Lacosamide for children with paroxysmal kinesigenic dyskinesia.
Lacosamide
Monotherapy
PRRT2 gene variant
Paroxysmal kinesigenic dyskinesia
Journal
Brain & development
ISSN: 1872-7131
Titre abrégé: Brain Dev
Pays: Netherlands
ID NLM: 7909235
Informations de publication
Date de publication:
Sep 2020
Sep 2020
Historique:
received:
22
03
2020
revised:
19
04
2020
accepted:
21
04
2020
pubmed:
21
5
2020
medline:
5
5
2021
entrez:
21
5
2020
Statut:
ppublish
Résumé
This study was performed to evaluate the efficacy and tolerability of lacosamide (LCM) for paroxysmal kinesigenic dyskinesia (PKD) in children. 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. 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. LCM is an effective and well-tolerated treatment for PKD in children, and low-dose treatment may be viable.
Identifiants
pubmed: 32430160
pii: S0387-7604(20)30136-4
doi: 10.1016/j.braindev.2020.04.009
pii:
doi:
Substances chimiques
Membrane Proteins
0
Nerve Tissue Proteins
0
PRRT2 protein, human
0
Carbamazepine
33CM23913M
Lacosamide
563KS2PQY5
Types de publication
Case Reports
Journal Article
Langues
eng
Sous-ensembles de citation
IM
Pagination
617-620Informations de copyright
Copyright © 2020 The Japanese Society of Child Neurology. Published by Elsevier B.V. All rights reserved.
Déclaration de conflit d'intérêts
Declaration of Competing Interest The authors declare that they have no known competing financial interests or personal relationships that could have appeared to influence the work reported in this paper.