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. 2019 Jan 30;10:492. doi: 10.1038/s41467-018-08283-7

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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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Fig. 6 — Phenotypic correction of muscular dystrophy in SCID/mdx mice. a SCID/mdx mice injected with ssAAV9-Sk-CRM4-Des-MD1Δ only, ssAAV9-Sk-CRM4-Des-FST only, and ssAAV9-Sk-CRM4-Des-MD1Δ with ssAAV9-Sk-CRM4-Des-FST combination therapy (2 × 10¹⁰ vg/mouse of each vector) were subjected to a treadmill assay. Physical performance of the 20-week-old SCID/mdx mice treated with the different therapeutic vectors compared to the untreated age-matched control SCID/mdx mice was determined by measuring the distance covered. Results were presented as mean ± standard error of the mean, (% increase in distance covered relative to untreated SCID/mdx was indicated. b–d Improvement of pathophysiological properties in SCID/mdx mice injected with ssAAV9-Sk-CRM4-Des-MD1Δ, ssAAV9-Sk-CRM4-Des-FST, ssAAV9-Sk-CRM4-Des-MD1Δ, and ssAAV9-Sk-CRM4-Des-FST combination therapy. b Hematoxylin and eosin staining of 5 μm thick transverse sections of the tibialis anterior muscle from mice treated with the different therapeutic vectors were compared with those of age-matched control C57BL/6 and SCID/mdx mice treated with PBS. The scale bars indicate 50 μm. c Graphical representation of the % of central nucleation in muscle fibers from treated sections versus the age-matched non-treated sections. Results were presented as mean ± standard error of the mean, ***p < 0.001 and ****p < 0.0001 using Student’s t-test (n = 4–9). d Muscle fiber cross-sectional area (expressed in pixels) of each of the different groups of SCID/mdx mice treated with the different therapeutic vectors (at a dose of 2 × 10¹⁰ vg/mouse). Untreated SCID/mdx and C57BL/6 mice were used as controls. Results were presented as mean ± standard error of the mean, *p < 0.05; ***p < 0.001, and ****p < 0.0001 using Student’s t-test (n = 4–5)