Child Neurology: A young child with an undiagnosed case of dystonia responsive to l-dopa

Neurology. 2020 Feb 18;94(7):326-328. doi: 10.1212/WNL.0000000000008963. Epub 2020 Feb 3.

Abstract

Childhood-onset dystonias are a heterogeneously diverse group. There exists a specific set of dystonias that respond profoundly well to low doses of l-dopa (dopa-responsive dystonia [DRD]). Classical DRD is caused by deficiency of GTP cyclohydrolase 1 or tyrosine hydroxylase, but other conditions can cause dystonias that are partially responsive to dopamine. The idea of a diagnostic therapeutic trial with l-dopa for children who present with dystonia has been around for decades and is frequently advocated for; however, l-dopa trials are not without risk.

Publication types

  • Case Reports

MeSH terms

  • Anti-Dyskinesia Agents / therapeutic use
  • Carbidopa / therapeutic use
  • Child, Preschool
  • Diagnosis, Differential
  • Drug Combinations
  • Dystonic Disorders / diagnosis*
  • Dystonic Disorders / drug therapy
  • Dystonic Disorders / genetics
  • Humans
  • Levodopa / therapeutic use
  • Male

Substances

  • Anti-Dyskinesia Agents
  • Drug Combinations
  • carbidopa, levodopa drug combination
  • Levodopa
  • Carbidopa

Supplementary concepts

  • Dystonia, Dopa-responsive