Sustained relief of generalized dystonia despite prolonged interruption of deep brain stimulation

Mov Disord. 2013 Sep;28(10):1431-4. doi: 10.1002/mds.25353. Epub 2013 Feb 11.

Abstract

Background: Pallidal deep brain stimulation (DBS) is an established treatment for disabling, medication-refractory generalized dystonia. Patients typically regress to their preoperative baseline when stimulation is discontinued.

Methods: Presented are case reports of 2 dystonia patients.

Results: Two patients with primary generalized dystonia (1 with the DYT1 mutation) who were treated successfully with bilateral pallidal DBS for periods of 18 months and 5 years retained motor benefit for several months after inadvertent interruption of stimulation. Stimulation was interrupted unilaterally for 3 and 7 months and bilaterally for 2 days and 2 months, respectively. Symptoms of dystonia returned only partially during the period of therapy interruption and rapidly and completely resolved after resuming stimulation.

Conclusions: We report unexpected and prolonged retention of motor benefits despite transient cessation of pallidal DBS in 2 dystonia patients. Factors that appear to differentiate these individuals are young age, short duration of disease, and chronic DBS therapy with relatively low energy of stimulation.

Keywords: deep brain stimulation; dystonia; globus pallidus; neuroplasticity.

Publication types

  • Case Reports

MeSH terms

  • Adolescent
  • Brain / pathology
  • Child
  • Deep Brain Stimulation / methods*
  • Dystonic Disorders / therapy*
  • Globus Pallidus / physiology
  • Humans
  • Male
  • Molecular Chaperones / genetics
  • Myoclonus / complications
  • Myoclonus / therapy
  • Neurologic Examination
  • Treatment Outcome
  • Young Adult

Substances

  • Molecular Chaperones
  • TOR1A protein, human