Longitudinal changes of SARA scale in Friedreich ataxia: Strong influence of baseline score and age at onset.

ABSTRACT:

Background

The Scale for Assessment and Rating of Ataxia (SARA) is widely used in different types of ataxias and has been chosen as the primary outcome measure in the European natural history study for Friedreich ataxia (FA).

Methods

To assess distribution and longitudinal changes of SARA scores and its single items, we analyzed SARA scores of 502 patients with typical-onset FA (<25 years) participating in the 4-year prospective European FA Consortium for Translational Studies (EFACTS). Pattern of disease progression was determined using linear mixed-effects regression models. The chosen statistical model was re-fitted in order to estimate parameters and predict disease progression. Median time-to-change and rate of score progression were estimated using the Kaplan-Meier met

SUBMITTER: Porcu L 

PROVIDER: S-EPMC10647003 | biostudies-literature | 2023 Nov

REPOSITORIES: biostudies-literature