TY - JOUR
T1 - Lacosamide for children with paroxysmal kinesigenic dyskinesia
AU - Furukawa, Gen
AU - Negishi, Yutaka
AU - Takeuchi, Tomoya
AU - Ishihara, Naoko
AU - Okumura, Akihisa
N1 - Publisher Copyright:
© 2020 The Japanese Society of Child Neurology
PY - 2020/9
Y1 - 2020/9
N2 - 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.
AB - 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.
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U2 - 10.1016/j.braindev.2020.04.009
DO - 10.1016/j.braindev.2020.04.009
M3 - Article
C2 - 32430160
AN - SCOPUS:85084608952
SN - 0387-7604
VL - 42
SP - 617
EP - 620
JO - Brain and Development
JF - Brain and Development
IS - 8
ER -