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Journal of Autism and Developmental Disorders

, Volume 48, Issue 6, pp 2112–2121 | Cite as

Rate and Risk Factors Associated with Autism Spectrum Disorder in Congenital Diaphragmatic Hernia

  • Enrico Danzer
  • Casey Hoffman
  • Jo Ann D’Agostino
  • Judith S. Miller
  • Lindsay N. Waqar
  • Marsha Gerdes
  • Judy C. Bernbaum
  • Hannah Rosenthal
  • Natalie E. Rintoul
  • Lisa M. Herkert
  • William H. Peranteau
  • Alan W. Flake
  • N. Scott Adzick
  • Holly L. Hedrick
Original Paper

Abstract

To determine the rate and predictors of autism spectrum disorder (ASD) in congenital diaphragmatic hernia (CDH). Between 06/2004 and 09/2015 a total of 110 CDH survivors underwent neurodevelopmental (ND) testing and screening for ASD, followed by a full autism diagnostic evaluation if indicated at our institution. We found a 9 time higher rate of ASD in CDH children compared to the general population (P = 0.0002). Multiple patient-related and clinical variables risk factors of ASD were identified by univariate analysis. However, only short-term and long-term neurodevelopmental delays were strongly associated with ASD in CDH by multivariate comparisons. There is a striking prevalence of ASD in CDH survivors and our findings suggest that all CDH children should be regularly screened for ASD.

Keywords

Congenital diaphragmatic hernia Autism spectrum disorder Autism diagnostic observation schedule Neurodevelopmental delay Pulmonary hypoplasia Pulmonary hypertension 

Notes

Acknowledgments

We greatly appreciate the statistical advice and analysis provided by Mark S. Cary, PhD, Sr. Staff Biostatistician, Biostatistics Analysis Center, Center for Clinical Epidemiology and Biostatistics, The Perelman School of Medicine at the University of Pennsylvania. We also thank Norma Rendon for superb administrative assistance. This study has been in part be presented as poster at the 2016 Annual Meeting of the American Pediatric Surgery Association in San Diego, California, USA.

Author Contributions

ED, CH, JSM, HLH—study design; ED, CH, JA, LNW, MG, JCB, HR, NER, LMH, WHP—data collection and analysis; ED, CH—writing the first draft; ED, CH, JA, JSM, LNW, MG, JCB, HR, NER, WHP, AWF, NSA, HLH—criticial review of manuscript and approval of final manuscript version.

Compliance with Ethical Standards

Conflict of interest

None of the authors have any conflict of interest to declare.

Ethical Approval

All procedures performed in studies involving human subjects were in accordance with the ethical standards of the institutional and/or national research committee and with the 1964 Helsinki declaration and its later amendments or comparable ethical standards.

Informed Consent

Informed consent was obtained from all individual participants (parents) included in the study.

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Copyright information

© Springer Science+Business Media, LLC, part of Springer Nature 2018

Authors and Affiliations

  • Enrico Danzer
    • 1
  • Casey Hoffman
    • 1
  • Jo Ann D’Agostino
    • 1
  • Judith S. Miller
    • 2
  • Lindsay N. Waqar
    • 1
  • Marsha Gerdes
    • 1
  • Judy C. Bernbaum
    • 1
  • Hannah Rosenthal
    • 1
  • Natalie E. Rintoul
    • 1
  • Lisa M. Herkert
    • 1
  • William H. Peranteau
    • 1
  • Alan W. Flake
    • 1
  • N. Scott Adzick
    • 1
  • Holly L. Hedrick
    • 1
  1. 1.The Center for Fetal Diagnosis and TreatmentThe Children’s Hospital of PhiladelphiaPhiladelphiaUSA
  2. 2.Center for Autism ResearchThe Children’s Hospital of PhiladelphiaPhiladelphiaUSA

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