title: Clinical and electroencephalographic effects of folinic acid treatment in Rett syndrome patients.
abstract: Rett syndrome is characterized by the development of stereotypic hand movements and seizures, which are often difficult to treat. Previous studies have shown conflicting results during add-on folinic acid. Here, the authors reevaluate the response to folinic acid in terms of epilepsy control and electroencephalography features. They performed a randomized, placebo-controlled, double-blind crossover trial, with a follow-up of more than 2 years. Twelve girls with Rett syndrome participated, comparable in clinical stage and disease severity. The Rett syndrome patients were given either folinic acid or placebo, for 1 year each. Only 3 girls benefited to some extent: 2 had a reduction and/or decrease in seizures, and all 3 showed some decreased epileptiform activity on electroencephalography during the addition of folinic acid. Despite this, antiepileptic drugs were adjusted. Because the effect of added folinic acid was limited and did not prevent antiepileptic drug increase, the authors do not recommend adding on folinic acid in Rett syndrome girls with epilepsy.
authors.affiliation: Academical Medical Center, Department of Paediatric Neurology, Amsterdam, the Netherlands. e.e.hagebeuk@amc.uva.nl
authors.forename: Eveline E O
authors.initials: EE
authors.lastname: Hagebeuk
authors.forename: Johannes H T M
authors.initials: JH
authors.lastname: Koelman
authors.forename: Marinus
authors.initials: M
authors.lastname: Duran
authors.forename: Nico G
authors.initials: NG
authors.lastname: Abeling
authors.forename: Arno
authors.initials: A
authors.lastname: Vyth
authors.forename: Bwee-Tien
authors.initials: BT
authors.lastname: Poll-The
mesh_terms: D058256:Brain Waves / Q000187:drug effects*; D002648:Child; D002675:Child, Preschool; D018592:Cross-Over Studies; D004311:Double-Blind Method; D004569:Electroencephalography; D005260:Female; D005500:Follow-Up Studies; D006801:Humans; D002955:Leucovorin / Q000627:therapeutic use*; D008297:Male; D015518:Rett Syndrome / Q000150:complications / Q000188:drug therapy*; D012640:Seizures / Q000188:drug therapy / Q000209:etiology; D012720:Severity of Illness Index; D013057:Spectrum Analysis
chemical_list: D002955:Leucovorin