Surgical Treatment of Anomalous Origin of the Left Coronary Artery from the Pulmonary Artery_Chinese Journal of Clinical Thoracic and Cardiovascular Surgery

Authors：

LIWen-xiu ,  GENGBin , LIXiao-feng , WUJiang , LIUYing-long , ZHANGGui-zhen

Pediatric Cardiovascular Center, Beijing Anzhen Hospital, Capital Medical University, Beijing 100029, P. R. China;

Corresponding?author：

GENGBin, Email: geng_bin1@163.com

Keywords：

Anomalous origin of the left coronary artery from the pulmonary artery; Direct coronary reimplantation of the anomalous artery; Intrapulmonary tunnel procedure; Low cardiac output syndrome

DOI：

10.7507/1007-4848.20140176

Video：

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Objective To explore surgical methods and risk factors of anomalous origin of the left coronary artery from the pulmonary artery (ALCAPA). Methods Clinical data of 28 ALCAPA patients who underwent surgical repair from October 1993 to September 2013 in Beijing Anzhen Hospital were retrospectively reviewed. There were 8 male and 20 female patients with their age of 0.6-l6.8 (4.3±0.7)years including 10 patients less than 1 years old. Surgical procedures included simple ligation of left coronary artery, intrapulmonary tunnel procedure (Takeuchi)and direct coronary reimplantation of the anomalous artery. Postoperative death, complication and cardiac function were observed. Results Among the 28 patients, 1 patient received simple ligation of left coronary artery, and 7 patients received intrapulmonary tunnel procedure (Takeuchi), among whom 2 patients died postoperatively. Twenty patients received direct implantation of the anomalous artery into the ascending aorta, and 3 patients died postoperatively. Five patients who died postoperatively were 10.20±3.27 months old, including 3 patients with moderate mitral regurgitation (MR)and 2 patients with mild MR preoperatively. Preoperative heart function of the patients who died postoperatively was significantly reduced. Preoperative left ventricular ejection fraction of the patients who died postoperatively was significantly lower than that of the patients who survived (36.6%±8.5% vs. 60.9%±10.7%, P=0.000). Low cardiac output syndrome was the reason for all postoperative death. All survival patients were followed up from 1 month to 18 years. One patient who underwent intra-pulmonary tunnel procedure (Takeuchi)received pulmonary artery balloon dilatation for pulmonary supravalvular stenosis 15 years after discharge. None of the other patients received a secondary operation. During follow-up, left ventricular function was improved. Growth and development of all the patients was normal. MR did not significantly aggravate in all the patients. Conclusion Patients with younger age and worse left ventricular function have greater surgical risks of ALCAPA.

Citation： LIWen-xiu, GENGBin, LIXiao-feng, WUJiang, LIUYing-long, ZHANGGui-zhen. Surgical Treatment of Anomalous Origin of the Left Coronary Artery from the Pulmonary Artery. Chinese Journal of Clinical Thoracic and Cardiovascular Surgery, 2014, 21(5): 619-623. doi: 10.7507/1007-4848.20140176 Copy

1.	Dodge-Khatamia D, 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(3):946-955.
2.	Pfannschmidt J, Ruskowski H, de Vivie ER. Bland-White-Garland syndrome. clinical aspects, diagnosis, therapy. Klin Padiatr, 1992, 204(5):328-334.
3.	梁家立, 姜冠華, 楊哲, 等.成人左冠狀動脈起源于肺動脈二例.中國胸心血管外科臨床雜志, 2011, 18(4):380-381.
4.	郭雷, 陳忠堂, 許運賓.左冠狀動脈異常起源于肺動脈一例.中國胸心血管外科臨床雜志, 2003, 10(1):28.
5.	Ilic S, Hercog D, Vucicevic M, et al. Anomalous origin of the left coronary artery from the pulmonary artery, scimitar syndrome, and aortic coarctation. Ann Thorac Surg, 2014, 97(2):707-710.
6.	朱曉東, 張寶仁, 主編.心臟外科學.北京:人民衛生出版社, 2007.533-542.
7.	Pe?a E, Nguye ET, Merchant N, et al. ALCAPA syndrome:not just a pediatric disease. Radiographics, 2009, 29(2):553-565.
8.	Wesselhoeft H, Fawcett JS, Johnson AL. Anomalous origin of the left coronary artery from the pulmonary trunk. Its clinical spectrum, pathology, and pathophysiology, based on a review of 140 cases with seven further cases. Circulation, 1968, 38(2):403-425.
9.	Ojala T, Salminen J, Happonen JM, et al. Excellent functional result in children after correction of anomalous origin of left coronary artery from the pulmonary artery-a population-based complete follow-up study. Interact Cardiovasc Thorac Surg, 2010, 10(1):70-75.
10.	Mainwaring RD, Reddy VM, Reinhartz O, et al. Surgical repair of anomalous aortic origin of a coronary artery. Eur J Cardiothorac Surg, 2014, 46(1):20-26.
11.	Jujjavarapu KR, Salhiyyah K, Witzke H, et al. Off-pump total arterial revascularization for anomalous origin of the left coronary artery from the pulmonary artery. Ann Thorac Surg, 2013, 96(6):2234-2236.
12.	Martín Raymondi D, Mejía Viana S, Berenguel Senén A, et al.Supravalvular pulmonary stenosis as a late complication of surgery for anomalous coronary origin. J Thorac Cardiovasc Surg, 2002, 123(6):1218-1220.
13.	Ginde S, Earing MG, Bartz PJ, et al. Late complications after Takeuchi repair of anomalous left coronary artery from the pulmonary artery:case series and review of literature. Pediatr Cardiol, 2012, 33(7):1115-1123.
14.	Kazmierczak PA, Ostrowska K, Dryzek P, et al. Repair of anomalous origin of the left coronary artery from the pulmonary artery in infants. Interact Cardiovasc Thorac Surg, 2013, 16(6):797-801.
15.	Alexi-Meskishvili V, Nasseri BA, Nordmeyer S, et al. Repair of anomalous origin of the left coronary artery from the pulmonary artery in infants and children. J Thorac Cardiovasc Surg, 2011, 142(4):868-874.
16.	Kottayil BP, Jayakumar K, Dharan BS, et al. Anomalous origin of left coronary artery from pulmonary artery in older children and adults:direct aortic implantation. Ann Thorac Surg, 2011, 91(2):549-553.
17.	Szmigielska A, Roszkowska-Blaim M, Go??bek-Dylewska M, et al. Bland-White-Garland syndrome-a rare and serious cause of failure to thrive. Am J Case Rep, 2013, 14(14):370-372.
18.	Azakie A, Russell JL, Mccrindle BW, et al. Anatomic repair of anomalous left coronary artery from the pulmonary artery by aortic reimplantation:early survival, patterns of ventricular recovery and late outcome. Ann Thorac Surg, 2003, 75(5):1535-1541.
19.	Brown JW, Ruzmetov M, Parent JJ, et al. Does the degree of preo-perative mitral regurgitation predict survival or the need for mitral valve repair or replacement in patients with anomalous origin of the left coronary artery from the pulmonary artery? J Thorac Cardiovasc Surg, 2008, 136(3):743-748.
20.	Kudumula V, Mehta C, Stumper O, et al. Twenty-year outcome of anomalous origin of left coronary artery from pulmonary artery:management of mitral regurgitation. Ann Thorac Surg, 2014, 97(3):938-944.
21.	Ben Ali W, Metton O, Roubertie F, et al. Anomalous origin of the left coronary artery from the pulmonary artery:late results with special attention to the mitral valve. Eur J Cardiothorac Surg, 2009, 36(2):244-249.
22.	Cochrane AD, Coleman DM, Davis AM, et al. Excellent long-term functional outcome after an operation for anomalous left coronary artery from the pulmonary artery. J Thorac Cardiovasc Surg, 1999, 117(2):332-342.
23.	Belli E, Roussin R, Ly M, et al. Anomalous origin of the left coronary artery from the pulmonary artery associated with severe left ventricular dysfunction:results in normothermia. Ann Thorac Surg, 2010, 90(3):856-860.
24.	Ramírez S, Curi-Curi PJ, Calderón-Colmenero J, et al. Outcomes of coronary reimplantation for correction of anomalous origin of left coronary artery from pulmonary artery. Rev Esp Cardiol, 2011, 64(8):681-687.
25.	Imamura M, Dossey AM, Jaquiss RD. Reoperation and mechanical circulatory support after repair of anomalous origin of the left coronary artery from the pulmonary artery:a twenty-year experience. Ann Thorac Surg, 2011, 92(1):167-173.

1. Dodge-Khatamia D, 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(3):946-955.
2. Pfannschmidt J, Ruskowski H, de Vivie ER. Bland-White-Garland syndrome. clinical aspects, diagnosis, therapy. Klin Padiatr, 1992, 204(5):328-334.
3. 梁家立, 姜冠華, 楊哲, 等.成人左冠狀動脈起源于肺動脈二例.中國胸心血管外科臨床雜志, 2011, 18(4):380-381.
4. 郭雷, 陳忠堂, 許運賓.左冠狀動脈異常起源于肺動脈一例.中國胸心血管外科臨床雜志, 2003, 10(1):28.
5. Ilic S, Hercog D, Vucicevic M, et al. Anomalous origin of the left coronary artery from the pulmonary artery, scimitar syndrome, and aortic coarctation. Ann Thorac Surg, 2014, 97(2):707-710.
6. 朱曉東, 張寶仁, 主編.心臟外科學.北京:人民衛生出版社, 2007.533-542.
7. Pe?a E, Nguye ET, Merchant N, et al. ALCAPA syndrome:not just a pediatric disease. Radiographics, 2009, 29(2):553-565.
8. Wesselhoeft H, Fawcett JS, Johnson AL. Anomalous origin of the left coronary artery from the pulmonary trunk. Its clinical spectrum, pathology, and pathophysiology, based on a review of 140 cases with seven further cases. Circulation, 1968, 38(2):403-425.
9. Ojala T, Salminen J, Happonen JM, et al. Excellent functional result in children after correction of anomalous origin of left coronary artery from the pulmonary artery-a population-based complete follow-up study. Interact Cardiovasc Thorac Surg, 2010, 10(1):70-75.
10. Mainwaring RD, Reddy VM, Reinhartz O, et al. Surgical repair of anomalous aortic origin of a coronary artery. Eur J Cardiothorac Surg, 2014, 46(1):20-26.
11. Jujjavarapu KR, Salhiyyah K, Witzke H, et al. Off-pump total arterial revascularization for anomalous origin of the left coronary artery from the pulmonary artery. Ann Thorac Surg, 2013, 96(6):2234-2236.
12. Martín Raymondi D, Mejía Viana S, Berenguel Senén A, et al.Supravalvular pulmonary stenosis as a late complication of surgery for anomalous coronary origin. J Thorac Cardiovasc Surg, 2002, 123(6):1218-1220.
13. Ginde S, Earing MG, Bartz PJ, et al. Late complications after Takeuchi repair of anomalous left coronary artery from the pulmonary artery:case series and review of literature. Pediatr Cardiol, 2012, 33(7):1115-1123.
14. Kazmierczak PA, Ostrowska K, Dryzek P, et al. Repair of anomalous origin of the left coronary artery from the pulmonary artery in infants. Interact Cardiovasc Thorac Surg, 2013, 16(6):797-801.
15. Alexi-Meskishvili V, Nasseri BA, Nordmeyer S, et al. Repair of anomalous origin of the left coronary artery from the pulmonary artery in infants and children. J Thorac Cardiovasc Surg, 2011, 142(4):868-874.
16. Kottayil BP, Jayakumar K, Dharan BS, et al. Anomalous origin of left coronary artery from pulmonary artery in older children and adults:direct aortic implantation. Ann Thorac Surg, 2011, 91(2):549-553.
17. Szmigielska A, Roszkowska-Blaim M, Go??bek-Dylewska M, et al. Bland-White-Garland syndrome-a rare and serious cause of failure to thrive. Am J Case Rep, 2013, 14(14):370-372.
18. Azakie A, Russell JL, Mccrindle BW, et al. Anatomic repair of anomalous left coronary artery from the pulmonary artery by aortic reimplantation:early survival, patterns of ventricular recovery and late outcome. Ann Thorac Surg, 2003, 75(5):1535-1541.
19. Brown JW, Ruzmetov M, Parent JJ, et al. Does the degree of preo-perative mitral regurgitation predict survival or the need for mitral valve repair or replacement in patients with anomalous origin of the left coronary artery from the pulmonary artery? J Thorac Cardiovasc Surg, 2008, 136(3):743-748.
20. Kudumula V, Mehta C, Stumper O, et al. Twenty-year outcome of anomalous origin of left coronary artery from pulmonary artery:management of mitral regurgitation. Ann Thorac Surg, 2014, 97(3):938-944.
21. Ben Ali W, Metton O, Roubertie F, et al. Anomalous origin of the left coronary artery from the pulmonary artery:late results with special attention to the mitral valve. Eur J Cardiothorac Surg, 2009, 36(2):244-249.
22. Cochrane AD, Coleman DM, Davis AM, et al. Excellent long-term functional outcome after an operation for anomalous left coronary artery from the pulmonary artery. J Thorac Cardiovasc Surg, 1999, 117(2):332-342.
23. Belli E, Roussin R, Ly M, et al. Anomalous origin of the left coronary artery from the pulmonary artery associated with severe left ventricular dysfunction:results in normothermia. Ann Thorac Surg, 2010, 90(3):856-860.
24. Ramírez S, Curi-Curi PJ, Calderón-Colmenero J, et al. Outcomes of coronary reimplantation for correction of anomalous origin of left coronary artery from pulmonary artery. Rev Esp Cardiol, 2011, 64(8):681-687.
25. Imamura M, Dossey AM, Jaquiss RD. Reoperation and mechanical circulatory support after repair of anomalous origin of the left coronary artery from the pulmonary artery:a twenty-year experience. Ann Thorac Surg, 2011, 92(1):167-173.

Chinese Journal of Clinical Thoracic and Cardiovascular Surgery

Surgical Treatment of Anomalous Origin of the Left Coronary Artery from the Pulmonary Artery

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