Are novel outcome measures for Charcot-Marie-Tooth disease sensitive to change? The 6-minute walk test and StepWatch™ Activity Monitor in a 12-month longitudinal study

Luca Padua, Costanza Pazzaglia, Daniele Coraci, Davide Pareyson, Angelo Schenone, Alessia Aiello, Gian Maria Fabrizi, Tiziana Cavallaro, Lucio Santoro, Fiore Manganelli, Franco Gemignani, Francesca Vitetta, Aldo Quattrone, Anna Mazzeo, Massimo Russo, Giuseppe Vita, L. Gentile, S. Messina, C. Stancanelli, C. IacovelliD. Figliolia, S. Silipo, I. Cabrini, C. Pisciotta, S. Tozza, M. Contini

Risultato della ricerca: Contributo in rivistaArticolo in rivista

2 Citazioni (Scopus)

Abstract

Charcot-Marie-Tooth (CMT) is the most common inherited neuropathy, yet has no available pharmacological therapy. Past pharmacotherapy trials failed to provide positive results, possibly due to a poor choice of outcome measures. We previously performed a study in which we validated the 6-minute walk test and StepWatch™ Activity Monitor in CMT. The aim of the current study was to determine if these outcome measures are sensitive to change over a 12-month period. In this longitudinal multicenter study, 149 out of 169 initially enrolled patients were re-evaluated after 12 months using the 6-minute walk test, StepWatch™ Activity Monitor and other outcome measures commonly adopted in CMT disease. Statistical analysis showed a worsening of the CMT-Neuropathy Score (p < 0.05), strength of distal muscles measured by myometry (p < 0.05) and StepWatch™ Activity Monitor outputs (p < 0.05). The 10 meter walking test (p > 0.05), muscular strength as detected by clinical evaluation (p > 0.05), 6-minute walk test (p > 0.05), pain (p > 0.05) and quality of life (p > 0.05) showed no change. In the current study, patients showed clinical worsening over 12 months, confirmed by a reduction of activity as detected by StepWatch™ Activity Monitor. The 6-minute walk test failed to detect change.
Lingua originaleEnglish
pagine (da-a)1-7
Numero di pagine7
RivistaNeuromuscular Disorders
DOI
Stato di pubblicazionePubblicato - 2019

Keywords

  • Charcot–Marie–Tooth
  • Outcome measures
  • Rehabilitation
  • Sensitivity to changes

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