Lacosamide for children with paroxysmal kinesigenic dyskinesia

Brain Dev. 2020 Sep;42(8):617-620. doi: 10.1016/j.braindev.2020.04.009. Epub 2020 May 16.

Abstract

Objectives: This study was performed to evaluate the efficacy and tolerability of lacosamide (LCM) for paroxysmal kinesigenic dyskinesia (PKD) in children.

Methods: We retrospectively reviewed the medical charts of pediatric PKD patients (aged <16 years) treated with LCM. Data regarding demographic characteristics, proline-rich transmembrane protein 2 (PRRT2) gene variant, clinical features of PKD, dose of LCM, efficacy, and adverse events were recorded.

Results: Four eligible patients (3 males, 1 female) were identified, with an age of onset ranging from 8.3 to 14.7 years. PRRT2 variant was evaluated in three children and a c.649dupC variant was identified in one child with a positive family history. Attacks were bilateral in three children and left-sided in one. Two children had a family history of PKD and one child had a family history of benign infantile epilepsy. Treatment with carbamazepine failed in two children due to drowsiness and auditory disturbance. The initial dose of LCM was 50 mg/day in three children and 100 mg/day in one. All patients were attack-free within a few days. The maintenance dose was mostly similar to the initial dose. No adverse events related to LCM were reported during follow-up.

Conclusions: LCM is an effective and well-tolerated treatment for PKD in children, and low-dose treatment may be viable.

Keywords: Lacosamide; Monotherapy; PRRT2 gene variant; Paroxysmal kinesigenic dyskinesia.

Publication types

  • Case Reports

MeSH terms

  • Adolescent
  • Carbamazepine / administration & dosage
  • Carbamazepine / therapeutic use
  • Child
  • Dystonia / drug therapy*
  • Dystonia / genetics
  • Female
  • Humans
  • Lacosamide / administration & dosage
  • Lacosamide / therapeutic use*
  • Male
  • Membrane Proteins / genetics
  • Mutation
  • Nerve Tissue Proteins / genetics
  • Retrospective Studies

Substances

  • Membrane Proteins
  • Nerve Tissue Proteins
  • PRRT2 protein, human
  • Carbamazepine
  • Lacosamide

Supplementary concepts

  • Familial paroxysmal dystonia