Neurophysiologic assessment of mucopolysaccharidosis III

Clin Neurophysiol. 2006 Sep;117(9):2059-63. doi: 10.1016/j.clinph.2006.05.031. Epub 2006 Aug 1.

Abstract

Objective: To describe finding of various neurophysiologic tests in patients with mucopolysaccharidosis III (MPS III) early in the disease course.

Methods: Patients were evaluated with flash visual evoked potentials (VEP), brainstem auditory evoked potentials (BAEP), electroencephalography (EEG), and nerve conduction studies (NCS) before they underwent hematopoietic stem cell transplantation (HSCT).

Results: Thirteen children underwent at least one neurophysiologic test before HSCT. The mean age at testing was 2.7 years. Ten of 11 (91%) patients had a normal flash VEP, and all 9 who had BAEP had normal central conduction. EEG was normal in 7/13 (54%), with the others showing diffuse slowing. NCS was normal in 10/11 (91%) patients.

Conclusions: Despite extensive central nervous system involvement in MPS III, flash VEP and BAEP are almost always normal. EEG is often abnormal early in the disease.

Significance: This is the first report of neurophysiologic tests in a large series of MPS III patients.

Publication types

  • Comparative Study

MeSH terms

  • Child, Preschool
  • Electroencephalography / methods
  • Evoked Potentials, Auditory, Brain Stem / physiology
  • Evoked Potentials, Visual / physiology
  • Female
  • Humans
  • Infant
  • Male
  • Mucopolysaccharidosis II / physiopathology*
  • Neural Conduction / physiology
  • Neurophysiology / methods*
  • Retrospective Studies