Validity and reliability of outcome measures assessing dexterity, coordination, and upper limb strength in Autosomal Recessive Spastic Ataxia of Charlevoix-Saguenay

Publication date: Available online 17 February 2018
Source:Archives of Physical Medicine and Rehabilitation
Author(s): Cynthia Gagnon, Isabelle Lessard, Bernard Brais, Isabelle Côté, Caroline Lavoie, Matthis Synofzik, Jean Mathieu
ObjectiveTo document in adults affected by Autosomal Recessive Spastic Ataxia of Charlevoix-Saguenay (ARSACS) the intra- and interrater reliability, standard error of measurement, agreement, minimal detectable change and construct validity of the Nine-Hole Peg Test (NHPT), Standardized Finger-to-Nose Test (SFNT), and grip strength.DesignMetrological study.SettingNeuromuscular rehabilitation clinic.ParticipantsGenetically confirmed adult patients with ARSACS (n = 42, 21 females, mean age = 38.6y for intrarater reliability, and n = 33, 18 females, mean age = 40.0y for interrater reliability).InterventionsNot applicable.Main outcome measuresIntra- and interrater reliability was determined using the intraclass correlation coefficient (ICC). Construct validity was determined by assessing the capacity of the NHPT, SFNT, and grip strength to distinguish between participants based on sex, mobility stages, and age groups, and on performance on the Archimedes spiral and Alternating hand movement tests.ResultsAll three tests have shown excellent reliability (ICC = 0.90-0.98). However, the limit of agreement was influenced by the participant's performance on the NHPT, and the minimal detectable change was very different for both hands (right = 9.7 vs left = 28.0). Construct validity was confirmed for the SFNT and NHPT, but was not demonstrated for grip strength.ConclusionsGiven the metrological properties assessed in this study, the SFNT is an excellent measure to assess upper limb coordination, while the NHPT must be used with caution. The grip strength is reliable but does not seem to reflect disease severity.



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