Title: Successful use of fenfluramine as an add-on treatment for Dravet syndrome
Authors: Ceulemans, Berten ×
Boel, Marc
Leyssens, Katrien
Van Rossem, Carolin
Neels, Pieter
Jorens, Philippe G
Lagae, Lieven #
Issue Date: Jul-2012
Publisher: Wiley-Blackwell Publishing, Inc.
Series Title: Epilepsia vol:53 issue:7 pages:1131-1139
Article number: 10.1111/j.1528-1167.2012.03495.x
Abstract: Purpose:  Despite the development of new antiepileptic drugs, Dravet syndrome frequently remains therapy resistant and is a catastrophic epilepsy syndrome. Fenfluramine is an amphetamine-like drug that has been used in the past as a part of antiobesity treatments. Because of the possible cardiac adverse effects (valve thickening, pulmonary hypertension) associated with use of fenfluramine, it was withdrawn from the market in 2001. In Belgium, a Royal Decree permitted examination of the potential anticonvulsive effects of fenfluramine in a clinical trial consisting of a small group of patients diagnosed with Dravet syndrome. Methods:  Herein, we report 12 patients, 7 female and 5 male, with a genetically proven (11 of 12) diagnosis of Dravet syndrome who received fenfluramine as add-on therapy. Key Findings:  Their ages at their last evaluation ranged from 3-35 years. The mean dosage of fenfluramine was 0.34 (0.12-0.90) mg/kg/day. Exposure duration to fenfluramine ranged from 1-19 years. Seven of the patients who were still receiving the fenfluramine treatment at the time of the last visit had been seizure-free for at least 1 year. In total, patients had been seizure-free for a mean of 6 (1-19) years. In seven patients, the fenfluramine treatment was interrupted once during the follow-up; seizures reappeared in three of the seizure-free patients. Subsequent reintroduction of fenfluramine controlled the seizures in these three patients again. Only two patients exhibited a mild thickening of one or two cardiac valves without clinical significance. Significance:  Compared with a recent long-term follow-up series in which a maximum of 16% of patients with Dravet syndrome were seizure-free, our result of 70% of patients with Dravet syndrome remaining seizure-free is noteworthy. Given the limitations of this observational study, a larger prospective study should be undertaken to confirm these promising results.
ISSN: 0013-9580
Publication status: published
KU Leuven publication type: IT
Appears in Collections:Pregnancy, Foetus and Newborn (-)
× corresponding author
# (joint) last author

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