Department of Pediatric Surgery and Intensive Care, Erasmus MC University Medical Centre - Sophia Children's Hospital.
Department of Child and Adolescent Psychiatry/Psychology, Erasmus Medical Centre - Sophia Children's Hospital, Rotterdam, The Netherlands.
J Pediatr Gastroenterol Nutr. 2021 Dec 1;73(6):695-702. doi: 10.1097/MPG.0000000000003293.
To longitudinally evaluate self-reported and proxy-reported health status (HS) and quality of life (QoL) of school-aged children born with esophageal atresia (EA).
We obtained Pediatric Quality of Life Inventory (HS) and DUX-25 (QoL) questionnaires from children born with EA between 1999 and 2011 at 8 and/or 12 years old. Children completed self-reports during neuropsychological assessments in a prospective longitudinal follow-up program. Parents filled out proxy-reports at home. Total and subscale scores were evaluated longitudinally and compared with sex-specific reference norms.
In total, 110 participants (62% boys) were included. Self-reported HS improved significantly between 8 and 12 years for both boys (mean difference [md] 4.35, effect size [ES] 0.54, P = 0.009) and girls (md 3.26, ES 0.63, P = 0.004). Proxy-reported HS tended to improve over time, while self-reported and proxy-reported QoL tended to decline. Self-reported HS at 8 years was below normal for both boys (md -5.44, ES -0.35, P < 0.001) and girls (md -7.61, ES -0.32, P < 0.001). Girls' self-reported QoL was below normal at 8 (md -5.00, ES -0.18, P = 0.019) and 12 years (md -10.50, ES -0.26, P = 0.001). Parents reported normal HS at both ages, whereas they rated the QoL of their daughters below normal at 12 years (md -10.00, ES -0.16, P = 0.022). All above results are total scores.
Self-reported and proxy-reported HS of children with EA improved between 8 and 12 years, while their QoL tended to decline. We recommend to consider HS and QoL as two separate concepts and to measure both simultaneously and longitudinally when evaluating the burden of disease.
纵向评估食管闭锁（EA）患儿的自我报告和代理报告健康状况（HS）和生活质量（QoL）。
我们从 1999 年至 2011 年出生的 EA 患儿中获得了儿科生活质量量表（HS）和 DUX-25（QoL）问卷，并在前瞻性纵向随访计划中于 8 岁和/或 12 岁时对其进行评估。儿童在神经心理学评估中完成自我报告。父母在家中填写代理报告。我们对总评分和子评分进行了纵向评估，并与性别特异性参考标准进行了比较。
共有 110 名参与者（62%为男孩）被纳入研究。男孩（平均差值[md]4.35，效应大小[ES]0.54，P=0.009）和女孩（md3.26，ES0.63，P=0.004）的自我报告 HS 均在 8 岁至 12 岁之间显著改善。代理报告的 HS 呈上升趋势，而自我报告和代理报告的 QoL 呈下降趋势。8 岁时，男孩（md-5.44，ES-0.35，P<0.001）和女孩（md-7.61，ES-0.32，P<0.001）的自我报告 HS 均低于正常值。8 岁（md-5.00，ES-0.18，P=0.019）和 12 岁（md-10.50，ES-0.26，P=0.001）时，女孩的自我报告 QoL 也低于正常值。父母在两个年龄段都报告了正常的 HS，而他们在 12 岁时则认为女儿的 QoL 低于正常值（md-10.00，ES-0.16，P=0.022）。以上所有结果均为总评分。
EA 患儿的自我报告和代理报告 HS 在 8 至 12 岁之间有所改善，而其 QoL 则呈下降趋势。我们建议将 HS 和 QoL 视为两个独立的概念，在评估疾病负担时同时进行纵向评估。
