Surgical treatment of patients with anomalous left coronary artery from the pulmonary artery and intramural left coronary artery_Chinese Journal of Clinical Thoracic and Cardiovascular Surgery

Authors：

DUN Yaojun , ZHANG Changwei , LIU Rui , MA Kai , RUI Lu , ZHANG Benqing , LIN Ye ,  LI Shoujun

Department of Cardiovascular Surgery, Fuwai Hospital, National Center for Cardiovascular Diseases, Chinese Academy of Medical Sciences and Peking Union Medical College, Beijing,100037, P. R. China;

Corresponding author：

LI Shoujun, Email: drlishoujun@126.com

Keywords：

Anomalous left coronary artery from the pulmonary artery; intramural coronary artery; coronary reimplantation; coronary unroofing

DOI：

10.7507/1007-4848.202112051

Video：

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Objective To summarize the surgical experience of patients with anomalous left coronary artery from the pulmonary artery (ALCAPA) and intramural left coronary artery and analyze the early and mid-term clinical outcomes. Methods The infants with ALCAPA and intramural left coronary artery who underwent surgical treatment in Fuwai Hospital from January 2014 to September 2020 were retrospectively enrolled, and the clinical data of the patients were analyzed. Results A total of 10 patients were included. There were 8 males and 2 females, with a median age of 7.5 (3-46) months at surgery. The surgical techniques included coronary unroofing in 7 patients, coronary unroofing with coronary reimplantation in 2 patients, and coronary unroofing with ligation of left coronary artery ostium in 1 patient. Seven infants received additional procedures, including 5 mitral valve repair. Delayed chest closure was required in 2 infants, and no operative death or major complications occurred in the whole group. Postoperative chest radiograph showed that the mean cardiothoracic ratio was lower than that before surgery (0.62±0.05 vs. 0.67±0.06, P=0.006). Postoperative echocardiography indicated that the mean left ventricular ejection fraction was increased than that before surgery, but there was no statistical difference (38.7%±15.9% vs. 30.0%±16.1%, P=0.066). The follow-up was available for all 10 survivors, with an average follow-up time of 13-92 (46.6±25.0) months. During the follow-up period, the patients had no obvious symptoms, death, coronary complications or other major complications. The chest radiograph at last follow-up showed that the mean cardiothoracic ratio was further decreased (0.60±0.07 vs. 0.62±0.05, P=0.024). The echocardiography at last follow-up showed that the mean left ventricular ejection fraction was further improved (60.1%±9.3% vs. 38.7%±15.9%, P=0.002). Conclusion ALCAPA with intramural left coronary artery is a rare malformation. It can be treated with different surgical techniques with satisfactory early and mid-term outcomes.

Citation： DUN Yaojun, ZHANG Changwei, LIU Rui, MA Kai, RUI Lu, ZHANG Benqing, LIN Ye, LI Shoujun. Surgical treatment of patients with anomalous left coronary artery from the pulmonary artery and intramural left coronary artery. Chinese Journal of Clinical Thoracic and Cardiovascular Surgery, 2023, 30(11): 1571-1579. doi: 10.7507/1007-4848.202112051 Copy

1.	Keith JD. The anomalous origin of the left coronary artery from the pulmonary artery. Br Heart J, 1959, 21(2): 149-161.
2.	Ma K, Wang L, Hua Z, et al. Outcomes of coronary transfer for anomalous origin of the left coronary artery from the pulmonary artery. Eur J Cardiothorac Surg, 2015, 47(4): 659-664.
3.	Karimi M, Kirshbom PM. Anomalous origins of coronary arteries from the pulmonary artery: A comprehensive review of literature and surgical options. World J Pediatr Congenit Heart Surg, 2015, 6(4): 526-540.
4.	Brothers JA, Frommelt MA, Jaquiss RDB, et al. Expert consensus guidelines: Anomalous aortic origin of a coronary artery. J Thorac Cardiovasc Surg, 2017, 153(6): 1440-1457.
5.	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.
6.	Day K, Avery R, Oliva I, et al. Coronary CTA appearance of anomalous left coronary artery arising from the pulmonary artery with intramural aortic route. Int J Cardiovasc Imaging, 2014, 30(2): 241-243.
7.	Barbero-Marcial M, Tanamati C, Atik E, et al. Anomalous origin of the left coronary artery from the pulmonary artery with intramural aortic route: Diagnosis and surgical treatment. J Thorac Cardiovasc Surg, 1999, 117(4): 823-825.
8.	Turley K, Szarnicki RJ, Flachsbart KD, et al. Aortic implantation is possible in all cases of anomalous origin of the left coronary artery from the pulmonary artery. Ann Thorac Surg, 1995, 60: 84-89.
9.	Sun H, Dun Y, Yan J, et al. Clinical outcome of patients with transposition of the great arteries and intramural coronary artery. Pediatr Cardiol, 2021, 42(2): 417-424.
10.	Fricke TA, Bulstra AE, Naimo PS, et al. Excellent long-term outcomes of the arterial switch operation in patients with intramural coronary arteries. Ann Thorac Surg, 2016, 101(2): 725-729.
11.	Adachi I, Kagisaki K, Yagihara T, et al. Unroofing aortic intramural left coronary artery arising from right pulmonary artery. Ann Thorac Surg, 2008, 85(2): 675-677.
12.	Agrawal V, Vaidhya N, Patel M, et al. Intramural aortic course should always be considered for anomalous origin of the left coronary artery from the right pulmonary artery. World J Pediatr Congenit Heart Surg, 2019, 10: 508-512.
13.	Atik E, Barbero-Marcial M, Tanamati C, et al. Anomalous origin of the left coronary artery from the right pulmonary artery with intramural aortic trajectory. Clinicosurgical diagnostic implications. Arq Bras Cardiol, 1999, 73: 181-190.
14.	Fu HY, Chou HW, Chen YS, et al. Anomalous origin of the left coronary artery from the right pulmonary artery with an intramural aortic course. Asian J Surg, 2020, 43: 376-377.
15.	Goldberg SP, Mitchell MB, Campbell DN, et al. Anomalous left coronary artery from the pulmonary artery with an intramural course within the aortic wall: Report of 3 surgical cases. J Thorac Cardiovasc Surg, 2008, 135: 696-698.
16.	Kumar TK, Sinha P, Donofrio MT, et al. Anomalous left coronary artery from the right pulmonary artery with aortic fusion. J Thorac Cardiovasc Surg, 2012, 143: 505-507.
17.	Marshall CD, Weigand J, Sambatakos P, et al. Repair of anomalous left coronary artery from the right pulmonary artery: A series of nine cases. World J Pediatr Congenit Heart Surg, 2015, 6: 38238-6.
18.	Zhang H, Cheng P, Jin G, et al. Surgical strategies for anomalous origin of the left coronary artery from the right pulmonary artery with an intramural aortic course: A report of 10 cases. J Thorac Cardiovasc Surg, 2017, 153: 648-653.
19.	Lange R, Cleuziou J, Krane M, et al. Long-term outcome after anomalous left coronary artery from the pulmonary artery repair: A 40-year single-centre experience. Eur J Cardiothorac Surg, 2018, 53: 732-739.
20.	Monge MC, Eltayeb O, Costello JM, et al. Aortic implantation of anomalous origin of the left coronary artery from the pulmonary artery: Long-term outcomes. Ann Thorac Surg, 2015, 100: 154-160.
21.	Weixler VHM, Zurakowski D, Baird CW, et al. Do patients with anomalous origin of the left coronary artery benefit from an early repair of the mitral valve? Eur J Cardiothorac Surg, 2020, 57: 72-77.
22.	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: 938-944.
23.	Naimo PS, Fricke TA, d'Udekem Y, et al. Surgical intervention for anomalous origin of left coronary artery from the pulmonary artery in children: A long-term follow-up. Ann Thorac Surg, 2016, 101: 1842-1848.
24.	Cabrera AG, Chen DW, Pignatelli RH, et al. Outcomes of anomalous left coronary artery from pulmonary artery repair: Beyond normal function. Ann Thorac Surg, 2015, 99: 1342-1347.
25.	张昌伟, 李守军, 张浩, 等. 左冠状动脉起源于肺动脉小儿患者的外科治疗. 中国胸心血管外科临床杂志, 2016, 23(1): 21-24.

1. Keith JD. The anomalous origin of the left coronary artery from the pulmonary artery. Br Heart J, 1959, 21(2): 149-161.
2. Ma K, Wang L, Hua Z, et al. Outcomes of coronary transfer for anomalous origin of the left coronary artery from the pulmonary artery. Eur J Cardiothorac Surg, 2015, 47(4): 659-664.
3. Karimi M, Kirshbom PM. Anomalous origins of coronary arteries from the pulmonary artery: A comprehensive review of literature and surgical options. World J Pediatr Congenit Heart Surg, 2015, 6(4): 526-540.
4. Brothers JA, Frommelt MA, Jaquiss RDB, et al. Expert consensus guidelines: Anomalous aortic origin of a coronary artery. J Thorac Cardiovasc Surg, 2017, 153(6): 1440-1457.
5. 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.
6. Day K, Avery R, Oliva I, et al. Coronary CTA appearance of anomalous left coronary artery arising from the pulmonary artery with intramural aortic route. Int J Cardiovasc Imaging, 2014, 30(2): 241-243.
7. Barbero-Marcial M, Tanamati C, Atik E, et al. Anomalous origin of the left coronary artery from the pulmonary artery with intramural aortic route: Diagnosis and surgical treatment. J Thorac Cardiovasc Surg, 1999, 117(4): 823-825.
8. Turley K, Szarnicki RJ, Flachsbart KD, et al. Aortic implantation is possible in all cases of anomalous origin of the left coronary artery from the pulmonary artery. Ann Thorac Surg, 1995, 60: 84-89.
9. Sun H, Dun Y, Yan J, et al. Clinical outcome of patients with transposition of the great arteries and intramural coronary artery. Pediatr Cardiol, 2021, 42(2): 417-424.
10. Fricke TA, Bulstra AE, Naimo PS, et al. Excellent long-term outcomes of the arterial switch operation in patients with intramural coronary arteries. Ann Thorac Surg, 2016, 101(2): 725-729.
11. Adachi I, Kagisaki K, Yagihara T, et al. Unroofing aortic intramural left coronary artery arising from right pulmonary artery. Ann Thorac Surg, 2008, 85(2): 675-677.
12. Agrawal V, Vaidhya N, Patel M, et al. Intramural aortic course should always be considered for anomalous origin of the left coronary artery from the right pulmonary artery. World J Pediatr Congenit Heart Surg, 2019, 10: 508-512.
13. Atik E, Barbero-Marcial M, Tanamati C, et al. Anomalous origin of the left coronary artery from the right pulmonary artery with intramural aortic trajectory. Clinicosurgical diagnostic implications. Arq Bras Cardiol, 1999, 73: 181-190.
14. Fu HY, Chou HW, Chen YS, et al. Anomalous origin of the left coronary artery from the right pulmonary artery with an intramural aortic course. Asian J Surg, 2020, 43: 376-377.
15. Goldberg SP, Mitchell MB, Campbell DN, et al. Anomalous left coronary artery from the pulmonary artery with an intramural course within the aortic wall: Report of 3 surgical cases. J Thorac Cardiovasc Surg, 2008, 135: 696-698.
16. Kumar TK, Sinha P, Donofrio MT, et al. Anomalous left coronary artery from the right pulmonary artery with aortic fusion. J Thorac Cardiovasc Surg, 2012, 143: 505-507.
17. Marshall CD, Weigand J, Sambatakos P, et al. Repair of anomalous left coronary artery from the right pulmonary artery: A series of nine cases. World J Pediatr Congenit Heart Surg, 2015, 6: 38238-6.
18. Zhang H, Cheng P, Jin G, et al. Surgical strategies for anomalous origin of the left coronary artery from the right pulmonary artery with an intramural aortic course: A report of 10 cases. J Thorac Cardiovasc Surg, 2017, 153: 648-653.
19. Lange R, Cleuziou J, Krane M, et al. Long-term outcome after anomalous left coronary artery from the pulmonary artery repair: A 40-year single-centre experience. Eur J Cardiothorac Surg, 2018, 53: 732-739.
20. Monge MC, Eltayeb O, Costello JM, et al. Aortic implantation of anomalous origin of the left coronary artery from the pulmonary artery: Long-term outcomes. Ann Thorac Surg, 2015, 100: 154-160.
21. Weixler VHM, Zurakowski D, Baird CW, et al. Do patients with anomalous origin of the left coronary artery benefit from an early repair of the mitral valve? Eur J Cardiothorac Surg, 2020, 57: 72-77.
22. 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: 938-944.
23. Naimo PS, Fricke TA, d'Udekem Y, et al. Surgical intervention for anomalous origin of left coronary artery from the pulmonary artery in children: A long-term follow-up. Ann Thorac Surg, 2016, 101: 1842-1848.
24. Cabrera AG, Chen DW, Pignatelli RH, et al. Outcomes of anomalous left coronary artery from pulmonary artery repair: Beyond normal function. Ann Thorac Surg, 2015, 99: 1342-1347.
25. 张昌伟, 李守军, 张浩, 等. 左冠状动脉起源于肺动脉小儿患者的外科治疗. 中国胸心血管外科临床杂志, 2016, 23(1): 21-24.

Chinese Journal of Clinical Thoracic and Cardiovascular Surgery

Surgical treatment of patients with anomalous left coronary artery from the pulmonary artery and intramural left coronary artery

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