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. 2019 Dec 10;9:18666. doi: 10.1038/s41598-019-53508-4

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© The Author(s) 2019

Open Access This article is licensed under a Creative Commons Attribution 4.0 International License, which permits use, sharing, adaptation, distribution and reproduction in any medium or format, as long as you give appropriate credit to the original author(s) and the source, provide a link to the Creative Commons license, and indicate if changes were made. The images or other third party material in this article are included in the article’s Creative Commons license, unless indicated otherwise in a credit line to the material. If material is not included in the article’s Creative Commons license and your intended use is not permitted by statutory regulation or exceeds the permitted use, you will need to obtain permission directly from the copyright holder. To view a copy of this license, visit http://creativecommons.org/licenses/by/4.0/.

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Gain of caz/FUS function in muscle impairs motor performance. (A) Overexpression of either caz or human FUS (hFUS) in muscle leads to adult flies with reduced mobility. Hence, on assessment, the first fly took significantly longer to reach a pre-set threshold. Furthermore, at a population level, climbing success was profoundly reduced. (B) Flies were in their majority non-fliers. Thus, when tested, a significant percentage dropped to the base (sector 1) of the Droso-Drome. Importantly, in combination with Gem3^BART, muscle-specific overexpression of either caz or hFUS induced lethality prior to eclosion. Data presented are the mean ± S.E.M. of at least 4 independent experiments, and for each time point measured, n ≥ 60 per genotype. Symbols indicate the sex of the genotype assessed: ♂ = males, ♀ = females, and ♀♂ = males + females. Adult-viable flies were assessed at day 5 post-eclosion. Significance as tested by the unpaired t-test (A) and two-way ANOVA, followed by Bonferroni’s post hoc tests (B) is indicated by the exact p-value.