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Asian Cardiovasc Thorac Ann 2008;16:324-326
© 2008 Asia Publishing EXchange Ltd


CASE STUDIES

Anomalous Left Coronary Artery from Non-facing Pulmonary Sinus

Masanori Hirota, MD, Masaaki Kawada, MD, Kozo Ishino, MD, Shunji Sano, MD

Department of Cardiovascular Surgery, Okayama University Graduate School of Medicine, Dentistry & Pharmaceutical Sciences, Okayama, Japan

For reprint information contact: Masanori Hirota, MD, Tel: 81 86 235 7359, Fax: 81 86 235 7431, Email: mhirota{at}md.okayama-u.ac.jp, Department of Cardiovascular Surgery, Okayama University Graduate School of Medicine, Dentistry and Pharmaceutical Sciences, 2-5-1 Shikata-cho, Okayama 700-8558, Japan.


    ABSTRACT
 TOP
 ABSTRACT
 INTRODUCTION
 CASE REPORT
 DISCUSSION
 REFERENCES
 
A case of anomalous left coronary artery originating from the non-facing pulmonary sinus, located at the most distal position from the aorta, is described. An intrapulmonary tunnel was created without causing undue stenosis or kinking. The Takeuchi procedure is considered to be a useful surgical technique when the origin of the anomalous left coronary artery is far from the aorta.


    INTRODUCTION
 TOP
 ABSTRACT
 INTRODUCTION
 CASE REPORT
 DISCUSSION
 REFERENCES
 
The gold standard therapy for anomalous left coronary artery originating from the pulmonary artery (ALCAPA) is surgical correction that establishes a 2-coronary system.1 Such operations include direct reimplantation, the Takeuchi procedure and saphenous vein grafting.2,3 Direct reimplantation is the most common procedure because the anomalous left coronary artery (LCA) usually originates from the right-facing pulmonary sinus.4 However, it cannot be carried out due to technical reasons in the following morphological variations: anomalous LCA originating distally from the aorta, short LCA, or anomalous LCA with an inappropriate angle. We describe a patient with ALCAPA originating from the non-facing pulmonary sinus that was located most distal from the aorta.


    CASE REPORT
 TOP
 ABSTRACT
 INTRODUCTION
 CASE REPORT
 DISCUSSION
 REFERENCES
 
A heart murmur was detected at the 3-month check-up in a girl born after a full-term normal pregnancy. At 6 months old, weighing 6.4 kg, she was referred to our unit. On physical examination, a grade 3/6 systolic murmur was heard at the apex. Chest radiography revealed no congestion and slight cardiomegaly with a cardiothoracic ratio of 0.62. Electrocardiography showed a normal QRS axis, left atrial overloading and left ventricular (LV) hypertrophy without ischemic ST-T changes. On echocardiography, a dilated LV (157% of normal) with severe mitral regurgitation, an ejection fraction of 53% and fractional shortening of 28% were noted. On detailed scanning, an anomalous LCA could be seen originating from the non-facing pulmonary sinus, which is the most distal position from the aorta (Figure 1AGo). Selective right coronary angiography revealed retrograde filling of the anomalous LCA via collateral septal arteries; pulmonary artery (PA) opacification was also noted. The tentative diagnosis was ALCAPA from the non-facing pulmonary sinus with severe mitral regurgitation. An elective operation was scheduled. The chest was opened through a median sternotomy. Inspection of the heart revealed a normal relationship of the great arteries and well-developed collaterals on the anterior wall of the right ventricle. The anomalous LCA was found to originate from the left anterolateral side of the PA. Cardiopulmonary bypass was established using bicaval drainage and return from the ascending aorta. The patient was cooled down to 28°C. The aorta and PA were cross clamped. Cold crystalloid cardioplegia was delivered via both the aorta and the pulmonary root. After cardiac arrest, the main PA was opened transversely, and the anomalous LCA was found to impinge on the middle of the non-facing sinus. A 5-mm aortopulmonary window was created. A long rectangular flap from the anterior wall of the main PA was fashioned with an incision parallel to the initial transverse incision, and its hinge was based on the created aortopulmonary window (Figure 2AGo). This flap was sutured to the posterior wall of the PA to create an intrapulmonary tunnel that ran transversely just above the commissure of the pulmonary valve (Figure 2BGo). The modified method of Reed and colleagues4 was used for mitral annuloplasty of both commissures through a right-sided left atrial incision. The main PA was reconstructed with a generous autologous pericardial patch. Weaning from cardiopulmonary bypass was uneventful. Bypass time was 131 min, and the aortic cross clamp time was 69 min. A postoperative aortogram showed an intrapulmonary tunnel without undue stenosis or kinking (Figure 1BGo). Echocardiography 6 months after the operation demonstrated that the LV had normal dimensions, there was no mitral regurgitation, and systolic LV function had improved (ejection fraction, 71%; fractional shortening, 39%).


Figure 1
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Figure 1. (A) The anomalous left coronary artery from the non-facing pulmonary sinus can be seen on cross-sectional echocardiography; (B) Postoperative aortogram showing the intrapulmonary tunnel without undue stenosis or kinking. Ao = aorta.

 

Figure 2
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Figure 2. (A) After transverse incision of the main pulmonary artery, the anomalous left coronary artery was observed in the middle of the non-facing pulmonary sinus; a 5-mm aortopulmonary window was created, and a long rectangular flap from the anterior wall of the main pulmonary artery was fashioned with an incision parallel to the initial transverse incision; (B) The hinge of the rectangular flap, based on the aortopulmonary window, was sutured to the pulmonary artery posterior wall to create an intrapulmonary tunnel running transversely just above the pulmonary valve commissure.

 

    DISCUSSION
 TOP
 ABSTRACT
 INTRODUCTION
 CASE REPORT
 DISCUSSION
 REFERENCES
 
An anomalous LCA usually originates from the right-hand pulmonary sinus. However, in rare cases, the anomalous LCA originates from the left-hand pulmonary sinus and runs above the commissure between the right-hand and non-facing sinuses, or originates from the posterior wall of the right or left branch of the PA.1,5 In this case, the anomalous LCA impinged on the non-facing pulmonary sinus, and direct reimplantation was considered technically difficult. Turley and colleagues2 reported that direct reimplantation using tube reconstruction to extend the anomalous LCA with a segment of the PA wall is feasible in all cases, regardless of the anomalous LCA origin. However, we chose the Takeuchi procedure because it would require a much shorter cross clamp time than tube reconstruction, given the need for post-surgical mechanical assist devices.6 When the Takeuchi procedure is used, in particular for the non-facing pulmonary sinus, attention should be given to the commissure between the right-hand and the non-facing sinuses because it has a curved course along the inner curvature of the main PA when the anomalous ostium is located deep in the sinus.

Based on preoperative imaging, which included the combined use of color-flow mapping and pulsed Doppler, the anomalous LCA originating from the non-facing pulmonary sinus was identified.7 The geometric analysis was performed based on the interrelationship of the orifice and the mobile cusps of the pulmonary valve and its commissures, especially on both sides of the non-facing sinus. When an anomalous coronary ostium is located at the most distal position from the aorta, the Takeuchi procedure is considered to be a useful surgical technique in such a rare case.


    REFERENCES
 TOP
 ABSTRACT
 INTRODUCTION
 CASE REPORT
 DISCUSSION
 REFERENCES
 

  1. Dodge-Khatami A, Mavroudis C, Backer CL. Anomalous origin of the left coronary artery from the pulmonary artery: collective review of surgical therapy. Ann Thorac Surg 2002;74:946–55.[Abstract/Free Full Text]

  2. Turley K, Szarnicki RJ, Flachsbart KD, Richter RC, Popper RW, Tarnoff H. Aortic implantation is possible in all cases of anomalous origin of the left coronary artery from the pulmonary artery. Ann Thorac Surg 1995;60:84–9.[Abstract/Free Full Text]

  3. Takeuchi S, Imamura H, Katsumoto K, Hayashi I, Katohgi T, Yozu R, et al. New surgical method for repair of anomalous left coronary artery from pulmonary artery. J Thorac Cardiovasc Surg 1979;78:7–11.[Abstract]

  4. Reed GE, Pooley RW, Moggio RA. Durability of measured mitral annuloplasty: seventeen-year study. J Thorac Cardiovasc Surg 1980;79:321–5.[Abstract]

  5. Smith A, Arnold R, Anderson RH, Wilkinson JL, Qureshi SA, Gerlis LM, et al. Anomalous origin of the left coronary artery from the pulmonary trunk. Anatomic findings in relation to pathophysiology and surgical repair. J Thorac Cardiovasc Surg 1989;98:16–24.[Abstract]

  6. del Nido PJ, Duncan BW, Mayer JE Jr, Wessel DL, LaPierre RA, Jonas RA. Left ventricular assist device improves survival in children with left ventricular dysfunction after repair of anomalous origin of the left coronary artery from the pulmonary artery. Ann Thorac Surg 1999;67:169–72.[Abstract/Free Full Text]

  7. Swensson RE, Murillo-Olivas A, Elias W, Bender R, Daily PO, Sahn DJ. Noninvasive Doppler color flow mapping for detection of anomalous origin of the left coronary artery from the pulmonary artery and for evaluation of surgical repair. J Am Coll Cardiol 1988;11:659–61.[Abstract]





This Article
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Kozo Ishino
Shunji Sano
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Right arrow Articles by Sano, S.


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