Proper neural networks rely on the neurons' ability to generate neurites (axons and dendrites) and form synaptic connections with appropriate partners. Impairments in neuronal morphology and synapse composition in specific subtypes of neurons have been described in various mental health conditions, ranging from schizophrenia and bipolar disorder to autism spectrum disorder (ASD). Such altered neuronal morphology and synapse assembly presumably underlie disrupted neural circuit function, and therefore disrupted behavioral patterns. The objective of Project 4 is to create a suite of cellular imaging-based assays in hIPSC-derived cultures of differentiated neurons. These assays are based on quantitative fluorescence microscopy, and will focus on specific aspects of neuritogenesis and synaptogenesis that are relevant to ASD. The assays will be compatible with high-content screening technology, and will be applied to engineered hIPSC ASD models.
| Mesci, Pinar; Macia, Angela; LaRock, Christopher N et al. (2018) Modeling neuro-immune interactions during Zika virus infection. Hum Mol Genet 27:41-52 |
| Chailangkarn, Thanathom; Noree, Chalongrat; Muotri, Alysson R (2018) The contribution of GTF2I haploinsufficiency to Williams syndrome. Mol Cell Probes 40:45-51 |
| Brito, Anita; Russo, Fabiele Baldino; Muotri, Alysson Renato et al. (2018) Autism spectrum disorders and disease modeling using stem cells. Cell Tissue Res 371:153-160 |
| Fernandes, Isabella R; Cruz, Ana C P; Ferrasa, Adriano et al. (2018) Genetic variations on SETD5 underlying autistic conditions. Dev Neurobiol 78:500-518 |
| Mesci, Pinar; Macia, Angela; Moore, Spencer M et al. (2018) Blocking Zika virus vertical transmission. Sci Rep 8:1218 |
| Beltrão-Braga, Patricia C B; Muotri, Alysson R (2017) Modeling autism spectrum disorders with human neurons. Brain Res 1656:49-54 |
| Song, Yan; Botvinnik, Olga B; Lovci, Michael T et al. (2017) Single-Cell Alternative Splicing Analysis with Expedition Reveals Splicing Dynamics during Neuron Differentiation. Mol Cell 67:148-161.e5 |
| de Souza, Janaina S; Carromeu, Cassiano; Torres, Laila B et al. (2017) IGF1 neuronal response in the absence of MECP2 is dependent on TRalpha 3. Hum Mol Genet 26:270-281 |
| Shiryaev, Sergey A; Mesci, Pinar; Pinto, Antonella et al. (2017) Repurposing of the anti-malaria drug chloroquine for Zika Virus treatment and prophylaxis. Sci Rep 7:15771 |
| Kakaradov, Boyko; Arsenio, Janilyn; Widjaja, Christella E et al. (2017) Early transcriptional and epigenetic regulation of CD8+ T cell differentiation revealed by single-cell RNA sequencing. Nat Immunol 18:422-432 |
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