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. 2020 Jul 16;10:11827. doi: 10.1038/s41598-020-68794-6

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© The Author(s) 2020

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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Mass-spectrometry analysis in FUS^WT and FUS^P525L motoneurons. (a) Outline of the generation of pure motoneuron samples from isogenic FUS^WT and FUS^P525L hiPSC lines. An Hb9::GFP reporter was used for isolation of motoneuron progenitors by FACS. After subsequent maturation, protein samples from four independent differentiation experiments were collected for proteome analysis by mass-spectrometry. This figure was drawn using the vector image bank of Servier Medical Art (https://smart.servier.com/). Servier Medical Art by Servier is licensed under a Creative Commons Attribution 3.0 Unported License. (b, c) Word clouds generated by FIDEA (https://omictools.com/fidea-tool)³⁴ representing GO “Biological Process”, “Molecular Function” and “Cellular Component” terms enriched in the set of proteins that are downregulated (b) or upregulated (c) in FUS^P525L motoneurons. The categories are represented with a character size proportional to the statistical significance of their enrichment. The images have been taken by Maria Giovanna Garone. (d) Table showing ALS-linked genes,^24,28–33 from the DISEASES web resource¹⁴, encoding for proteins that are significantly up- or downregulated in FUS^P525L motoneurons.