Colony-Stimulating Factor 1 Receptor (CSF1R) Regulates Microglia Density and Distribution, but Not Microglia Differentiation In Vivo

Oosterhof, Nynke, Kuil, Laura E., van der Linde, Herma C., Burm, Saskia M., Berdowski, Woutje, van Ijcken, Wilfred F.J., van Swieten, John C., Hol, Elly M. +2 more·2018·Cell Reports

DOI 10.1016/j.celrep.2018.06.113

Cited by (182)

Items 101–182 of 182

Genetic basis of orange spot formation in the guppy (Poecilia reticulata)
Mayuko Kawamoto, Yuu Ishii, Masakado Kawata · 2021 · BMC Ecology and Evolution
Leukoencephalopathy with calcifications, developmental brain abnormalities and skeletal dysplasia due to homozygosity for a hypomorphic CSF1R variant: A report of three siblings
Shanice Beerepoot, Jonathan I. M. L. Verbeke, Maud Plantinga, Stefan Nierkens, Petra J. W. Pouwels, Nicole I. Wolf, Cas Simons, Marjo S. van der Knaap · 2024 · American Journal of Medical Genetics Part A
Macrophage transplantation rescues RNASET2-deficient leukodystrophy by replacing deficient microglia in a zebrafish model
Holly A. Rutherford, Diogo Candeias, Christopher James Arthur Duncan, Stephen A. Renshaw, Noémie Hamilton · 2024 · Proceedings of the National Academy of Sciences
Transcriptome analysis reveals the contribution of oligodendrocyte and radial glia‐derived cues for maintenance of microglia identity
Raissa Timmerman, Ella Alwine Zuiderwijk-Sick, Nynke Oosterhof, Anke E. J. 't Jong, Jennifer Veth, Saskia Maria Burm, Tjakko J. van Ham, Jeffrey John Bajramovic · 2021 · Glia
Dynamic changes in microglia in the mouse hippocampus during administration and withdrawal of the CSF1R inhibitor PLX3397
Qirun Wang, Yiyan Wang, Wen‐Jun Pu, Xiaohong Ma, Rong‐Jun Ni · 2023 · Journal of Anatomy
Microglia Stimulate Zebrafish Brain Repair Via a Tumor Necrosis Factor- α -Initiated Inflammatory Cascade
Palsamy Kanagaraj, Jessica Y. Chen, Kaia Skaggs, Yusuf Qadeer, Meghan Connors, Noah S. Cutler, Joshua Richmond, Vineeth Kommidi, Allison Poles, Danielle Affrunti et al. · 2020 · bioRxiv (Cold Spring Harbor Laboratory)
Glial Cell Development and Function in the Zebrafish Central Nervous System
Tim Czopka, Kelly R. Monk, Francesca Peri · 2024 · Cold Spring Harbor Perspectives in Biology
Two Novel Intronic Mutations in the CSF1R Gene in Two Families With CSF1R-Microglial Encephalopathy
Jiwei Jiang, Wenyi Li, Xiaohong Wang, Zhongli Du, Jinlong Chen, Yaou Liu, Wei Li, Zhonghua Lu, Yanli Wang, Jun Liang Xu · 2022 · Frontiers in Cell and Developmental Biology
Role of Glia in the Regulation of Sleep in Health and Disease
Stefano Garofalo, Katherine Picard, Cristina Limatola, Agnès Nadjar, Olivier Pascual, Marie‐Ève Tremblay · 2020 · Comprehensive physiology
Ethanol induces neuroimmune dysregulation and soluble TREM2 generation in a human iPSC neuron, astrocyte, microglia triculture model
Andrew J. Boreland, Yara Abbo, Xindi Li, Alessandro C. Stillitano, Siwei Zhang, Jubao Duan, Zhiping P. Pang, Ronald P. Hart · 2025 · Alcohol
CSF1R+ macrophage and osteoclast depletion impairs neural crest proliferation and craniofacial morphogenesis
Felix Ma, Ru Zhou, Matthew Rosin, Iris Zhou, Sabrina Ownsworth, Rouzbeh Ostadsharif Memar, Vincent B. Wong, Jessica M. Rosin · 2026 · Development
The microglia-derived protein Sema4ab attenuates regenerative neurogenesis after spinal cord injury in zebrafish
Alberto Docampo-Seara, Mehmet İlyas Coşacak, Kim Heilemann, Friederike Kessel, Ana‐Maria Oprişoreanu, Markus Westphal, Özge Çark, Daniela Zöller, Josi Arnold, Anja Bretschneider et al. · 2026 · PLoS Biology
Clinical, genetic, and molecular characteristics in a central‐southern Chinese cohort of genetic leukodystrophies
Yingjie Li, Jiaming Xu, Xu Yan, Chuanzhou Li, Yan Wu, Zhijun Liu · 2023 · Annals of Clinical and Translational Neurology
Glial Diversity and Evolution: Insights from Teleost Fish
Carla Lucini, Claudia Gatta · 2025 · Brain Sciences
Implications of fractalkine on glial function, ablation and glial proteins/receptors/markers—understanding its therapeutic usefulness in neurological settings: a narrative review
Deepraj Paul, Duraiswamy Basavan · 2022 · Future Journal of Pharmaceutical Sciences
A single-cell transcriptomic atlas reveals resident dendritic-like cells in the zebrafish brain parenchyma
Mireia Rovira, Giuliano Ferrero, Magali Miserocchi, Alice Montanari, Ruben Lattuca, Valérie Wittamer · 2024 · eLife
Pancreatic exocrine damage induces beta cell stress in zebrafish larvae
Noura Faraj, Willem M.H. Hoogaars, B. H. Peter Duinkerken, Anouk H. G. Wolters, Kim Kats, Mette C. Dekkers, Arnaud Zaldumbide, Ben N. G. Giepmans · 2025 · Diabetologia
Macrophage/microglia-dependent mechanisms drive retinal pigment epithelium regeneration in zebrafish
Lyndsay L. Leach, Rebecca G. Gonzalez, Sayuri U. Jayawardena, Jeffrey M. Gross · 2025 · Cell Reports
Temporal dynamics of a CSF1R signaling gene regulatory network involved in epilepsy
Claude Gérard, Laurane De Mot, Sabine Cordi, Jonathan M. van Eyll, Frédéric P. Lemaigre · 2021 · PLoS Computational Biology
Case Report: Novel CSF1R Variant in a Patient With Behavioral Variant Frontotemporal Dementia Syndrome With Prodromal Repetitive Scratching Behavior
Adit Friedberg, Eliana Marisa Ramos, Zhongan Yang, Luke W. Bonham, Jennifer S. Yokoyama, Peter Alexander Ljubenkov, Kyan Younes, Daniel H. Geschwind, Bruce L. Miller · 2022 · Frontiers in Neurology
Macrophage activation drives ovarian failure and masculinization
Paloma Bravo, Yulong Liu, Bruce W. Draper, Florence L. Marlow · 2023 · bioRxiv (Cold Spring Harbor Laboratory)
Swimming Through Dendritic Cell Biology: Insights From Fish Models
Changlong Zhao, Guanzhen Lin, Kefan Cheng, Shaoli Hassen, Hongru Pan, Zilong Wen · 2025 · Immunological Reviews
Blood vessel-resident macrophages safeguard blood and vessel integrity in zebrafish
Bart G. M. W. Weijts, Jeroen Demmers, Catherine Robin · 2026 · Nature Immunology
Progress in leukodystrophies with zebrafish
Hung‐Yu Shih, Quentin Raas, Joshua L. Bonkowsky · 2024 · Development Growth & Differentiation
Pericytes and Microglia
Nicola Marchi, Amy L. Brewster · 2024 · Oxford University Press eBooks
Heterozygous missense CSF1R variants hamper in vitro CD34+-derived dendritic cell generation but not in vivo dendritic cell development
Shanice Beerepoot, Nicole I. Wolf, Marjo S. van der Knaap, Stefan Nierkens, Maud Plantinga · 2024 · Molecular Immunology
Microglial knockdown does not affect acute withdrawal but delays analgesic tolerance from oxycodone in male and female C57BL/6J mice
Omar El Jordi, Kathryn D. Fischer, Timothy B. Meyer, Brady K. Atwood, Adrian L. Oblak, Raymond W. Pan, David L. McKinzie · 2022 · Advances in Drug and Alcohol Research
Cranial radiotherapy profoundly affects glia without inducing widespread cellular senescence
Laura E. Kuil, Roan H van Scheppingen, Yoran M Leter, Berend H Röring, Mark C. de Gooijer, Caroline L. Van Heijningen, Stefan Preković, Annette Compter, Olaf van Tellingen, Sanne B. Schagen · 2025 · Neuro-Oncology
Regulation of LPS-Induced Inflammatory Responses in HMC3 Microglia by Nicotine and its Neuroprotective Effects
Yuhan Qin, Yan-Bo Luo, Hongjuan Wang, Yushan Tian, Xiaqing Wu, Huan Chen, Hong-Wei Hou, Qingyuan Hu · 2023 · Research Square
Defining mononuclear phagocyte distribution and behaviour in the zebrafish heart
Bethany Moyse, Joanna J. Moss, Laura Bevan, Aaron Scott, Valérie Wittamer, Rebecca J. Richardson · 2024 · eLife
A Dynamic Change of Microglial States Occurs During the Transition From Photoreceptor Degeneration to Regeneration in Zebrafish pde6c Mutants
Darshini Ravishankar, Yuki Takeuchi, Ichiro Masai · 2026 · Glia
Müller glia–microglia cross talk reprograms the Müller glia transcriptome for cell division–related processes during retina regeneration
Soumitra Mitra, Sulochana Devi, Mi‐Sun Lee, Aresh Sahu, Jonathan Jui, Emil Kriukov, Petr Y. Baranov, Daniel J. Goldman · 2026 · Proceedings of the National Academy of Sciences
Loss of Ramified Microglia Precedes Axonal Spheroid Formation in Adult-Onset Leukoencephalopathy with Axonal Spheroids.
Murad A. Alturkustani, Zhang, Qi, Basma M Alyamany, Lee-Cyn Ang · 2020 · Free Neuropathology
Radial astroglia cooperate with microglia to clear neuronal cell bodies during zebrafish optic tectum development
Heather M. Barber, Cassidy G. Robbins, Zachary Cutler, Robin Isadora Brown, Lauren E. Jung, Inge L. Werkman, Sarah Kucenas · 2025 · Cell Reports
From seed to flower: blossoming of microglia in development and brain repair
Victoria N. Neckles, David M. Feliciano · 2021 · Cell and Tissue Research
Microglia in Epilepsy: From Molecular Mechanism to Therapeutic Strategy
Yam Nath Paudel, Efthalia Angelopoulou, Sai Kulkarni, Robert E. Blair, Laxmikant S. Deshpande · 2026 · Cells
Cone photoreceptor ablation in microglia-deficient larval zebrafish retina elicits a regenerative response alongside a compensatory immune cell response
Jordan E. Rumford, Ashley A. Farre, Justin Mai, Halle V. Weimar, Claire D Shelton, M Castaneda Morales, Diana M. Mitchell · 2026 · PLoS ONE
Aging-related inflammatory and metabolic disorder in the novel mutation of colony-stimulating factor-1 receptor (csf1r)P853T/+ in CSF1R-microglial encephalopathy
Xiaohong Wang, Yanli Wang, Tianlin Jiang, Jiwei Jiang, Linlin Wang, Shi‐Yi Yang, Mengfan Sun, Yuan Zhang, Ziyan Jia, Wenyi Li et al. · 2024 · Genes & Diseases
Olfactory detection of viruses shapes brain immunity and behavior in zebrafish
Aurora Kraus, Benjamin Garcia, Jie Ma, Kristian J. Herrera, Hanna Zwaka, Roy Harpaz, Ryan Y. Wong, Florian Engert, Irene Salinas · 2023
A single-cell transcriptomic atlas reveals resident dendritic-like cells in the zebrafish brain parenchyma
Mireia Rovira, Giuliano Ferrero, Magali Miserocchi, Alice Montanari, Valérie Wittamer · 2024 · eLife
Defining mononuclear phagocyte distribution and behaviour in the zebrafish heart
Bethany Moyse, Joanna J. Moss, Laura Bevan, Aaron Scott, Valérie Wittamer, Rebecca J. Richardson · 2024 · eLife
Microglia are not required for normal postnatal brain development and cognitive function in the rat
Emma K. Green, Xinyi Li, Yang Guo, Dylan Carter‐Cusack, Rocío Rojo, Maia Lyall, Annie Bell, Helen Zheng, Oliver Teenan, Sally M. Till et al. · 2025 · bioRxiv (Cold Spring Harbor Laboratory)
Investigation of CSF1-CSF1R Signaling in AML-Bone Marrow Stromal Cell Interactions
Ayesha Rashid · 2019 · TSpace (University of Toronto)
Faculty Opinions recommendation of Homozygous Mutations in CSF1R Cause a Pediatric-Onset Leukoencephalopathy and Can Result in Congenital Absence of Microglia.
David H. Rowitch · 2019 · Faculty Opinions – Post-Publication Peer Review of the Biomedical Literature
Transcriptome-guided optimization of in vitro culture conditions to study microglia in health and disease
Raissa Timmerman · 2022
Olfactory detection of viruses shapes brain immunity and behavior in zebrafish
Aurora Kraus, Benjamin Garcia, Jie Ma, Kristian J. Herrera, Hanna Zwaka, Roy Harpaz, Ryan Y. Wong, Florian Engert, Irene Salinas · 2023 · bioRxiv (Cold Spring Harbor Laboratory)
Other diseases of the CNS
Alexei Verkhratsky, Arthur Morgan Butt · 2023 · Elsevier eBooks
Olfactory detection of viruses shapes brain immunity and behavior in zebrafish
Aurora Kraus, Benjamin Garcia, Jie Ma, Kristian J. Herrera, Hanna Zwaka, Roy Harpaz, Ryan Y. Wong, Florian Engert, Irene Salinas · 2023
An adapted stem cell-derived microglia protocol for the study of microgliopathies and other neurological disorders
Marie‐France Dorion, Diana Casas, Moein Yaqubi, Peter Fleming, Carol X.‐Q. Chen, Valerio E. C. Piscopo, Michaël Nicouleau, Taylor Goldsmith, Irina Shlaifer, Adam MacDonald et al. · 2023 · bioRxiv (Cold Spring Harbor Laboratory)
Defining mononuclear phagocyte distribution and behaviour in the zebrafish heart
Bethany Moyse, Joanna J. Moss, Laura Bevan, Aaron Scott, Valérie Wittamer, Rebecca J. Richardson · 2024 · bioRxiv (Cold Spring Harbor Laboratory)
Reviewer #1 (Public Review): Defining mononuclear phagocyte distribution and behaviour in the zebrafish heart
2024 · eLife
Reviewer #2 (Public Review): Defining mononuclear phagocyte distribution and behaviour in the zebrafish heart
2024 · eLife
Reviewer #3 (Public Review): Defining mononuclear phagocyte distribution and behaviour in the zebrafish heart
2024 · eLife
eLife Assessment: Defining mononuclear phagocyte distribution and behaviour in the zebrafish heart
Shih-Lei Lai · 2024 · eLife
Advances in induced pluripotent stem cell-based in vitro disease modeling for rare neurodegenerative disease: A narrative review
Seung‐Yeon Lee, Hyo Jin Son, Sang Won Seo, Duk Lyul Na, Hee Jin Kim · 2024 · Precision and Future Medicine
Interleukin-34 and debris clearance by mononuclear phagocytes drive retinal pigment epithelium regeneration in zebrafish
Lyndsay L. Leach, Rebecca G. Gonzalez, Sayuri U. Jayawardena, Jeffrey M. Gross · 2025 · bioRxiv (Cold Spring Harbor Laboratory)
Csf2rb-Dependent Development of Yolk Sac-Derived Macrophages Drives Persistent Pro-Tumorigenic Activity
Yong Xiang Zhao, Tong Lei, Qian Zhang, Zhaoqi Zhang · 2025 · Research Square
TFEB-depletion rescues TMEM106B-related myelin defects in zebrafish
Woutje M. Berdowski, Herma C. van der Linde, Nicole I. Wolf, Tjakko J. van Ham · 2025 · bioRxiv (Cold Spring Harbor Laboratory)
A single-cell transcriptomic atlas reveals resident dendritic-like cells in the zebrafish brain parenchyma
Mireia Rovira, Giuliano Ferrero, Magali Miserocchi, Alice Montanari, Ruben Lattuca, Valérie Wittamer · 2025 · eLife
Additional file 5 of Microglia specific deletion of miR-155 in Alzheimer’s disease mouse models reduces amyloid-β pathology but causes hyperexcitability and seizures
Macarena S. Aloi, Katherine E. Prater, Sánchez, Raymond E. A., Asad Beck, Jasmine L. Pathan, Stephanie Davidson, Angela M. Wilson, Keene, C. Dirk, Horacio O. de la Iglesia, Suman Jayadev et al. · 2024 · Figshare
Additional file 3 of Microglia specific deletion of miR-155 in Alzheimer’s disease mouse models reduces amyloid-β pathology but causes hyperexcitability and seizures
Macarena S. Aloi, Katherine E. Prater, Sánchez, Raymond E. A., Asad Beck, Jasmine L. Pathan, Stephanie Davidson, Angela M. Wilson, Keene, C. Dirk, Horacio O. de la Iglesia, Suman Jayadev et al. · 2024 · Figshare
Additional file 5 of An adapted protocol to derive microglia from stem cells and its application in the study of CSF1R-related disorders
Dorion, Marie-France, Diana Casas, Irina Shlaifer, Moein Yaqubi, Fleming, Peter, Nathan C. Karpilovsky, Carol X.‐Q. Chen, Michaël Nicouleau, Piscopo, Valerio E. C., Emma J. MacDougall et al. · 2024 · Figshare
Additional file 1 of An adapted protocol to derive microglia from stem cells and its application in the study of CSF1R-related disorders
Dorion, Marie-France, Diana Casas, Irina Shlaifer, Moein Yaqubi, Fleming, Peter, Nathan C. Karpilovsky, Carol X.‐Q. Chen, Michaël Nicouleau, Piscopo, Valerio E. C., Emma J. MacDougall et al. · 2024 · Figshare
Additional file 3 of An adapted protocol to derive microglia from stem cells and its application in the study of CSF1R-related disorders
Dorion, Marie-France, Diana Casas, Irina Shlaifer, Moein Yaqubi, Fleming, Peter, Nathan C. Karpilovsky, Carol X.‐Q. Chen, Michaël Nicouleau, Piscopo, Valerio E. C., Emma J. MacDougall et al. · 2024 · Figshare
Additional file 1 of Microglia specific deletion of miR-155 in Alzheimer’s disease mouse models reduces amyloid-β pathology but causes hyperexcitability and seizures
Macarena S. Aloi, Katherine E. Prater, Sánchez, Raymond E. A., Asad Beck, Jasmine L. Pathan, Stephanie Davidson, Angela M. Wilson, Keene, C. Dirk, Horacio O. de la Iglesia, Suman Jayadev et al. · 2024 · Figshare
Additional file 2 of An adapted protocol to derive microglia from stem cells and its application in the study of CSF1R-related disorders
Dorion, Marie-France, Diana Casas, Irina Shlaifer, Moein Yaqubi, Fleming, Peter, Nathan C. Karpilovsky, Carol X.‐Q. Chen, Michaël Nicouleau, Piscopo, Valerio E. C., Emma J. MacDougall et al. · 2024 · Figshare
Additional file 4 of Microglia specific deletion of miR-155 in Alzheimer’s disease mouse models reduces amyloid-β pathology but causes hyperexcitability and seizures
Macarena S. Aloi, Katherine E. Prater, Sánchez, Raymond E. A., Asad Beck, Jasmine L. Pathan, Stephanie Davidson, Angela M. Wilson, Keene, C. Dirk, Horacio O. de la Iglesia, Suman Jayadev et al. · 2024 · Figshare
Additional file 2 of Microglia specific deletion of miR-155 in Alzheimer’s disease mouse models reduces amyloid-β pathology but causes hyperexcitability and seizures
Macarena S. Aloi, Katherine E. Prater, Sánchez, Raymond E. A., Asad Beck, Jasmine L. Pathan, Stephanie Davidson, Angela M. Wilson, Keene, C. Dirk, Horacio O. de la Iglesia, Suman Jayadev et al. · 2024 · Figshare
Additional file 1 of Early postnatal microglial ablation in the Ccdc39 mouse model reveals adverse effects on brain development and in neonatal hydrocephalus
Farrah N. Brown, Eri Iwasawa, Crystal Shula, Elizabeth M. Fugate, Diana M. Lindquist, Francesco T. Mangano, June Goto · 2023 · Figshare
Additional file 4 of An adapted protocol to derive microglia from stem cells and its application in the study of CSF1R-related disorders
Dorion, Marie-France, Diana Casas, Irina Shlaifer, Moein Yaqubi, Fleming, Peter, Nathan C. Karpilovsky, Carol X.‐Q. Chen, Michaël Nicouleau, Piscopo, Valerio E. C., Emma J. MacDougall et al. · 2024 · Figshare
Additional file 6 of Microglia specific deletion of miR-155 in Alzheimer’s disease mouse models reduces amyloid-β pathology but causes hyperexcitability and seizures
Macarena S. Aloi, Katherine E. Prater, Sánchez, Raymond E. A., Asad Beck, Jasmine L. Pathan, Stephanie Davidson, Angela M. Wilson, Keene, C. Dirk, Horacio O. de la Iglesia, Suman Jayadev et al. · 2024 · Figshare
CSF1R-related leukoencephalopathy presenting with early apathy, hypoactivity, and cognitive flattening: a case report of a diagnostic challenge
Yang Wen, Chunli Li, Hongjiang Zhang, Yangjia Zhang · 2026 · Frontiers in Human Neuroscience
Nicotine Regulates LPS‐Induced Inflammatory Responses in HMC3 Microglia and Exerts Neuronal Protection
Yuhan Qin, Xiaohui Yan, Yanbo Luo, Hongjuan Wang, Yushan Tian, Xiaqing Wu, Huan Chen, Hongwei Hou, Qingyuan Hu · 2026 · Mediators of Inflammation
Inactivation génique des transporteurs ABC peroxysomaux ABCD1 et ABCD2 dans les cellules microgliales BV-2 : étude de la physiopathogenèse de l’adrénoleucodystrophie liée à l’X.
Quentin Raas · 2018 · theses.fr (ABES)
Dynamics of macrophage polarization in zebrafish during the tissue injury and infection
Tamara Sipka · 2020 · theses.fr (ABES)
Statistical signatures of learning in the olfactory bulb and a new approach to image stack segmentation
Corentin Guérinot · 2022 · theses.fr (ABES)
Mechanisms controlling microglial colonization of the forebrain
Cécile Bridlance · 2023 · theses.fr (ABES)
Diverse origins and transcriptional profiles of macrophages in a spinal lesion in zebrafish
Kim Heilemann, Elena Vanessa Jidav, Oezge Cark, Stephen J. Enos, Bastian Wolf, Catherina Gwynne Becker, Thomas Becker, Alberto Docampo-Seara · 2026 · bioRxiv (Cold Spring Harbor Laboratory)
CSF1R-related leukoencephalopathy: experimental models and potential for treatment
David Arthur Hume, Katharine M. Irvine · 2026 · Disease Models & Mechanisms
Accumulation of lipid droplets in microglia following neonatal brain hypoxia-ischemia
Li Fan, Yuqing Lei, Sihao Li, Gui Zhang, Yinuo Li, Beiyan Wu, Donna Marie Ferriero, Peipei Pan, Zhonghui Guan, Xiangning Jiang · 2026 · Journal of Neuroinflammation
Spatially distinct microglia coordinate the response to photoreceptor injury in zebrafish
Mikiko Nagashima, Lu Jiang, Sriram Garapati, Thanh Hoang, Peter F. Hitchcock · 2026 · bioRxiv (Cold Spring Harbor Laboratory)
Spatial transcriptomics analysis of post-mortem brain tissues from Adult-onset leukoencephalopathy with axonal spheroids and pigmented glia (ALSP) patients
Siling Du · 2025 · Zenodo (CERN European Organization for Nuclear Research)