Corticosteroids in Duchenne muscular dystrophy: impact on the motor function measure sensitivity to change and implications for clinical trials

Dev Med Child Neurol. 2018 Feb;60(2):185-191. doi: 10.1111/dmcn.13590. Epub 2017 Oct 9.

Abstract

Aim: To monitor the evolution of the motor function of ambulatory patients with Duchenne muscular dystrophy (DMD) treated by corticosteroids for 2 years in comparison with untreated patients.

Method: This observational, multicentre cohort study explores the evolution of the motor function measure (MFM) over a 24-month period for 29 ambulant corticosteroids-treated and 45 ambulant untreated patients with DMD.

Results: Significant differences were found between mean MFM scores in corticosteroids-treated and untreated groups for domain 1 of the MFM (standing position and transfers; D1), domain 2 of the MFM (axial and proximal motor function; D2), and domain 3 of the MFM (distal motor function; D3). Subscores were between 0 months and 6 months, and 0 months and 24 months. For the D1 subscore specifically, there was a significant increase in the corticosteroids-treated group (mean±standard deviation [SD] slope of change=12.6±15.5%/y), while a decrease was observed in the untreated group (-17.8±17.7%/y) between 0 months and 6 months (p<0.001). Sensitivity to change as assessed by standardized response means was high between 12 months and 24 months for D1 of both corticosteroids-treated and untreated groups (1.0 and 1.2 respectively), and low for D2 and D3 of both treated and untreated groups.

Interpretation: Patients with DMD treated by corticosteroids present a different course of the disease as assessed by MFM, confirming the sensitivity to change of the MFM in this population.

What this paper adds: Corticosteroids have a quantitative impact on muscle strength 6 to 24 months after starting treatment. Motor function measure is a valid outcome measure in Duchenne muscular dystrophy patients under corticosteroid treatment.

Publication types

  • Multicenter Study
  • Observational Study

MeSH terms

  • Adrenal Cortex Hormones / therapeutic use*
  • Child
  • Cohort Studies
  • Disability Evaluation*
  • Female
  • Humans
  • Male
  • Movement Disorders / drug therapy*
  • Movement Disorders / etiology*
  • Muscle Strength / drug effects
  • Muscular Dystrophy, Duchenne / complications*
  • Muscular Dystrophy, Duchenne / drug therapy
  • Sensitivity and Specificity
  • Severity of Illness Index
  • Statistics, Nonparametric
  • Time Factors

Substances

  • Adrenal Cortex Hormones