A New Perspective on Drug-Resistant Epilepsy in Children with Focal Cortical Dysplasia Type 1: From Challenge to Favourable Outcome

Kód výsledku v IS VaVaI

<a href="https://www.isvavai.cz/riv?ss=detail&h=RIV%2F68407700%3A21230%2F25%3A00378751" target="_blank" >RIV/68407700:21230/25:00378751 - isvavai.cz</a>

Výsledek na webu

<a href="https://doi.org/10.1111/epi.18237" target="_blank" >https://doi.org/10.1111/epi.18237</a>

DOI - Digital Object Identifier

<a href="http://dx.doi.org/10.1111/epi.18237" target="_blank" >10.1111/epi.18237</a>

Jazyk výsledku

angličtina

Název v původním jazyce

A New Perspective on Drug-Resistant Epilepsy in Children with Focal Cortical Dysplasia Type 1: From Challenge to Favourable Outcome

Popis výsledku v původním jazyce

Objectives: We comprehensively characterised a large paediatric cohort with focal cortical dysplasia (FCD) type 1 to expand phenotypic spectrum and to identify predictors of postsurgical outcomes. Methods: We included paediatric patients with histopathological diagnosis of isolated FCD type 1 and at least one year of postsurgical follow-up. We systematically re-analysed clinical, electrophysiological, and radiological features. The results of this re-analysis served as independent variables for subsequent statistical analyses of outcome predictors. Results: All children (N=31) had drug-resistant epilepsy with varying impacts on neurodevelopment and cognition (presurgical intelligence quotient (IQ)/developmental quotient scores: 32–106). Low presurgical IQ was associated with abnormal slow background EEG activity and disrupted sleep architecture. Scalp EEG showed predominantly multiregional and often bilateral epileptiform activity. Advanced epilepsy MRI protocols identified FCD-specific features in 74.2% of patients (23/31), 17 of whom were initially evaluated as MRI-negative. In six out of eight MRI-negative cases, fluorodeoxyglucose-PET and subtraction ictal single-photon emission computed tomography co-registered to MRI (SISCOM) helped localise the dysplastic cortex. Sixteen patients (51.6%) underwent invasive EEG. By the last follow-up (median 5 years, interquartile range 3.3–9 years), seizure freedom was achieved in 71% of patients (22/31), including 7 out of 8 MRI-negative patients. Anti-seizure medications were reduced in 21 patients, with complete withdrawal in six. Seizure outcome was predicted by a combination of the following descriptors: age at epilepsy onset, epilepsy duration, long-term invasive EEG, and specific MRI, and PET findings. Significance: This study highlights the broad phenotypic spectrum of FCD type 1, which spans far beyond the narrow descriptions of previous studies. The applied multi-layered presurgical approach helped localise the epileptogenic zone in many previously non-lesional cases, resulting in improved postsurgical seizure outcomes, which are more favourable than previously reported for FCD type 1 patients.

Název v anglickém jazyce

A New Perspective on Drug-Resistant Epilepsy in Children with Focal Cortical Dysplasia Type 1: From Challenge to Favourable Outcome

Popis výsledku anglicky

Objectives: We comprehensively characterised a large paediatric cohort with focal cortical dysplasia (FCD) type 1 to expand phenotypic spectrum and to identify predictors of postsurgical outcomes. Methods: We included paediatric patients with histopathological diagnosis of isolated FCD type 1 and at least one year of postsurgical follow-up. We systematically re-analysed clinical, electrophysiological, and radiological features. The results of this re-analysis served as independent variables for subsequent statistical analyses of outcome predictors. Results: All children (N=31) had drug-resistant epilepsy with varying impacts on neurodevelopment and cognition (presurgical intelligence quotient (IQ)/developmental quotient scores: 32–106). Low presurgical IQ was associated with abnormal slow background EEG activity and disrupted sleep architecture. Scalp EEG showed predominantly multiregional and often bilateral epileptiform activity. Advanced epilepsy MRI protocols identified FCD-specific features in 74.2% of patients (23/31), 17 of whom were initially evaluated as MRI-negative. In six out of eight MRI-negative cases, fluorodeoxyglucose-PET and subtraction ictal single-photon emission computed tomography co-registered to MRI (SISCOM) helped localise the dysplastic cortex. Sixteen patients (51.6%) underwent invasive EEG. By the last follow-up (median 5 years, interquartile range 3.3–9 years), seizure freedom was achieved in 71% of patients (22/31), including 7 out of 8 MRI-negative patients. Anti-seizure medications were reduced in 21 patients, with complete withdrawal in six. Seizure outcome was predicted by a combination of the following descriptors: age at epilepsy onset, epilepsy duration, long-term invasive EEG, and specific MRI, and PET findings. Significance: This study highlights the broad phenotypic spectrum of FCD type 1, which spans far beyond the narrow descriptions of previous studies. The applied multi-layered presurgical approach helped localise the epileptogenic zone in many previously non-lesional cases, resulting in improved postsurgical seizure outcomes, which are more favourable than previously reported for FCD type 1 patients.

Druh

J_imp - Článek v periodiku v databázi Web of Science

CEP obor

—

OECD FORD obor

30210 - Clinical neurology

Projekt

Výsledek vznikl pri realizaci vícero projektů. Více informací v záložce Projekty.

Návaznosti

P - Projekt vyzkumu a vyvoje financovany z verejnych zdroju (s odkazem do CEP)

Rok uplatnění

2025

Kód důvěrnosti údajů

S - Úplné a pravdivé údaje o projektu nepodléhají ochraně podle zvláštních právních předpisů

Název periodika

Epilepsia

ISSN

0013-9580

e-ISSN

1528-1167

Svazek periodika

66

Číslo periodika v rámci svazku

3

Stát vydavatele periodika

US - Spojené státy americké

Počet stran výsledku

16

Strana od-do

632-647

Kód UT WoS článku

001383241100001

EID výsledku v databázi Scopus

2-s2.0-85213012062