CONGENITAL HEART
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score and time to normalization of lactate. The long-term predictive ability of the Bayley-III scores measured at 18 to 24 months is unclear [21]. This is an observational study and, therefore, we can only establish an association between clinical score and Bayley-III scores, not a cause and effect relationship. The clinical outcome score may not distinguish moderate from severely impaired brain function. In the multiple regression model, it was not possible to adjust for ICU LOS or total hospital LOS as these variables are highly correlated with the clinical outcome score. We have not demonstrated that interventions aimed at lowering the clinical outcome score in any class of patients are associated with improved neurodevelopmental outcomes. In summary, the clinical outcome score is associated with adverse Bayley-III scores at 18 to 24 months of age and correlated strongly with other markers of postoperative events, including time to normalization of serum lactate and ICU and hospital LOS. This score is easy to calculate, has no cost, and can be used to identify children who may benefit from early and comprehensive neurodevelopmental intervention. The score may be valuable as an outcome variable in future studies aimed at evaluating novel therapies in this high-risk population. The members of the Western Canadian Complex Pediatric Therapies Program Follow-Up Group are Charlene M. T. Robertson, Ari R. Joffe, David B. Ross, and Ivan M. Rebeyka, Edmonton, Alberta; Reg Sauve, Calgary, Alberta; Patricia Blakley, Saskatoon, Saskatchewan; Anne Synnes, Vancouver, British Columbia; and Jaya Bodani, Regina, Saskatchewan, Canada.
References 1. Bellinger DC, Wypij D, duPlessis AJ, et al. Neurodevelopmental status at eight years in children with dextrotransposition of the great arteries: the Boston Circulatory Arrest Trial. J Thorac Cardiovasc Surg 2003;126:1385–96. 2. Shillingford AJ, Glanzman MM, Ittenbach RF, Clancy RR, Gaynor JW, Wernovsky G. Inattention, hyperactivity, and school performance in a population of school-age children with complex congenital heart disease. Pediatrics 2008;121:e759–67. 3. Mackie AS, Alton GY, Dinu IA, et al. Clinical outcome score predicts the need for neurodevelopmental intervention after infant heart surgery. J Thorac Cardiovasc Surg 2013;145: 1248–54.e2. 4. Bayley N. Bayley scales of infant and toddler development. 3rd ed. San Antonio, TX: Harcourt Assessment; 2006.
Ann Thorac Surg 2015;99:2124–33
5. Robertson CMT, Joffe AR, Sauve RS, et al. Outcomes from an interprovincial program of newborn open heart surgery. J Pediatr 2004;144:86–92. 6. Robertson CMT, Sauve RS, Joffe AR, et al. The registry and follow-up of complex pediatric therapies program of western Canada: a mechanism for service, audit, and research after life-saving therapies for young children. Cardiol Res Pract 2011;2011:965740. 7. Wernovsky G, Wypij D, Jonas RA, et al. Postoperative course and hemodynamic profile after the arterial switch operation in neonates and infants. A comparison of low-flow cardiopulmonary bypass and circulatory arrest. Circulation 1995;92:2226–35. 8. Cheung PY, Chui N, Joffe AR, Rebeyka IM, Robertson CM. Postoperative lactate concentrations predict the outcome of infants aged 6 weeks or less after intracardiac surgery: a cohort follow-up to 18 months. J Thorac Cardiovasc Surg 2005;130:837–43. 9. Norman GR, Sloan JA, Wyrwich KW. Interpretation of changes in health-related quality of life: the remarkable universality of half a standard deviation. Med Care 2003;41: 582–92. 10. Hunter JE. Cognitive ability, cognitive aptitudes, job knowledge, and job performance. J Vocat Behav 1986;29:340–62. 11. McCall RB. Childhood IQ’s as predictors of adult educational and occupational status. Science 1977;197:482–3. 12. Moffitt TE, Gabrielli WF, Mednick SA, Schulsinger F. Socioeconomic status, IQ, and delinquency. J Abnorm Psychol 1981;90:152–6. 13. Fagan J. Intelligence in infancy. In: Sternberg RJ, Kaufman SB (eds). New York: Cambridge Press; 2011:130–43. 14. Wechsler D. Wechsler preschool and primary scale of intelligence-fourth edition (WPPSI-IV). San Antonio, TX: Pearson; 2012. 15. Bayley N (ed). Bayley scales of infant and toddler development-third edition. Technical manual. San Antonio, TX: Harcourt Assessment; 2006. 16. Newburger JW, Wypij D, Bellinger DC, et al. Length of stay after infant heart surgery is related to cognitive outcome at age 8 years. J Pediatr 2003;143:67–73. 17. Zobel G, Rodl S, Rigler B, et al. Prospective evaluation of clinical scoring systems in infants and children with cardiopulmonary insufficiency after cardiac surgery. J Cardiovasc Surg 1993;34:333–7. 18. Kang N, Cole T, Tsang V, Elliott M, De Leval M. Risk stratification in paediatric open-heart surgery. Eur J Cardiothorac Surg 2004;26:3–11. 19. Shime N, Kageyama K, Ashida H, Tanaka Y. Application of modified sequential organ failure assessment score in children after cardiac surgery. J Cardiothorac Vasc Anesth 2001;15:463–8. 20. Gillespie M, Kuijpers M, Van Rossem M, et al. Determinants of intensive care unit length of stay for infants undergoing cardiac surgery. Congenit Heart Dis 2006;1:152–60. 21. Aylward GP. Continuing issues with the Bayley-III: where to go from here. J Develop Behav Pediatr 2013;34:697–701.
INVITED COMMENTARY Neurodevelopmental outcome is one of the most important factors that parents of children born with congenital heart defects are concerned about once the initial anxieties about the diagnosis and operation have settled. Long-term studies have shown that developmental and functional abnormalities across multiple domains are present at school entry in these children. Among the many predictors of later developmental
Ó 2015 by The Society of Thoracic Surgeons Published by Elsevier
difficulties (microcephaly, genetic predisposition, age at operation, palliation, circulatory arrest time, abnormal postoperative neurologic features, and maternal education), very few are modifiable. Recent advances in the understanding of the fetal mechanisms of impaired cerebral blood flow in the context of congenital heart disease suggest that immediate postoperative events may be only an indicator and may not explain causality
0003-4975/$36.00 http://dx.doi.org/10.1016/j.athoracsur.2015.02.072
Ann Thorac Surg 2015;99:2124–33
MACKIE ET AL NEURODEVELOPMENTAL OUTCOME POST INFANT CHD SURGERY
examination, or both would add to the predictive validity of the 18- to 24-month Bayley-III score for late neurodevelopment. Most certainly, delayed recognition, for example at school age, is clearly too late. Early identification and intervention, right from infancy, can mitigate some of these risks, help the family to seek help, and help the community services to target interventions for these vulnerable children. Victor Tsang, FRCS Aparna Hoskote, MD Department of Cardiothoracic Surgery Great Ormond Street Hospital for Children NHS Trust Great Ormond St London WC1N 3JH, United Kingdom e-mail: [email protected]
Reference 1. Mackie AS, Vatanpour S, Alton GY, et al. Clinical outcome score predicts adverse neurodevelopmental outcome after infant heart surgery. Ann Thorac Surg 2015;99:2124–33.
CONGENITAL HEART
for neurodevelopmental morbidity. Nonetheless, modifying the neurodevelopmental outcome is an area of intense research leading to the development of dedicated cardiac neurodevelopmental programs. Understandably, it is very challenging to enroll infants in clinical trials such as randomized trials of neuroprotective strategies, track the long-term progress of particular cohorts, and wait years before one can determine the consequences of current management strategies. Hence, a postoperative score reflecting the patient’s progress in the first few days after a cardiac operation does hold promise [1]. An easy-to-measure score, one not needing complex data collection and validated against formal neurodevelopmental testing, is of practical value. However, certain caveats have to be recognized. Most intensive care units (ICU) have traditional unit-specific criteria to determine the timing of chest closure and extubation. The thresholds to drive diuresis and use renal replacement therapy differ widely. Furthermore, the presence of extracardiac comorbidities may have an impact on these decisions. Validation of this score in similar representative cardiac programs with a certain degree of standardization in ICU practices would add power to the conclusions. Furthermore, the provisions of neuroimaging, neurologic
2133
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MACKIE ET AL NEURODEVELOPMENTAL OUTCOME POST INFANT CHD SURGERY
score and time to normalization of lactate. The long-term predictive ability of the Bayley-III scores measured at 18 to 24 months is unclear [21]. This is an observational study and, therefore, we can only establish an association between clinical score and Bayley-III scores, not a cause and effect relationship. The clinical outcome score may not distinguish moderate from severely impaired brain function. In the multiple regression model, it was not possible to adjust for ICU LOS or total hospital LOS as these variables are highly correlated with the clinical outcome score. We have not demonstrated that interventions aimed at lowering the clinical outcome score in any class of patients are associated with improved neurodevelopmental outcomes. In summary, the clinical outcome score is associated with adverse Bayley-III scores at 18 to 24 months of age and correlated strongly with other markers of postoperative events, including time to normalization of serum lactate and ICU and hospital LOS. This score is easy to calculate, has no cost, and can be used to identify children who may benefit from early and comprehensive neurodevelopmental intervention. The score may be valuable as an outcome variable in future studies aimed at evaluating novel therapies in this high-risk population. The members of the Western Canadian Complex Pediatric Therapies Program Follow-Up Group are Charlene M. T. Robertson, Ari R. Joffe, David B. Ross, and Ivan M. Rebeyka, Edmonton, Alberta; Reg Sauve, Calgary, Alberta; Patricia Blakley, Saskatoon, Saskatchewan; Anne Synnes, Vancouver, British Columbia; and Jaya Bodani, Regina, Saskatchewan, Canada.
References 1. Bellinger DC, Wypij D, duPlessis AJ, et al. Neurodevelopmental status at eight years in children with dextrotransposition of the great arteries: the Boston Circulatory Arrest Trial. J Thorac Cardiovasc Surg 2003;126:1385–96. 2. Shillingford AJ, Glanzman MM, Ittenbach RF, Clancy RR, Gaynor JW, Wernovsky G. Inattention, hyperactivity, and school performance in a population of school-age children with complex congenital heart disease. Pediatrics 2008;121:e759–67. 3. Mackie AS, Alton GY, Dinu IA, et al. Clinical outcome score predicts the need for neurodevelopmental intervention after infant heart surgery. J Thorac Cardiovasc Surg 2013;145: 1248–54.e2. 4. Bayley N. Bayley scales of infant and toddler development. 3rd ed. San Antonio, TX: Harcourt Assessment; 2006.
Ann Thorac Surg 2015;99:2124–33
5. Robertson CMT, Joffe AR, Sauve RS, et al. Outcomes from an interprovincial program of newborn open heart surgery. J Pediatr 2004;144:86–92. 6. Robertson CMT, Sauve RS, Joffe AR, et al. The registry and follow-up of complex pediatric therapies program of western Canada: a mechanism for service, audit, and research after life-saving therapies for young children. Cardiol Res Pract 2011;2011:965740. 7. Wernovsky G, Wypij D, Jonas RA, et al. Postoperative course and hemodynamic profile after the arterial switch operation in neonates and infants. A comparison of low-flow cardiopulmonary bypass and circulatory arrest. Circulation 1995;92:2226–35. 8. Cheung PY, Chui N, Joffe AR, Rebeyka IM, Robertson CM. Postoperative lactate concentrations predict the outcome of infants aged 6 weeks or less after intracardiac surgery: a cohort follow-up to 18 months. J Thorac Cardiovasc Surg 2005;130:837–43. 9. Norman GR, Sloan JA, Wyrwich KW. Interpretation of changes in health-related quality of life: the remarkable universality of half a standard deviation. Med Care 2003;41: 582–92. 10. Hunter JE. Cognitive ability, cognitive aptitudes, job knowledge, and job performance. J Vocat Behav 1986;29:340–62. 11. McCall RB. Childhood IQ’s as predictors of adult educational and occupational status. Science 1977;197:482–3. 12. Moffitt TE, Gabrielli WF, Mednick SA, Schulsinger F. Socioeconomic status, IQ, and delinquency. J Abnorm Psychol 1981;90:152–6. 13. Fagan J. Intelligence in infancy. In: Sternberg RJ, Kaufman SB (eds). New York: Cambridge Press; 2011:130–43. 14. Wechsler D. Wechsler preschool and primary scale of intelligence-fourth edition (WPPSI-IV). San Antonio, TX: Pearson; 2012. 15. Bayley N (ed). Bayley scales of infant and toddler development-third edition. Technical manual. San Antonio, TX: Harcourt Assessment; 2006. 16. Newburger JW, Wypij D, Bellinger DC, et al. Length of stay after infant heart surgery is related to cognitive outcome at age 8 years. J Pediatr 2003;143:67–73. 17. Zobel G, Rodl S, Rigler B, et al. Prospective evaluation of clinical scoring systems in infants and children with cardiopulmonary insufficiency after cardiac surgery. J Cardiovasc Surg 1993;34:333–7. 18. Kang N, Cole T, Tsang V, Elliott M, De Leval M. Risk stratification in paediatric open-heart surgery. Eur J Cardiothorac Surg 2004;26:3–11. 19. Shime N, Kageyama K, Ashida H, Tanaka Y. Application of modified sequential organ failure assessment score in children after cardiac surgery. J Cardiothorac Vasc Anesth 2001;15:463–8. 20. Gillespie M, Kuijpers M, Van Rossem M, et al. Determinants of intensive care unit length of stay for infants undergoing cardiac surgery. Congenit Heart Dis 2006;1:152–60. 21. Aylward GP. Continuing issues with the Bayley-III: where to go from here. J Develop Behav Pediatr 2013;34:697–701.
INVITED COMMENTARY Neurodevelopmental outcome is one of the most important factors that parents of children born with congenital heart defects are concerned about once the initial anxieties about the diagnosis and operation have settled. Long-term studies have shown that developmental and functional abnormalities across multiple domains are present at school entry in these children. Among the many predictors of later developmental
Ó 2015 by The Society of Thoracic Surgeons Published by Elsevier
difficulties (microcephaly, genetic predisposition, age at operation, palliation, circulatory arrest time, abnormal postoperative neurologic features, and maternal education), very few are modifiable. Recent advances in the understanding of the fetal mechanisms of impaired cerebral blood flow in the context of congenital heart disease suggest that immediate postoperative events may be only an indicator and may not explain causality
0003-4975/$36.00 http://dx.doi.org/10.1016/j.athoracsur.2015.02.072
Ann Thorac Surg 2015;99:2124–33
MACKIE ET AL NEURODEVELOPMENTAL OUTCOME POST INFANT CHD SURGERY
examination, or both would add to the predictive validity of the 18- to 24-month Bayley-III score for late neurodevelopment. Most certainly, delayed recognition, for example at school age, is clearly too late. Early identification and intervention, right from infancy, can mitigate some of these risks, help the family to seek help, and help the community services to target interventions for these vulnerable children. Victor Tsang, FRCS Aparna Hoskote, MD Department of Cardiothoracic Surgery Great Ormond Street Hospital for Children NHS Trust Great Ormond St London WC1N 3JH, United Kingdom e-mail: [email protected]
Reference 1. Mackie AS, Vatanpour S, Alton GY, et al. Clinical outcome score predicts adverse neurodevelopmental outcome after infant heart surgery. Ann Thorac Surg 2015;99:2124–33.
CONGENITAL HEART
for neurodevelopmental morbidity. Nonetheless, modifying the neurodevelopmental outcome is an area of intense research leading to the development of dedicated cardiac neurodevelopmental programs. Understandably, it is very challenging to enroll infants in clinical trials such as randomized trials of neuroprotective strategies, track the long-term progress of particular cohorts, and wait years before one can determine the consequences of current management strategies. Hence, a postoperative score reflecting the patient’s progress in the first few days after a cardiac operation does hold promise [1]. An easy-to-measure score, one not needing complex data collection and validated against formal neurodevelopmental testing, is of practical value. However, certain caveats have to be recognized. Most intensive care units (ICU) have traditional unit-specific criteria to determine the timing of chest closure and extubation. The thresholds to drive diuresis and use renal replacement therapy differ widely. Furthermore, the presence of extracardiac comorbidities may have an impact on these decisions. Validation of this score in similar representative cardiac programs with a certain degree of standardization in ICU practices would add power to the conclusions. Furthermore, the provisions of neuroimaging, neurologic
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