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Comparing Parent Perception of Neurodevelopment after Primary versus Staged Repair of Neonatal Symptomatic Tetralogy of Fallot - 11/12/24

Doi : 10.1016/j.jpeds.2024.114357 
Jeffrey D. Zampi, MD 1, , Dawn L. Ilardi, PhD, ABPP-CN 2, 3, Courtney E. McCracken, PhD 4, Yun Zhang, PhD 5, Andrew C. Glatz, MD, MSCE 6, Bryan H. Goldstein, MD 7, Christopher J. Petit, MD 5, Athar M. Qureshi, MD 8, Caren S. Goldberg, MD, MS 1, Mark A. Law, MD 9, Jeffery J. Meadows, MD 10, Shabana Shahanavaz, MD 11, Sarosh P. Batlivala, MD, MSCI 11, Shiraz A. Maskatia, MD 12, Michael L. O'Byrne, MD, MSCE 13, R. Allen Ligon, MD 3, Joelle A. Pettus, MPH, MS 3, Asaad Beshish, MD 3, Jennifer C. Romano, MD 14, Kathryn O. Stack, MD 13, Hala Q. Khan, MD 8, Shalin Parekh, MD 10, George T. Nicholson, MD 15
1 Division of Cardiology, Department of Pediatrics, C.S. Mott Children's Hospital, University of Michigan School of Medicine, Ann Arbor, MI 
2 Pediatric Neurodevelopmental Center, Atlanta, GA 
3 Children's Heart Center Cardiology, Department of Pediatrics, Children's Healthcare of Atlanta, Emory University School of Medicine, Atlanta, GA 
4 Center for Research and Evaluation, Kaiser Permanente, Atlanta, GA 
5 Morgan Stanley Children's Hospital, Columbia University Vagelos College of Physicians & Surgeons, New York, NY 
6 Division of Cardiology, Department of Pediatrics, Washington University, St Louis, MO 
7 Department of Pediatrics, Heart Institute, UPMC Children's Hospital of Pittsburgh, University of Pittsburgh School of Medicine, Pittsburgh, PA 
8 Lillie Frank Abercrombie Division of Cardiology, Department of Pediatrics, Texas Children's Hospital, Baylor College of Medicine, Houston, TX 
9 Division of Pediatric Cardiology, Department of Pediatrics, Children's of Alabama, University of Alabama Birmingham School of Medicine, Birmingham, AL 
10 Division of Cardiology, Department of Pediatrics, University of California San Francisco School of Medicine, San Francisco, CA 
11 The Heart Institute, Cincinnati Children's Hospital Medical Center and Division of Pediatric Cardiology, University of Cincinnati College of Medicine, Cincinnati, OH 
12 Division of Pediatric Cardiology, Department of Pediatrics, Stanford University School of Medicine, Stanford, CA 
13 The Cardiac Center at The Children's Hospital of Philadelphia, Philadelphia, PA 
14 Division of Congenital Cardiothoracic Surgery, Department of Cardiothoracic Surgery, C.S. Mott Children's Hospital, University of Michigan School of Medicine, Ann Arbor, MI 
15 Division of Cardiology, Department of Pediatrics, Vanderbilt University School of Medicine, Nashville, TN 

Reprint requests: Jeffrey D. Zampi, MD, Associate Professor, Pediatric Cardiology University of Michigan Congenital Heart Center, C.S. Mott Children's Hospital Floor 11, Room 715Z, 1540 E. Hospital Dr, Ann Arbor, MI 48109-4204.Associate ProfessorPediatric Cardiology University of Michigan Congenital Heart CenterC.S. Mott Children's Hospital Floor 11Room 715Z1540 E. Hospital DrAnn ArborMI48109-4204

Abstract

Objective

To assess the association between primary and staged repair of neonatal symptomatic tetralogy of Fallot (sTOF) and neurodevelopmental outcomes in preschool through school-age children.

Study design

Multicenter cohort (n = 9 sites) study of patients with sTOF who underwent neonatal intervention between 2005 and 2017. The neurodevelopmental outcomes measures included caregivers’ ratings of executive function with the Behavior Rating Inventory of Executive Function, and psychosocial functioning with the Behavior Assessment System for Children – third Edition (BASC-3). Results were compared with normative data and by treatment strategy (primary repair vs staged repair). A parent survey assessed history of disabilities and access to services related to neurodevelopment.

Results

Although the majority of patients (median age 8.3 years, IQR 5.7-11.2) had median Behavior Rating Inventory of Executive Function and BASC-3 scores within the normal range, a proportion had clinically elevated (abnormal) scores, especially in the school-age patient subgroup (Behavior Rating Inventory of Executive Function 24%-30% and BASC 20%-37%). There were no statistically significant differences based on treatment strategy for either the Behavior Rating Inventory of Executive Function or BASC-3. However, lower birth weight, genetic syndrome, and medical complexity were significantly associated with worse executive function, and lower maternal education was associated in school-age children with lower executive and psychosocial functioning. Ongoing disabilities were relatively common (learning disability 35%, speech delay 33%, developmental delay 31%), although up to 50% of children were not receiving educational or developmental services.

Conclusions

Elevated executive and psychosocial concerns are present in the patient population with sTOF. Although initial treatment strategy appears unrelated to neurodevelopmental outcomes, lower birth weight, genetic syndrome, and medical complexity and lower maternal education are risk factors. Early recognition of neurodevelopmental concerns can facilitate access to appropriate neurodevelopmental services in this high-risk group.

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Keywords : congenital heart disease, tetralogy of Fallot, neurodevelopment, executive functioning, psychosocial functioning

Abbreviations : ADHD, AS, BASC-3, PR, RI, SR, sTOF, TOF


Plan


 Ethical Standards: A single institutional review board was utilized for this multicenter study. Informed consent was indicated by return of completed study materials.


© 2024  Elsevier Inc. Tous droits réservés.
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