Division of Cardiology, Children's Hospital of Philadelphia, Philadelphia, PA, 19104, USA.
Department of Pediatrics, Children's Hospital of Philadelphia, Philadelphia, PA, USA.
Pediatr Cardiol. 2021 Mar;42(3):643-653. doi: 10.1007/s00246-020-02525-6. Epub 2021 Feb 3.
Neurodevelopmental sequelae are prevalent among patients with congenital heart defects (CHD). In a study of infants and children with repaired tetralogy of Fallot (TOF), we sought to identify those at risk for abnormal neurodevelopment and to test associations between socioeconomic and medical factors with neurodevelopment deficits. Single-center retrospective observational study of patients with repaired TOF that were evaluated at the institution's Cardiac Kids Developmental Follow-up Program (CKDP) between 2012 and 2018. Main outcomes included neurodevelopmental test scores from the Bayley Infant Neurodevelopmental Screener (BINS), Peabody Developmental Motor Scale (PDMS), and Bayley Scales of Infant and Toddler Development, Third Edition (Bayley-III). Mixed effects linear regression and marginal logistic regression models tested relationships between patient characteristics and outcomes. Sub-analyses were conducted to test correlations between initial and later neurodevelopment tests. In total, 49 patients were included, predominantly male (n = 33) and white (n = 28), first evaluated at a median age of 4.5 months. Forty-three percent of patients (n = 16) had deficits in the BINS, the earliest screening test. Several socioeconomic parameters and measures of disease complexity were associated with neurodevelopment, independently of genetic syndrome. Early BINS and PDMS performed in infancy were associated with Bayley-III scores performed after 1 year of age. Early screening identifies TOF patients at risk for abnormal neurodevelopment. Socioeconomic factors and disease complexity are associated with abnormal neurodevelopment and should be taken into account in the risk stratification and follow-up of these patients. Early evaluation with BINS and PDMS is suggested for detection of early deficits.
神经发育后遗症在先天性心脏病 (CHD) 患者中较为常见。在一项对法洛四联症 (TOF) 修复后的婴儿和儿童的研究中，我们试图确定那些有神经发育异常风险的患者，并检验社会经济和医疗因素与神经发育缺陷之间的关联。这是一项单中心回顾性观察研究，研究对象为 2012 年至 2018 年在该机构的心脏宝贝发育随访计划 (CKDP) 接受评估的修复后 TOF 患者。主要结局包括来自贝利婴幼儿神经发育筛查量表 (BINS)、佩皮发育运动量表 (PDMS) 和贝利婴幼儿发展量表第三版 (Bayley-III) 的神经发育测试分数。混合效应线性回归和边缘逻辑回归模型检验了患者特征与结局之间的关系。进行了亚分析以检验初始和后续神经发育测试之间的相关性。共有 49 名患者被纳入研究，其中男性居多 (n = 33)，白人为主 (n = 28)，中位首次评估年龄为 4.5 个月。43%的患者 (n = 16) 在最早的筛查测试 BINS 中存在缺陷。几种社会经济参数和疾病复杂性指标与神经发育相关，独立于遗传综合征。婴儿期进行的早期 BINS 和 PDMS 与 1 岁后进行的 Bayley-III 评分相关。早期筛查可识别出有神经发育异常风险的 TOF 患者。社会经济因素和疾病复杂性与神经发育异常相关，应在这些患者的风险分层和随访中考虑到这些因素。建议早期使用 BINS 和 PDMS 进行评估，以发现早期缺陷。
