Trihexyphenidyl for acute life-threatening episodes due to a dystonic movement disorder in Rett syndrome

Mov Disord. 2010 Feb 15;25(3):385-9. doi: 10.1002/mds.22926.

Abstract

In Rett syndrome (RS), acute life-threatening episodes (ALTEs) are usually attributed to epilepsy or autonomic dysfunction but they can represent a movement disorder (MD). We describe three girls with RS who experienced ALTEs from an early age. These were long considered epileptic until video-EEG in Patients 1 and 3 revealed their non-epileptic nature. A primary dystonic mechanism was suspected and Patients 1 and 2 were treated with Trihexyphenidyl with significantly reduced frequency of the ALTEs. Patient 3 died before Trihexyphenidyl was tried. Trihexyphenidyl in RS patients with similar presentations can modify the dystonia and prevent ALTEs.

MeSH terms

  • Adolescent
  • Antiparkinson Agents / therapeutic use*
  • Child
  • Dystonic Disorders / drug therapy*
  • Dystonic Disorders / etiology*
  • Electrocardiography / methods
  • Electroencephalography / methods
  • Electromyography / methods
  • Female
  • Humans
  • Rett Syndrome / complications*
  • Trihexyphenidyl / therapeutic use*
  • Video Recording / methods
  • Young Adult

Substances

  • Antiparkinson Agents
  • Trihexyphenidyl