TY - JOUR
T1 - What is the optimal duration for vigabatrin monotherapy in patients with infantile spasms
T2 - 6 months or longer?
AU - Desnous, Béatrice
AU - Lenoir, Marien
AU - Bitton, Jonathan Y.
AU - Arbour, Mélina
AU - Villeneuve, Nathalie
AU - Whiting, Sharon
AU - Mohammed, Ismail
AU - Wirrell, Elaine C.
AU - Bello-Espinosa, Luis
AU - Ronen, Gabriel M.
AU - Lortie, Anne
AU - Birca, Ala
N1 - Funding Information:
The authors have no conflict of interest to disclose. This study was funded by the Canadian Institutes of Health Research grant #MCT/53573, the CURE Foundation, and the Savoy Foundation.
Publisher Copyright:
© 2021 British Epilepsy Association
PY - 2021/10
Y1 - 2021/10
N2 - Vigabatrin (VGB) is approved as monotherapy for pediatric patients with Infantile Spasms (IS). Duration of VGB use should be limited because of the risk of retinal and neurotoxicity, but the optimal length of treatment is unknown. Our study aimed to determine the risk of spasms relapse after 6 months of VGB as first-line therapy in IS patients deemed VGB good responders. The participants were 44 infants with IS who demonstrated both absence of clinical spasms and hypsarrhythmia four weeks after starting VGB, obtained from two cohorts: 29 patients from a multicenter prospective cohort and 15 patients from a retrospective single-center cohort. We divided them post hoc into two groups according to the duration of VGB treatment: 6-month group (n=34) and >6-month group (n=10) and compared outcome between the two groups. No patient in either group had a relapse of spasms. For patients with non-identified etiology (NIE) in the 6 months treatment group, no other seizure types were observed. Late epilepsy, in the form of focal seizures, emerged in only 5/37 patients (3/30 in the 6-month treatment group; 2/7 in the extended treatment group); all within the first 6-9 months after VGB initiation. Our study provides substantial evidence that a shortened VGB course of 6 months could be sufficient to treat and prevent relapse of spasms in children with IS, particularly those with NIE.
AB - Vigabatrin (VGB) is approved as monotherapy for pediatric patients with Infantile Spasms (IS). Duration of VGB use should be limited because of the risk of retinal and neurotoxicity, but the optimal length of treatment is unknown. Our study aimed to determine the risk of spasms relapse after 6 months of VGB as first-line therapy in IS patients deemed VGB good responders. The participants were 44 infants with IS who demonstrated both absence of clinical spasms and hypsarrhythmia four weeks after starting VGB, obtained from two cohorts: 29 patients from a multicenter prospective cohort and 15 patients from a retrospective single-center cohort. We divided them post hoc into two groups according to the duration of VGB treatment: 6-month group (n=34) and >6-month group (n=10) and compared outcome between the two groups. No patient in either group had a relapse of spasms. For patients with non-identified etiology (NIE) in the 6 months treatment group, no other seizure types were observed. Late epilepsy, in the form of focal seizures, emerged in only 5/37 patients (3/30 in the 6-month treatment group; 2/7 in the extended treatment group); all within the first 6-9 months after VGB initiation. Our study provides substantial evidence that a shortened VGB course of 6 months could be sufficient to treat and prevent relapse of spasms in children with IS, particularly those with NIE.
KW - Infantile spasms
KW - Relapse
KW - Vigabatrin treatment duration
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U2 - 10.1016/j.seizure.2021.07.032
DO - 10.1016/j.seizure.2021.07.032
M3 - Article
C2 - 34371246
AN - SCOPUS:85111979565
SN - 1059-1311
VL - 91
SP - 503
EP - 506
JO - Seizure : the journal of the British Epilepsy Association
JF - Seizure : the journal of the British Epilepsy Association
ER -