Caren S Goldberg1, Minmin Lu2, Lynn A Sleeper2, William T Mahle3, J William Gaynor4, Ismee A Williams5, Kathleen A Mussatto6, Richard G Ohye7, Eric M Graham8, Deborah U Frank9, Jeffrey P Jacobs10, Catherine Krawczeski11, Linda Lambert9, Alan Lewis12, Victoria L Pemberton13, Renee Sananes14, Erica Sood15, Stephanie B Wechsler16, David C Bellinger17, Jane W Newburger17. 1. University of Michigan Medical School and CS Mott Children's Hospital, Ann Arbor, MI. Electronic address: cgoldber@umich.edu. 2. New England Research Institute, Watertown, MA. 3. Children's Healthcare of Atlanta and Emory University School of Medicine, Atlanta, GA. 4. Childrens Hospital of Philadelphia, Philadelphia, PA. 5. Columbia University and Morgan Stanley Children's Hospital, New York, NY. 6. Children's Hospital of Wisconsin and Medical College of Wisconsin, Milwaukee, WI. 7. University of Michigan Medical School and CS Mott Children's Hospital, Ann Arbor, MI. 8. Medical University of South Carolina, Charleston, SC. 9. University of Utah Primary Children's Medical Center, Salt Lake City, UT. 10. All Children's Hospital, Johns Hopkins Medicine, St Petersburg, FL. 11. Cincinnati Children's Hospital Medical Center, Cincinnati, OH. 12. Children's Hospital of Los Angeles, Los Angeles, CA. 13. National Heart, Lung, and Blood Institute, Bethesda, MD. 14. Hospital for Sick Children, Toronto, Ontario, Canada. 15. Alfred I. duPont Hospital for Children, Wilmington, DE. 16. Duke University Medical Center, Durham, NC. 17. Boston Children's Hospital, Boston, MA.
Abstract
OBJECTIVE: To measure neurodevelopment at 3 years of age in children with single right-ventricle anomalies and to assess its relationship to Norwood shunt type, neurodevelopment at 14 months of age, and patient and medical factors. STUDY DESIGN:All subjects in the Single Ventricle Reconstruction Trial who were alive without cardiac transplant were eligible for inclusion. The Ages and Stages Questionnaire (ASQ, n = 203) and other measures of behavior and quality of life were completed at age 3 years. Medical history, including measures of growth, feeding, and complications, was assessed through annual review of the records and phone interviews. The Bayley Scales of Infant Development, Second Edition (BSID-II) scores from age 14 months were also evaluated as predictors. RESULTS: Scores on each ASQ domain were significantly lower than normal (P < .001). ASQ domain scores at 3 years of age varied nonlinearly with 14-month BSID-II. More complications, abnormal growth, and evidence of feeding, vision, or hearing problems were independently associated with lower ASQ scores, although models explained <30% of variation. Type of shunt was not associated with any ASQ domain score or with behavior or quality-of-life measures. CONCLUSION:Children with single right-ventricle anomalies have impaired neurodevelopment at 3 years of age. Lower ASQ scores are associated with medical morbidity, and lower BSID-II scores but not with shunt type. Because only a modest percentage of variation in 3-year neurodevelopmental outcome could be predicted from early measures, however, all children with single right-ventricle anomalies should be followed longitudinally to improve recognition of delays.
RCT Entities:
OBJECTIVE: To measure neurodevelopment at 3 years of age in children with single right-ventricle anomalies and to assess its relationship to Norwood shunt type, neurodevelopment at 14 months of age, and patient and medical factors. STUDY DESIGN: All subjects in the Single Ventricle Reconstruction Trial who were alive without cardiac transplant were eligible for inclusion. The Ages and Stages Questionnaire (ASQ, n = 203) and other measures of behavior and quality of life were completed at age 3 years. Medical history, including measures of growth, feeding, and complications, was assessed through annual review of the records and phone interviews. The Bayley Scales of Infant Development, Second Edition (BSID-II) scores from age 14 months were also evaluated as predictors. RESULTS: Scores on each ASQ domain were significantly lower than normal (P < .001). ASQ domain scores at 3 years of age varied nonlinearly with 14-month BSID-II. More complications, abnormal growth, and evidence of feeding, vision, or hearing problems were independently associated with lower ASQ scores, although models explained <30% of variation. Type of shunt was not associated with any ASQ domain score or with behavior or quality-of-life measures. CONCLUSION:Children with single right-ventricle anomalies have impaired neurodevelopment at 3 years of age. Lower ASQ scores are associated with medical morbidity, and lower BSID-II scores but not with shunt type. Because only a modest percentage of variation in 3-year neurodevelopmental outcome could be predicted from early measures, however, all children with single right-ventricle anomalies should be followed longitudinally to improve recognition of delays.
Authors: Catherine L Dent; James P Spaeth; Blaise V Jones; Steven M Schwartz; Tracy A Glauser; Barbara Hallinan; Jeffrey M Pearl; Philip R Khoury; C Dean Kurth Journal: J Thorac Cardiovasc Surg Date: 2006-01 Impact factor: 5.209
Authors: A V Diez Roux; S S Merkin; D Arnett; L Chambless; M Massing; F J Nieto; P Sorlie; M Szklo; H A Tyroler; R L Watson Journal: N Engl J Med Date: 2001-07-12 Impact factor: 91.245
Authors: Darren H Freed; Charlene M T Robertson; Reginald S Sauve; Ari R Joffe; Ivan M Rebeyka; David B Ross; John D Dyck Journal: J Thorac Cardiovasc Surg Date: 2006-10 Impact factor: 5.209
Authors: David Wypij; Jane W Newburger; Leonard A Rappaport; Adre J duPlessis; Richard A Jonas; Gil Wernovsky; Ming Lin; David C Bellinger Journal: J Thorac Cardiovasc Surg Date: 2003-11 Impact factor: 5.209
Authors: Jane W Newburger; David Wypij; David C Bellinger; Adre J du Plessis; Karl C K Kuban; Leonard A Rappaport; Daniel Almirall; David L Wessel; Richard A Jonas; Gil Wernovsky Journal: J Pediatr Date: 2003-07 Impact factor: 4.406
Authors: John Beca; Julia K Gunn; Lee Coleman; Ayton Hope; Peter W Reed; Rodney W Hunt; Kirsten Finucane; Christian Brizard; Brieana Dance; Lara S Shekerdemian Journal: Circulation Date: 2013-01-31 Impact factor: 29.690
Authors: Kathleen A Mussatto; Danielle Hollenbeck-Pringle; Felicia Trachtenberg; Erica Sood; Renee Sananes; Nancy A Pike; Linda M Lambert; William T Mahle; David J Goldberg; Caren S Goldberg; Carolyn Dunbar-Masterson; Michelle Otto; Bradley S Marino; Bronwyn H Bartle; Ismee A Williams; Jeffrey P Jacobs; Sinai C Zyblewski; Victoria L Pemberton Journal: Cardiol Young Date: 2017-08-29 Impact factor: 1.093
Authors: Sean J Cooney; Kristen Campbell; Kelly Wolfe; Michael V DiMaria; Christopher M Rausch Journal: Pediatr Cardiol Date: 2020-11-15 Impact factor: 1.655
Authors: Jason Homsy; Samir Zaidi; Yufeng Shen; James S Ware; Kaitlin E Samocha; Konrad J Karczewski; Steven R DePalma; David McKean; Hiroko Wakimoto; Josh Gorham; Sheng Chih Jin; John Deanfield; Alessandro Giardini; George A Porter; Richard Kim; Kaya Bilguvar; Francesc López-Giráldez; Irina Tikhonova; Shrikant Mane; Angela Romano-Adesman; Hongjian Qi; Badri Vardarajan; Lijiang Ma; Mark Daly; Amy E Roberts; Mark W Russell; Seema Mital; Jane W Newburger; J William Gaynor; Roger E Breitbart; Ivan Iossifov; Michael Ronemus; Stephan J Sanders; Jonathan R Kaltman; Jonathan G Seidman; Martina Brueckner; Bruce D Gelb; Elizabeth Goldmuntz; Richard P Lifton; Christine E Seidman; Wendy K Chung Journal: Science Date: 2015-12-04 Impact factor: 47.728
Authors: Eric M Graham; Renee' H Martin; Andrew M Atz; Kasey Hamlin-Smith; Minoo N Kavarana; Scott M Bradley; Bahaaldin Alsoufi; William T Mahle; Allen D Everett Journal: J Thorac Cardiovasc Surg Date: 2019-01-23 Impact factor: 5.209
Authors: Maia M Noeder; Beth A Logan; Kari L Struemph; Nancy Condon; Isabel Mueller; Barbara Sands; Ryan R Davies; Erica Sood Journal: Cardiol Young Date: 2017-04-05 Impact factor: 1.093
Authors: Cynthia M Ortinau; Caitlin K Rollins; Ali Gholipour; Hyuk Jin Yun; Mackenzie Marshall; Borjan Gagoski; Onur Afacan; Kevin Friedman; Wayne Tworetzky; Simon K Warfield; Jane W Newburger; Terrie E Inder; P Ellen Grant; Kiho Im Journal: Cereb Cortex Date: 2019-07-22 Impact factor: 5.357
Authors: J William Gaynor; Christian Stopp; David Wypij; Dean B Andropoulos; Joseph Atallah; Andrew M Atz; John Beca; Mary T Donofrio; Kim Duncan; Nancy S Ghanayem; Caren S Goldberg; Hedwig Hövels-Gürich; Fukiko Ichida; Jeffrey P Jacobs; Robert Justo; Beatrice Latal; Jennifer S Li; William T Mahle; Patrick S McQuillen; Shaji C Menon; Victoria L Pemberton; Nancy A Pike; Christian Pizarro; Lara S Shekerdemian; Anne Synnes; Ismee Williams; David C Bellinger; Jane W Newburger Journal: Pediatrics Date: 2015-05 Impact factor: 7.124