Duong Tina, Krosschell Kristin J, James Meredith K, Nelson Leslie, Alfano Lindsay N, Eichinger Katy, Mazzone Elena, Rose Kristy, Lowes Linda P, Mayhew Anna, Florence Julaine, King Wendy, Senesac Claudia R, Eagle Michelle
Department of Neurology, Stanford University, Palo Alto, CA, United States.
Children's National Hospital, Washington, DC, United States.
Front Genet. 2021 Nov 10;12:735936. doi: 10.3389/fgene.2021.735936. eCollection 2021.
Critical components of successful evaluation of clinical outcome assessments (COAs) in multisite clinical trials and clinical practice are standardized training, administration, and documented reliability of scoring. Experiences of evaluators, alongside patient differences from regional standards of care, may contribute to heterogeneity in clinical center's expertise. Achieving low variability and high reliability of COA is fundamental to clinical research and to give confidence in our ability to draw rational, interpretable conclusions from the data collected. The objective of this manuscript is to provide a framework to guide the learning process for COAs for use in clinics and clinical trials to maximize reliability and validity of COAs in neuromuscular disease (NMD). This is a consensus-based guideline with contributions from fourteen leading experts in clinical outcomes and the field of clinical outcome training in NMD. This framework should guide reliable and valid assessments in NMD specialty clinics and clinical trials. This consensus aims to expedite study start up with a progressive training pathway ranging from research naïve to highly experienced clinical evaluators. This document includes recommendations for education guidelines and roles and responsibilities of key stakeholders in COA assessment and implementation to ensure quality and consistency of outcome administration across different settings.
在多中心临床试验和临床实践中,成功评估临床结局评估指标(COA)的关键要素包括标准化培训、实施以及有记录的评分可靠性。评估者的经验以及患者与地区护理标准的差异,可能会导致临床中心专业水平的异质性。实现COA的低变异性和高可靠性对于临床研究以及让我们有信心从收集的数据中得出合理、可解释的结论至关重要。本文的目的是提供一个框架,以指导在诊所和临床试验中使用COA的学习过程,从而最大限度地提高神经肌肉疾病(NMD)中COA的可靠性和有效性。这是一份基于共识的指南,由临床结局和NMD临床结局培训领域的十四位顶尖专家共同撰写。该框架应指导NMD专科诊所和临床试验中的可靠且有效的评估。这一共识旨在通过从毫无研究经验到经验丰富的临床评估者的渐进式培训路径,加快研究启动。本文档包括教育指南建议以及COA评估和实施中关键利益相关者的角色和职责,以确保不同环境下结局管理的质量和一致性。