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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. 4 — Neurons lacking FMRP have reduced I_NaP densities, and their burst properties are insensitive to riluzole. A Persistent sodium currents (I_NaP) evoked by a slow depolarizing ramp (−100 to −20 mV, 20 mV/s) either before (traces a) or during (traces b) application of TTX. The TTX-sensitive current (traces c) was isolated by subtracting traces a and b. B I–V curves plotted from the ramp-evoked I_NaP, showing the decreased I_NaP in neurons lacking FMRP. Currents are normalized to the corresponding cell capacitance. C Quantification of current density at −20 mV show significant decrease of the persistent sodium current in neurons lacking FMRP. Overall, the mean current densities are −1.44 ± 0.17 (FMR1^+/y, n = 18, N = 3); −1.73 ± 0.33 (CON2, n = 10, N = 3); −0.27 ± 0.03 (FMR1^−/y, n = 18, N = 3); −0.35 ± 0.098 (FXS3, n = 9, N = 3). **p < 0.01, ***p < 0.001, two-way repeated measures ANOVA with post hoc Tukey’s test, paired t test and Wilcoxon test. D Representative traces illustrating spontaneous bursts recorded from either a FMR1^+/y neuron or a FMR1^−/y neuron before and after the application of riluzole (0.1 μM). E Quantification of the number of bursts recorded from FMR1^+/y, CON2, FMR1^−/y, or FXS3 neurons before (open symbols) or after (closed symbols) application of riluzole. Overall, the mean number of bursts recorded are 5.733 ± 0.72 (FMR1^+/y, n = 15, N = 3); 5.733 ± 0.65 (FMR1^+/y + riluzole, n = 15, N = 3); 6.3 ± 0.9 (CON2, n = 10, N = 3); 7 ± 1.12 (CON2 + riluzole, n = 10, N = 3); 42.33 ± 8.92 (FMR1^−/y, n = 9, N = 3); 38.89 ± 8.49 (FMR1^−/y + riluzole, n = 9, N = 3); 34.1 ± 9.52 (FXS3, n = 10, N = 3); 34.3 ± 9.62 (FXS3 + riluzole, n = 10, N = 3). F Quantification of the duration of bursts recorded from FMR1^+/y , CON2, FMR1^−/y, or FXS3 neurons before (open symbols) or after (closed symbols) application of riluzole. Overall, the mean burst durations are 47.86 ± 6.27 s (FMR1^+/y); 14.14 ± 1.33 s (FMR1^+/y + riluzole); 47.81 ± 12.14 s (CON2); 25.55 ± 9.54 s (CON2 + riluzole); 8.77 ± 1.34 s (FMR1^−/y ); 9.43 ± 1.62 s (FMR1^−/y + riluzole); 8.91 ± 2.19 s (FXS3); 9.01 ± 2.52 (FXS3 + riluzole) **p < 0.01; ***p < 0.001, paired t test, Wilcoxon test