Cited by 210Epilepsy research and treatmentPharmacological Effects and Toxicity StudiesDiet and metabolism studies

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Successful use of fenfluramine as an add‐on treatment for Dravet syndrome

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Ceulemans, Berten, Boel, Marc, Leyssens, Katrien, Van Rossem, Carolin, Neels, Pieter, Jorens, Philippe G., Lagae, Lieven·2012·Epilepsia

DOI 10.1111/j.1528-1167.2012.03495.x

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Abstract

Summary: 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possible cardiac adverse effects (valve thickening, pulmonary hypertension) associated with use of fenfluramine, it was withdrawn from the market in 2001.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.

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Self-citation
Uji ketelitian pengukur massa di laboratprium fisika FPMIPA IKIP Malang / I Nengah Simpan
Simpen, I. N. (1989)
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Lieven Lagae
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Authors: Ceulemans, B., Boel, M., Leyssens, K., Van Rossem, C., Neels, P., Jorens, P. G., & Lagae, L.
Affiliations: University of Antwerp (BE)
KU Leuven (BE)
Corresponding author: Ceulemans, Berten
ORCID: Ceulemans, Berten
Leyssens, Katrien
Jorens, Philippe G.
Lagae, Lieven
Venue: Epilepsia
Publisher: Wiley
Published: 2012-05-03
Volume: 53
Issue: 7
Pages: 1131-1139
ISSN: 0013-9580 (ISSN-L)
1528-1167
DOI: 10.1111/j.1528-1167.2012.03495.x
OpenAlex: W1926532831
Cited by: 210
References: 51
Citations by year: 2026: 7 · 2025: 10 · 2024: 18 · 2023: 13 · 2022: 20 · 2021: 28 · 2020: 27 · 2019: 23 · 2018: 18 · 2017: 13 · 2016: 15 · 2015: 8 · 2014: 5 · 2013: 3 · 2012: 1
FWCI: 3.88
Type: journal article
Language: en
Open access: bronze
doi.org
Licence: onlinelibrary.wiley.com/termsAndConditions#vor (vor)
Locations: Epilepsia (publishedVersion)
PubMed (publishedVersion)
Topics: Health Sciences › Medicine › Clinical Neurology › Epilepsy research and treatment
Health Sciences › Medicine › Pediatrics, Perinatology and Child Health › Pharmacological Effects and Toxicity Studies
Health Sciences › Medicine › Physiology (medical) › Diet and metabolism studies
Keywords: Dravet syndrome, fenfluramine, adjunctive therapy, seizure freedom, drug repurposing, amphetamine-type stimulants, pulmonary hypertension, drug withdrawal, seizure recurrence, long-term follow-up
MeSH terms: Adolescent, Adult, Anticonvulsants, Child, Preschool, Electroencephalography, Epilepsies, Myoclonic, Female, Fenfluramine, Follow-Up Studies, Humans, Male, Selective Serotonin Reuptake Inhibitors, Young Adult

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Successful use of fenfluramine as an add‐on treatment for Dravet syndrome

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