Gen Furukawa1, Yutaka Negishi2, Tomoya Takeuchi3, Naoko Ishihara4, Akihisa Okumura5. 1. Department of Pediatrics, Toyokawa City Hospital, Japan. Electronic address: gen.furukawa@fujita-hu.ac.jp. 2. Department of Pediatrics, Gifu Prefectural Tajimi Hospital, Japan. 3. Department of Pediatrics, Japanese Red Cross Nagoya Daiichi Hospital, Japan. 4. Department of Pediatrics, Fujita Health University School of Medicine, Japan. 5. Department of Pediatrics, Aichi Medical University, Japan.
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.
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 PKDpatients (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.
Authors: Michel Sáenz-Farret; Marina A J Tijssen; Dawn Eliashiv; Robert S Fisher; Kapil Sethi; Alfonso Fasano Journal: CNS Drugs Date: 2022-07-21 Impact factor: 6.497