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J Pediatr. 2014 Sep;165(3):490-496.e8. doi: 10.1016/j.jpeds.2014.05.019. Epub 2014 Jun 19.

Factors associated with neurodevelopment for children with single ventricle lesions.

Collaborators (138)

Pearson G, Pemberton V, Kavey RE, Stylianou M, Mathis M, Mahony L, Sleeper L, Tennstedt S, Colan S, Virzi L, Connell P, Muratov V, Wruck L, Lu M, Gallagher D, Devine A, Travison T, Teitel DF, Newburger JW, Laussen P, del Nido P, Breitbart R, Levine J, McGrath E, Dunbar-Masterson C, Lai W, Printz B, Hsu D, Hellenbrand W, Williams I, Prakash A, Mosca R, Servedio D, Corda R, Korsin R, Nash M, Vetter VL, Tabbutt S, Gaynor J, Ravishankar C, Spray T, Cohen M, Nolan M, Piacentino S, DiLullo S, Mirarchi N, Benson D, Krawczeski CD, Bogenschutz L, Barnard T, Hamstra M, Griffiths R, Hogan K, Schwartz S, Nelson D, Anderson PA, Li J, Covitz W, Crawford K, Hines M, Jaggers J, Koutlas T, Sang C Jr, Sutton LJ, Xu M, Saul J, Atz A, Shirali G, Bradley S, Graham E, Atz T, Infinger P, Minich L, Hawkins J, Puchalski M, Williams R, Lambert L, Porter J, Shearrow M, McCrindle B, Kirsh J, Caldarone C, Radojewski E, Khaikin S, McIntyre S, Slater N, Goldberg CS, Ohye RG, Nowak C, Ghanayem N, Tweddell J, Mussatto K, Frommelt M, Young- Borkowski L, Lewis A, Starnes V, Pike N, Jacobs JP, Quintessenza JA, Chai PJ, Cooper DS, John J, Huhta JC, Merola T, Cox T, Kanter K, Mahle W, Bond J, French L, Huckaby J, Pizarro C, Prospero C, Simons J, Baffa G, Zeltzer I, Tortoriello T, McElroy D, Town D, Rhodes J, Fudge J, Frommelt P, Marx G, Stolle C, Artman M, Austin E, Feltes T, Johnson J, Klitzner T, Krischer J, Matherne G, Kugler J, Kavey RE, Driscoll DJ, Galantowicz M, Hunsberger SA, Knight TJ, Taylor H, Webb CL.

Author information

University of Michigan Medical School and CS Mott Children's Hospital, Ann Arbor, MI. Electronic address:
New England Research Institute, Watertown, MA.
Children's Healthcare of Atlanta and Emory University School of Medicine, Atlanta, GA.
Childrens Hospital of Philadelphia, Philadelphia, PA.
Columbia University and Morgan Stanley Children's Hospital, New York, NY.
Children's Hospital of Wisconsin and Medical College of Wisconsin, Milwaukee, WI.
University of Michigan Medical School and CS Mott Children's Hospital, Ann Arbor, MI.
Medical University of South Carolina, Charleston, SC.
University of Utah Primary Children's Medical Center, Salt Lake City, UT.
All Children's Hospital, Johns Hopkins Medicine, St Petersburg, FL.
Cincinnati Children's Hospital Medical Center, Cincinnati, OH.
Children's Hospital of Los Angeles, Los Angeles, CA.
National Heart, Lung, and Blood Institute, Bethesda, MD.
Hospital for Sick Children, Toronto, Ontario, Canada.
Alfred I. duPont Hospital for Children, Wilmington, DE.
Duke University Medical Center, Durham, NC.
Boston Children's Hospital, Boston, MA.



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.


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.


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.


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.


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