Comparing Parent Perception of Neurodevelopment after Primary versus Staged Repair of Neonatal Symptomatic Tetralogy of Fallot

IF 3.9 2区医学 Q1 PEDIATRICS

Journal of Pediatrics Pub Date : 2024-10-16 DOI:10.1016/j.jpeds.2024.114357

Jeffrey D. Zampi MD , Dawn L. Ilardi PhD, ABPP-CN , Courtney E. McCracken PhD , Yun Zhang PhD , Andrew C. Glatz MD, MSCE , Bryan H. Goldstein MD , Christopher J. Petit MD , Athar M. Qureshi MD , Caren S. Goldberg MD, MS , Mark A. Law MD , Jeffery J. Meadows MD , Shabana Shahanavaz MD , Sarosh P. Batlivala MD, MSCI , Shiraz A. Maskatia MD , Michael L. O'Byrne MD, MSCE , R. Allen Ligon MD , Joelle A. Pettus MPH, MS , Asaad Beshish MD , Jennifer C. Romano MD , Kathryn O. Stack MD , George T. Nicholson MD

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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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比较新生儿症状性法洛氏四联症初次修复与分期修复后家长对神经发育的看法。

目的评估新生儿无症状法洛氏四联症（sTOF）的初次修复（PR）和分期修复（SR）与学龄前至学龄期儿童神经发育结果之间的关联：研究设计：对2005年至2017年间接受新生儿干预的法洛氏四联症患者进行多中心队列研究（n=9个地点）。神经发育结果测量包括护理人员使用执行功能行为评分量表（BRIEF）对执行功能的评分，以及使用儿童行为评估系统--第三版（BASC-3）对社会心理功能的评分。研究结果与常模数据以及治疗策略（PR 与 SR）进行了比较。一项家长调查对残疾史和获得神经发育相关服务的情况进行了评估：虽然大多数患者（中位年龄为 8.3 岁，四分位数间距为 5.7-11.2 岁）的 BRIEF 和 BASC-3 中位分数在正常范围内，但仍有一部分患者的分数出现临床升高（异常），尤其是学龄患者亚组（BRIEF 24-30% 和 BASC 20-37%）。无论是 BRIEF 还是 BASC-3，治疗策略的不同在统计学上都没有显著差异。然而，较低的出生体重、遗传综合征和医疗复杂性与较差的执行功能显著相关，而在学龄儿童中，较低的母亲教育程度与较差的执行功能和社会心理功能相关。尽管多达50%的儿童没有接受教育或发育服务，但持续性残疾相对常见（学习障碍35%、语言发育迟缓33%、发育迟缓31%）：结论：在 sTOF 患者群体中，存在着较高的执行力和社会心理问题。尽管最初的治疗策略似乎与神经发育结果无关，但出生体重较轻、遗传综合征、医疗复杂性以及产妇受教育程度较低都是风险因素。及早发现神经发育方面的问题可以帮助这类高危人群获得适当的神经发育服务。

本文章由计算机程序翻译，如有差异，请以英文原文为准。

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来源期刊

Journal of Pediatrics 医学-小儿科

CiteScore

6.00

自引率

2.00%

发文量

696

审稿时长

31 days

期刊介绍： The Journal of Pediatrics is an international peer-reviewed journal that advances pediatric research and serves as a practical guide for pediatricians who manage health and diagnose and treat disorders in infants, children, and adolescents. The Journal publishes original work based on standards of excellence and expert review. The Journal seeks to publish high quality original articles that are immediately applicable to practice (basic science, translational research, evidence-based medicine), brief clinical and laboratory case reports, medical progress, expert commentary, grand rounds, insightful editorials, “classic” physical examinations, and novel insights into clinical and academic pediatric medicine related to every aspect of child health. Published monthly since 1932, The Journal of Pediatrics continues to promote the latest developments in pediatric medicine, child health, policy, and advocacy. Topics covered in The Journal of Pediatrics include, but are not limited to: General Pediatrics Pediatric Subspecialties Adolescent Medicine Allergy and Immunology Cardiology Critical Care Medicine Developmental-Behavioral Medicine Endocrinology Gastroenterology Hematology-Oncology Infectious Diseases Neonatal-Perinatal Medicine Nephrology Neurology Emergency Medicine Pulmonology Rheumatology Genetics Ethics Health Service Research Pediatric Hospitalist Medicine.