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. 2019 Oct 4;9:14324. doi: 10.1038/s41598-019-50550-0

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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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Muscle function in dy^3K/dy^3K mice throughout the disease course. (a,b) Righting reflex and hind limb suspension test do not show significant impairment of neuromuscular function in 2- and 7-day-old dy^3K/dy^3K pups. Righting reflex in 2-day-old mice was scored as follows: 0: no attempt to right itself, very passive; 1: poor attempt to right itself; 2: fair attempt to right itself, active; 3: managed to turn. (c) Righting reflex, hind limb suspension and grip strength (hang-wire) test in 11-day-old animals. No striking difficulties in performance of dy^3K/dy^3K mice were noted during righting reflex and hind limb suspension tests. Hang-wire test showed dysfunction of dy^3K/dy^3K neuromuscular system when attempting a more complex physical activity. (d) Two grip strength tests (hang-wire and mesh grid) and open field test (activity in a new cage) in 14-day-old mice. Both grip strength tests show enormous impairment of muscle function in dy^3K/dy^3K mice. The performance of dy^3K/dy^3K animals in the open field test, on the other hand, was not significantly diminished.