A Single-Center Retrospective Study of Patients with ALCAPA

Yıl 2022, Cilt: 25 Sayı: 2, 200 - 207, 20.08.2022

Berra Zümrüt Tan Recep Aybala Tongut Ali Can Hatemi Nihat Cine Eylem Tunçer Hakan Ceyran

Öz

Introduction: Anomalous left coronary artery from the pulmonary artery (ALCAPA) is a rare but fatal disease. Untreated cases have a first-year mortality rate of 90%. We aimed to evaluate the early outcomes of patients who were operated on for ALCAPA in our clinic.

Patients and Methods: We retrospectively reviewed the cases of seven patients who were operated on in our clinic for ALCAPA between 2013 and 2019. Preoperative, early postoperative, and first year echocardiography results were compared.

Results: Five patients (71.4%) underwent reimplantation and two patients (28.6%) underwent Takeuchi repair. The median age was 25 months and 71.4% (n= 5) of the patients were aged ≤1 year. In preoperative echocardiograms, mean ejection fraction (EF) was 32.1 ± 4.9% and mean preoperative MR grade was 2.1 ± 0.7. Two patients with severe MR (grade 3) underwent simultaneous mitral annuloplasty. The mean EF % significantly increased (p= 0.023) and the mean MR grade significantly decreased (p= 0.039, p< 0.05) in the early postoperative period. This finding was not statistically associated with surgical technique or age. The mortality rate was 14.3% (n= 1). The patient who died had severe preoperative LV dysfunction and MR, which did not improve after surgical intervention.

Conclusion: Early improvement in LV function and MR grade after ALCAPA repair is crucial for survival regardless of age. Moderate or mild functional MR may spontaneously improve after the surgical correction of LV dysfunction. Simultaneous mitral valve intervention is needed in cases of severe MR and patients aged ≥1 year.

Anahtar Kelimeler

Left coronary artery, pulmonary artery, coronary artery reimplantation

Kaynakça

• 1. Bland EF, White PD, Garland J. Congenital anomalies of the coro- nary arteries: Report of an unusual case associated with cardiac hypertrophy. Am Heart J 1933;8:787-801. [Crossref]
• 2. Ali DK, Constantine M, 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. [Crossref]
• 3. Jin Z, Berger F, Uhlemann F, Schroder C, Hetzer R, Alexi-Meskhishvili V, et al. Improvement in left ventricular dysfunction after aortic reimplantation in 11 consecutive paediatric patients with anomalous origin of the left coronary artery from the pulmonary artery. Early results of a serial echocardiographic follow- up. Eur Heart J 1994;15:1044-9. [Crossref]
• 4. Fornwalt BK, Mahle WT, Fyfe DA. Resynchronization and strain recovery following reimplantation of anomalous coronary artery: A tissue Doppler and three-dimensional echocardiographic study. Congenit Heart Dis 2007;2:58-63. [Crossref]
• 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;1968:403-25. [Crossref]
• 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. [Crossref]
• 7. Muzaffar T, Ahmad Ganie F, Gpoal Swamy S, Wani NU. The surgical outcome of anomalous origin of the left coronary artery from the pulmonary artery. Int Cardiovasc Res J 2014;8:57-60.
• 8. Mongé MC, Eltayeb O, Costello JM, Sarwark AE, Carr MR, Backer CL. Aortic implantation of anomalous origin of the left coronary artery from the pulmonary artery: Long-term outcomes. Ann Thorac Surg 2015;100:154-60. [Crossref]
• 9. Cabrera AG, Chen DW, Pignatelli RH, Khan MS, Jeewa A, Mery CM, et al. Outcomes of anomalous left coronary artery from pulmonary artery repair: Beyond Normal Function. Ann Thorac Surg 2015;99:1342-7. [Crossref]
• 10. Fratz S, Hager A, Schreiber C, Schwaiger M, Hess J, Stern HC. Long-term myocardial scarring after operation for anomalous left coronary artery from the pulmonary artery. Ann Thorac Surg 2011;92(5):1761-5. [Crossref]
• 11. Shivalkar B, Borgers M, Daenen W, Gewillig M, Flameng W. ALCAPA syndrome: An example of chronic myocardial hypoperfusion? J Am Coll Cardiol 1994;23:(3):772-8. [Crossref]
• 12. Bergmann O, Bhardwaj RD, Bernard S, Zdunek S, Barnabé-Heider F, Zupicich J, et al. Evidence for cardiomyocyte renewal in humans. Science 2009;324:98-102. [Crossref]
• 13. Latus H, Gummel K, Rupp S, Mueller M, Jux C, Kerst G, et al. Cardiovascular magnetic resonance assessment of ventricular function and myocardial scarring before and early after repair of anomalous left coronary artery from the pulmonary artery. J Carvasc Magn Reson 2014;16:3. [Crossref]
• 14. Schmitt B, Bauer S, Kutty S, Nordmeyer S, Nasseri B, Berger F, et al. Myocardial perfusion, scarring, and function in anomalous left coronary artery from the pulmonary artery syndrome: A long-term analysis using magnetic resonance imaging. Ann Thorac Surg 2014;98(4):1425-36. [Crossref]
• 15. Secinaro A, Ntsinjana H, Tann O, Schuler PK, Muthurangu V, Hughes M, et al. Cardiovascular magnetic resonance findings in repaired anomalous left coronary artery to pulmonary artery connection (ALCAPA). J Cardiovasc Magn Reson 2001;13:27. [Crossref]
• 16. Kudumula V, Mehta C, Stumper O, Desai T, Chikermane A, Miller P, et al. Twenty-year outcome of anomalous origin of left coronary artery from pulmonary artery: Management of mitral regurgitation. Ann Thorac Surg 2014;97:938-44. [Crossref]
• 17. Zhang HL, Li SJ, Yan J, Hua ZD. Preoperative evaluation and midterm outcomes after the surgical correction of anomalous origin of the left coronary artery from the pulmonary artery in 50 infants and children. Chin Med J 2017;130:2816-22. [Crossref]
• 18. Zhang W, Hu R, Zhu Y, Zhang W, Yu X, Sun Y, et al. Surgical outcomes for anomalous left coronary artery from the pulmonary artery: Influence of late presentation. J Thorac Cardiovasc Surg 2020;159:1945-52. [Crossref]
• 19. Lange R, Vogt M, Horer J, Cleuziou J, Menzel A, Holper K, et al. Long- term results of repair of anomalous origin of the left coronary artery from the pulmonary artery. Ann Thorac Surg 2007;83:1463-71. [Crossref]
• 20. Huddleston CB, Balzer DT, Mendeloff EN. Repair of anomalous left main coronary artery arising from the pulmonary artery in infants: Long-term impact on the mitral valve. Ann Thorac Surg 2001;71:1985-8. [Crossref]
• 21. Guzeltas A, Ozturk E, Tanidir IC, Kasar T, Haydin S. Evaluation of anomalous coronary arteries from the pulmonary artery. Braz J Cardiovasc Surg 2017;32:29-37. [Crossref]
• 22. Lange R, Cleuziou J, Krane M, Ewert P, Pabst von Ohain J, Beran E, 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;53:732-9. [Crossref]
• 23. Stern H, Sauer U, Locher D, Bauer R, Meisner H, Sebening F, et al. Left ventricular function assessed with echocardiography and myocardial perfusion assessed with scintigraphy under dipyridamole stress in pediatric patients after repair for anomalous origin of the left coronary artery from the pulmonary artery. J Thorac Cardiovasc Surg 1993;106(4):723-32. [Crossref]
• 24. Alexi-Meskishvili V, Nasseri BA, Nordmeyer S, Schmitt B, Weng YG, Böttcher W, 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):868-74. [Crossref]
• 25. Jinmei Z, Yunfei L, Yue W, Yongjun Q. Anomalous origin of the left coronary artery from the pulmonary artery (ALCAPA) diagnosed in children and adolescents. Journal of Cardiothorac Surg 2020;15:90. [Crossref]
• 26. Cochrane AD, Coleman DM, Davis AM, Brizard CP, Wolfe R, Karl TR. Excellent long-term functional outcome after an operation for anomalous left coronary artery from the pulmonary artery. J Thorac Cardiovasc Surg 1999;117:332-42. [Crossref]
• 27. Isomatsu Y, Imai Y, Shin’oka T, Aoki M, Iwata Y. Surgical intervention for anomalous origin of the left coronary artery from the pulmonary artery: The Tokyo experience. J Thorac Cardiovasc Surg 2001;121:792-7. [Crossref]
• 28. Alsoufi B, Sallehuddin A, Bulbul Z, Joufan M, Khouqeer F, Canver CC, et al. Surgical strategy to establish a dual-coronary system for the management of anomalous left coronary artery origin from the pulmonary artery. Ann Thorac Surg 2008; 86:170-6. [Crossref]
• 29. Neumann A, Sarikouch S, Bobylev D, Meschenmoser L, Breymann T, Westhoff-Bleck M, et al. Long-term results after repair of anomalous origin of left coronary artery from the pulmonary artery: Takeuchi repair versus coronary transfer. Eur J Cardiothorac Surg 2017;51:308-15. [Crossref]
• 30. Azakie A, Russell JL, McCrindle BW, Van Arsdell GS, Benson LN, Coles JG, 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;75:1535-41. [Crossref]
• 31. Schwartz ML, Jonas RA, Colan SD. Anomalous origin of left coronary artery from pulmonary artery: Recovery of left ventricular function after dual coronary repair. J Am Coll Cardiol 1997;30:547-53. [Crossref]
• 32. Caspi J, Pettitt TW, Sperazza C, Mulder T, Stopa A. Reimplantation of anomalous left coronar artery from the pulmonary artery without mitral valve repair. Ann Thorac Surg 2007;84:619-23. [Crossref]