC.
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Fig. 1. Preoperative electrocardiogram showed atrial flutter (a) and postoperative electrocardiogram showed normal sinus rhythm (b).
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Fig. 2. Preoperative transthoracic echocardiogram showed a coronary sinus atrial septal defect (arrow).
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The operation was performed through the midline sternotomy under standard cardiopulmonary bypass with cannulation of the ascending aorta and bicaval veins. After aortic cross-clamping, the right atriotomy revealed the large defect laterally extending to a small orifice of the coronary sinus in the atrial septum. The lower rim of coronary sinus ASD was defect and the remnant part of coronary sinus in the left atrium was intact. The vestigial duct which might be the left superior vena cava was found left to the pulmonary trunk and left. Mitral and tricuspid regurgitation were due to annular dilatation with no cusp prolapse. Bipolar radiofrequency – modified Cox maze procedure [2] with an isthmus ablation was carried out in the right and left atrium at first. Then the coronary sinus ASD was closed using a 3.0x3.5-cm equine patch so that the small orifice of the coronary sinus could drain into the right atrium. After mitral and tricuspid annuloplasty using prosthetic rings, revascularization of the LAD with a left internal thoracic artery was performed. The patient was easily weaned from cardiopulmonary bypass. Aortic cross-clamp and cardiopulmonary bypass time were 111 min and 148 min, respectively. The postoperative course was uneventful. The postoperative echocardiogram showed no regurgitation of the mitral and tricuspid valves and no residual shunt of the atrial septum. The patient has been in normal sinus rhythm (Fig. 1b) and in NYHA functional class I postoperatively.
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3. Discussion
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Coronary sinus ASD is a rare congenital anomaly, which emerges in the process of development of the sinoatrial region of the heart. The diagnosis of this anomaly is usually difficult to be done preoperatively and some cases have been accidentally found during surgery. With development of echocardiography the precise diagnosis has been possible and transthoracic echocardiography was useful in this present case. Coronary sinus ASD should be suspected preoperatively when ASD cannot be clearly detected on the echocardiogram although the physical examination and cardiac catheterization show similar findings as those of ASD.
PLSVC has been often complicated with an unroofed coronary sinus syndrome [3]. In this patient the left SVC was found as a vestige laterally to the pulmonary trunk during surgery. Closure methods for coronary sinus ASD vary depending on its position and form [4]. Return of the coronary sinus was reconstructed to the right atrium with the patch which was also used in closure of ASD in this patient.
This patient has suffered from the supraventricular tachycardia including atrial flutter and paroxysmal AF because of a long-standing volume overload on the right-side heart. Arrhythmia associated with right heart failure could be sometimes lethal, resulting in a sudden death. Maze procedure and isthmus ablation were dramatically effective for this patient.
Tricuspid regurgitation in patients with ASD develops more significantly with annular dilatation due to a volume overload in the right heart. In contrast, the pathogenesis of significant mitral regurgitation in ASD is unclear. Significant mitral regurgitation associated with ASD has been found in 2–10% of operation cases [1] and this trend has appeared to be strong in patients older than 50 years of age (15%) and rare in those younger than 20 years of age (0.4%) [5]. Most causes of mitral regurgitation in ASD are mitral prolapses because of alteration in left ventricular configuration, which is a response to RV enlargement secondary to volume overload that results from left-to-right shunts at the atrial level. In this patient, annular dilatation was the main cause of mitral regurgitation, which was successfully repaired with mitral annular plication with a prosthetic ring.
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References
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- Kirklin JW, Barratt-Boyes BG. Unroofed coronary sinus syndrome, Cardiac Surgery, 3rd ed. New York. 2003:790–799.
- Gaynor SL, Schuessler RB, Bailey MS, Ishii Y, Boineau JP, Gleva MJ, Cox JL, Damiano RJ. Surgical treatment of atrial fibrillation: predictors of late recurrence. J Thorac Cardiovasc Surg 2005;129:104–111.[Abstract/Free Full Text]
- Ootaki Y, Yamaguchi M, Yoshimura N, Oka S, Yoshida M, Hasegawa T. Unroofed coronary simus syndrome: diagnosis, classification, and surgical treatment. J Thorac Cardiovasc Surg 2003;126:1655–1656.[Free Full Text]
- Nakano K, Ayusawa K, Gome A, Nakatani H, Nakamura Y, Sugimoto K, Sato A. Creation of coronary sinus using left atrial diaphragm in the patient with Cor Triatriatum and unroofed coronary sinus. Ann Thorac Surg 2006;81:1893–1895.[Abstract/Free Full Text]
- Konstantinides S, van Tournout FAM, Bennett JM. Unroofed coronary sinus associated with atrial septal defect and significant mitral regurgitation. J Thorac Cardiovasc Surg 1990;99:377–378.[Medline]
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Abstract
1. Introduction