Hostname: page-component-8448b6f56d-t5pn6 Total loading time: 0 Render date: 2024-04-19T10:47:14.164Z Has data issue: false hasContentIssue false

Quality of life and exercise performance in unoperated children with anomalous aortic origin of a coronary artery from the opposite sinus of valsalva

Published online by Cambridge University Press:  26 September 2016

Alan C. Sing*
Affiliation:
The Children’s Hospital of Philadelphia, Division of Cardiology, Philadelphia, Pennsylvania, United States of America
Stephen Tsaur
Affiliation:
Perelman School of Medicine at the University of Pennsylvania, Philadelphia, Pennsylvania, United States of America
Stephen M. Paridon
Affiliation:
The Children’s Hospital of Philadelphia, Division of Cardiology, Philadelphia, Pennsylvania, United States of America
Julie A. Brothers
Affiliation:
The Children’s Hospital of Philadelphia, Division of Cardiology, Philadelphia, Pennsylvania, United States of America
*
Correspondence to: A. Sing, MD, Pediatric Heart Specialists, 7777 Forest Lane, Suite C-855, Dallas, TX 75230, United States of America. Tel: 972-331-9700; Fax: 972-331-9833; E-mail: acsing@gmail.com

Abstract

Background

Anomalous aortic origin of a coronary artery is a congenital cardiac condition that can be associated with increased risk of sudden death. To date, quality of life and exercise performance have not been evaluated in patients with this condition who do not undergo surgical repair.

Methods

We carried out a cross-sectional analysis of patients with unoperated anomalous aortic origin of a coronary artery at our institution from 1 January, 2000 to 31 January, 2016. We prospectively assessed quality of life using standardised questionnaires. Medical records were reviewed for clinical and exercise stress test data. Statistical analyses were performed using Student’s t-tests and Spearman’s correlation coefficients.

Results

In total, 56 families completed the questionnaires. The average age at enrolment was 14.7±6 years. The majority were male (n=44, 78.6%) and had interarterial anomalous right coronary artery (n=38, 67.9%). Patients had normal quality of life on the PedsQL 4.0 Report, Child Health Questionnaire Child Form 87, and SF-36v2. Their parents had normal quality of life on the PedsQL 4.0 Parent Report, but parents of exercise-restricted patients had decreased Physical Functioning, General Health Perception, Emotional Impact on Parent, and Physical Summary scores (p<0.001–0.048) on the Child Health Questionnaire Parent Form 50.

Conclusions

Patients with unoperated anomalous aortic origin of a coronary artery appear to have normal quality of life, but parents of exercise-restricted patients have decreased general health and emotional and physical quality of life scores. Improved counselling of families may be beneficial in this group. Future studies with more patients should evaluate quality of life and exercise performance over time.

Type
Original Articles
Copyright
© Cambridge University Press 2016 

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

1. Hoffman, JI. Abnormal origins of the coronary arteries from the aortic root. Cardiol Young 2014; 24: 774791.CrossRefGoogle ScholarPubMed
2. Davis, JA, Cecchin, F, Jones, TK, et al. Major coronary artery anomalies in a pediatric population: incidence and clinical importance. J Am Coll Cardiol 2001; 37: 593597.CrossRefGoogle Scholar
3. Zeppilli, P, dello Russo, A, Santini, C, et al. In vivo detection of coronary artery anomalies in asymptomatic athletes by echocardiographic screening. Chest 1998; 114: 8993.CrossRefGoogle ScholarPubMed
4. Pelliccia, A, Spataro, A, Maron, BJ. Prospective echocardiographic screening for coronary artery anomalies in 1,360 elite competitive athletes. Am J Cardiol 1993; 72: 978979.CrossRefGoogle Scholar
5. Topaz, O, deMarchena, EJ, Perin, E, et al. Anomalous coronary arteries: angiographic findings in 80 patients. Int J Cardiol 1992; 34: 129138.CrossRefGoogle ScholarPubMed
6. Yamanaka, O, Hobbs, RE. Coronary artery anomalies in 126,595 patients undergoing coronary arteriography. Cathet Cardiovasc Diagn 1990; 21: 2840.CrossRefGoogle ScholarPubMed
7. Waters, EB, Salmon, LA, Wake, M, et al. The health and well-being of adolescents: a school-based population study of the self-report child health questionnaire. J Adolesc Health 2001; 29: 140149.CrossRefGoogle ScholarPubMed
8. Tuo, G, Marasini, M, Brunelli, C, et al. Incidence and clinical relevance of primary congenital anomalies of the coronary arteries in children and adults. Cardiol Young 2013; 23: 381386.CrossRefGoogle ScholarPubMed
9. Maron, BJ, Doerer, JJ, Haas, TS, et al. Sudden deaths in young competitive athletes: analysis of 1866 deaths in the United States, 1980-2006. Circulation 2009; 119: 10851092.CrossRefGoogle ScholarPubMed
10. Taylor, AJ, Rogan, KM, Virmani, R. Sudden cardiac death associated with isolated congenital coronary artery anomalies. J Am Coll Cardiol 1992; 20: 640647.CrossRefGoogle ScholarPubMed
11. Liberthson, RR, Dinsmore, RE, Fallon, JT. Aberrant coronary artery origin from the aorta. Report of 18 patients, review of literature and delineation of natural history and management. Circulation 1979; 59: 748754.CrossRefGoogle ScholarPubMed
12. Basso, C, Maron, BJ, Corrado, D, et al. Clinical profile of congenital coronary artery anomalies with origin from the wrong aortic sinus leading to sudden death in young competitive athletes. J Am Coll Cardiol 2000; 35: 14931501.CrossRefGoogle ScholarPubMed
13. Cheitlin, MD, De Castro, CM, McAllister, HA. Sudden death as a complication of anomalous left coronary origin from the anterior sinus of Valsalva, A not-so-minor congenital anomaly. Circulation 1974; 50: 780787.CrossRefGoogle ScholarPubMed
14. 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: 689695.CrossRefGoogle Scholar
15. Kragel, AH, Roberts, WC. Anomalous origin of either the right or left main coronary artery from the aorta with subsequent coursing between aorta and pulmonary trunk: analysis of 32 necropsy cases. Am J Cardiol 1988; 62 (10 Pt 1): 771777.CrossRefGoogle ScholarPubMed
16. Corrado, D, Thiene, G, Nava, A, et al. Sudden death in young competitive athletes: clinicopathologic correlations in 22 cases. Am J Med 1990; 89: 588596.CrossRefGoogle Scholar
17. Maron, BJ, Shirani, J, Poliac, LC, et al. Sudden death in young competitive athletes. Clinical, demographic, and pathological profiles. JAMA 1996; 276: 199204.CrossRefGoogle ScholarPubMed
18. Taylor, AJ, Byers, JP, Cheitlin, MD, et al. Anomalous right or left coronary artery from the contralateral coronary sinus: “high-risk” abnormalities in the initial coronary artery course and heterogeneous clinical outcomes. Am Heart J 1997; 133: 428435.CrossRefGoogle ScholarPubMed
19. Brothers, JA, McBride, MG, Seliem, MA, et al. Evaluation of myocardial ischemia after surgical repair of anomalous aortic origin of a coronary artery in a series of pediatric patients. J Am Coll Cardiol 2007; 50: 20782082.CrossRefGoogle Scholar
20. Turner, II, Turek, JW, Jaggers, J, et al. Anomalous aortic origin of a coronary artery: preoperative diagnosis and surgical planning. World J Pediatr Congenit Heart Surg 2011; 2: 340345.CrossRefGoogle ScholarPubMed
21. Graham, TP Jr, Driscoll, DJ, Gersony, WM, et al. Task Force 2: congenital heart disease. J Am Coll Cardiol 2005; 45: 13261333.CrossRefGoogle ScholarPubMed
22. Van Hare, GF, Ackerman, MJ, Evangelista, JK, et al. Eligibility and disqualification recommendations for competitive athletes with cardiovascular abnormalities: Task Force 4: congenital heart disease: a scientific statement from the American Heart Association and American College of Cardiology. J Am Coll Cardiol 2015; 66: 23722384.CrossRefGoogle ScholarPubMed
23. Van Hare, GF, Ackerman, MJ, Evangelista, JK, et al. Eligibility and disqualification recommendations for competitive athletes with cardiovascular abnormalities: Task Force 4: congenital heart disease: a scientific statement from the American Heart Association and American College of Cardiology. Circulation 2015; 132: e281e291.CrossRefGoogle ScholarPubMed
24. Brothers, J, Gaynor, JW, Paridon, S, et al. Anomalous aortic origin of a coronary artery with an interarterial course: understanding current management strategies in children and young adults. Pediatr Cardiol 2009; 30: 911921.CrossRefGoogle ScholarPubMed
25. Vinocur, JM, Menk, JS, Connett, J, et al. Surgical volume and center effects on early mortality after pediatric cardiac surgery: 25-year North American experience from a multi-institutional registry. Pediatr Cardiol 2013; 34: 12261236.CrossRefGoogle ScholarPubMed
26. Brothers, JA, Mcbride, MG, Marino, BS, et al. Exercise performance and quality of life following surgical repair of anomalous aortic origin of a coronary artery in the pediatric population. J Thorac Cardiovasc Surg 2009; 137: 380384.CrossRefGoogle ScholarPubMed
27. Wittlieb-Weber, CA, Paridon, SM, Gaynor, JW, et al. Medium-term outcome after anomalous aortic origin of a coronary artery repair in a pediatric cohort. J Thorac Cardiovasc Surg 2014; 147: 15801586.CrossRefGoogle Scholar
28. Dean, PN, Gillespie, CW, Greene, E, et al. Sports participation and quality of life in adolescents and young adults with congenital heart disease. Congenit Heart Dis 2014; 63(12S): 111.Google Scholar
29. Varni, JW, Limbers, CA, Burwinkle, TM. How young can children reliably and validly self-report their health-related quality of life?: an analysis of 8,591 children across age subgroups with the PedsQL 4.0 generic core scales. Health Qual Life Outcomes 2007; 5: 1.CrossRefGoogle Scholar
30. Ware, JE Jr. SF-36 health survey update. Spine (Phila Pa 1976) 2000; 25: 31303139.CrossRefGoogle ScholarPubMed
31. HealthActCHQ. The CHQ Scoring and Interpretation Manual. The Health Institute, New England Medical Center, Boston, MA, 2008, pp. 1–213.Google Scholar
32. Landgraf, J, Abetz, L. Functional status and well-being of children representing three cultural groups: initial self-reports using the CHQ-CF87. J Psychol Health 1997; 12: 839854.Google Scholar
33. Varni, JW, Seid, M, Rode, CA. The PedsQL: measurement model for the pediatric quality of life inventory. Med Care 1999; 37: 126139.CrossRefGoogle ScholarPubMed
34. Varni, JW, Burwinkle, TM, Seid, M, et al. The PedsQL 4.0 as a pediatric population health measure: feasibility, reliability, and validity. Ambul Pediatr 2003; 3: 329341.2.0.CO;2>CrossRefGoogle ScholarPubMed
35. Friess, MR, Marino, BS, Cassedy, A, et al. Health-related quality of life assessment in children followed in a cardiomyopathy clinic. Pediatr Cardiol 2014; 36: 516523.CrossRefGoogle Scholar
36. Ware, J. QualityMetric Health OutcomesTM Scoring Software 4.5 – User’s Guide. Optum, Eden Prairie, MN, 2011.Google Scholar
37. Raat, H, Mangunkusumo, RT, Landgraf, JM, et al. Feasibility, reliability, and validity of adolescent health status measurement by the Child Health Questionnaire Child Form (CHQ-CF): internet administration compared with the standard paper version. Qual Life Res 2007; 16: 675685.CrossRefGoogle ScholarPubMed
38. Raat, H, Landgraf, JM, Bonsel, GJ, et al. Reliability and validity of the Child Health Questionnaire-Child Form (CHQ-CF87) in a Dutch adolescent population. Qual Life Res 2002; 11: 575581.CrossRefGoogle Scholar
39. Lindstrom, C, Aman, J, Norberg, AL. Increased prevalence of burnout symptoms in parents of chronically ill children. Acta Paediatr 2010; 99: 427432.CrossRefGoogle ScholarPubMed
40. Vance, YH, Morse, RC, Jenney, ME, et al. Issues in measuring quality of life in childhood cancer: measures, proxies, and parental mental health. J Child Psychol Psychiatry 2001; 42: 661667.CrossRefGoogle ScholarPubMed
41. Brothers, JA, Gaynor, JW, Jacobs, JP, et al. The registry of anomalous aortic origin of the coronary artery of the Congenital Heart Surgeons’ Society. Cardiol Young 2010; 20 (Suppl 3): 5058.CrossRefGoogle ScholarPubMed