Tai Geneieve, Corben Louise A, Woodcock Ian R, Yiu Eppie M, Delatycki Martin B
Bruce Lefroy Centre for Genetic Health Research Murdoch Children's Research Institute Parkville Victoria Australia.
Department of Paediatrics University of Melbourne Parkville Victoria Australia.
Mov Disord Clin Pract. 2021 Apr 6;8(5):688-693. doi: 10.1002/mdc3.13204. eCollection 2021 Jul.
The Friedreich Ataxia Rating Scale (FARS) and the Scale for the Assessment and Rating of Ataxia (SARA) are commonly used neurological rating scales in Friedreich ataxia (FRDA). The modified Friedreich Ataxia Rating Scale (mFARS) has been accepted as an appropriate outcome measure for clinical trials in FRDA.
The COVID-19 pandemic has resulted in limited face-to-face interactions with individuals involved in natural history studies and clinical trials. The aim of this study was to determine the validity of conducting the mFARS and SARA through video.
Individuals who had the mFARS administered face-to-face in the previous 6 months were invited to participate. Participants were sent instructions and asked to have a carer present to assist. The mFARS and SARA were then administered by video. Differences between face-to-face and video scores and the reliability between scores obtained face-to-face and by video were examined.
The mFARS and SARA were conducted by video with 19 individuals. Excellent test-retest reliability was seen in the mFARS lower limb coordination (ICC = 0.96, 95% CI 0.90-0.98) and upright stability sections (ICC = 0.97, 95% CI 0.93-0.99), total mFARS (ICC = 0.97, 95% CI 0.92-0.99) and SARA scores (ICC = 0.98, 95% CI 0.95-0.99).
Excellent test-retest reliability was demonstrated in the majority of the mFARS sections, and in the total mFARS and SARA scores, suggesting that video is a valid method of conducting these scales. This method enables inclusion of participants who are unable to travel to study sites. A larger cohort will be required to further validate the use of video mFARS and SARA for future studies.
弗里德赖希共济失调评定量表（FARS）和共济失调评估与评定量表（SARA）是弗里德赖希共济失调（FRDA）中常用的神经学评定量表。改良弗里德赖希共济失调评定量表（mFARS）已被认可为FRDA临床试验的合适结局指标。
2019冠状病毒病大流行导致与参与自然史研究和临床试验的个体进行面对面互动的机会有限。本研究的目的是确定通过视频进行mFARS和SARA评定的有效性。
邀请在过去6个月内接受过面对面mFARS评定的个体参与。向参与者发送了指导说明，并要求有护理人员在场协助。然后通过视频进行mFARS和SARA评定。检查了面对面评分与视频评分之间的差异以及面对面获得的评分与视频获得的评分之间的可靠性。
对19名个体通过视频进行了mFARS和SARA评定。在mFARS下肢协调性（组内相关系数ICC = 0.96，95%置信区间CI 0.90 - 0.98）和直立稳定性部分（ICC = 0.97, 95% CI 0.93 - 0.99）、mFARS总分（ICC = 0.97, 95% CI 0.92 - 0.99）以及SARA评分（ICC = 0.98, 95% CI 0.95 - 0.99）中观察到了极好的重测信度。
在大多数mFARS部分以及mFARS总分和SARA评分中都证明了极好的重测信度，这表明视频是进行这些量表评定的有效方法。这种方法能够纳入无法前往研究地点的参与者。未来的研究需要更大的队列来进一步验证视频mFARS和SARA的使用。
