Observational studyEpilepsy research2018
High vigabatrin dosage is associated with lower risk of infantile spasms relapse among children with tuberous sclerosis complex.
Observational study in Epilepsy research, 2018. The graph could read no effect estimate from its abstract, so it casts no vote on the map. Cited by 16 papers.
What it found
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The abstract states no effect estimate the extractor could read, or names no intervention and outcome on the map, so this paper lights no cell and moves no belief. It is still indexed, cited and linked below.
The trial behind it
Trials whose registry record cites this paper, or whose number appears in the abstract. A trial that started after this paper was published is citing it as background, not reporting it.
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Who cites it
16 citing papers in PubMed, 32 citations in OpenAlex.
- Long-term outcome in children with infantile epileptic spasms syndrome: a multicenter retrospective study in Korea.Clinical and experimental pediatrics · 2026Article
- Molecular Basis of GABA Aminotransferase Inhibition in Epilepsy: Structure, Mechanisms, and Drug Development.Current issues in molecular biology · 2025Review
- Preventative treatment of tuberous sclerosis complex with sirolimus: Phase I safety and efficacy results.Annals of the Child Neurology Society · 2024Article
- Epileptic spasms relapse is associated with response latency but not conventional attributes of post-treatment EEG.Epilepsia open · 2024Article
- The Evolving Landscape of Therapeutics for Epilepsy in Tuberous Sclerosis Complex.Biomedicines · 2023Review
- Brazilian experts' consensus on the treatment of infantile epileptic spasm syndrome in infants.Arquivos de neuro-psiquiatria · 2023Article
- Treatment-Resistant Epilepsy and Tuberous Sclerosis Complex: Treatment, Maintenance, and Future Directions.Neuropsychiatric disease and treatment · 2023Review
- The state of pediatric tuberous sclerosis complex epilepsy care: Results from a national survey.Epilepsia open · 2022Article
- Epilepsy Is Heterogeneous in Early-Life Tuberous Sclerosis Complex.Pediatric neurology · 2021Article
- Pharmacotherapy for Seizures in Tuberous Sclerosis Complex.CNS drugs · 2021Review
- Review of the treatment options for epilepsy in tuberous sclerosis complex: towards precision medicine.Therapeutic advances in neurological disorders · 2021Review
- Modeling epileptic spasms during infancy: Are we heading for the treatment yet?Pharmacology & therapeutics · 2020Review
- Etiologic classification of infantile spasms using positron emission/magnetic resonance imaging and the efficacy of adrenocorticotropic hormone therapy.European journal of nuclear medicine and molecular imaging · 2020Article
- Update on Drug Management of Refractory Epilepsy in Tuberous Sclerosis Complex.Paediatric drugs · 2020Review
- Metabolomic analyses of vigabatrin (VGB)-treated mice: GABA-transaminase inhibition significantly alters amino acid profiles in murine neural and non-neural tissues.Neurochemistry international · 2019Article
- Preclinical tissue distribution and metabolic correlations of vigabatrin, an antiepileptic drug associated with potential use-limiting visual field defects.Pharmacology research & perspectives · 2019Article
Corrections and comments
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Authors and funding
10 authors at 7 institutions in 1 country.
Funding
Abstract
After initially successful treatment of infantile spasms, the long-term cumulative risk of relapse approaches 50%, and there is no established protocol to mitigate this risk. Although vigabatrin may be an effective means to prevent relapse, there is little guidance as to ideal duration and dosage. Using a cohort of children with infantile spasms and tuberous sclerosis complex (TSC), we evaluated the potential association of post-response VGB treatment and the rate of infantile spasms relapse. Patients with infantile spasms and clinical response to vigabatrin were identified among a multicenter prospective observational cohort of children with TSC. For each patient we recorded dates of infantile spasms onset, response to vigabatrin, relapse (if any), and quantified duration and dosage of vigabatrin after response. Time to relapse as a function of vigabatrin exposure was evaluated using survival analyses. We identified 50 children who responded to VGB. During a median follow-up of 16.6 months (IQR 10.3-22.9), 12 (24%) patients subsequently relapsed after a median of 7.8 months (IQR 3.1-9.6). Relapse occurred after VGB discontinuation in four patients, and during continued VGB treatment in the remaining eight cases. In survival analyses, risk of relapse was unaffected by the presence or absence of VGB treatment (HR 0.31, 95%CI 0.01-28.4, P = 0.61), but weighted-average dosage was associated with marked reduction in relapse risk: Each 50 mg/kg/d increment in dosage was associated with 61% reduction in risk (HR 0.39, 95%CI 0.17 - 0.90, P = 0.026). This study suggests that the risk of infantile spasms relapse in TSC may be reduced by high-dose vigabatrin treatment.
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Read under generation 80e0d062 · epoch 390. Bibliography from PubMed, PubMed Central and OpenAlex; grants from NIH RePORTER; trial links from ClinicalTrials.gov; estimates, votes and beliefs from the OpenQuestion graph.