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Dopaminergic dysfunction in neurodevelopmental disorders: recent advances and synergistic technologies to aid basic research

Robinson, J. Elliott and Gradinaru, Viviana (2018) Dopaminergic dysfunction in neurodevelopmental disorders: recent advances and synergistic technologies to aid basic research. Current Opinion in Neurobiology, 48 . pp. 17-29. ISSN 0959-4388. http://resolver.caltech.edu/CaltechAUTHORS:20170918-075519928

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Abstract

Neurodevelopmental disorders (NDDs) represent a diverse group of syndromes characterized by abnormal development of the central nervous system and whose symptomatology includes cognitive, emotional, sensory, and motor impairments. The identification of causative genetic defects has allowed for creation of transgenic NDD mouse models that have revealed pathophysiological mechanisms of disease phenotypes in a neural circuit- and cell type-specific manner. Mouse models of several syndromes, including Rett syndrome, Fragile X syndrome, Angelman syndrome, Neurofibromatosis type 1, etc., exhibit abnormalities in the structure and function of dopaminergic circuitry, which regulates motivation, motor behavior, sociability, attention, and executive function. Recent advances in technologies for functional circuit mapping, including tissue clearing, viral vector-based tracing methods, and optical readouts of neural activity, have refined our knowledge of dopaminergic circuits in unperturbed states, yet these tools have not been widely applied to NDD research. Here, we will review recent findings exploring dopaminergic function in NDD models and discuss the promise of new tools to probe NDD pathophysiology in these circuits.


Item Type:Article
Related URLs:
URLURL TypeDescription
https://doi.org/10.1016/j.conb.2017.08.003DOIArticle
http://www.sciencedirect.com/science/article/pii/S0959438817301605PublisherArticle
ORCID:
AuthorORCID
Gradinaru, Viviana0000-0001-5868-348X
Additional Information:© 2017 Elsevier Ltd. Available online 30 August 2017. This review comes from a themed issue on Neurobiology of disease Edited by Anatol Kreitzer and Claudia Bagni. We would like to acknowledge support from the Children's Tumor Foundation (Young Investigator Award 2016-01-006 to JER), the National Institutes of Health (IDP20D017782 and R01AG047664 to VG), the Heritage Medical Research Institute (VG), and the Tianqiao and Chrissy Chen Institute for Neuroscience at Caltech. We would like to thank Jennifer Treweek, Benjamin Deverman, Ken Chan, Min Jang, Alon Greenbaum, and Ryan Cho for histological images used in the manuscript figures. Conflict of interest statement: Nothing declared.
Funders:
Funding AgencyGrant Number
Children’s Tumor Foundation2016-01-006
NIHIDP20D017782-01
NIHR01AG047664
Record Number:CaltechAUTHORS:20170918-075519928
Persistent URL:http://resolver.caltech.edu/CaltechAUTHORS:20170918-075519928
Official Citation:J Elliott Robinson, Viviana Gradinaru, Dopaminergic dysfunction in neurodevelopmental disorders: recent advances and synergistic technologies to aid basic research, In Current Opinion in Neurobiology, Volume 48, 2018, Pages 17-29, ISSN 0959-4388, https://doi.org/10.1016/j.conb.2017.08.003. (http://www.sciencedirect.com/science/article/pii/S0959438817301605)
Usage Policy:No commercial reproduction, distribution, display or performance rights in this work are provided.
ID Code:81511
Collection:CaltechAUTHORS
Deposited By: Ruth Sustaita
Deposited On:18 Sep 2017 21:11
Last Modified:18 Sep 2017 21:11

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