Department of General Paediatrics, Erasmus MC-Sophia Children's Hospital, Rotterdam, the Netherlands; The ENCORE Expertise Centre for Neurodevelopmental Disorders, Erasmus MC, Rotterdam, the Netherlands.
The ENCORE Expertise Centre for Neurodevelopmental Disorders, Erasmus MC, Rotterdam, the Netherlands; Department of Child and Adolescent Psychiatry/Psychology, Erasmus MC-Sophia Children's Hospital, Rotterdam, the Netherlands.
Eur J Paediatr Neurol. 2023 Jul;45:36-46. doi: 10.1016/j.ejpn.2023.05.010. Epub 2023 May 30.
This study aims to (1) investigate health-related quality of life (HRQoL) in children with Neurofibromatosis Type 1 (NF1) using the Pediatric Quality of Life inventory (PedsQL) and the Child Health Questionnaire (CHQ); and (2) compare the psychometric properties and content of these questionnaires in NF1 patients. PedsQL and CHQ proxy-reports were administered to parents/caregivers of 160 patients with NF1 aged 5-12 years. HRQoL scores were compared with Dutch population norms using independent t-tests. Psychometric properties (feasibility and reliability) were assessed by floor/ceiling effects and Cronbach's alpha coefficient. A principal component analysis (PCA) with varimax rotation was performed to identify the data's internal structure. By content mapping, we identified unique constructs of each questionnaire. Proxy-reported HRQoL was significantly lower on all PedsQL subscales for children aged 5-7 years, and on 4/6 subscales for children aged 8-12 years compared to norms. Significantly lower HRQoL was reported on 6/14 CHQ subscales (children 5-7 years) and 9/14 subscales (children 8-12 years). The PedsQL showed slightly better feasibility and reliability. The PCA identified two components, representing psychosocial and physical aspects of HRQoL, explaining 63% of total variance. Both questionnaires showed relevant loadings on both components. The CHQ subscales concerning parents and family were considered unique contributions. Proxy-reported HRQoL of children with NF1 is significantly lower compared to norms on multiple domains. Both questionnaires adequately measure HRQoL in children with NF1. However, the PedsQL has slightly better psychometric properties, while the CHQ covers a unique dimension of HRQoL associated with disease impact on parents and family.
（1）使用儿科生活质量问卷（PedsQL）和儿童健康问卷（CHQ）调查 1 型神经纤维瘤病（NF1）患儿的健康相关生活质量（HRQoL）；（2）比较这些问卷在 NF1 患者中的心理测量特性和内容。向 160 名 5-12 岁 NF1 患儿的父母/照顾者发放 PedsQL 和 CHQ 代理报告。使用独立 t 检验将 HRQoL 评分与荷兰人群正常值进行比较。通过地板/天花板效应和克朗巴赫α系数评估心理测量特性（可行性和可靠性）。采用最大方差旋转的主成分分析（PCA）来确定数据的内部结构。通过内容映射，确定了每个问卷的独特结构。与正常值相比，所有 PedsQL 子量表的 5-7 岁儿童和 8-12 岁儿童的代理报告 HRQoL 均显著降低。6/14 CHQ 子量表（5-7 岁儿童）和 9/14 子量表（8-12 岁儿童）的 HRQoL 报告显著降低。PedsQL 显示出稍好的可行性和可靠性。PCA 确定了两个分量，代表 HRQoL 的心理社会和身体方面，解释了总方差的 63%。两个问卷在两个分量上都有相关的负荷。CHQ 子量表涉及父母和家庭被认为是独特的贡献。与多个领域的正常值相比，NF1 患儿的代理报告 HRQoL 显著降低。两个问卷都能充分测量 NF1 患儿的 HRQoL。然而，PedsQL 的心理测量特性稍好，而 CHQ 则涵盖了与疾病对父母和家庭影响相关的独特 HRQoL 维度。
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