Isokinetic muscle strength differences in patients with mucopolysaccharidosis I, II, and VI

J Pediatr Rehabil Med. 2014;7(4):353-60. doi: 10.3233/PRM-140305.

Abstract

Purpose: To determine muscular strength differences in patients with MPS-I, II, and VI versus age- and sex-matched healthy controls.

Methods: Dominant leg isokinetic knee extension strength was measured at 90 and 120 degrees per second (d/s) using a dynamometer in 30 subjects with MPS and 42 controls (5-16 yrs). MPS-I was further divided into MPS-IA (attenuated) and MPS-IH (severe). Strength measures analyzed were peak torque (PkT), peak torque per unit body weight (PkT/BW) and per unit lean body mass (PkT/LBM), and average power (AP).

Results: Following adjusting strength measures for age, MPS-IH and MPS-II had significantly lower strength measures for all variables at both angular velocities. MPS-VI had significantly lower PkT, PkT/LBM, and AP compared to controls at 90 and 120d/s. In contrast, MPS-IA was not significantly different from controls for any strength variable at either angular velocity.

Conclusion: The results of this study suggest that decrements in skeletal muscle strength depend on MPS diagnosis and severity of disease. Children with MPS-IH demonstrate the greatest difference in muscular strength compared to healthy controls.

Keywords: Mucopolysaccharidosis; isokinetic; strength.

Publication types

  • Research Support, N.I.H., Extramural
  • Research Support, Non-U.S. Gov't

MeSH terms

  • Adolescent
  • Case-Control Studies
  • Child
  • Child, Preschool
  • Cohort Studies
  • Female
  • Humans
  • Knee Joint / physiology
  • Knee Joint / physiopathology
  • Male
  • Mucopolysaccharidosis I / diagnosis
  • Mucopolysaccharidosis I / physiopathology*
  • Mucopolysaccharidosis II / diagnosis
  • Mucopolysaccharidosis II / physiopathology*
  • Mucopolysaccharidosis VI / diagnosis
  • Mucopolysaccharidosis VI / physiopathology*
  • Muscle Strength* / physiology
  • Muscle, Skeletal / physiology
  • Muscle, Skeletal / physiopathology*
  • Severity of Illness Index
  • Torque