Towards a 3D spheroid system for modelling leukodystrophies 139 4 FUNDING This work was supported by the European Joint Programme on Rare Diseases (EJPRD19201, NG4LEUKO). COMPETING INTERESTS The authors report no competing interests. AUTHOR CONTRIBUTIONS EM, CR generated spheroids and collected samples with assistance of LMLK and GC. FC, RA and IM analysed scRNA-seq data. VMH, AG and OB supervised the project. LMLK visualized the data and wrote the chapter in consultation with other authors. REFERENCES Dooves, S., Kok, L. M. L., Holmes, D. B., Breeuwsma, N., Breur, M., Bugiani, M., Wolf, N. I., & Heine, V. M. (2023). Cortical interneuron development is affected in 4H leukodystrophy. Brain, 146(7), 2846-2860. https://doi.org/10.1093/brain/awad017 Holmes, D. B., & Heine, V. M. (2017). Simplified 3D protocol capable of generating early cortical neuroepithelium. Biol Open, 6(3), 402-406. https://doi.org/10.1242/bio.021725 Mangiameli, E., Cecchele, A., Morena, F., Sanvito, F., Matafora, V., Cattaneo, A., Della Volpe, L., Gnani, D., Paulis, M., Susani, L., Martino, S., Di Micco, R., Bachi, A., & Gritti, A. (2021). Human iPSC-based neurodevelopmental models of globoid cell leukodystrophy uncover patient- and cell type-specific disease phenotypes. Stem Cell Reports, 16(6), 1478-1495. https://doi.org/10.1016/j.stemcr.2021.04.011 Marton, R. M., Miura, Y., Sloan, S. A., Li, Q., Revah, O., Levy, R. J., Huguenard, J. R., & Pasca, S. P. (2019). Differentiation and maturation of oligodendrocytes in human three-dimensional neural cultures. Nat Neurosci, 22(3), 484-491. https://doi.org/10.1038/s41593-018-0316-9 Naldini, M. M., Casirati, G., Barcella, M., Rancoita, P. M. V., Cosentino, A., Caserta, C., Pavesi, F., Zonari, E., Desantis, G., Gilioli, D., Carrabba, M. G., Vago, L., Bernardi, M., Di Micco, R., Di Serio, C., Merelli, I., Volpin, M., Montini, E., Ciceri, F., & Gentner, B. (2023). Longitudinal single-cell profiling of chemotherapy response in acute myeloid leukemia. Nat Commun, 14(1), 1285. https://doi.org/10.1038/s41467-023-36969-0 Pollen, A. A., Nowakowski, T. J., Chen, J., Retallack, H., Sandoval-Espinosa, C., Nicholas, C. R., Shuga, J., Liu, S. J., Oldham, M. C., Diaz, A., Lim, D. A., Leyrat, A. A., West, J. A., & Kriegstein, A. R. (2015). Molecular identity of human outer radial glia during cortical development. Cell, 163(1), 55-67. https://doi.org/10.1016/j.cell.2015.09.004 Spaas, J., van Veggel, L., Schepers, M., Tiane, A., van Horssen, J., Wilson, D. M., 3rd, Moya, P. R., Piccart, E., Hellings, N., Eijnde, B. O., Derave, W., Schreiber, R., & Vanmierlo, T. (2021). Oxidative stress and impaired oligodendrocyte precursor cell differentiation in neurological disorders. Cell Mol Life Sci, 78(10), 4615-4637. https://doi.org/10.1007/s00018-021-03802-0
RkJQdWJsaXNoZXIy MTk4NDMw