Dopaminergic loss of cyclin-dependent kinase-like 5 recapitulates methylphenidate-remediable hyperlocomotion in mouse model of CDKL5 deficiency disorder Downloaded from https://academic.oup.com/hmg/article-abstract/doi/10.1093/hmg/ddaa122/5863032 by University of Exeter user on 26 June 2020 Cian-Ling Jhang1, Hom-Yi Lee3,4, Jinchung Chen5, Wenlin Liao1,2 * 1 Institute of Neuroscience, 2 Research Center for Mind, Brain and Learning, National Cheng-Chi University, Taipei 116, Taiwan 3 Department of Psychology, 4 Department of Speech Language Pathology and Audiology, Chung Shan Medical University, Taichung 402, Taiwan 5 Graduate Institute of Biomedical Sciences, Chang Gung University, Taoyuan 333, Taiwan UNCORRECTED MANUSCRIPT © The Author(s) 2020. Published by Oxford University Press. All rights reserved. For Permissions, please email:
[email protected] page 1 Downloaded from https://academic.oup.com/hmg/article-abstract/doi/10.1093/hmg/ddaa122/5863032 by University of Exeter user on 26 June 2020 * To whom correspondence should be addressed: Wenlin Liao, PhD Associate Professor Institute of Neuroscience, National Cheng-Chi University 64, Sec. 2, Chi-Nan Road, Wen-Shan District Taipei 116, Taiwan Phone: 886-2-29393091 ext. 89621 UNCORRECTEDEmail:
[email protected] MANUSCRIPT page 2 ABSTRACT Cyclin-dependent kinase-like 5 (CDKL5), a serine-threonine kinase encoded by an X-linked gene, is highly expressed in mammalian forebrain. Mutations in this gene Downloaded from https://academic.oup.com/hmg/article-abstract/doi/10.1093/hmg/ddaa122/5863032 by University of Exeter user on 26 June 2020 cause CDKL5 deficiency disorder, a neurodevelopmental encephalopathy characterized by early-onset seizures, motor dysfunction and intellectual disability.