School of Health and Related Research (ScHARR), University of Sheffield, Regent Court, 30 Regent Street, Sheffield, S1 4DA, UK.
Health Qual Life Outcomes. 2022 Apr 2;20(1):57. doi: 10.1186/s12955-022-01964-4.
Duchenne muscular dystrophy is a rare, progressive, life-limiting genetic neuromuscular condition that significantly impacts the quality of life of informal caregivers. Carer quality of life is measured using heterogeneous self-report scales, yet their suitability for Duchenne remains unclear. This review aimed to identify and evaluate the reliability and validity of quality of life instruments in Duchenne carers.
Systematic searches were conducted in Embase, MEDLINE, CINAHL, PsycINFO, Cochrane Library and Google Scholar. Full research articles reporting data on multiple-item self-report quality of life instruments in informal Duchenne carers were included. Extracted evidence was qualitatively synthesised and evaluated, including risk of bias, against the Consensus-based Standards for the selection of health Measurement Instruments. Duchenne carer collaborators (N = 17) helped rate the instruments' content validity.
Thirty-one articles featuring thirty-two quality of life instruments were included. Content validity was rated as "inconsistent" based on very low quality evidence. For Duchenne carer collaborators, the best instrument was PedsQL Family Impact Module. Only one instrument had evidence for structural validity (rated "unsatisfactory") and measurement invariance (rated "satisfactory"). Instruments received "satisfactory" ratings for internal consistency and mixed ratings for construct validity and responsiveness. There was no evidence for reliability, measurement error, or criterion validity.
Instruments used to measure Duchenne carer quality of life have limited and often inconsistent supportive psychometric evidence. Further work must investigate instruments' measurement properties in Duchenne carers and/or the development of new tools. In the interim, we recommend considering the PedsQL Family Impact Module based on Duchenne carer ratings.
杜氏肌营养不良症是一种罕见的、进行性的、危及生命的遗传性神经肌肉疾病，严重影响非专业护理人员的生活质量。护理人员的生活质量是通过使用各种自评量表来衡量的，但这些量表在杜氏肌营养不良症中的适用性尚不清楚。本综述旨在确定和评估杜氏肌营养不良症护理人员生活质量量表的信度和效度。
在 Embase、MEDLINE、CINAHL、PsycINFO、Cochrane 图书馆和 Google Scholar 中进行了系统搜索。纳入了报告非专业杜氏肌营养不良症护理人员使用的多项目自评生活质量量表数据的全文研究文章。提取的证据进行了定性综合评估，包括对基于共识的健康测量仪器选择标准的偏倚风险评估。杜氏肌营养不良症护理人员（N=17）帮助评估了这些工具的内容效度。
共纳入 31 篇文章，涉及 32 种生活质量量表。基于极低质量证据，内容效度被评为“不一致”。对于杜氏肌营养不良症护理人员，最佳的工具是 PedsQL 家庭影响模块。只有一种工具具有结构有效性（评为“不满意”）和测量不变性（评为“满意”）的证据。量表在内部一致性方面获得了“满意”的评分，在结构有效性和反应度方面获得了混合评分。没有关于可靠性、测量误差或效标效度的证据。
用于测量杜氏肌营养不良症护理人员生活质量的工具的心理测量学证据有限且往往不一致。必须进一步研究这些工具在杜氏肌营养不良症护理人员中的测量特性，或者开发新的工具。在这期间，我们建议根据杜氏肌营养不良症护理人员的评分，考虑使用 PedsQL 家庭影响模块。
