Six-Year Neurodevelopmental Outcomes for Children With Single-Ventricle Physiology

被引:27
作者
Sananes, Renee [1 ,2 ]
Goldberg, Caren S. [3 ]
Newburger, Jane W. [4 ,5 ]
Hu, Chenwei [6 ]
Trachtenberg, Felicia [6 ]
Gaynor, J. William [7 ]
Mahle, William T. [8 ]
Miller, Thomas [9 ,10 ]
Uzark, Karen [3 ]
Mussatto, Kathleen A. [11 ]
Pizarro, Christian [14 ]
Jacobs, Jeffrey P. [15 ]
Cnota, James [16 ]
Atz, Andrew M. [17 ]
Lai, Wyman W. [18 ]
Burns, Kristin M. [19 ]
Milazzo, Angelo [20 ,21 ,22 ]
Votava-Smith, Jodie [23 ]
Brosig, Cheryl L. [12 ,13 ]
机构
[1] Univ Toronto, Hosp Sick Children, Dept Pediat, Toronto, ON, Canada
[2] Univ Toronto, Hosp Sick Children, Labatt Family Heart Ctr, Toronto, ON, Canada
[3] Univ Michigan, Dept Pediat, Med Sch, Ann Arbor, MI 48109 USA
[4] Harvard Univ, Boston Childrens Hosp, Dept Cardiol, Boston, MA 02115 USA
[5] Harvard Univ, Dept Pediat, Harvard Med Sch, Boston, MA 02115 USA
[6] New England Res Inst, 9 Galen St, Watertown, MA 02172 USA
[7] Univ Penn, Childrens Hosp Philadelphia, Perelman Sch Med, Div Pediat Cardiothorac Surg, Philadelphia, PA 19104 USA
[8] Emory Univ, Dept Pediat, Childrens Healthcare Atlanta, Atlanta, GA 30322 USA
[9] Univ Utah, Dept Pediat, Primary Childrens Hosp, Salt Lake City, UT USA
[10] Maine Med Ctr, Div Pediat Cardiol, Portland, ME 04102 USA
[11] Childrens Wisconsin, Herma Heart Inst, Dept Surg, Milwaukee, WI USA
[12] Childrens Wisconsin, Herma Heart Inst, Dept Pediat, Milwaukee, WI USA
[13] Med Coll Wisconsin, Milwaukee, WI 53226 USA
[14] Alfred I du Pont Hosp Children, Dept Surg, Nemours Cardiac Ctr, Delaware, OH USA
[15] Heart Inst Florida, St Petersburg, FL USA
[16] Cincinnati Childrens Hosp Med Ctr, Dept Pediat, Cincinnati, OH 45229 USA
[17] Med Univ South Carolina, Dept Pediat, Charleston, SC 29425 USA
[18] Childrens Hosp Orange Cty, Childrens Heart Inst, Orange, CA 92668 USA
[19] NHLBI, Bldg 10, Bethesda, MD 20892 USA
[20] Duke Univ, Sch Med, Dept Pediat, Durham, NC USA
[21] East Carolina Univ, Dept Pediat, Greenville, NC 27858 USA
[22] Wake Forest Univ, Dept Pediat, Winston Salem, NC USA
[23] Childrens Hosp Los Angeles, Dept Pediat, Los Angeles, CA 90027 USA
基金
美国国家卫生研究院;
关键词
D O I
10.1542/peds.2020-014589
中图分类号
R72 [儿科学];
学科分类号
100202 ;
摘要
We report on patterns of neurodevelopment in children with single-ventricle anomalies and the predictive value of early evaluations for behavior at age 6. OBJECTIVES: To determine if neurodevelopmental deficits in children with single-ventricle physiology change with age and early developmental scores predict 6-year outcomes. METHODS: In the Single Ventricle Reconstruction Trial, Bayley Scales of Infant Development, Second Edition, were administered at 14 months of age, and parents completed the Behavior Assessment System for Children, Second Edition (BASC-2) annually from the ages of 2 to 6 years. Scores were classified as average, at risk, or impaired. We calculated sensitivities, specificities, and positive and negative predictive values of earlier tests on 6-year outcomes. RESULTS: Of 291 eligible participants, 244 (84%) completed the BASC-2 at 6 years; more Single Ventricle Reconstruction participants than expected on the basis of normative data scored at risk or impaired on the BASC-2 Adaptive Skills Index at that evaluation (28.7% vs 15.9%; P < .001). Children with Adaptive Skills Composite scores <2 SD below the mean at the age of 6 were more likely to have had delayed development at 14 months, particularly on the Psychomotor Development Index (sensitivity of 79%). However, the positive predictive value of the 14-month Mental Development Index and Psychomotor Development Index for 6-year BASC-2 Adaptive Scores was low (44% and 36%, respectively). Adaptive Skills Composite score impairments at the age of 6 were poorly predicted by using earlier BASC-2 assessments, with low sensitivities at the ages of 3 (37%), 4 (48%), and 5 years (55%). CONCLUSIONS: Many children with hypoplastic left heart syndrome who have low adaptive skills at the age of 6 years will not be identified by screening at earlier ages. With our findings, we highlight the importance of serial evaluations for children with critical congenital heart disease throughout development.
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页数:8
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