Hostname: page-component-586b7cd67f-vdxz6 Total loading time: 0 Render date: 2024-11-22T15:31:43.038Z Has data issue: false hasContentIssue false
  • English
  • Français

Effective myocardial perfusion and concomitant haemodynamic status determine the clinical diversity of anomalous left coronary artery from the pulmonary artery

Published online by Cambridge University Press:  22 January 2020

Amr A. Matoq Open the ORCID record for Amr A. Matoq [Opens in a new window]  and
Takeshi Tsuda Open the ORCID record for Takeshi Tsuda [Opens in a new window]
Amr A. Matoq
Affiliation:
Nemours Cardiac Center, Alfred I. duPont Hospital for Children, Wilmington, DE 19803, USA
Takeshi Tsuda*
Affiliation:
Nemours Cardiac Center, Alfred I. duPont Hospital for Children, Wilmington, DE 19803, USA Department of Pediatrics, Sidney Kimmel Medical College at Thomas Jefferson University, Philadelphia, PA 19107, USA
*
Author for correspondence: T. Tsuda, MD, FAAP, FACC, Nemours Cardiac Center, Nemours/Alfred I. duPont Hospital for Children, 1600 Rockland Rd, Wilmington, DE 19803, USA. Tel: (302) 651-6677; Fax: (302) 651-6601; E-mail: [email protected]
Get access Rights & Permissions [Opens in a new window]

Abstract

Background:

Anomalous left coronary artery from the pulmonary artery is a rare congenital heart disease (CHD) with diverse clinical presentations despite the same anatomy. Factors determining this heterogeneous presentation are not well understood.

Method and Results:

We retrospectively investigated 14 patients (12 females) who underwent surgical repair of anomalous left coronary artery from the pulmonary artery. These patients were divided into three groups based upon the severity of initial presentation: (1) severe, life-threatening condition (n = 5), (2) mild-to-moderate distress (n = 6), and (3) asymptomatic (n = 3). All patients presented with left ventricular dilation and retrograde flow in left coronary artery by echocardiogram. Eight patients in (1) and (2) presented with severe left ventricular dysfunction. All but one showed abnormal ECG consistent with myocardial ischemia or infarction. Asymptomatic patients had preserved left ventricular systolic function despite ischemic findings on ECG. In 13 patients after surgical repair, all but one normalised left ventricular geometry and systolic function, suggesting nearly full myocardial recovery upon improvement of myocardial perfusion; 8 patients had residual echogenic papillary muscle with variable degree of mitral regurgitation.

Conclusions

Evidence of myocardial ischemic injury was present in all patients with anomalous left coronary artery from the pulmonary artery regardless of their initial presentation. Retrograde flow in left coronary artery, implying collateral vessel development from right coronary artery to left coronary artery, was noted in all patients, yet only few patients had preserved systolic function at the time of diagnosis. The balance between effective myocardial perfusion and a deleterious fistulous flow provided by these collaterals and the simultaneous haemodynamic status are what determine the clinical diversity of anomalous left coronary artery from the pulmonary artery.

Keywords

Anomalous left coronary artery from pulmonary arterymyocardial infarctionischemiacollateralcoronary stealmyocardial regeneration
Type
Original Article
Information
Cardiology in the Young , Volume 30 , Issue 3 , March 2020 , pp. 362 - 368
Copyright
© Cambridge University Press 2020

Access options

Get access to the full version of this content by using one of the access options below. (Log in options will check for institutional or personal access. Content may require purchase if you do not have access.)

References

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: 946–955.10.1016/S0003-4975(02)03633-0CrossRefGoogle ScholarPubMed
Keith, JD.The anomalous origin of the left coronary artery from the pulmonary artery. Br Heart J 1959; 21: 149–161.CrossRefGoogle ScholarPubMed
Askenazi, J, Nadas, AS.Anomalous left coronary artery originating from the pulmonary artery. Report on 15 cases. Circulation 1975; 51: 976–987.CrossRefGoogle ScholarPubMed
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: 403–425.CrossRefGoogle ScholarPubMed
Brooks, HS.Two cases of an abnormal coronary artery of the heart arising from the pulmonary artery: with some remarks upon the effect of this anomaly in producing cirsoid dilatation of the vessels. J Anat Physiol 1885; 20: 26–29.Google ScholarPubMed
Yau, JM, Singh, R, Halpern, EJ, Fischman, D.Anomalous origin of the left coronary artery from the pulmonary artery in adults: a comprehensive review of 151 adult cases and a new diagnosis in a 53-year-old woman. Clin Cardiol 2011; 34: 204–210.CrossRefGoogle Scholar
Laux, D, Bertail, C, Bajolle, F, Houyel, L, Boudjemline, Y, Bonnet, D.Anomalous left coronary artery connected to the pulmonary artery associated with other cardiac defects: a difficult joint diagnosis. Pediatr Cardiol 2014; 35: 1198–1205.10.1007/s00246-014-0916-4CrossRefGoogle ScholarPubMed
Bland, EF, White, PD, Garland, J.Congenital anomalies of the coronary arteries: report of an unusual case associated with cardiac hypertrophy. Am Heart J 1933; 8: 787–801.10.1016/S0002-8703(33)90140-4CrossRefGoogle Scholar
Levitas, A, Krymko, H, Ioffe, V, Zalzstein, E, Broides, A.Anomalous left coronary artery from the pulmonary artery in infants and toddlers misdiagnosed as myocarditis. Pediatr Emerg Care 2016; 32: 232–234.CrossRefGoogle ScholarPubMed
Rodriguez-Gonzalez, M, Tirado, AM, Hosseinpour, R, de Soto, JS.Anomalous origin of the left coronary artery from the pulmonary artery: diagnoses and surgical results in 12 pediatric patients. Tex Heart Inst J 2015; 42: 350–356.CrossRefGoogle ScholarPubMed
Zheng, JY, Han, L, Ding, WH, et al.Clinical features and long-term prognosis of patients with anomalous origin of the left coronary artery from the pulmonary artery. Chin Med J 2010; 123: 2888–2894.Google ScholarPubMed
Berre, LL, Baruteau, AE, Fraisse, A, et al.Anomalous origin of the left coronary artery from the pulmonary artery presenting in adulthood: a French Nationwide Retrospective Study. Semin Thorac Cardiovasc Surg 2017; 29: 486–490.CrossRefGoogle Scholar
Pachon, R, Bravo, C, Niemiera, M.Sudden cardiac death as a presentation of anomalous origin of the left coronary artery from pulmonary artery in a young adult. Eur Heart J Acute Cardiovasc Care 2015; 4: 589–590.10.1177/2048872614562969CrossRefGoogle Scholar
Boutsikou, M, Shore, D, Li, W, et al.Anomalous left coronary artery from the pulmonary artery (ALCAPA) diagnosed in adulthood: varied clinical presentation, therapeutic approach and outcome. Int J Cardiol 2018; 261: 49–53.10.1016/j.ijcard.2018.02.082CrossRefGoogle ScholarPubMed
Brotherton, H, Philip, RK.Anomalous left coronary artery from pulmonary artery (ALCAPA) in infants: a 5-year review in a defined birth cohort. Eur J Pediatr 2008; 167: 43–46.10.1007/s00431-007-0423-1CrossRefGoogle Scholar
Dilawar, M, Ahmad, Z.Anomalous left coronary artery from pulmonary artery: case series and brief review. Open J Pediatr 2012; 02: 77–81.CrossRefGoogle Scholar
Michielon, G, Di Carlo, D, Brancaccio, G, et al.Anomalous coronary artery origin from the pulmonary artery: correlation between surgical timing and left ventricular function recovery. Ann Thorac Surg 2003; 76: 581–588; discussion 588.10.1016/S0003-4975(03)00344-8CrossRefGoogle ScholarPubMed
Rein, AJ, Colan, SD, Parness, IA, Sanders, SP.Regional and global left ventricular function in infants with anomalous origin of the left coronary artery from the pulmonary trunk: preoperative and postoperative assessment. Circulation 1987; 75: 115–123.10.1161/01.CIR.75.1.115CrossRefGoogle ScholarPubMed
Schaper, W.Collateral circulation: past and present. Basic Res Cardiol 2009; 104: 5–21.CrossRefGoogle ScholarPubMed
Zimarino, M, D’Andreamatteo, M, Waksman, R, Epstein, SE, De Caterina, R.The dynamics of the coronary collateral circulation. Nat Rev Cardiol 2014; 11: 191–197.CrossRefGoogle ScholarPubMed
Seiler, C, Stoller, M, Pitt, B, Meier, P.The human coronary collateral circulation: development and clinical importance. Eur Heart J 2013; 34: 2674–2682.10.1093/eurheartj/eht195CrossRefGoogle ScholarPubMed
Schaper, W.Collateral anatomy and blood flow: its potential role in sudden coronary death. Ann N Y Acad Sci 1982; 382: 69–75.CrossRefGoogle ScholarPubMed
Risau, W.Mechanisms of angiogenesis. Nature 1997; 386: 671–674.10.1038/386671a0CrossRefGoogle ScholarPubMed
Aurora, AB, Porrello, ER, Tan, W, et al.Macrophages are required for neonatal heart regeneration. J Clin Invest 2014; 124: 1382–1392.CrossRefGoogle ScholarPubMed
Lavine, KJ, Epelman, S, Uchida, K, et al., Distinct macrophage lineages contribute to disparate patterns of cardiac recovery and remodeling in the neonatal and adult heart. Proc Natl Acad Sci U S A 2014; 111: 16029–16034.10.1073/pnas.1406508111CrossRefGoogle ScholarPubMed
Fudulu, DP, Tulloh, RM, Wolf, AR, Parry, AJ, Stoica, SC.Anomalous left coronary from the pulmonary artery presenting as ventricular fibrillation after persistent ductus arteriosus ligation. Ann Thorac Surg 2015; 100: e9–e10.CrossRefGoogle ScholarPubMed
Bafani, E, Shukla, AC, DiNardo, JA.Unrecognized anomalous origin of the left coronary artery from the pulmonary artery as a cause of ventricular fibrillation after patent ductus arteriosus ligation in an infant. Anesth Analg 2007; 104: 81–83.CrossRefGoogle ScholarPubMed
Holst, LM, Helvind, M, Andersen, HO.Diagnosis and prognosis of anomalous origin of the left coronary artery from the pulmonary artery. Danish Med J 2015; 62: pii: A5125.Google ScholarPubMed
Galal, MO, Amin, M, Hussein, A, Kouatli, A, Al-Ata, J, Jamjoom, A.Left ventricular dysfunction after closure of large patent ductus arteriosus. Asian Cardiovasc Thorac Ann 2005; 13: 24–29.10.1177/021849230501300106CrossRefGoogle ScholarPubMed
Tilahun, B, Tefera, E.Transient left ventricular systolic dysfunction following surgical closure of large patent ductus arteriosus among children and adolescents operated at the cardiac centre, Ethiopia. J Cardiothorac Surg 2013; 8: 139.CrossRefGoogle ScholarPubMed
Weber, KT, Janicki, JS.Myocardial oxygen consumption: the role of wall force and shortening. Am J Physiol 1977; 233: H421–430.Google ScholarPubMed
Gao, Y, Zhang, J, Huang, GY, Liang, XC, Jia, B, Ma, XJ.Surgical outcomes of anomalous origin of the left coronary artery from the pulmonary artery in children: an echocardiography follow-up. Chinese Med J 2017; 130: 2333–2338.Google ScholarPubMed
Kakou Guikahue, M, Sidi, D, Kachaner, J, et al.Anomalous left coronary artery arising from the pulmonary artery in infancy: is early operation better? Br Heart J 1988; 60: 522–526.10.1136/hrt.60.6.522CrossRefGoogle ScholarPubMed
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.CrossRefGoogle ScholarPubMed
Weigand, J, Marshall, CD, Bacha, EA, Chen, JM, Richmond, ME.Repair of anomalous left coronary artery from the pulmonary artery in the modern era: preoperative predictors of immediate postoperative outcomes and long term cardiac follow-up. Pediatr cardiol 2015; 36: 489–497.10.1007/s00246-014-1038-8CrossRefGoogle ScholarPubMed
van Royen, N, Piek, JJ, Buschmann, I, Hoefer, I, Voskuil, M, Schaper, W.Stimulation of arteriogenesis; a new concept for the treatment of arterial occlusive disease. Cardiovasc Res 2001; 49: 543–553.10.1016/S0008-6363(00)00206-6CrossRefGoogle ScholarPubMed
Stoller, M, Seiler, C.Pathophysiology of coronary collaterals. Curr Cardiol Rev 2014; 10: 38–56.CrossRefGoogle ScholarPubMed