Genetics and Genome Biology (GGB) Program, The Hospital for Sick Children Research Institute, Toronto, Ontario, Canada.
Department of Ophthalmology and Vision Sciences, The Hospital for Sick Children, University of Toronto, Toronto, Ontario, Canada.
Ophthalmic Genet. 2023 Jun;44(3):218-225. doi: 10.1080/13816810.2023.2179074. Epub 2023 Feb 22.
To determine the validity of the validate the adult patient-reported outcome measure tools, the Michigan Retinal Degeneration Questionnaire (MRDQ) and Michigan Vision-Related Anxiety Questionnaire (MVAQ), in adolescent patients with inherited retinal diseases (IRDs).
Ninety-one adolescent patients diagnosed with IRDs were recruited at the Hospital for Sick Children (University of Toronto) and the Kellogg Eye Center (University of Michigan). The patients were administered the MRDQ, MVAQ, and Patient Health Questionnaire-4 (PHQ-4). Test-retest variability was assessed in eighteen patients within 14 days of the initial administration. Adolescent responses were analyzed for validity and reliability. As a further validation step, comparisons were made to adult data from the original MRDQ and MVAQ studies to ensure consistency in response ranges.
The existing MRDQ and MVAQ content and format could accurately detect the impact of IRD on activities of daily living in adolescents with IRDs. No floor/ceiling effects were identified, test-retest reliability was established (r = 0.73-0.86), and no items were excluded after differential item functioning analysis. Domain and trait associations with visual acuity and IRD phenotypes were similar between adolescents and adults.
The MRDQ and MVAQ are psychometrically validated questionnaires for which we have shown validity for use in adolescent patients with IRDs.
为了确定密歇根视网膜变性问卷（MRDQ）和密歇根视觉相关焦虑问卷（MVAQ）这两种成人患者报告结局测量工具在青少年遗传性视网膜疾病（IRD）患者中的有效性。
在多伦多大学 SickKids 医院和密歇根大学 Kellogg 眼科中心招募了 91 名被诊断患有 IRD 的青少年患者。患者接受了 MRDQ、MVAQ 和患者健康问卷-4（PHQ-4）的测试。在初始管理后 14 天内，对 18 名患者进行了复测以评估测试-重测的变异性。对青少年的反应进行了有效性和可靠性分析。作为进一步的验证步骤，将与原始 MRDQ 和 MVAQ 研究中的成人数据进行比较，以确保反应范围的一致性。
现有的 MRDQ 和 MVAQ 内容和格式可以准确地检测到青少年遗传性视网膜疾病患者的日常生活活动受到疾病的影响。没有发现地板/天花板效应，建立了测试-重测的可靠性（r=0.73-0.86），并且在进行差异项目功能分析后没有删除任何项目。在青少年和成人中，与视力和 IRD 表型相关的领域和特质关联相似。
MRDQ 和 MVAQ 是具有心理计量学有效性的问卷，我们已经证明了它们在青少年遗传性视网膜疾病患者中的有效性。
