Hyperactive locomotion in a Drosophila model is a functional readout for the synaptic abnormalities underlying fragile X syndrome
Year: 2017
Type: article
Abstract: FXS model. Genetic or pharmacological intervention on the BMPR2-LIMK pathway ameliorated the synaptic abnormality and locomotion phenotypes of FXS larvae, as well as hyperactivity in an FXS mouse model. Our study demonstrates that (i) the BMPR2-LIMK pathway is a promising therapeutic target for FXS and (ii) the locomotion phenotype of FXS larvae is a quantitative functional readout for the neuromorphological phenotype associated with FXS and is amenable to the screening novel FXS therapeutics.
Source: Science Signaling
Authors: Risa Kashima, Patrick L. Redmond, Prajakta Ghatpande, Sougata Roy, Thomas B. Kornberg, Thomas Hanke, Stefan Knapp, Giorgio Lagna, Akiko Hata
Language: English
Subfield: Genetics
Domain: Life Sciences
Open Access status: green
Funders: Boehringer Ingelheim, Pfizer, Merck, University of Pennsylvania, Wellcome Trust, Ontario Ministry of Economic Development and Innovation, Canada Foundation for Innovation, National Institutes of Health, University of California, San Francisco, Japan Society for the Promotion of Science, National Heart, Lung, and Blood Institute, National Institute of General Medical Sciences, Janssen Biotech, Eshelman Institute for Innovation, University of North Carolina at Chapel Hill
Awards: G1515482456, G172828412, G2051503454, Role of miRNAs in Vascular Physiology, A UK Hub to Catalyse Open Target Discovery., G2656761119, Molecular Mechanisms in Development, G345611779, G3708366301, Mechanisms of TGFB Signal Transduction, Identification of a novel modulator of Pulmonary Artery Hypertension, Role of BMPRII Mutants in the Pathogenesis of Pulmonary Hypertension, G7392968182, G7510464700, G7654023209, G8210223503, Molecular mechanisms in development
OpenAlex ID: W2611360577