摘要：SummarySTXBP1mutations are associated with encephalopathy, developmental delay, intellectual disability, and epilepsy. While neural networks are known to operate at a critical state in the healthy brain, network behavior during pathological epileptic states remains unclear. Examining activity during periods between well-characterized ictal-like events (i.e., interictal period) could provide a valuable step toward understanding epileptic networks. To study these networks in the context ofSTXBP1mutations, we combine a larval zebrafish model within vivofast confocal calcium imaging and extracellular local field potential recordings.Stxbp1bmutants display transient periods of elevated activity among local clusters of interacting neurons. These network “cascade” events were significantly larger in size and duration in mutants. At mesoscale resolution, cascades exhibit neurodevelopmental abnormalities. At single-cell scale, we describe spontaneous hyper-synchronized neuronal ensembles. That calcium imaging reveals uniquely disordered brain states during periods between pathological ictal-like seizure events is striking and represents a potential interictal biomarker.Graphical abstractDisplay OmittedHighlights•Calcium imaging in genetic epilepsy zebrafish model to study the interictal state•Network cascade activity was more prominent instxbp1bmutant zebrafish•stxbp1bmutants characterized by spontaneous hyper-synchronized neuronal ensemblesOptical imaging; Molecular neuroscience; Cellular neuroscience
