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Behavioral Characterization of an Angelman Syndrome Mouse Model
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SYSNO ASEP 0581626 Document Type J - Journal Article R&D Document Type Journal Article Subsidiary J Článek ve SCOPUS Title Behavioral Characterization of an Angelman Syndrome Mouse Model Author(s) Kubik-Zahorodna, Agnieszka (UMG-J)
Procházka, Jan (UMG-J) ORCID
Sedláček, Radislav (UMG-J) RIDNumber of authors 0 Article number e65182 Source Title Jove-Journal of Visualized Experiments - ISSN 1940-087X
Roč. 2023, č. 200 (2023)Number of pages 18 s. Language eng - English Country US - United States Keywords Angelman Syndrome ; Animals ; Behavior ; Animal ; Disease Models OECD category Biochemistry and molecular biology R&D Projects LM2018126 GA MŠMT - Ministry of Education, Youth and Sports (MEYS) EF16_013/0001789 GA MŠMT - Ministry of Education, Youth and Sports (MEYS) EF18_046/0015861 GA MŠMT - Ministry of Education, Youth and Sports (MEYS) ED2.1.00/19.0395 GA MŠMT - Ministry of Education, Youth and Sports (MEYS) LM2023036 GA MŠMT - Ministry of Education, Youth and Sports (MEYS) Method of publishing Limited access Institutional support UMG-J - RVO:68378050 EID SCOPUS 85175006976 DOI 10.3791/65182 Annotation This manuscript describes a battery of behavioral tests available to characterize Angelman syndrome (AS)-like phenotypes in an established murine model of AS. We use the rotarod learning paradigm, detailed gait analysis, and nest building test to detect and characterize animal motor impairments. We test animal emotionality in the open field and elevated plus maze tests, as well as the affect in the tail suspension test. When AS mice are tested in the open field test, the results should be interpreted with care, since motor dysfunctions influence mouse behavior in the maze and alter activity scores. The reproducibility and effectiveness of the presented behavioral tests has already been validated in several independent Uba3a mouse lines with different knockout variants, establishing this set of tests as an excellent validation tool in AS research. Models with the relevant construct and face validity will warrant further investigations to elucidate the pathophysiology of the disease and grant the development of causal treatments. Workplace Institute of Molecular Genetics Contact Nikol Škňouřilová, nikol.sknourilova@img.cas.cz, Tel.: 241 063 217 Year of Publishing 2024 Electronic address https://www.jove.com/t/65182/behavioral-characterization-of-an-angelman-syndrome-mouse-model
Number of the records: 1