Ventricular-subventricular zone stem cell niche adaptations in a mouse model of post-infectious hydrocephalus
Herman J, Rittenhouse N, Mandino F, Majid M, Wang Y, Mezger A, Kump A, Kadian S, Lake E, Verardi P, Conover J. Ventricular-subventricular zone stem cell niche adaptations in a mouse model of post-infectious hydrocephalus. Frontiers In Neuroscience 2024, 18: 1429829. PMID: 39145299, PMCID: PMC11322059, DOI: 10.3389/fnins.2024.1429829.Peer-Reviewed Original ResearchPost-infectious hydrocephalusVentricular-subventricular zoneInfluenza virusMouse modelMouse-adapted influenza virusIntact ependymaStem cellsPostnatal day 4Stem cell numbersStem cell nicheCell-to-cell transmissionOlfactory bulbMultiorgan hemorrhageLateral ventricleIntracerebroventricular injectionVentriculomegalyEmbryonic dayVentricular systemDay 4Cell nicheNo significant reductionHydrocephalusSignificant reductionVentricleCell numberPathogenic variants in autism gene KATNAL2 cause hydrocephalus and disrupt neuronal connectivity by impairing ciliary microtubule dynamics
DeSpenza T, Singh A, Allington G, Zhao S, Lee J, Kiziltug E, Prina M, Desmet N, Dang H, Fields J, Nelson-Williams C, Zhang J, Mekbib K, Dennis E, Mehta N, Duy P, Shimelis H, Walsh L, Marlier A, Deniz E, Lake E, Constable R, Hoffman E, Lifton R, Gulledge A, Fiering S, Moreno-De-Luca A, Haider S, Alper S, Jin S, Kahle K, Luikart B. Pathogenic variants in autism gene KATNAL2 cause hydrocephalus and disrupt neuronal connectivity by impairing ciliary microtubule dynamics. Proceedings Of The National Academy Of Sciences Of The United States Of America 2024, 121: e2314702121. PMID: 38916997, PMCID: PMC11228466, DOI: 10.1073/pnas.2314702121.Peer-Reviewed Original ResearchConceptsCongenital hydrocephalusCerebral ventriculomegalyPathogenic variantsPrefrontal pyramidal neuronsGenetic subsets of patientsDevelopment of ventriculomegalyRadial gliaSubsets of patientsHigh-frequency firingNeuronal connectivityHeterozygous germline variantsAutism spectrum disorderVentricular-subventricular zoneMicrotubule dynamicsImpaired spermatogenesisCSF shuntingExcitatory driveMicrotubule-severing ATPasePyramidal neuronsDisrupt neuronal connectivityGermline variantsVentriculomegalyCSF homeostasisDisrupt microtubule dynamicsPlanar cell polarity