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Purkinje neuron differentiation

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https://www.readbyqxmd.com/read/28317875/chd7-is-indispensable-for-mammalian-brain-development-through-activation-of-a-neuronal-differentiation-programme
#1
Weijun Feng, Daisuke Kawauchi, Huiqin Körkel-Qu, Huan Deng, Elisabeth Serger, Laura Sieber, Jenna Ariel Lieberman, Silvia Jimeno-González, Sander Lambo, Bola S Hanna, Yassin Harim, Malin Jansen, Anna Neuerburg, Olga Friesen, Marc Zuckermann, Vijayanad Rajendran, Jan Gronych, Olivier Ayrault, Andrey Korshunov, David T W Jones, Marcel Kool, Paul A Northcott, Peter Lichter, Felipe Cortés-Ledesma, Stefan M Pfister, Hai-Kun Liu
Mutations in chromatin modifier genes are frequently associated with neurodevelopmental diseases. We herein demonstrate that the chromodomain helicase DNA-binding protein 7 (Chd7), frequently associated with CHARGE syndrome, is indispensable for normal cerebellar development. Genetic inactivation of Chd7 in cerebellar granule neuron progenitors leads to cerebellar hypoplasia in mice, due to the impairment of granule neuron differentiation, induction of apoptosis and abnormal localization of Purkinje cells, which closely recapitulates known clinical features in the cerebella of CHARGE patients...
March 20, 2017: Nature Communications
https://www.readbyqxmd.com/read/28315396/ischemia-deteriorates-spike-encoding-at-cortical-gabaergic-neurons-and-cerebellar-purkinje-cells-by-increasing-the-intracellular-ca-2
#2
Li Huang, Chun Wang, Rongjing Ge, Hong Ni, Shidi Zhao
GABAergic neurons play a critical role in the central nervous system, such as well-organized behaviors. The ischemic cell death is presumably initiated by neuronal excitotoxicity resulted from the dysfunction of GABAergic neurons. It is not clear how ischemia influences different types of GABAergic neurons and whether intracellular Ca(2+) plays a key role in the ischemic excitotoxicity. We have investigated this issue at cortical GABAergic neurons and cerebellar Purkinje cells by whole-cell recording in mouse brain slices, and the roles of intracellular Ca(2+) are examined by BABTA infusion...
March 14, 2017: Brain Research Bulletin
https://www.readbyqxmd.com/read/28299779/the-related-neuronal-endosomal-proteins-neep21-nsg1-and-p19-nsg2-have-divergent-expression-profiles-in-vivo
#3
Kelly Barford, Chan Choo Yap, Noelle D Dwyer, Bettina Winckler
Endosomal maturation and transport constitutes a complex trafficking system present in all cell types. Neurons have adapted their endosomal system to meet their unique and complex needs. These adaptations include repurposing existing proteins to diversify endocytosis and trafficking, as well as preferential expression of certain regulators more highly in neurons than other cell types. These neuronal regulators include the family of Neuron-Specific Gene family members (Nsg), NEEP21 (Nsg1), and P19 (Nsg2). NEEP21/Nsg1 plays a role in the trafficking of multiple receptors, including the cell adhesion molecule L1/NgCAM, the neurotransmitter receptor GluA2, and β-APP...
March 15, 2017: Journal of Comparative Neurology
https://www.readbyqxmd.com/read/28219683/the-c1q-complement-family-of-synaptic-organizers-not-just-complementary
#4
REVIEW
Michisuke Yuzaki
Molecules that regulate formation, differentiation, and maintenance of synapses are called synaptic organizers. Recently, various 'C1q family' proteins have been shown to be released from neurons, and serve as a new class of synaptic organizers. Cbln1 and C1ql1 proteins regulate the formation and maintenance of parallel fiber-Purkinje cell and climbing fiber-Purkinje cell synapses, respectively, in the cerebellum. Cbln1 also modulates the function of postsynaptic delta2 glutamate receptors to regulate synaptic plasticity...
February 17, 2017: Current Opinion in Neurobiology
https://www.readbyqxmd.com/read/28198698/a-role-for-cerebellum-in-the-hereditary-dystonia-dyt1
#5
Rachel Fremont, Ambika Tewari, Chantal Angueyra, Kamran Khodakhah
DYT1 is a debilitating movement disorder caused by loss-of-function mutations in torsinA. How these mutations cause dystonia remains unknown. Mouse models which have embryonically targeted torsinA have failed to recapitulate the dystonia seen in patients, possibly due to differential developmental compensation between rodents and humans. To address this issue, torsinA was acutely knocked down in select brain regions of adult mice using shRNAs. TorsinA knockdown in the cerebellum, but not in the basal ganglia, was sufficient to induce dystonia...
February 15, 2017: ELife
https://www.readbyqxmd.com/read/28092268/phenotypic-outcomes-in-mouse-and-human-foxc1-dependent-dandy-walker-cerebellar-malformation-suggest-shared-mechanisms
#6
Parthiv Haldipur, Derek Dang, Kimberly A Aldinger, Olivia K Janson, Fabien Guimiot, Homa Adle-Biasette, William B Dobyns, Joseph R Siebert, Rosa Russo, Kathleen J Millen
FOXC1 loss contributes to Dandy-Walker malformation (DWM), a common human cerebellar malformation. Previously, we found that complete Foxc1 loss leads to aberrations in proliferation, neuronal differentiation and migration in the embryonic mouse cerebellum (Haldipur et al., 2014). We now demonstrate that hypomorphic Foxc1 mutant mice have granule and Purkinje cell abnormalities causing subsequent disruptions in postnatal cerebellar foliation and lamination. Particularly striking is the presence of a partially formed posterior lobule which echoes the posterior vermis DW 'tail sign' observed in human imaging studies...
January 16, 2017: ELife
https://www.readbyqxmd.com/read/27878595/cell-type-specific-expression-of-nfix-in-the-developing-and-adult-cerebellum
#7
James Fraser, Alexandra Essebier, Richard M Gronostajski, Mikael Boden, Brandon J Wainwright, Tracey J Harvey, Michael Piper
Transcription factors from the nuclear factor one (NFI) family have been shown to play a central role in regulating neural progenitor cell differentiation within the embryonic and post-natal brain. NFIA and NFIB, for instance, promote the differentiation and functional maturation of granule neurons within the cerebellum. Mice lacking Nfix exhibit delays in the development of neuronal and glial lineages within the cerebellum, but the cell-type-specific expression of this transcription factor remains undefined...
November 23, 2016: Brain Structure & Function
https://www.readbyqxmd.com/read/27802273/fusion-of-human-fetal-mesenchymal-stem-cells-with-degenerating-cerebellar-neurons-in-spinocerebellar-ataxia-type-1-model-mice
#8
Fathul Huda, Yiping Fan, Mamiko Suzuki, Ayumu Konno, Yasunori Matsuzaki, Nobutaka Takahashi, Jerry K Y Chan, Hirokazu Hirai
Mesenchymal stem cells (MSCs) migrate to damaged tissues, where they participate in tissue repair. Human fetal MSCs (hfMSCs), compared with adult MSCs, have higher proliferation rates, a greater differentiation capacity and longer telomeres with reduced senescence. Therefore, transplantation of quality controlled hfMSCs is a promising therapeutic intervention. Previous studies have shown that intravenous or intracortical injections of MSCs result in the emergence of binucleated cerebellar Purkinje cells (PCs) containing an MSC-derived marker protein in mice, thus suggesting a fusion event...
2016: PloS One
https://www.readbyqxmd.com/read/27782041/neonicotinoid-insecticides-alter-the-gene-expression-profile-of-neuron-enriched-cultures-from-neonatal-rat-cerebellum
#9
Junko Kimura-Kuroda, Yasumasa Nishito, Hiroko Yanagisawa, Yoichiro Kuroda, Yukari Komuta, Hitoshi Kawano, Masaharu Hayashi
Neonicotinoids are considered safe because of their low affinities to mammalian nicotinic acetylcholine receptors (nAChRs) relative to insect nAChRs. However, because of importance of nAChRs in mammalian brain development, there remains a need to establish the safety of chronic neonicotinoid exposures with regards to children's health. Here we examined the effects of longterm (14 days) and low dose (1 μM) exposure of neuron-enriched cultures from neonatal rat cerebellum to nicotine and two neonicotinoids: acetamiprid and imidacloprid...
October 4, 2016: International Journal of Environmental Research and Public Health
https://www.readbyqxmd.com/read/27756668/near-infrared-confocal-laser-reflectance-cytoarchitectural-imaging-of-the-substantia-nigra-and-cerebellum-in-the-fresh-human-cadaver
#10
Cletus Cheyuo, Walter Grand, Lucia L Balos
BACKGROUND: Cytoarchitectural neuroimaging remains critical for diagnosis of many brain diseases. Fluorescent dye-enhanced, near-infrared confocal in situ cellular imaging of the brain has been reported. However, impermeability of the blood-brain barrier to most fluorescent dyes limits clinical utility of this modality. The differential degree of reflectance from brain tissue with unenhanced near-infrared imaging may represent an alternative technique for in situ cytoarchitectural neuroimaging...
January 2017: World Neurosurgery
https://www.readbyqxmd.com/read/27737697/loss-of-cerebellar-neurons-in-the-progression-of-lentiviral-disease-effects-of-cns-permeant-antiretroviral-therapy
#11
Christian Wächter, Lee E Eiden, Nedye Naumann, Candan Depboylu, Eberhard Weihe
BACKGROUND: The majority of investigations on HIV-associated neurocognitive disorders (HAND) neglect the cerebellum in spite of emerging evidence for its role in higher cognitive functions and dysfunctions in common neurodegenerative diseases. METHODS: We systematically investigated the molecular and cellular responses of the cerebellum as contributors to lentiviral infection-induced neurodegeneration, in the simian immunodeficiency virus (SIV)-infected rhesus macaque model for HIV infection and HAND...
October 14, 2016: Journal of Neuroinflammation
https://www.readbyqxmd.com/read/27729409/caspase-mediated-apoptosis-induction-in-zebrafish-cerebellar-purkinje-neurons
#12
Thomas Weber, Kazuhiko Namikawa, Barbara Winter, Karina Müller-Brown, Ralf Kühn, Wolfgang Wurst, Reinhard W Köster
The zebrafish is a well-established model organism in which to study in vivo mechanisms of cell communication, differentiation and function. Existing cell ablation methods are either invasive or they rely on the cellular expression of prokaryotic enzymes and the use of antibiotic drugs as cell death-inducing compounds. We have recently established a novel inducible genetic cell ablation system based on tamoxifen-inducible Caspase 8 activity, thereby exploiting mechanisms of cell death intrinsic to most cell types...
November 15, 2016: Development
https://www.readbyqxmd.com/read/27709457/defining-trends-in-global-gene-expression-in-arabian-horses-with-cerebellar-abiotrophy
#13
E Y Scott, M C T Penedo, J D Murray, C J Finno
Equine cerebellar abiotrophy (CA) is a hereditary neurodegenerative disease that affects the Purkinje neurons of the cerebellum and causes ataxia in Arabian foals. Signs of CA are typically first recognized either at birth to any time up to 6 months of age. CA is inherited as an autosomal recessive trait and is associated with a single nucleotide polymorphism (SNP) on equine chromosome 2 (13074277G>A), located in the fourth exon of TOE1 and in proximity to MUTYH on the antisense strand. We hypothesize that unraveling the functional consequences of the CA SNP using RNA-seq will elucidate the molecular pathways underlying the CA phenotype...
April 2017: Cerebellum
https://www.readbyqxmd.com/read/27540357/the-intracellular-signaling-molecule-darpp-32-is-a-marker-for-principal-neurons-in-the-cerebellum-and-cerebellum-like-circuits-of-zebrafish
#14
Lena Robra, Vatsala Thirumalai
The dopamine and cAMP regulated phosphoprotein of apparent molecular weight 32 kDa (Darpp-32) is an inhibitory subunit of protein phosphatase-1 (PP-1). Darpp-32 activity is regulated by multiple ligand-activated G-protein coupled receptors (GPCRs). This protein is coded for by the protein phosphatase-1 regulatory subunit 1b (ppp1r1b) gene. Here, we provide experimental evidence for the presence of multiple isoforms of ppp1r1b in zebrafish. We show that these isoforms are differentially expressed during development with the full-length isoform being maternally deposited...
2016: Frontiers in Neuroanatomy
https://www.readbyqxmd.com/read/27469598/olig2-regulates-purkinje-cell-generation-in-the-early-developing-mouse-cerebellum
#15
Jun Ju, Qian Liu, Yang Zhang, Yuanxiu Liu, Mei Jiang, Liguo Zhang, Xuelian He, Chenchen Peng, Tao Zheng, Q Richard Lu, Hedong Li
The oligodendrocyte transcription factor Olig2 plays a crucial role in the neurogenesis of both spinal cord and brain. In the cerebellum, deletion of both Olig2 and Olig1 results in impaired genesis of Purkinje cells (PCs) and Pax2(+) interneurons. Here, we perform an independent study to show that Olig2 protein is transiently expressed in the cerebellar ventricular zone (VZ) during a period when PCs are specified. Further analyses demonstrate that Olig2 is expressed in both cerebellar VZ progenitors and early-born neurons...
2016: Scientific Reports
https://www.readbyqxmd.com/read/27068104/polycomb-ezh2-controls-the-fate-of-gabaergic-neurons-in-the-embryonic-cerebellum
#16
Xuesong Feng, Aster H Juan, Hongjun A Wang, Kyung Dae Ko, Hossein Zare, Vittorio Sartorelli
Although the genetic interactions between signaling pathways and transcription factors have been largely decoded, much remains to be learned about the epigenetic regulation of cerebellar development. Here, we report that cerebellar deletion of Ezh2, the methyltransferase subunit of the PRC2 complex, results in reduced H3K27me3 and profound transcriptional dysregulation, including that of a set of transcription factors directly involved in cerebellar neuronal cell-type specification and differentiation. Such transcriptional changes lead to increased GABAergic interneurons and decreased Purkinje cells...
June 1, 2016: Development
https://www.readbyqxmd.com/read/26935062/primary-cilia-in-the-murine-cerebellum-and-in-mutant-models-of-medulloblastoma
#17
Chiara Di Pietro, Daniela Marazziti, Gina La Sala, Zeinab Abbaszadeh, Elisabetta Golini, Rafaele Matteoni, Glauco P Tocchini-Valentini
Cellular primary cilia crucially sense and transduce extracellular physicochemical stimuli. Cilium-mediated developmental signaling is tissue and cell type specific. Primary cilia are required for cerebellar differentiation and sonic hedgehog (Shh)-dependent proliferation of neuronal granule precursors. The mammalian G-protein-coupled receptor 37-like 1 is specifically expressed in cerebellar Bergmann glia astrocytes and participates in regulating postnatal cerebellar granule neuron proliferation/differentiation and Bergmann glia and Purkinje neuron maturation...
January 2017: Cellular and Molecular Neurobiology
https://www.readbyqxmd.com/read/26777474/an-optimized-surgical-approach-for-obtaining-stable-extracellular-single-unit-recordings-from-the-cerebellum-of-head-fixed-behaving-mice
#18
Joshua J White, Tao Lin, Amanda M Brown, Marife Arancillo, Elizabeth P Lackey, Trace L Stay, Roy V Sillitoe
BACKGROUND: Electrophysiological recording approaches are essential for understanding brain function. Among these approaches are various methods of performing single-unit recordings. However, a major hurdle to overcome when recording single units in vivo is stability. Poor stability results in a low signal-to-noise ratio, which makes it challenging to isolate neuronal signals. Proper isolation is needed for differentiating a signal from neighboring cells or the noise inherent to electrophysiology...
March 15, 2016: Journal of Neuroscience Methods
https://www.readbyqxmd.com/read/26613348/rapid-generation-of-sub-type-region-specific-neurons-and-neural-networks-from-human-pluripotent-stem-cell-derived-neurospheres
#19
Aynun N Begum, Caleigh Guoynes, Jane Cho, Jijun Hao, Kabirullah Lutfy, Yiling Hong
Stem cell-based neuronal differentiation has provided a unique opportunity for disease modeling and regenerative medicine. Neurospheres are the most commonly used neuroprogenitors for neuronal differentiation, but they often clump in culture, which has always represented a challenge for neurodifferentiation. In this study, we report a novel method and defined culture conditions for generating sub-type or region-specific neurons from human embryonic and induced pluripotent stem cells derived neurosphere without any genetic manipulation...
November 2015: Stem Cell Research
https://www.readbyqxmd.com/read/26581874/classifying-neuronal-subclasses-of-the-cerebellum-through-constellation-pharmacology
#20
Kigen J Curtice, Lee S Leavitt, Kevin Chase, Shrinivasan Raghuraman, Martin P Horvath, Baldomero M Olivera, Russell W Teichert
A pressing need in neurobiology is the comprehensive identification and characterization of neuronal subclasses within the mammalian nervous system. To this end, we used constellation pharmacology as a method to interrogate the neuronal and glial subclasses of the mouse cerebellum individually and simultaneously. We then evaluated the data obtained from constellation-pharmacology experiments by cluster analysis to classify cells into neuronal and glial subclasses, based on their functional expression of glutamate, acetylcholine, and GABA receptors, among other ion channels...
February 1, 2016: Journal of Neurophysiology
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