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Войти в исследования Epilepsia · 2026
Diagnosis and management guidelines for infantile epileptic spasms syndrome around the world: A scoping review and comparative study of international approaches.
СтатьяScoping ReviewСравнительное исследованиеНазвание и аннотация: Перевод в очереди
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Erdemir Gozde, Rao Chethan K, Pino Anne Francine, Briscoe Christina, Samanta Debopam, Wilmshurst Jo M, Bhatia Sonal, Carpenter Jessica L, Hoei-Hansen Christina, Jain Puneet, Stodberg Tommy, Whitney Robyn, Matsuura Ryuki, Astorga Karina Rosso, Gallinato Keryma Acevedo, Singhi Pratibha, Hadijinicolaou Aristides, Melikishvili Gia, Smeyers Patricia, Specchio Nicola, Auvin Stéphane, Pediatric Epilepsy Research Consortium Infantile Spasm Work Group and International Committee
Аннотация
OBJECTIVE: Infantile epileptic spasms syndrome (IESS) is an epileptic encephalopathy requiring rapid diagnosis and treatment to optimize neurodevelopmental outcomes. Although multiple national and regional guidelines exist, recommendations vary. We conducted a comparative scoping review of international IESS guidelines to synthesize current recommendations, evaluate trends, and examine regional differences in diagnosis and treatment.
METHODS: Published guidelines were identified through MEDLINE, Embase, and Cochrane CENTRAL, using PRISMA-ScR (Preferred Reporting Items of Systematic Reviews and Meta-Analyses, extension for scoping reviews) methodology and PROSPERO (Prospective Register of Systematic Reviews) registration. Unpublished institutional protocols were obtained from pediatric neurologists worldwide. Extracted variables included diagnostic evaluation, treatment recommendations, dosing, response assessment, and monitoring. Descriptive analysis compared recommendations across regions and time periods.
RESULTS: Twenty-eight guidelines were analyzed (16 published, 12 unpublished), including 15 from high-income countries (HICs) and 11 from low- and middle-income countries (LMICs). Electroencephalography was recommended in nearly all guidelines (27/28, 96%), whereas brain magnetic resonance imaging (20/28, 71%) and genetic testing (15/28, 54%) were less consistent. Vigabatrin was universally recommended as first-line therapy for IESS-associated tuberous sclerosis complex. Earlier guidelines (2004-2015) favored adrenocorticotropic hormone (ACTH), whereas recent guidelines (2016-2024) increasingly endorsed prednisolone and vigabatrin. Combination therapy emerged after 2017 (~25%). LMIC guidelines uniformly recommended prednisolone, whereas HIC guidelines more frequently endorsed ACTH, vigabatrin, and combination therapy. Treatment response was most often assessed at ~14 days.
SIGNIFICANCE: International IESS guidelines share core principles but demonstrate evolving practices and persistent global disparities. HIC and LMIC recommendation differences likely reflect variation in medication access, cost, and diagnostic resources. Harmonized international guidance may reduce disparities and improve equitable care. Further research is needed to develop standardized treatment protocols and explore novel therapeutic options to optimize outcomes for infants with IESS.
METHODS: Published guidelines were identified through MEDLINE, Embase, and Cochrane CENTRAL, using PRISMA-ScR (Preferred Reporting Items of Systematic Reviews and Meta-Analyses, extension for scoping reviews) methodology and PROSPERO (Prospective Register of Systematic Reviews) registration. Unpublished institutional protocols were obtained from pediatric neurologists worldwide. Extracted variables included diagnostic evaluation, treatment recommendations, dosing, response assessment, and monitoring. Descriptive analysis compared recommendations across regions and time periods.
RESULTS: Twenty-eight guidelines were analyzed (16 published, 12 unpublished), including 15 from high-income countries (HICs) and 11 from low- and middle-income countries (LMICs). Electroencephalography was recommended in nearly all guidelines (27/28, 96%), whereas brain magnetic resonance imaging (20/28, 71%) and genetic testing (15/28, 54%) were less consistent. Vigabatrin was universally recommended as first-line therapy for IESS-associated tuberous sclerosis complex. Earlier guidelines (2004-2015) favored adrenocorticotropic hormone (ACTH), whereas recent guidelines (2016-2024) increasingly endorsed prednisolone and vigabatrin. Combination therapy emerged after 2017 (~25%). LMIC guidelines uniformly recommended prednisolone, whereas HIC guidelines more frequently endorsed ACTH, vigabatrin, and combination therapy. Treatment response was most often assessed at ~14 days.
SIGNIFICANCE: International IESS guidelines share core principles but demonstrate evolving practices and persistent global disparities. HIC and LMIC recommendation differences likely reflect variation in medication access, cost, and diagnostic resources. Harmonized international guidance may reduce disparities and improve equitable care. Further research is needed to develop standardized treatment protocols and explore novel therapeutic options to optimize outcomes for infants with IESS.