Aug 2026· Asian Journal of Pediatric Research· 0 citations
TL;DR
This critical narrative review synthesises evidence on the clinical features, aetiologies, and therapeutic options for these syndromes, and evaluates the strength, consistency, and limitations of that evidence rather than cataloguing individual studies.
Abstract
Severe childhood epilepsy syndromes, encompassing the developmental and epileptic encephalopathies together with related drug-resistant electroclinical constellations, remain among the most challenging conditions in paediatric neurology. They combine frequent, treatment-resistant seizures with developmental impairment, substantial comorbidity, and elevated premature mortality. The past two decades have transformed the field: a revised syndrome classification, an expanding catalogue of monogenic causes, and a succession of syndrome-specific and mechanism-based therapies have altered both diagnosis and management. This critical narrative review synthesises evidence on the clinical features, aetiologies, and therapeutic options for these syndromes, and evaluates the strength, consistency, and limitations of that evidence rather than cataloguing individual studies. Literature was selected from bibliographic searching, citation chaining, and appraisal of consensus statements and clinical guidelines, with every cited reference and its digital object identifier independently verified. Several themes emerge. Aetiological diagnosis, particularly through broad genetic testing, now carries direct management consequences, yet a diagnostic gap persists and genotype does not map cleanly onto phenotype or treatment response. Syndrome-specific pharmacotherapy is supported by robust randomised evidence for a small number of agents in Dravet syndrome, Lennox-Gastaut syndrome, and CDKL5 deficiency disorder, but head-to-head comparisons, long-term developmental outcomes, and effects on the encephalopathy itself remain poorly characterised. Mechanism-based and disease-modifying strategies, including mammalian target of rapamycin inhibition, pre-emptive treatment, and antisense oligonucleotides, represent a conceptual shift from seizure suppression toward disease modification, though the durability and developmental impact of these approaches are not yet established. Sudden unexpected death in epilepsy and other causes of early mortality remain insufficiently mitigated. The available evidence supports cautious optimism: outcomes are improving, but confidence in many conclusions is constrained by small samples, heterogeneous endpoints, short follow-up, and reliance on seizure count as the dominant outcome. Priorities include earlier aetiological diagnosis, developmentally meaningful outcome measures, and trials designed to test disease modification rather than seizure frequency alone.
Comprehensive care extends beyond seizure management and includes addressing developmental, educational, and psychosocial needs, and a multidisciplinary, family-centred approach involving neurologists, dietitians, psychologists, and educators is essential.
Jayesh S. Patil, Hitendra S. Chaudhari, S. Pawar et al.· Research and reviews : a jou...· 0 citations
Current and emerging pharmacotherapeutic options for selected representative pediatric focal epilepsies are summarized using a syndrome- and localization-oriented approach, highlighting conventional antiseizure medications, newer agents, and investigational or repurposed treatments.
L. Perilli, G. Dell’Isola, Pietro Ferrara et al.· Expert Opinion on Pharmacoth...· 0 citations
Critical unmet needs include earlier molecular diagnosis, precision therapies targeting developmental outcomes beyond seizure control, and prospective international registries to characterize the long-term natural history of EIDEE.
Debopam Samanta· Brain & development (Tokyo....· 0 citations
Current evidence regarding diagnosis, typical and atypical radiological findings, aetiologies, tertiary-care experiences, pathophysiological mechanisms, emerging genetic associations, and an algorithmic approach to management are summarized.
Rachna Sehgal, Archana Kashyap, Bhavna Anand et al.· International Journal of Con...· 0 citations
This review examines the evolving landscape of pediatric OLE, highlighting the shift from syndromic to etiological classification and management strategies and the need to monitor cognitive comorbidities and syndrome evolution.
Preeti Srivastava, D. Nag, Shikha Swaroop et al.· World Journal of Clinical Pe...· 0 citations
BACKGROUND
Epilepsy of infancy with migrating focal seizures (EIMFS) is among the most severe developmental and epileptic encephalopathies (DEEs), marked by intractable multifocal seizures migrating across both hemispheres, profound developmental arrest, and high early mortality. Advances in next-generation sequencing have revealed a heterogeneous genetic architecture dominated by KCNT1 gain-of-function variants across more than 30 implicated genes, creating opportunities for precision therapeutics.
OBJECTIVE
To systematically map published evidence on the clinical, electrophysiological, neuroimaging, genetic, and therapeutic landscape of EIMFS, and to delineate critical knowledge gaps and future research priorities.
METHODS
A scoping review was conducted following the Arksey and O'Malley framework, searching PubMed, Ovid MEDLINE, Embase, Cochrane Library/CENTRAL, and ClinicalTrials.gov.
RESULTS
Of 643 articles screened, 89 met inclusion criteria. Beyond confirmation of the canonical electroclinical phenotype, several gaps emerged: neonatal versus post-neonatal onset stratification by genetic etiology remains largely uncharacterized; genotype-specific EEG biomarkers are lacking except for a single small KCNT1 study; and the clinical significance of atypical EEG features-including burst suppression and hypsarrhythmia-is undefined. Neuroimaging literature documents progressive cerebral atrophy and myelination abnormalities without quantitative volumetry, diffusion tractography markers, or attribution to seizure burden, medication effects, or underlying etiology. Genetic diagnostic yield was 70-80%, with KCNT1 accounting for 30-50% of solved cases; however, genotype-outcome stratification is limited. Seizures were broadly refractory; potassium bromide, ketogenic diet, cannabidiol, and quinidine (in KCNT1-confirmed cases) showed partial efficacy. Emerging precision approaches include sodium channel blockers for SCN2A gain-of-function variants, novel small molecules, fluoxetine, antisense oligonucleotides, and divalent siRNA targeting KCNT1. Systemic-to-pulmonary collateral circulation causing severe cardiopulmonary complications was reported across multiple cases, yet no consensus screening protocol exists.
CONCLUSIONS
EIMFS remains one of the most refractory epilepsy syndromes of infancy. Precision genetic diagnosis is essential to guide targeted therapy. International collaborative registries, standardized outcome measures, genotype-stratified biomarker studies, and rapid point-of-care genomic testing are urgently needed to advance evidence-based care for this highly vulnerable population.