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Modeling hereditary diffuse leukoencephalopathy with axonal spheroids using microglia-sufficient brain organoidsPreprint

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Modeling hereditary diffuse leukoencephalopathy with axonal spheroids using microglia-sufficient brain organoids

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Reviewer #2 (Public Review): Modeling Hereditary Diffuse Leukoencephalopathy with Axonal Spheroids using microglia-sufficient brain organoids

Wong, W. J., Zhu, Y., Wang, H. T., et al. (2024) · eLife · doi:10.7554/elife.96693.2.sa0 · Cited by 3 · see citing works

Loss of Ramified Microglia Precedes Axonal Spheroid Formation in Adult-Onset Leukoencephalopathy with Axonal Spheroids.

Alturkustani, M. A., Zhang, Q., Alyamany, B. M., & Ang, L.-C. (2020) · Free Neuropathology · doi:10.17879/freeneuropathology-2020-2971 · Cited by 2 · see citing works

Loss of homeostatic microglia in rare neurological disorders: implications for cell transplantation

Ikeuchi, T., Fitrah, Y. A., & Shu, B. (2021) · Folia Pharmacologica Japonica · doi:10.1254/fpj.21017 · Cited by 2 · see citing works

Cerebellar white matter development is regulated by fractalkine-dependent microglia phagocytosis of oligodendrocyte progenitor cells

Chappell, M. K., Shelestak, J., Irfan, M., Shelestak, E., Nemes, A. D., Mey, G. M., Venkat, L., Surabhi, A., & DeSilva, T. M. (2025) · iScience · doi:10.1016/j.isci.2025.113458 · Cited by 2 · see citing works

Soluble CSF1R alleviates microgliopathy in a CSF1R-related leukoencephalopathy (CRL) mouse model

Zhou, Y., Hu, B., Luo, J., Wang, X., Huang, X., Li, Y., Zhang, X., Bu, G., Zhang, H., Zhang, Y., et al. (2025) · Journal of Neuroinflammation · doi:10.1186/s12974-025-03648-4 · Cited by 1 · see citing works

Adult-onset leukoencephalopathy with axonal spheroids and pigmented glia – An unusual cause of dementia

Klotz, S., Riederer, F., Hergovich, N., Schlager, T., Steinkellner, L., Fertl, E., Baumgartner, C., Zimprich, A. A., & Gelpí, E. (2019) · Clinical Neuropathology · doi:10.5414/np301253 · Cited by 1 · see citing works

A kinase-dead Csf1r mutation associated with adult-onset leukoencephalopathy has a dominant-negative impact on CSF1R signalingPreprint

Stables, J., Green, E. K., Sehgal, A., Patkar, O. L., Keshvari, S., Taylor, I., Ashcroft, M. E., Grabert, K., Wollscheid-Lengeling, E., Szymkowiak, S., et al. (2021) · bioRxiv (Cold Spring Harbor Laboratory) · doi:10.1101/2021.09.29.462493 · Cited by 1 · see citing works

Microglia are not required for normal postnatal brain development and cognitive function in the ratPreprint

Green, E. K., Li, X., Guo, Y., Carter‐Cusack, D., Rojo, R., Lyall, M., Bell, A., Zheng, H., Teenan, O., Till, S. M., et al. (2025) · bioRxiv (Cold Spring Harbor Laboratory) · doi:10.64898/2025.12.22.694658 · Cited by 1 · see citing works

Anti-GABAbR antibody positivity in a patient with hereditary diffuse leukoencephalopathy with spheroids (HDLS): a case reportPreprint

Cao, H., Yang, L., Yu, L., Qin, W. D., Zhang, J., Li, Y., Yang, S., & Hu, W. L. (2019) · Research Square · doi:10.21203/rs.2.9165/v1

An adapted stem cell-derived microglia protocol for the study of microgliopathies and other neurological disordersPreprint

Dorion, M., Casas, D., Yaqubi, M., Fleming, P., Chen, C. X. ‐., Piscopo, V. E. C., Nicouleau, M., Goldsmith, T., Shlaifer, I., MacDonald, A., et al. (2023) · bioRxiv (Cold Spring Harbor Laboratory) · doi:10.1101/2023.09.08.556933

Modeling Hereditary Diffuse Leukoencephalopathy with Axonal Spheroids using microglia-sufficient brain organoidsPreprint

Wong, W. J., Zhu, Y., Wang, H. T., Qian, J. W., Li, Z., Li, S., Liu, Z. Y., Guo, W., Zhang, S. Y., Su, B., et al. (2024) · eLife · doi:10.7554/elife.96693.1

Author response: Modeling Hereditary Diffuse Leukoencephalopathy with Axonal Spheroids using microglia-sufficient brain organoids

Wong, W. J., Zhu, Y. W., Wang, H. T., Qian, J. W., Li, Z., Li, S., Liu, Z. Y., Guo, W., Zhang, S. Y., Su, B., et al. (2024) · eLife · doi:10.7554/elife.96693.2.sa3

Reviewer #1 (Public Review): Modeling Hereditary Diffuse Leukoencephalopathy with Axonal Spheroids using microglia-sufficient brain organoids

Wong, W. J., Zhu, Y., Wang, H. T., et al. (2024) · eLife · doi:10.7554/elife.96693.2.sa1

Modeling Hereditary Diffuse Leukoencephalopathy with Axonal Spheroids using microglia-sufficient brain organoidsPreprint

Wong, W. J., Zhu, Y. W., Wang, H. T., Qian, J. W., Li, Z., Li, S., Liu, Z. Y., Guo, W., Zhang, S. Y., Su, B., et al. (2024) · eLife · doi:10.7554/elife.96693.2

eLife Assessment: Modeling Hereditary Diffuse Leukoencephalopathy with Axonal Spheroids using microglia-sufficient brain organoids

Takeda, K. (2024) · eLife · doi:10.7554/elife.96693.2.sa2

Additional file 3 of Epigenetic modulation of AREL1 and increased HLA expression in brains of multiple system atrophy patients

Rydbirk, R., Folke, J., Busato, F., Roché, E., Chauhan, A. S., Løkkegaard, A., Hejl, A.-M., Bode, M., Blaabjerg, M., Møller, M. L., et al. (2020) · Figshare · doi:10.6084/m9.figshare.11960322

Additional file 7 of Minocycline protects against microgliopathy in a Csf1r haplo-insufficient mouse model of adult-onset leukoencephalopathy with axonal spheroids and pigmented glia (ALSP)

Li, X., Hu, B., Xiaoyan, G., Wang, Z., Zhou, Y., Sun, H., Zhang, X., Li, Y., Huang, X., Zhao, Y., et al. (2024) · Figshare · doi:10.6084/m9.figshare.26593178

Additional file 2 of Minocycline protects against microgliopathy in a Csf1r haplo-insufficient mouse model of adult-onset leukoencephalopathy with axonal spheroids and pigmented glia (ALSP)

Li, X., Hu, B., Xiaoyan, G., Wang, Z., Zhou, Y., Sun, H., Zhang, X., Li, Y., Huang, X., Zhao, Y., et al. (2024) · Figshare · doi:10.6084/m9.figshare.26593163

Additional file 5 of An adapted protocol to derive microglia from stem cells and its application in the study of CSF1R-related disorders

Dorion, M.-F., Casas, D., Shlaifer, I., Yaqubi, M., Fleming, P., Karpilovsky, N. C., Chen, C. X. ‐., Nicouleau, M., Piscopo, V. E. C., MacDougall, E. J., et al. (2024) · Figshare · doi:10.6084/m9.figshare.26702383

Additional file 3 of Minocycline protects against microgliopathy in a Csf1r haplo-insufficient mouse model of adult-onset leukoencephalopathy with axonal spheroids and pigmented glia (ALSP)

Li, X., Hu, B., Xiaoyan, G., Wang, Z., Zhou, Y., Sun, H., Zhang, X., Li, Y., Huang, X., Zhao, Y., et al. (2024) · Figshare · doi:10.6084/m9.figshare.26593166

Additional file 1 of An adapted protocol to derive microglia from stem cells and its application in the study of CSF1R-related disorders

Dorion, M.-F., Casas, D., Shlaifer, I., Yaqubi, M., Fleming, P., Karpilovsky, N. C., Chen, C. X. ‐., Nicouleau, M., Piscopo, V. E. C., MacDougall, E. J., et al. (2024) · Figshare · doi:10.6084/m9.figshare.25546980

Additional file 1 of Minocycline protects against microgliopathy in a Csf1r haplo-insufficient mouse model of adult-onset leukoencephalopathy with axonal spheroids and pigmented glia (ALSP)

Li, X., Hu, B., Xiaoyan, G., Wang, Z., Zhou, Y., Sun, H., Zhang, X., Li, Y., Huang, X., Zhao, Y., et al. (2024) · Figshare · doi:10.6084/m9.figshare.26593160

Additional file 3 of An adapted protocol to derive microglia from stem cells and its application in the study of CSF1R-related disorders

Dorion, M.-F., Casas, D., Shlaifer, I., Yaqubi, M., Fleming, P., Karpilovsky, N. C., Chen, C. X. ‐., Nicouleau, M., Piscopo, V. E. C., MacDougall, E. J., et al. (2024) · Figshare · doi:10.6084/m9.figshare.26702377

Additional file 2 of An adapted protocol to derive microglia from stem cells and its application in the study of CSF1R-related disorders

Dorion, M.-F., Casas, D., Shlaifer, I., Yaqubi, M., Fleming, P., Karpilovsky, N. C., Chen, C. X. ‐., Nicouleau, M., Piscopo, V. E. C., MacDougall, E. J., et al. (2024) · Figshare · doi:10.6084/m9.figshare.25547144

Additional file 2 of Epigenetic modulation of AREL1 and increased HLA expression in brains of multiple system atrophy patients

Rydbirk, R., Folke, J., Busato, F., Roché, E., Chauhan, A. S., Løkkegaard, A., Hejl, A.-M., Bode, M., Blaabjerg, M., Møller, M. L., et al. (2020) · Figshare · doi:10.6084/m9.figshare.11960316

Additional file 4 of Minocycline protects against microgliopathy in a Csf1r haplo-insufficient mouse model of adult-onset leukoencephalopathy with axonal spheroids and pigmented glia (ALSP)

Li, X., Hu, B., Xiaoyan, G., Wang, Z., Zhou, Y., Sun, H., Zhang, X., Li, Y., Huang, X., Zhao, Y., et al. (2024) · Figshare · doi:10.6084/m9.figshare.26593169

Additional file 4 of An adapted protocol to derive microglia from stem cells and its application in the study of CSF1R-related disorders

Dorion, M.-F., Casas, D., Shlaifer, I., Yaqubi, M., Fleming, P., Karpilovsky, N. C., Chen, C. X. ‐., Nicouleau, M., Piscopo, V. E. C., MacDougall, E. J., et al. (2024) · Figshare · doi:10.6084/m9.figshare.26702380

Additional file 6 of Minocycline protects against microgliopathy in a Csf1r haplo-insufficient mouse model of adult-onset leukoencephalopathy with axonal spheroids and pigmented glia (ALSP)

Li, X., Hu, B., Xiaoyan, G., Wang, Z., Zhou, Y., Sun, H., Zhang, X., Li, Y., Huang, X., Zhao, Y., et al. (2024) · Figshare · doi:10.6084/m9.figshare.26593175

Additional file 5 of Minocycline protects against microgliopathy in a Csf1r haplo-insufficient mouse model of adult-onset leukoencephalopathy with axonal spheroids and pigmented glia (ALSP)

Li, X., Hu, B., Xiaoyan, G., Wang, Z., Zhou, Y., Sun, H., Zhang, X., Li, Y., Huang, X., Zhao, Y., et al. (2024) · Figshare · doi:10.6084/m9.figshare.26593172

Impact de la consommation de fructose sur la microglie et la santé cérébrale

Coursan, A. (2024) · theses.fr (ABES) · doi:10.70675/c509f961z7098z4c0bz92b0z8ba14be2e725

Spatial transcriptomics analysis of post-mortem brain tissues from Adult-onset leukoencephalopathy with axonal spheroids and pigmented glia (ALSP) patients

Du, S. (2025) · Zenodo (CERN European Organization for Nuclear Research) · doi:10.5281/zenodo.15186189

BindingDB Entry 50022497: Sustained microglial depletion with CSF1R inhibitor impairs parenchymal plaque development in an Alzheimer's disease model.

BindingDB (2025) · UC San Diego · doi:10.7270/q2xw4qks

Hereditary Diffuse Leukoencephalopathy With Spheroids (HDLS): Histological Features Of Microglia (P1.245)

Tada, M., Konno, T., Tada, M., Okazaki, K.-I., Arakawa, M., Itoh, K., Yamamoto, T., Yokoo, H., Yoshikura, N., Ishihara, K., et al. (2014) · Neurology · doi:10.1212/wnl.82.10_supplement.p1.245

Characteristic microglial features in patients with hereditary diffuse leukoencephalopathy with spheroids caused by mutations of the gene encoding the colony stimulating factor-1 receptor

Tada, M., Konno, T., Tada, M., Tezuka, T., Miura, T., Mezaki, N., Arakawa, M., Itoh, K., Yamamoto, T., Yokoo, H., et al. (2017) · Journal of the Neurological Sciences · doi:10.1016/j.jns.2017.08.2876

Loss of homeostatic microglial phenotype in CSF1R-related Leukoencephalopathy

Kempthorne, L., Yoon, H., Madore, C., Smith, S., Wszolek, Z. K., Rademakers, R., Kim, J., Butovsky, O., & Dickson, D. W. (2020) · Acta Neuropathologica Communications · doi:10.1186/s40478-020-00947-0 · Cited by 77 · see citing works

Early Pathologic Changes in Hereditary Diffuse Leukoencephalopathy With Spheroids

Riku, Y., Ando, T., Goto, Y., Mano, K., Iwasaki, Y., Sobue, G., & Yoshida, M. (2014) · Journal of Neuropathology & Experimental Neurology · doi:10.1097/nen.0000000000000139 · Cited by 28 · see citing works

[Hereditary diffuse leukoencephalopathy with spheroids (HDLS): a review of the literature on its clinical characteristics and mutations in the colony-stimulating factor-1 receptor gene].

Konno, T., Tada, M., Tada, M., Nishizawa, M., & Ikeuchi, T. (2014) · PubMed · Cited by 10 · see citing works