Antero-ventral internal pallidum stimulation improves behavioral disorders in Lesch-Nyhan disease

Mov Disord. 2007 Oct 31;22(14):2126-9. doi: 10.1002/mds.21723.

Abstract

The Lesch-Nyhan syndrome is an X-linked recessive disorder caused by a deficiency in hypoxanthine-guanine phosphoribosyl transferase, a purine salvage enzyme. Affected individuals exhibit a characteristic neurobehavioral disorder with delayed acquisition of motor skills, dystonia, severe self-mutilations, and aggressive behavior. Deep brain stimulation has been previously proposed for controlling isolated involuntary movements and psychiatric disorders. We applied a double bilateral simultaneous stimulation to limbic and motor internal pallidum in one patient for controlling both behavioral and movement disorders, respectively. The injurious compulsions disappeared; dystonia and dyskinesia were decreased at 28 months follow-up.

Publication types

  • Case Reports

MeSH terms

  • Adolescent
  • Behavioral Symptoms / etiology
  • Behavioral Symptoms / therapy*
  • Electric Stimulation Therapy / methods*
  • Globus Pallidus / physiopathology
  • Globus Pallidus / radiation effects*
  • Humans
  • Lesch-Nyhan Syndrome / complications
  • Male