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Developmental outcomes of children with congenital diaphragmatic hernia: A multicenter prospective study
Authors:Julia Wynn  Gudrun Aspelund  Annette Zygmunt  Charles J.H. Stolar  George Mychaliska  Jennifer Butcher  Foong-Yen Lim  Teresa Gratton  Douglas Potoka  Kate Brennan  Ken Azarow  Barbara Jackson  Howard Needelman  Timothy Crombleholme  Yuan Zhang  Jimmy Duong  Marc S. Arkovitz  Wendy K. Chung  Christiana Farkouh
Affiliation:1. Department of Pediatrics, Columbia University, New York, NY 10032, USA;2. Division of Pediatric Surgery, Department of Surgery, Columbia University, New York, NY, USA;3. Division of Pediatric Surgery, Department of Surgery, University of Michigan School of Medicine, Ann Arbor, MI, USA;4. Division of Child Behavioral Health, Department of Pediatrics and Communicable Diseases, University of Michigan School of Medicine, Ann Arbor, MI, USA;5. Division of Pediatric General, Thoracic, and Fetal Surgery, Center for Molecular Fetal Therapy, Cincinnati Children''s Hospital Medical Center, and Cincinnati, OH, USA;6. University of Cincinnati, Department of Pediatrics, Cincinnati, OH, USA;g Cincinnati Children''s Hospital Medical Center, Department of Pediatrics, Division of Neonatology and Pulmonary Biology, Cincinnati, OH, USA;h Division of Pediatric Surgery, Department of Surgery, University of Pittsburgh School of Medicine, Pittsburgh, PA, USA;i Department of Instruction and Learning-Early Intervention Program, University of Pittsburgh, Pittsburgh, PA, USA;j Division of Pediatric Surgery, Department of Surgery, University of Nebraska College of Medicine/Children''s Hospital and Medical Center, Omaha, NE, USA;k Department of Education, Munroe-Meyer Institute, University of Nebraska Medical Center Omaha, NE, USA;l Section of Developmental/Behavioral Pediatrics, Munroe-Meyer Institute, University of Nebraska Medical Center Omaha, NE, USA;m Colorado Fetal Care Center, Division of Pediatric General, Thoracic, and Fetal Surgery, Children''s Hospital Colorado and the University of Colorado School of Medicine, Denver, CO, USA;n Department of Biostatistics, Mailman School of Public Health, Columbia University, New York, NY, USA;o Department of Pediatric Surgery, Tel Hashomer Medical Center, Tel Aviv, Israel
Abstract:

Purpose

To determine developmental outcomes and associated factors in patients with congenital diaphragmatic hernia (CDH) at 2 years of age.

Methods

This is a multicenter prospective study of a CDH birth cohort. Clinical and socioeconomic data were collected. Bayley Scales of Infant Development (BSID-III) and Vineland Adaptive Behavior Scales (VABS-II) were performed at 2 years of age.

Results

BSID-III and VABS-II assessments were completed on 48 and 49 children, respectively. The BSID-III mean cognitive, language, and motor scores were significantly below the norm mean with average scores of 93 ± 15, 95 ± 16, and 95 ± 11. Ten percent (5/47) scored more than 2 standard deviations below the norm on one or more domains. VABS-II scores were similar to BSID-III scores with mean communication, daily living skills, social, motor, adaptive behavior scores of 97 ± 14, 94 ± 16, 93 ± 13, 97 ± 10, and 94 ± 14. For the BSID-III, supplemental oxygen at 28 days, a prenatal diagnosis, need for extracorporeal membrane oxygenation (ECMO) and exclusive tube feeds at time of discharge were associated with lower scores. At 2 years of age, history of hospital readmission and need for tube feeds were associated with lower scores. Lower socioeconomic status correlated with lower developmental scores when adjusted for significant health factors.

Conclusion

CDH patients on average have lower developmental scores at 2 years of age compared to the norm. A need for ECMO, oxygen at 28 days of life, ongoing health issues and lower socioeconomic status are factors associated with developmental delays.
Keywords:BSID-III, Bayley Scales of Infant Development third edition   CI, confidence interval   CDH, congenital diaphragmatic hernia   DHREAMS, Diaphragmatic Hernia Research &   Exploration, Advancing Molecular Science   ECMO, extracorporeal membrane oxygenation   HI, Hollingshead Four Factor Index of socioeconomic status   REDCap, Research Electronic Data Capture   SD, standard deviation   SES, socioeconomic status   VABS-II, Vineland Adaptive Behavior Scales second edition
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