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J. Dev. Biol. 2018, 6(3), 21; https://doi.org/10.3390/jdb6030021

Drosophila as a Model for Assessing the Function of RNA-Binding Proteins during Neurogenesis and Neurological Disease

Department of Biology, University of Colorado Colorado Springs, 1420 Austin Bluffs Parkway, Colorado Springs, CO 80918, USA
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Received: 6 July 2018 / Revised: 15 August 2018 / Accepted: 15 August 2018 / Published: 18 August 2018
(This article belongs to the Special Issue Drosophila - A Model System for Developmental Biology)
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Abstract

An outstanding question in developmental neurobiology is how RNA processing events contribute to the regulation of neurogenesis. RNA processing events are increasingly recognized as playing fundamental roles in regulating multiple developmental events during neurogenesis, from the asymmetric divisions of neural stem cells, to the generation of complex and diverse neurite morphologies. Indeed, both asymmetric cell division and neurite morphogenesis are often achieved by mechanisms that generate asymmetric protein distributions, including post-transcriptional gene regulatory mechanisms such as the transport of translationally silent messenger RNAs (mRNAs) and local translation of mRNAs within neurites. Additionally, defects in RNA splicing have emerged as a common theme in many neurodegenerative disorders, highlighting the importance of RNA processing in maintaining neuronal circuitry. RNA-binding proteins (RBPs) play an integral role in splicing and post-transcriptional gene regulation, and mutations in RBPs have been linked with multiple neurological disorders including autism, dementia, amyotrophic lateral sclerosis (ALS), spinal muscular atrophy (SMA), Fragile X syndrome (FXS), and X-linked intellectual disability disorder. Despite their widespread nature and roles in neurological disease, the molecular mechanisms and networks of regulated target RNAs have been defined for only a small number of specific RBPs. This review aims to highlight recent studies in Drosophila that have advanced our knowledge of how RBP dysfunction contributes to neurological disease. View Full-Text
Keywords: Drosophila; RNA-binding proteins; neurons; neurodegeneration; amyotrophic lateral sclerosis; Fmr1; spinal muscular atrophy; TDP-43; FUS; C9orf72; shep; brat Drosophila; RNA-binding proteins; neurons; neurodegeneration; amyotrophic lateral sclerosis; Fmr1; spinal muscular atrophy; TDP-43; FUS; C9orf72; shep; brat
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This is an open access article distributed under the Creative Commons Attribution License which permits unrestricted use, distribution, and reproduction in any medium, provided the original work is properly cited. (CC BY 4.0).
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Olesnicky, E.C.; Wright, E.G. Drosophila as a Model for Assessing the Function of RNA-Binding Proteins during Neurogenesis and Neurological Disease. J. Dev. Biol. 2018, 6, 21.

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