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. 2020 Jun 19;11:52. doi: 10.1186/s13229-020-00351-4

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© The Author(s) 2020

Open AccessThis 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 licence, and indicate if changes were made. The images or other third party material in this article are included in the article's Creative Commons licence, unless indicated otherwise in a credit line to the material. If material is not included in the article's Creative Commons licence 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 licence, visit http://creativecommons.org/licenses/by/4.0/. The Creative Commons Public Domain Dedication waiver (http://creativecommons.org/publicdomain/zero/1.0/) applies to the data made available in this article, unless otherwise stated in a credit line to the data.

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Fig. 5 — In neurons lacking FMRP activation of voltage-dependent Na⁺ channels decreases action potential burst frequencies but increases their duration. a Representative traces of a FMR1^+/y neuron illustrating spontaneous action potential bursting before and after the application of veratridine (0.5 μM). b As in a but illustrating traces obtained from a FMR1^−/y neuron. c Quantification of burst number in FMR1^+/y, CON2, FMR1^−/y and FXS3 lines before and after the application of veratridine. Veratridine reduced significantly the frequency of bursts in lines lacking FMRP. Overall, the mean number of bursts are 6 ± 0.92 (FMR1^+/y, n = 10, N = 3); 7.3 ± 0.76 (FMR1^+/y + veratridine, n = 10, N = 3); 7.87 ± 1.46 (CON2, n = 8, N = 3); 8 ± 1.45 (CON2 + veratridine, n = 8, N = 3); 39.29 ± 5.27 (FMR1^−/y , n = 14, N = 3); 8.857 ± 0.98 (FMR1^−/y + veratridine, n = 14, N = 3); 24.45 ± 4.37 (FXS3, n = 11, N = 3); 7.818 ± 1.32 (FXS3 + veratridine, n = 11, N = 3). d Quantification of action potential burst durations in each of the four lines examined. Veratridine increased significantly the duration of bursts in FMR1^−/y and FXS3 lines and had no overall effect on FMR1^+/y and CON2 lines. Overall, the mean burst durations are 39.09 ± 6.47 s (FMR1^+/y); 34.34 ± 4.5 s (FMR1^+/y + veratridine); 37.42 ± 7.39 s (CON2); 35.77 ± 7.25 s (CON2 + veratridine); 7.318 ± 0.64 s (FMR1^−/y ); 25.5 ± 3.23 s (FMR1^−/y + veratridine); 7.004 ± 1.28 s (FXS3); 32.26 ± 5.66 (FXS3 + veratridine). For both c and d, **p < 0.01, ***p < 0.001, paired t test, Wilcoxon test