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. 2022 Jun 8;13:3180. doi: 10.1038/s41467-022-30778-7

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© The Author(s) 2022

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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Fig. 7 — a Adult miR-1/206/133 tKO (n = 4) and adult Crk-SPH (n = 3) show muscle weakness, abnormal locomotion in footprint assays, and hindlimb clasping during tail suspension (n = 3, control = miR-1-1/133a-2^−/−//miR-1-2/133a-1^lox/lox//miR-206/133b^−/−//HSA-rtTA-TRE-Cre^+/+ [control and tKO female ~44 weeks; adult Crk-SPH 13-16 weeks, females]). Scale-bar: 1 cm. b, c Adult miR-1/206/133 tKO (n = 4) show impaired motor function in Rotarod tests (n = 3, control = miR-1-1/133a-2^−/−//miR-1-2/133a-1^lox/lox//miR-206/133b^−/−//HSA-rtTA-TRE-Cre^+/+ [male, female, ~44 weeks]; Mann-Whitney-U test, one-tailed, *p = 0.0286). d-e Adult Crk-SPH mice show impaired motor functions in Rotarod tests (n = 6, control = CAG-LSL-dCas9-SPH^+/+, n = 7 Crk-SPH animals [female, 13–16 weeks]; Mann-Whitney-U test, one-tailed, **p = 0.0051 (d), **p = 0.0029 (e)). f, g GSEA of adult miR-1/206/133 tKO (n = 3) and control (n = 2) TA (control = miR-1-1/133a-2^−/−//miR-1-2/133a-1^lox/lox//miR-206/133b^−/−//HSA-rtTA-TRE-Cre^+/+, familywise-error rate method (FWER), p = < 0.004 (f), false discovery rate (FDR) p = 0.001 (g) [male, ~44 weeks]). h, i Quantification of BTX-stained postsynaptic regions in TA transversal sections from adult miR-1/206/133 tKO (n = 3) and adult Crk-SPH (n = 3) mice in comparison to controls (n = 3, control of adult tKO= miR-1-1/133a-2^−/−//miR-1-2/133a-1^lox/lox//miR-206/133b^−/−//HSA-rtTA-TRE- Cre^+/+, [~44 weeks, male]; n = 3, control of Crk-SPH = HSA-rtTA-TRE-Cre^+/+ or CAG-LSL-dCas9-SPH^+/+[10–14 weeks, female], Mann-Whitney-U tests, one tailed, *p = 0.05). Scale-bar in r: 50 µm. j BTX staining (red) of isolated single myofibers of flexor digitorum brevis muscle from adult miR-1/206/133 tKO (n = 3) in comparison to controls (n = 3, control of adult miR-1/206/133 tKO = miR-1-1/133a-2^−/−//miR-1-2/133a-1^lox/lox//miR-206/133b^−/−//HSA-rtTA-TRE- Cre^+/+, [~44 weeks, male]). DAPI staining in blue. Z-stacks. Scale-bar: 20 µm. Source data are provided as a Source Data file. Data are presented as mean values +/− SEM in b–e and i.