Developmental and epileptic encephalopathies

IE Scheffer, S Zuberi, HC Mefford, R Guerrini… - Nature Reviews …, 2024‏ - nature.com
Developmental and epileptic encephalopathies, the most severe group of epilepsies, are
characterized by seizures and frequent epileptiform activity associated with developmental …

The current landscape of epilepsy genetics: where are we, and where are we going?

SM Ruggiero, J **an, I Helbig - Current opinion in neurology, 2023‏ - journals.lww.com
New approaches to gene discovery and characterization are advancing rapidly our
understanding of the genetic and phenotypic architecture of the epilepsies. This review …

SNAP25 disease mutations change the energy landscape for synaptic exocytosis due to aberrant SNARE interactions

A Kádková, J Murach, M Østergaard, A Malsam… - Elife, 2024‏ - elifesciences.org
SNAP25 is one of three neuronal SNAREs driving synaptic vesicle exocytosis. We studied
three mutations in SNAP25 that cause epileptic encephalopathy: V48F, and D166Y in the …

Delineating clinical and developmental outcomes in STXBP1-related disorders

J **an, KM Thalwitzer, J McKee, KR Sullivan, E Brimble… - Brain, 2023‏ - academic.oup.com
STXBP1-related disorders are among the most common genetic epilepsies and
neurodevelopmental disorders. However, the longitudinal epilepsy course and …

SCN1A gain‐of‐function mutation causing an early onset epileptic encephalopathy

J Clatot, S Parthasarathy, S Cohen, JL McKee… - …, 2023‏ - Wiley Online Library
Objective Loss‐of‐function variants in SCN1A cause Dravet syndrome, the most common
genetic developmental and epileptic encephalopathy (DEE). However, emerging evidence …

Epilepsy Course and Developmental Trajectories in STXBP1-DEE

G Balagura, J **an, A Riva, F Marchese… - Neurology …, 2022‏ - neurology.org
Background and Objectives Clinical manifestations in STXBP1 developmental and epileptic
encephalopathy (DEE) vary in severity and outcome, and the genotypic spectrum is diverse …

Enriching representation learning using 53 million patient notes through human phenotype ontology embedding

M Daniali, PD Galer, D Lewis-Smith… - Artificial intelligence in …, 2023‏ - Elsevier
Abstract The Human Phenotype Ontology (HPO) is a dictionary of> 15,000 clinical
phenotypic terms with defined semantic relationships, developed to standardize phenotypic …

A recurrent de novo splice site variant involving DNM1 exon 10a causes developmental and epileptic encephalopathy through a dominant-negative mechanism

S Parthasarathy, SMK Ruggiero, A Gelot… - The American Journal of …, 2022‏ - cell.com
Heterozygous pathogenic variants in DNM1 cause developmental and epileptic
encephalopathy (DEE) as a result of a dominant-negative mechanism impeding vesicular …

Clinical trials for Lennox–Gastaut syndrome: Challenges and priorities

JK Knowles, AEL Warren, IS Mohamed… - Annals of clinical …, 2024‏ - Wiley Online Library
Abstract Objective: Lennox‐Gastaut syndrome (LGS) is a severe, childhood‐onset epilepsy
that is typically refractory to treatment. We surveyed the current landscape of LGS treatment …

Computational analysis of neurodevelopmental phenotypes: Harmonization empowers clinical discovery

D Lewis‐Smith, S Parthasarathy, J **an… - Human …, 2022‏ - Wiley Online Library
Making a specific diagnosis in neurodevelopmental disorders is traditionally based on
recognizing clinical features of a distinct syndrome, which guides testing of its possible …