Neuron · 2016
Psychiatric Risk Gene Transcription Factor 4 Regulates Intrinsic Excitability of Prefrontal Neurons via Repression of SCN10a and KCNQ1.
СтатьяПоддержка NIH: внешняя программаПоддержка исследования: вне правительства СШАНазвание и аннотация: Перевод в очереди
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Rannals Matthew D, Hamersky Gregory R, Page Stephanie Cerceo, Campbell Morganne N, Briley Aaron, Gallo Ryan A, Phan BaDoi N, Hyde Thomas M, Kleinman Joel E, Shin Joo Heon, Jaffe Andrew E, Weinberger Daniel R, Maher Brady J
Аннотация
Transcription Factor 4 (TCF4) is a clinically pleiotropic gene associated with schizophrenia and Pitt-Hopkins syndrome (PTHS). To gain insight about the neurobiology of TCF4, we created an in vivo model of PTHS by suppressing Tcf4 expression in rat prefrontal neurons immediately prior to neurogenesis. This cell-autonomous genetic insult attenuated neuronal spiking by increasing the afterhyperpolarization. At the molecular level, using a novel technique called iTRAP that combined in utero electroporation and translating ribosome affinity purification, we identified increased translation of two ion channel genes, Kcnq1 and Scn10a. These ion channel candidates were validated by pharmacological rescue and molecular phenocopy. Remarkably, similar excitability deficits were observed in prefrontal neurons from a Tcf4(+/tr) mouse model of PTHS. Thus, we identify TCF4 as a regulator of neuronal intrinsic excitability in part by repression of Kcnq1 and Scn10a and suggest that this molecular function may underlie pathophysiology associated with neuropsychiatric disorders.
Предметные рубрики MeSH
AnimalsBasic Helix-Loop-Helix Leucine Zipper Transcription FactorsCRISPR-Cas SystemsDNA-Binding ProteinsDisease Models, AnimalElectroporationFaciesFemaleGene Expression Regulation, DevelopmentalGenetic Predisposition to DiseaseHaploinsufficiencyHyperventilationIntellectual DisabilityKCNQ1 Potassium ChannelMiceNAV1.8 Voltage-Gated Sodium ChannelPatch-Clamp TechniquesPrefrontal CortexPregnancyRNA, MessengerRNA, Small InterferingRatsRats, WistarSchizophreniaTranscription Factor 4Transcription Factors