Efficacy and tolerability of add-on stiripentol in real-world clinical practice: An observational study in Dravet syndrome and non-Dravet developmental and epileptic encephalopathies

To assess efficacy and tolerability of stiripentol (STP) as adjunctive treatment in Dravet syndrome and non-Dravet refractory developmental and epileptic encephalopathies (DREEs). Retrospective observational study of all children and adults with DREE and prescribed adjunctive STP at Hospital Ruber I...

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Published in:Epilepsia open 2024-02, Vol.9 (1), p.164-175
Main Authors: Gil-Nagel, Antonio, Aledo-Serrano, Angel, Beltrán-Corbellini, Álvaro, Martínez-Vicente, Laura, Jimenez-Huete, Adolfo, Toledano-Delgado, Rafael, Gacía-Morales, Irene, Valls-Carbó, Adrián
Format: Article
Language:English
Subjects:
Adolescent
Adult
Anticonvulsants - therapeutic use
antiseizure medication
Child
Child, Preschool
Clinical medicine
Convulsions & seizures
developmental and epileptic encephalopathy
Dioxolanes - therapeutic use
Dravet syndrome
Drug dosages
Epilepsies, Myoclonic - drug therapy
Epilepsy
Humans
Infant
Middle Aged
Observational studies
Original
Patients
Retention
Seizures - drug therapy
status epilepticus
stiripentol
Survival analysis
Young Adult
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creator Gil-Nagel, Antonio
Aledo-Serrano, Angel
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Gacía-Morales, Irene
Valls-Carbó, Adrián
description To assess efficacy and tolerability of stiripentol (STP) as adjunctive treatment in Dravet syndrome and non-Dravet refractory developmental and epileptic encephalopathies (DREEs). Retrospective observational study of all children and adults with DREE and prescribed adjunctive STP at Hospital Ruber Internacional from January 2000 to February 2023. Outcomes were retention rate, responder rate (proportion of patients with ≥50% reduction in total seizure frequency relative to baseline), seizure freedom rate, responder rate for status epilepticus, rate of adverse event and individual adverse events, reported at 3, 6, and 12 months and at final visit. Seizure outcomes are reported overall, and for Dravet and non-Dravet subgroups. A total of 82 patients (55 Dravet syndrome and 27 non-Dravet DREE) were included. Median age was 5 years (range 1-59 years), and median age of epilepsy onset was younger in the Dravet group (4.9 [3.6-6] months) than non-Dravet (17.9 [6-42.3], P 
doi_str_mv 10.1002/epi4.12847
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Retrospective observational study of all children and adults with DREE and prescribed adjunctive STP at Hospital Ruber Internacional from January 2000 to February 2023. Outcomes were retention rate, responder rate (proportion of patients with ≥50% reduction in total seizure frequency relative to baseline), seizure freedom rate, responder rate for status epilepticus, rate of adverse event and individual adverse events, reported at 3, 6, and 12 months and at final visit. Seizure outcomes are reported overall, and for Dravet and non-Dravet subgroups. A total of 82 patients (55 Dravet syndrome and 27 non-Dravet DREE) were included. Median age was 5 years (range 1-59 years), and median age of epilepsy onset was younger in the Dravet group (4.9 [3.6-6] months) than non-Dravet (17.9 [6-42.3], P &lt; 0.001). Median follow-up time STP was 24.1 months (2 years; range 0.3-164 months) and was longer in the Dravet group (35.9 months; range 0.8-164) than non-Dravet (17 months range 0.3-62.3, P &lt; 0.001). At 12 months, retention rate, responder rate and seizure free rate was 68.3% (56/82), 65% [48-77%] and 18% [5.7-29%], respectively. There were no statistically significant differences between groups on these seizure outcomes. Adverse events were reported in 46.3% of patients (38/82), without differences between groups. 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Retrospective observational study of all children and adults with DREE and prescribed adjunctive STP at Hospital Ruber Internacional from January 2000 to February 2023. Outcomes were retention rate, responder rate (proportion of patients with ≥50% reduction in total seizure frequency relative to baseline), seizure freedom rate, responder rate for status epilepticus, rate of adverse event and individual adverse events, reported at 3, 6, and 12 months and at final visit. Seizure outcomes are reported overall, and for Dravet and non-Dravet subgroups. A total of 82 patients (55 Dravet syndrome and 27 non-Dravet DREE) were included. Median age was 5 years (range 1-59 years), and median age of epilepsy onset was younger in the Dravet group (4.9 [3.6-6] months) than non-Dravet (17.9 [6-42.3], P &lt; 0.001). Median follow-up time STP was 24.1 months (2 years; range 0.3-164 months) and was longer in the Dravet group (35.9 months; range 0.8-164) than non-Dravet (17 months range 0.3-62.3, P &lt; 0.001). At 12 months, retention rate, responder rate and seizure free rate was 68.3% (56/82), 65% [48-77%] and 18% [5.7-29%], respectively. There were no statistically significant differences between groups on these seizure outcomes. Adverse events were reported in 46.3% of patients (38/82), without differences between groups. 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ispartof Epilepsia open, 2024-02, Vol.9 (1), p.164-175
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source Wiley Online Library Open Access; Publicly Available Content Database; PubMed Central
subjects Adolescent
Adult
Anticonvulsants - therapeutic use
antiseizure medication
Child
Child, Preschool
Clinical medicine
Convulsions & seizures
developmental and epileptic encephalopathy
Dioxolanes - therapeutic use
Dravet syndrome
Drug dosages
Epilepsies, Myoclonic - drug therapy
Epilepsy
Humans
Infant
Middle Aged
Observational studies
Original
Patients
Retention
Seizures - drug therapy
status epilepticus
stiripentol
Survival analysis
Young Adult
title Efficacy and tolerability of add-on stiripentol in real-world clinical practice: An observational study in Dravet syndrome and non-Dravet developmental and epileptic encephalopathies
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