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Paediatric-onset coronary artery anomalies in pregnancy: a single-centre experience and systematic literature review

  • Michelle Keir (a1), Catriona Bhagra (a2), Debra Vatenmakher (a1), Francisca Arancibia-Galilea (a1), Katrijn Jansen (a1), Norihisa Toh (a1), Candice K. Silversides (a1) (a2), Jack Colman (a1) (a2), Samuel C. Siu (a2) (a3), Mathew Sermer (a4), Andrew M. Crean (a1) (a5) and Rachel M. Wald (a1) (a2)...

Abstract

Objectives

Individuals with childhood-onset coronary artery anomalies are at increased risk of lifelong complications. Although pregnancy is thought to confer additional risk, a few data are available regarding outcomes in this group of women. We sought to define outcomes of pregnancy in this unique population.

Methods

We performed a retrospective survey of women with paediatric-onset coronary anomalies and pregnancy in our institution, combined with a systematic review of published cases. We defined paediatric-onset coronary artery anomalies as congenital coronary anomalies and inflammatory arteriopathies of childhood that cause coronary aneurysms. Major cardiovascular events were defined as pulmonary oedema, sustained arrhythmia requiring treatment, stroke, myocardial infarction, cardiac arrest, or death.

Results

A total of 25 surveys were mailed, and 20 were returned (80% response rate). We included 46 articles from the literature, which described cardiovascular outcomes in 82 women (138 pregnancies). These data were amalgamated for a total of 102 women and 194 pregnancies; 59% of women were known to have paediatric-onset coronary artery anomalies before pregnancy. In 23%, the anomaly was unmasked during or shortly after pregnancy. The remainder, 18%, was diagnosed later in life. Major cardiovascular events occurred in 14 women (14%) and included heart failure (n=5, 5%), myocardial infarction (n=7, 7%), maternal death (n=2, 2%), cardiac arrest secondary to ventricular fibrillation (n=1, 1%), and stroke (n=1, 1%). The majority of maternal events (13/14, 93%) occurred in women with no previous diagnosis of coronary disease.

Conclusions

Women with paediatric-onset coronary artery anomalies have a 14% risk of adverse cardiovascular events in pregnancy, indicating the need for careful assessment and close follow-up. Prospective, multicentre studies are required to better define risk and predictors of complications during pregnancy.

Copyright

Corresponding author

Correspondence to: R. M. Wald, Toronto Congenital Cardiac Centre for Adults, Toronto General Hospital, University of Toronto, 585 University Avenue, 5N 517, Toronto, Ontario, Canada, M5G 2N2. Tel: +416 340 5502; Fax: +416 340 5014; E-mail: rachel.wald@uhn.ca

References

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1. Grewal, J, Silversides, CK, Colman, JM. Pregnancy in women with heart disease: risk assessment and management of heart failure. Heart Failure Clin 2014; 10: 117129.
2. Akpinar, I, Sayin, M, Karabag, T, et al. Differences in sex, angiographic frequency, and parameters in patients with coronary artery anomalies: single-center screening of 25368 patients by coronary angiography. Coron Artery Dis 2013; 24: 266271.
3. Ripley, D, Saha, A, Teis, A, et al. The distribution and prognosis of anomalous coronary arteries identified by cardiovascular magnetic resonance: 15 year experience from two tertiary centres. J Cardiovasc Magn Reson 2014; 16: 18.
4. Rajbanshi, BG, Burkhart, HM, Schaff, HV, Daly, RC, Phillips, SD, Dearani, JD. Surgical strategies for anomalous origin of coronary artery from pulmonary artery in adults. Congit Heart Dis 2014; 148: 220224.
5. Sharma, V, Burkhart, HM, Dearani, JD, et al. Surgical unroofing of anomalous aortic origin of a coronary artery: a single-center experience. Ann Thorac Surg 2014; 98: 941946.
6. Kato, H. Cardiovascular complications in Kawasaki disease: coronary artery lumen and long-term consequences. Progress Pediatr Cardiol 2004; 19: 137145.
7. Holmes, A, Laney, D. A retrospective survey studying the impact of Fabry disease on pregnancy. JIMD Rep 2015; 21: 5763.
8. Siu, SC, Sermer, M, Colman, JM, et al. Prospective multicenter study of pregnancy outcomes in women with heart disease. Circulation 2001; 104: 515521.
9. Cauldwell, M, Swan, L, von Klemperer, K, Patel, R, Steer, P. Management of ALCAPA in two pregnancies. Int J Cardiol 2015; 181: 353354.
10. Kanno, K, Sakai, H, Nakajima, M, Satake, A, Konishi, T, Hajime, I. An adult case of Kawasaki disease in a pregnant Japanese woman: a case report. Case Rep Dermatol 2011; 3: 98102.
11. McAndrew, P, Hughes, D, Adams, P. Pregnancy and Kawasaki disease. Int J Obstet Anesth 2000; 9: 279281.
12. Gordon, CT, Jimenez-Fernandez, S, Daniels, LB, et al. Pregnancy in women with a history of Kawasaki disease: management and outcomes. BJOG 2014; 121: 14311438.
13. Youssef, A, Ben Amara, F, M’barki, M, Reziga, H, Magherbi, H, Neji, K. Grossesse et accrouchement associée a une insuffisance coronarienne due a une maladie de Kawaski. Tunise Med 2013; 91: 217218.
14. Roscani, MG, Zanati, SG, Salmazo, PS, et al. Congenital aneurysmal circumflex coronary artery fistula in a pregnant woman. Clinics 2012; 67: 15231525.
15. Wanczura, P, Orlowski, M, Stecko, W, et al. Niedomykalnosc zastawki mitralnej w przebiegu zespolu Blanda-White’a-Garlanda u chorej po czterech neipowiklanych porodach fizjolgicznych. Pol Merkur Lekarski 2011; 31: 103105.
16. Samada, K, Shiraishi, H, Sato, A, Momoi, MY. Grown-up Kawasaki disease patients who have giant coronary aneurysms. World J Pediatr 2010; 6: 3842.
17. Lazar, D, Enriquez, LJ, Rozental, T. Anomalous origin of the left coronary artery from the pulmonary artery in a pregnant woman with preeclampsia. J Cardiothorac Vasc Anesth 2010; 24: 112115.
18. Radomska, E, Polewyczyk, A, Sadowski, M, Janion, M. Acute myocardial infarction due to thrombosis of coronary artery aneurysm in a 29-year-old women during postpartum period. Kardiol Pol 2008; 66: 13021305.
19. Wojciuk, J, Bury, RW, Roberts, DH. Large bilateral coronary artery fistulae. Int J Cardiol 2008; 128: e93e94.
20. Hibbard, JU, Fajardo, JE, Briller, J. Kawasaki disease with coronary artery sequelae. Obstet Gynecol 2007; 109: 517519.
21. Lee, AC, Foster, E, Yeghiazarians, Y. Anomalous origin of the left coronary artery from the pulmonary artery: a case series and brief review. Congenit Heart Dis 2006; 1: 111115.
22. Adamo, L, Adamo, M. Giant single coronary artery with terminal fistulous aneurysm: a rare entity. Heart 2006; 92: 1419.
23. Choong, CK, Martinez, C, Barner, HB, Ludbrook, PA. Bland-White-Garland syndrome in pregnancy: reoperation of ALCAPA with an internal thoracic radial artery “Y”-graft. Ann Thorac Surg 2006; 81: 15121514.
24. Tsuda, E, Kawamata, K, Neki, R, Echigo, S, Chiba, Y. Nationwide survey of pregnancy and delivery in patients with coronary arterial lesions caused by Kawasaki disease in Japan. Cardiol Young 2006; 16: 173178.
25. Tsuda, E, Ishihara, Y, Kawamata, K, et al. Pregnancy and delivery in patients with coronary artery lesions caused by Kawasaki disease. Heart 2005; 91: 14811482.
26. Zavalloni, D, Belli, G, Caratti, A, Presbitero, P. Anomalous origin of the left coronary artery from the pulmonary artery in an adult pregnant patient: surgical and percutaneous myocardial revascularization. Ital Heart J 2005; 6: 348352.
27. Brown, B, Xie, GY, Booth, D. Images in cardiology: multiple coronary fistulae. Clin Cardiol 2005; 28: 143.
28. Maeder, M, Vogt, PR, Ammann, P, Rickli, H. Bland-White-Garland syndrome in a 39-year-old mother. Ann Thorac Surg 2004; 78: 14511453.
29. Tay, SM, Ong, BC, Tan, SA. Cesarean section in a mother with uncorrected congenital coronary to pulmonary artery fistula. Can J Anesth 1999; 46: 368371.
30. Shear, R, Leduc, L. Successful pregnancy following Kawasaki disease. Obstet Gynecol 1999; 94: 841.
31. Nightingale, AK, Burrell, CJ, Marshall, AJ. Anomalous origin of the left coronary artery from the pulmonary artery: natural history and normal pregnancies. Heart 1998; 80: 629631.
32. Hayakawa, H, Katoh, T. Successful pregnancy after coronary artery bypass grafting for Kawasaki disease. Acta Paediatr Jpn 1998; 40: 275277.
33. Espinosa, R, Badui, E, Rangel, A. A case of a coronary artery arising from the pulmonary artery. Rev Invest Clin 1997; 49: 225226.
34. Daniels, C, Bacon, J, Fontana, ME, Eaton, G, Cohen, D, Leier, CV. Anomalous origin of the left main coronary artery from the pulmonary trunk masquerading as peripartum cardiomyopathy. Am J Cardiol 1997; 79: 13071308.
35. Arakawa, K, Akita, T, Nizhizawa, K, et al. Anticoagulant therapy during successful pregnancy and delivery in a Kawasaki disease patient with coronary aneurysm: a case report. Jpn Circ J 1997; 61: 197200.
36. Sbraggia, P, Habib, G, Garcia, M, et al. Naissance anormale de la coronaire gauche a partir de l’artère pulmonaire: apport diagnostique des techniques échographiques. Ann Cardiol Angéiol 1996; 45: 507511.
37. Alam, S, Sakura, S, Kosaka, Y. Anaesthetic management for Caesarean section in a patient with Kawasaki disease. Can J Anaesth 1995; 42: 10241026.
38. Ruszkiewicz, A, Opeskin, K. Sudden death in pregnancy from congenital malformation of the coronary arteries. Pathology 1993; 25: 236239.
39. Puppo, A, Fournier, JA, Leal, R, et al. Infarto agudo de miocardio y aneurisma arterial coronario durante el embarazo. Rev Esp Cardiol 1993; 46: 4748.
40. Nolan, TE, Savage, RW. Peripartum myocardial infarction from presumed Kawasaki’s disease. South Med J 1990; 83: 13601361.
41. Goldberg, N, Zisbrod, Z, Kipperman, R, et al. Congenital aneurysm of the left coronary sinus and left main coronary artery with fistulous communication to the right atrium in pregnancy. J Am Soc Echocardiogr 1990; 3: 125130.
42. Suarez Ortega, S, Franco Cebrian, J, Medina Fernandez-Aceytuno, A, Armas Portela, M, Mendez Mora, D, Zarazaga, E. Infarto de miocardio no arteriosclerotico en enferma de 24 anos y uso accidental de amiodarona durante el embarazo sin efectos embriotoxicos. Med Clin (Barc) 1988; 90: 586588.
43. Chestnut, DH, Roberts, SL, Laube, DW, Martins, JB. Pregnancy in a patient with aneurysms of the right coronary artery and an arterioventricular fistula: a case report. J Reprod Med 1986; 31: 528530.
44. Klein, VR, Repke, JT, Marquette, GP, Niebyl, JR. The Bland-White-Garland syndrome in pregnancy. Am J Obset Gyencol 1984; 150: 106107.
45. Sabharwal, MS, Tak, J, Chatterjee, S, et al. Anomalous left coronary artery arising from the pulmonary artery in a 38-year pregnant female. J Am Med Assoc 2015; 65: A635.
46. Holliday, L, Misenheimer, J, Sharma, G. Peripartum diagnosis and management of coronary arterial fistula. J Am Med Assoc 2015; 65: A582.
47. Juraszek, A, Drohomirecka, A, Kolsut, P, et al. Takeuchi’s repair in adult women with Bland-White-Garland syndrome and silent myocardial ischemia. Eur Surg 2014; 46: S23S24.
48. Ganigara, M, Collins, N, Tanous, D, Clermajer, D, Puranik, R. Anomalous left anterior descending coronary artery from the pulmonary artery – the role of cardiac MRI. Int J Cardiol 2014; 172: e172e174.
49. Ginghina, C, Popescu, BA, Craciunescu, I, et al. Associated coronary artery anomalies: a diagnostic and therapeutic challenge. Arch Balk Med Un 2006; 41: 132134.
50. Taniguchi, K, Ono, H, Sato, A, et al. Strict management of a pregnant patient with giant coronary aneurysm due to Kawasaki disease. Pediatr Int 2015; 57: 990992.
51. Bruckheimer, E, Bulbul, Z, McCarthy, P, Madri, JA, Friedman, AH, Hellenbrand, WE. Kawasaki disease: coronary aneurysms in mother and son. Circulation 1998; 97: 410411.
52. Simsek, H, Tuncer, M, Yaman, M, Celik, M. Presentation of adult Bland-White-Garland syndrome in a 32-year-old female. Int J Cardiovasc Acad 2015; 1: 5658.
53. Patel, H, Swirsky, B. Alcapa syndrome: a rare congenital anomaly diagnosed postpregnancy in an adult. Chest 2015; 148: 37A.
54. Saaid, R, Balchin, I, Azman, W. Coronary artery fistula in pregnancy. J Obstet Gynaecol Res 2015; 41: 5152.
55. Burchill, LJ, Lameijer, H, Roos-Hesselink, JW, et al. Pregnancy risks in women with pre-existing coronary artery disease, or following acute coronary syndrome. Heart 2015; 101: 525529.
56. Chang, AM, Mumma, B, Sease, KL, Robey, JL, Shofer, FS, Hollander, JE. Gender bias in cardiovascular testing persists after adjustment for presenting characteristics and cardiac risk. Acad Emerg Med 2007; 14: 599605.
57. Henderson, JT, Whitlock, EP, O’Connor, E, Senger, CA, Thompson, JH, Rowland, MG. Low-dose aspirin for prevention of morbidity and mortality from preeclampsia: a systematic evidence review for the U.S. Preventive Services Task Force. Ann Intern Med 2014; 160: 695703.
58. Newburger, JW, Takahashi, M, Gerber, MA, et al. Diagnosis, treatment, and long-term management of Kawasaki disease: a statement for health professionals from the committee on rheumatic fever, endocarditis, and Kawasaki disease, Council on Cardiovascular Disease in the Young, American Heart Association. Pediatrics 2004; 114: 17081733.
59. Warnes, CA, Williams, RG, Bashore, TM, et al. ACC/AHA 2008 guidelines for the management of adults with congenital heart disease. J Am Coll Cardiol 2008; 52: e143e263.
60. Newport, DJ, Brennan, PA, Green, P, et al. Maternal depression and medication exposure during pregnancy: comparison of maternal retrospective recall to prospective documentation. BJOG 2008; 115: 681688.
61. Davies, JE, Burkhart, HM, Dearani, JA, et al. Surgical management of anomalous aortic origin of a coronary artery. Ann Thorac Surg 2009; 89: 844847.
62. Angelini, P. Coronary fistulae: which ones deserve treatment and what kind of treatment do they need? Tex Heart Inst J 2007; 34: 202203.

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