REVIEW PAPER
Anomalous left coronary artery from the pulmonary artery - what we know about the disease 90 years after its discovery by Bland, White, and Garland
1
Department of Cardiovascular Diseases, Centre of Postgraduate Medical Education, Warsaw, Poland
2
Department of Cardiology, Polish-Mother’s Memorial Hospital Research Institute, Łódź, Poland
3
National Medical Institute of the Ministry of the Interior and Administration, Warsaw, Poland
4
Jerzy Popiełuszko Bielański Hospital- Independent Public Healthcare Centre, Poland
5
Mazowiecki Szpital Brodnowski Warsaw, Poland
6
Central Clinical Hospital, Warsaw, Poland
7
Infant Jesus Clinical Hospital, Warsaw, Poland
8
Medical University of Warsaw, Warsaw, Poland
9
Department of Pulmonary Circulation, Thromboembolic Diseases and Cardiology, Centre of Postgraduate Medical Education,
European Health Centre, ERN-LUNG Member, Otwock, Poland
Submission date: 2024-06-24
Final revision date: 2024-08-19
Acceptance date: 2024-08-31
Publication date: 2024-10-16
Corresponding author
Marcin Wasilewski
Department of Cardiovascular Diseases, Centre of Postgraduate Medical Education, 01-813 Warsaw, Poland
Department of Cardiovascular Diseases, Centre of Postgraduate Medical Education, 01-813 Warsaw, Poland
Pediatr Pol 2024;99(4):340-346
KEYWORDS
ALCAPAcoronary artery anomalyanomalous left coronary artery from the pulmonary arteryBland-White-Garland syndromeadult congenital heart disease
TOPICS
ABSTRACT
Anomalous left coronary artery from the pulmonary artery (ALCAPA) is a rare congenital coronary anomaly associated with a mortality rate of 90% within the first year of life, with diagnosis occurring in only 10% to 15% of patients during adulthood. ALCAPA is subdivided into infant type and adult type. While angiography remains the diagnostic gold standard, echocardiography serves as a convenient, non-invasive, and highly reliable initial step in suspected cases of ALCAPA. Computed tomography and magnetic resonance imaging are also capable of providing a definitive diagnosis. Surgical management aims to establish a dual-coronary system through various techniques, with goals including gradual improvement in left ventricular function and reduction in mitral regurgitation. In selected cases, consideration may also be given to mitral valvuloplasty or placement of an implantable cardioverter defibrillator. Postoperatively, patients should undergo regular follow-up evaluations to monitor cardiac function and detect any complications.
REFERENCES (49)
1.
Angelini P, Velasco JA, Flamm S. Coronary anomalies: incidence, pathophysiology, and clinical relevance. Circulation 2002; 105: 2449-2454.
2.
Villa AD, Sammut E, Nair A, et al. Coronary artery anomalies overview: the normal and the abnormal. World J Radiol 2016; 8: 537-555.
3.
Yamanaka O, Hobbs RE. Coronary artery anomalies in 126,595 patients undergoing coronary arteriography. Cathet Cardiovasc Diagn 1990; 21: 28-40.
4.
Frescura C, Basso C, Thiene G, et al. Anomalous origin of coronary arteries and risk of sudden death: a study based on an autopsy population of congenital heart disease. Hum Pathol 1998; 29: 689-695.
5.
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.
6.
Konstantinowitsch W. Ein seltener Fall von Herzmissbildung. Prager Med Wchnschr 1906; 31: 657-660.
7.
Abrikossoff A. Aneurysma des linken Herzventrikels mit abnormer Abgangsstelle der linken Koronararterie von der Pulmonalis bei einem funfmonatlichen Kinde. Arch Pathol Anat 1911; 203: 413-420.
8.
Bland EF, White PD, Garland J. Congenital anomalies of coronary arteries. Report of unusual case associated with cardiac hypertrophy. Am Heart J 1932–1933; 8: 787-801.
9.
Tomanek R, Angelini P. Embryology of coronary arteries and anatomy/pathophysiology of coronary anomalies. A comprehensive update. Int J Cardiol 2019; 281: 28-34.
10.
Cowles RA, Berdon WE. Bland-White-Garland syndrome of nomalous left coronary artery arising from the pulmonary artery (ALCAPA): a historical review. Pediatr Radiol 2007; 37: 890-895.
11.
Peña E, Nguyen ET, Merchant N, et al. ALCAPA syndrome: not just a pediatric disease. Radiographics 2009; 29: 553-565.
12.
Niu M, Zhang J, Ge Y, et al. Acute myocardial infarction in the elderly with anomalous origin of the left coronary artery from the pulmonary artery (ALCAPA): a case report and literature review. Medicine (Baltimore) 2022; 101: e32219.
13.
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.
14.
Laux D, Bertail C, Bajolle F, et al. Anomalous left coronary artery connected to the pulmonary artery associated with other cardiac defects: a difficult joint diagnosis. Pediatr Cardiol 2014; 35: 1198-1205.
15.
Nathan M, Emani S, Marx G, et al. Anomalous left coronary artery arising from the pulmonary artery in hypoplastic left hearts: case series and review of literature. J Thorac Cardiovasc Surg 2011; 142: 225-227.
16.
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.
17.
Yau JM, Singh R, Halpern EJ, et al. 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.
18.
Blickenstaff EA, Smith SD, Cetta F, et al. Anomalous left coronary artery from the pulmonary artery: how to diagnose and treat. J Pers Med 2023; 13: 1561.
19.
Beasley GS, Stephens EH, Backer CL, et al. Anomalous left coronary artery from the pulmonary artery (ALCAPA): a systematic review and historical perspective. Curr Pediatr Rep 2019; 7: 45-52.
20.
Kim BH, Park YW, Hong SP, et al. Anomalous origin of the left coronary artery from the pulmonary artery initially visualized by echocardiography and multidetector computed tomography coronary angiography. J Cardiovasc Ultrasound 2012; 20: 197-200.
21.
Zou JN, Gao YH, Huang B, et al. The diagnostic value of dynamic volume computed tomography angiography in children with anomalous origin of the left coronary artery from the pulmonary artery. Am J Transl Res 2021; 13: 10348-10355.
22.
Karr SS, Parness IA, Spevak PJ, et al. Diagnosis of anomalous left coronary artery by Doppler color flow mapping: distinction from other causes of dilated cardiomyopathy. J Am Coll Cardiol 1992; 19: 1271-1275.
23.
Kerut EK, Kogos PG, Anderson JH, et al. Adult presentation of ALCAPA: echo and CT diagnosis. Echocardiography 2018; 35: 1045-1048.
24.
Ajam A, Rahnamoun Z, Sahebjam M, et al. Cardiac imaging findings in anomalous origin of the coronary arteries from the pulmonary artery; narrative review of the literature. Echo Res Pract 2022; 9: 12.
25.
Yu Y, Wang QS, Wang XF, et al. Diagnostic value of echocardiography on detecting the various types of anomalous origin of the left coronary artery from the pulmonary artery. J Thorac Dis 2020; 12: 319-328.
26.
Amer A, Shai H, Assa S, et al. Aberrant left coronary artery from the pulmonary artery with patent ductus arteriosus – a case report and review of the literature. J Cardiothorac Surg 2024; 19: 319.
27.
Mazurak M, Kusa J. The radiologist’s tragedy, or Bland-White-Garland syndrome (BWGS). On the 80(th) anniversary of the first clinical description of ALCAPA (anomalous left coronary artery from the pulmonary artery). Kardiochir Torakochir Pol 2014; 11: 225-229.
28.
Stout KK, Daniels CJ, Aboulhosn JA, et al. 2018 AHA/ACC guideline for the management of adults with congenital heart disease: executive summary: a report of the American College of Cardiology/American heart association task force on clinical practice guidelines. J Am Coll Cardiol 2019; 73: 1494-1563. Erratum in: J Am Coll Cardiol 2019; 73: 2361.
29.
Baumgartner H, De Backer J, Babu-Narayan SV, et al. ESC Scientific Document Group. 2020 ESC Guidelines for the management of adult congenital heart disease. Eur Heart J 2021; 42: 563-645.
30.
Lange R, Vogt M, Hörer J, et al. Long-term results of repair of anomalous origin of the left coronary artery from the pulmonary artery. Ann Thorac Surg 2007; 83: 1463-1471.
31.
Backer CL, Stout MJ, Zales VR, et al. Anomalous origin of the left coronary artery. A twenty-year review of surgical management. J Thorac Cardiovasc Surg 1992; 103: 1049-1057.
32.
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: 549-553.
33.
Parizek P, Haman L, Harrer J, et al. Bland-White-Garland syndrome in adults: sudden cardiac death as a first symptom and long-term follow-up after successful resuscitation and surgery. Europace 2010; 12: 1338-1340.
34.
Hu R, Zhang W, Yu X, et al. Midterm surgical outcomes for ALCAPA repair in infants and children. Thorac Cardiovasc Surg 2022; 70: 2-9.
35.
Sinha SK, Verma CM, Krishna V, et al. ALCAPA in an octogenarian woman: an enigma. Cardiol Res 2015; 6: 289-291.
36.
Kanoh M, Inai K, Shinohara T, et al. Outcomes from anomalous origin of the left coronary artery from the pulmonary artery repair: Long-term complications in relation to residual myocardial abnormalities. J Cardiol 2017; 70: 498-503.
37.
Schmitt B, Bauer S, Kutty S, 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: 1425-1436.
38.
Browne LP, Kearney D, Taylor MD, et al. ALCAPA: the role of myocardial viability studies in determining prognosis. Pediatr Radiol 2010; 40: 163-167.
39.
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: 547-553.
40.
Mishra A. Surgical management of anomalous origin of coronary artery from pulmonary artery. Indian J Thorac Cardiovasc Surg 2021; 37: 131-143.
41.
Harky A, Noshirwani A, Karadakhy O, et al. Comprehensive literature review of anomalies of the coronary arteries. J Card Surg 2019; 34: 1328-1343.
42.
Radman M, Mastropietro CW, Costello JM, et al. Collaborative research from the Pediatric Cardiac Intensive Care Society (CoRe-PCICS) investigators. Intermediate outcomes after repair of anomalous left coronary artery from the pulmonary artery. Ann Thorac Surg 2021; 112: 1307-1315.
43.
Flores S, Riley CM, Sassalos P, et al. Collaborative research from the Pediatric Cardiac Intensive Care Society (CoRe-PCICS) investigators. ALCAPA in children with complex congenital heart disease: a multicenter study. Pediatr Cardiol 2024.
44.
Sundara Rao CY, Aravind A, Nelam D. Anomalous origin of left coronary artery from the pulmonary artery: our 30 years of surgical experience and outcomes. Turk Gogus Kalp Damar Cerrahisi Derg 2023; 32: 26-36.
45.
Straka N, Gauvreau K, Huang Y, et al. Analysis of perioperative and long-term outcomes among presentations of anomalous left coronary artery from the pulmonary artery diagnosed beyond infancy versus during infancy. Pediatr Cardiol 2023.
46.
Kubota H, Endo H, Ishii H, et al. Adult ALCAPA: from histological picture to clinical features. J Cardiothorac Surg 2020; 15: 14.
47.
Kreutzer U, Krülls-Münch J, Angres M, et al. Erfolgreiche Reanimation einer Patientin mit Kammerflimmern beim Bland-White-Garland-Syndrom im Erwachsenenalter. Ein Fallbericht [Successful resuscitation of a patient with ventricular fibrillation in Bland-White-Garland syndrome in adulthood. A case report]. Z Kardiol 1998; 87: 560-565.
48.
Hsu WF, Lee PC, Li HY, et al. Diagnosis and surgical outcomes of patients with anomalous left coronary artery from the pulmonary artery: a single taiwanese medical center experience. Heart Surg Forum 2020; 23: E101-E106.
49.
Warnes CA, Williams RG, Bashore TM, et al. ACC/AHA 2008 guidelines for the management of adults with congenital heart disease: a report of the American College of Cardiology/American Heart Association Task Force on Practice Guidelines (Writing Committee to Develop Guidelines on the Management of Adults With Congenital Heart Disease). Developed in Collaboration With the American Society of Echocardiography, Heart Rhythm Society, International Society for Adult Congenital Heart Disease, Society for Cardiovascular Angiography and Interventions, and Society of Thoracic Surgeons. J Am Coll Cardiol 2008; 52: e143-e263.
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