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. 2019 Dec 12;9:19006. doi: 10.1038/s41598-019-54314-8

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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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Single-breath ILC in LOPD and control subjects. Single-breath ILC responses differed according to the group (control: closed circles, Pompe: open squares) and load magnitude. During loaded breaths, peak inspiratory flow (a) progressively decreased in LOPD as compared to control subjects (3-way interaction, F = 2.794, p < 0.05). Compared to recovery breaths, peak expiratory flow (b) was lower during loaded breaths, but neither group assignment nor load magnitude affected the loaded peak expiratory flow. The inspired volume of loaded breaths (c) was similar for both groups at the lowest load magnitude, but in LOPD subjects, volume progressively decreased with larger load magnitudes (breath-load interaction, F = 19.403, p < 0.005). Inspiratory time (d) did not vary by breath type, load magnitude, or group, largely due to variability of breath timing in the control subjects. Expiratory time (e) was lower for loaded breaths, compared to unloaded controls and recovery breaths (F = 5.767, p < 0.05). Despite the group differences in flow and volume ILC, the maximal rate of inspiratory pressure development (dP/dt) of the loaded breaths (f), an estimate of neuromuscular activation, did not differ between the groups. (Mean, SEM depicted).