Dominant-acting CSF1R variants cause microglial depletion and altered astrocytic phenotype in zebrafish and adult-onset leukodystrophy

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

(2022) Jennifer Stables et al.   DEVELOPMENT

Reactive astrocyte nomenclature, definitions, and future directions

(2021) Carole Escartin et al.   NATURE NEUROSCIENCE

Further expanding the mutational spectrum of brain abnormalities, neurodegeneration, and dysosteosclerosis : A rare disorder with neurologic regression and skeletal features

(2021) Erdem Kındış et al.   AMERICAN JOURNAL OF MEDICAL GENETICS PART A

Microglia as hackers of the matrix: sculpting synapses and the extracellular space

(2021) Joshua D. Crapser et al.   Cellular & Molecular Immunology

The multicellular interplay of microglia in health and disease: lessons from leukodystrophy

(2021) Woutje M. Berdowski et al.   Disease Models & Mechanisms

Skull and vertebral bone marrow are myeloid cell reservoirs for the meninges and CNS parenchyma

(2021) Andrea Cugurra et al.   SCIENCE

Microglial dyshomeostasis drives perineuronal net and synaptic loss in a CSF1R+/− mouse model of ALSP, which can be rescued via CSF1R inhibitors

(2021) Miguel A. Arreola et al.   Science Advances

Microglia Depletion-Induced Remodeling of Extracellular Matrix and Excitatory Synapses in the Hippocampus of Adult Mice

(2021) Luisa Strackeljan et al.   Cells

Three novel mutations in Chinese patients with CSF1R-related leukoencephalopathy

(2021) Min Chu et al.   Annals of Translational Medicine

In situ and transcriptomic identification of microglia in synapse-rich regions of the developing zebrafish brain

(2021) Nicholas J. Silva et al.   Nature Communications

Insights Into Central Nervous System Glial Cell Formation and Function From Zebrafish

(2021) Sarah A. Neely et al.   Frontiers in Cell and Developmental Biology

Pro-inflammatory activation following demyelination is required for myelin clearance and oligodendrogenesis

(2020) Maria Inês Cunha et al.   JOURNAL OF EXPERIMENTAL MEDICINE

Microglia depletion rapidly and reversibly alters amyloid pathology by modification of plaque compaction and morphologies

(2020) Brad T. Casali et al.   NEUROBIOLOGY OF DISEASE

A Novel Hypomorphic CSF1R Gene Mutation in the Biallelic State Leading to Fatal Childhood Neurodegeneration

(2020) Parag Mohan Tamhankar et al.   NEUROPEDIATRICS

Astrocytes and Microglia as Major Players of Myelin Production in Normal and Pathological Conditions

(2020) Elisabeth Traiffort et al.   Frontiers in Cellular Neuroscience

Zebrafish macrophage developmental arrest underlies depletion of microglia and reveals Csf1r-independent metaphocytes

(2020) Laura E Kuil et al.   eLife

Loss of homeostatic microglial phenotype in CSF1R-related Leukoencephalopathy

(2020) Liam Kempthorne et al.   Acta Neuropathologica Communications

Microglial Remodeling of the Extracellular Matrix Promotes Synapse Plasticity

(2020) Phi T. Nguyen et al.   CELL

Microglia phagocytose myelin sheaths to modify developmental myelination

(2020) Alexandria N. Hughes et al.   NATURE NEUROSCIENCE

Altered features of monocytes in adult onset leukoencephalopathy with axonal spheroids and pigmented glia: A clue to the pathomechanism of microglial dyshomeostasis

(2020) Mio Hamatani et al.   NEUROBIOLOGY OF DISEASE

Alzheimer’s Patient Microglia Exhibit Enhanced Aging and Unique Transcriptional Activation

(2020) Karpagam Srinivasan et al.   Cell Reports

Astrocytes and microglia play orchestrated roles and respect phagocytic territories during neuronal corpse removal in vivo

(2020) Eyiyemisi C. Damisah et al.   Science Advances

Astrocytic phagocytosis is a compensatory mechanism for microglial dysfunction

(2020) Hiroyuki Konishi et al.   EMBO JOURNAL

Negative feedback control of neuronal activity by microglia

(2020) Ana Badimon et al.   NATURE

Live-imaging of astrocyte morphogenesis and function in zebrafish neural circuits

(2020) Jiakun Chen et al.   NATURE NEUROSCIENCE

A csf1rb mutation uncouples two waves of microglia development in zebrafish

(2020) Giuliano Ferrero et al.   DEVELOPMENT

Glutathione S-transferases promote proinflammatory astrocyte-microglia communication during brain inflammation

(2019) Shin-ichi Kano et al.   Science Signaling

Microglia damage precedes major myelin breakdown in X-linked adrenoleukodystrophy and metachromatic leukodystrophy

(2019) Caroline G. Bergner et al.   GLIA

Reverse genetic screen reveals that Il34 facilitates yolk sac macrophage distribution and seeding of the brain

(2019) Laura E. Kuil et al.   Disease Models & Mechanisms

Bi-allelic CSF1R Mutations Cause Skeletal Dysplasia of Dysosteosclerosis-Pyle Disease Spectrum and Degenerative Encephalopathy with Brain Malformation

(2019) Long Guo et al.   AMERICAN JOURNAL OF HUMAN GENETICS

Homozygous Mutations in CSF1R Cause a Pediatric-Onset Leukoencephalopathy and Can Result in Congenital Absence of Microglia

(2019) Nynke Oosterhof et al.   AMERICAN JOURNAL OF HUMAN GENETICS

Hexb enzyme deficiency leads to lysosomal abnormalities in radial glia and microglia in zebrafish brain development

(2019) Laura E. Kuil et al.   GLIA

Glia Accumulate Evidence that Actions Are Futile and Suppress Unsuccessful Behavior

(2019) Yu Mu et al.   CELL

Phenotypic impacts of CSF1R deficiencies in humans and model organisms

(2019) David A. Hume et al.   JOURNAL OF LEUKOCYTE BIOLOGY

Haematopoietic stem cell transplantation in CSF1R-related adult-onset leukoencephalopathy with axonal spheroids and pigmented glia

(2019) Fanny Mochel et al.   JOURNAL OF NEUROLOGY NEUROSURGERY AND PSYCHIATRY

Zebrafish: Housing and husbandry recommendations

(2019) Peter Aleström et al.   LABORATORY ANIMALS

Sustained microglial depletion with CSF1R inhibitor impairs parenchymal plaque development in an Alzheimer’s disease model

(2019) Elizabeth Spangenberg et al.   Nature Communications

Allogeneic HSCT for adult-onset leukoencephalopathy with spheroids and pigmented glia

(2019) Jeffrey M Gelfand et al.   BRAIN

Microglia monitor and protect neuronal function through specialized somatic purinergic junctions

(2019) Csaba Cserép et al.   SCIENCE

Adult-onset leukoencephalopathy with axonal spheroids and pigmented glia (ALSP): Integrating the literature on hereditary diffuse leukoencephalopathy with spheroids (HDLS) and pigmentary orthochromatic leukodystrophy (POLD)

(2018) Scott J. Adams et al.   JOURNAL OF CLINICAL NEUROSCIENCE

A Combination of Ontogeny and CNS Environment Establishes Microglial Identity

(2018) F. Chris Bennett et al.   NEURON

Adult-Onset Leukoencephalopathy with Axonal Spheroids and Pigmented Glia: An MRI Study of 16 French Cases

(2018) P. Codjia et al.   AMERICAN JOURNAL OF NEURORADIOLOGY

Pleiotropic Impacts of Macrophage and Microglial Deficiency on Development in Rats with Targeted Mutation of the Csf1r Locus

(2018) Clare Pridans et al.   JOURNAL OF IMMUNOLOGY

TREM2 — a key player in microglial biology and Alzheimer disease

(2018) Tyler K. Ulland et al.   Nature Reviews Neurology

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

(2018) Nynke Oosterhof et al.   Cell Reports

Effective CRISPR/Cas9-based nucleotide editing in zebrafish to model human genetic cardiovascular disorders

(2018) Federico Tessadori et al.   Disease Models & Mechanisms

CSF1R-related leukoencephalopathy

(2018) Takuya Konno et al.   NEUROLOGY

Autozygosity reveals recessive mutations and novel mechanisms in dominant genes: implications in variant interpretation

(2017) Dorota Monies et al.   GENETICS IN MEDICINE

Transcriptomic analysis of purified human cortical microglia reveals age-associated changes

(2017) Thais F Galatro et al.   NATURE NEUROSCIENCE

Oligodendroglial myelination requires astrocyte-derived lipids

(2017) Nutabi Camargo et al.   PLOS BIOLOGY

A comparative strategy for single-nucleus and single-cell transcriptomes confirms accuracy in predicted cell-type expression from nuclear RNA

(2017) Blue B. Lake et al.   Scientific Reports

Characteristic microglial features in patients with hereditary diffuse leukoencephalopathy with spheroids

(2016) Mari Tada et al.   ANNALS OF NEUROLOGY

Myelin phagocytosis by astrocytes after myelin damage promotes lesion pathology

(2016) Gerald Ponath et al.   BRAIN

Maximizing mutagenesis with solubilized CRISPR-Cas9 ribonucleoprotein complexes

(2016) Alexa Burger et al.   DEVELOPMENT

Clinical and genetic characterization of adult-onset leukoencephalopathy with axonal spheroids and pigmented glia associated withCSF1Rmutation

(2016) T. Konno et al.   EUROPEAN JOURNAL OF NEUROLOGY

Identification of a conserved and acute neurodegeneration-specific microglial transcriptome in the zebrafish

(2016) Nynke Oosterhof et al.   GLIA

CSF1R-related leukoencephalopathy mimicking primary progressive multiple sclerosis

(2016) Carol Prieto-Morin et al.   JOURNAL OF NEUROLOGY

The extracellular matrix: Tools and insights for the “omics” era

(2016) Alexandra Naba et al.   MATRIX BIOLOGY

New tools for studying microglia in the mouse and human CNS

(2016) Mariko L. Bennett et al.   PROCEEDINGS OF THE NATIONAL ACADEMY OF SCIENCES OF THE UNITED STATES OF AMERICA

Emerging Roles for CSF-1 Receptor and its Ligands in the Nervous System

(2016) Violeta Chitu et al.   TRENDS IN NEUROSCIENCES

slan-defined subsets of CD16-positive monocytes: impact of granulomatous inflammation and M-CSF receptor mutation

(2015) T. P. Hofer et al.   BLOOD

Efficient Intracellular Delivery of Native Proteins

(2015) Diego S. D’Astolfo et al.   CELL

Most Tissue-Resident Macrophages Except Microglia Are Derived from Fetal Hematopoietic Stem Cells

(2015) Jianpeng Sheng et al.   IMMUNITY

Low-pH Solid-Phase Amino Labeling of Complex Peptide Digests with TMTs Improves Peptide Identification Rates for Multiplexed Global Phosphopeptide Analysis

(2015) Gitte Böhm et al.   JOURNAL OF PROTEOME RESEARCH

HISAT: a fast spliced aligner with low memory requirements

(2015) Daehwan Kim et al.   NATURE METHODS

Synaptic vesicle release regulates myelin sheath number of individual oligodendrocytes in vivo

(2015) Sigrid Mensch et al.   NATURE NEUROSCIENCE

Cell type– and brain region–resolved mouse brain proteome

(2015) Kirti Sharma et al.   NATURE NEUROSCIENCE

limma powers differential expression analyses for RNA-sequencing and microarray studies

(2015) Matthew E. Ritchie et al.   NUCLEIC ACIDS RESEARCH

In vivo characterization of microglial engulfment of dying neurons in the zebrafish spinal cord

(2015) Marco Morsch et al.   Frontiers in Cellular Neuroscience

HTSeq--a Python framework to work with high-throughput sequencing data

(2014) S. Anders et al.   BIOINFORMATICS

Enhanced uptake of multiple sclerosis-derived myelin by THP-1 macrophages and primary human microglia

(2014) Debbie AE Hendrickx et al.   Journal of Neuroinflammation

Neuronal morphometry directly from bitmap images

(2014) Tiago A Ferreira et al.   NATURE METHODS

Microglia and brain macrophages in the molecular age: from origin to neuropsychiatric disease

(2014) Marco Prinz et al.   NATURE REVIEWS NEUROSCIENCE

CSF-1 Receptor Signaling in Myeloid Cells

(2014) E. R. Stanley et al.   Cold Spring Harbor Perspectives in Biology

An immunoaffinity purification method for the proteomic analysis of ubiquitinated protein complexes

(2013) Petra Schwertman et al.   ANALYTICAL BIOCHEMISTRY

Individual Oligodendrocytes Have Only a Few Hours in which to Generate New Myelin Sheaths In Vivo

(2013) Tim Czopka et al.   DEVELOPMENTAL CELL

A novel A781V mutation in the CSF1R gene causes hereditary diffuse leucoencephalopathy with axonal spheroids

(2013) Rebekah Ahmed et al.   JOURNAL OF THE NEUROLOGICAL SCIENCES

Whole-brain functional imaging at cellular resolution using light-sheet microscopy

(2013) Misha B Ahrens et al.   NATURE METHODS

M2 microglia and macrophages drive oligodendrocyte differentiation during CNS remyelination

(2013) Veronique E Miron et al.   NATURE NEUROSCIENCE

White matter astrocytes in health and disease

(2013) I. Lundgaard et al.   NEUROSCIENCE

CSF1R mutations in hereditary diffuse leukoencephalopathy with spheroids are loss of function

(2013) Clare Pridans et al.   Scientific Reports

Astrocytes: Emerging stars in leukodystrophy pathogenesis

(2013) Angela Lanciotti et al.   Translational Neuroscience

Improvement of White Matter Changes on Neuroimaging Modalities After Stem Cell Transplant in Metachromatic Leukodystrophy

(2013) Martje E. van Egmond et al.   JAMA Neurology

Reciprocal Regulation between Resting Microglial Dynamics and Neuronal Activity In Vivo

(2012) Ying Li et al.   DEVELOPMENTAL CELL

Fiji: an open-source platform for biological-image analysis

(2012) Johannes Schindelin et al.   NATURE METHODS

Microglia Sculpt Postnatal Neural Circuits in an Activity and Complement-Dependent Manner

(2012) Dorothy P. Schafer et al.   NEURON

Regeneration of the adult zebrafish brain from neurogenic radial glia-type progenitors

(2011) V. Kroehne et al.   DEVELOPMENT

Parenchymal accumulation of CD163+ macrophages/microglia in multiple sclerosis brains

(2011) Zhiren Zhang et al.   JOURNAL OF NEUROIMMUNOLOGY

Andromeda: A Peptide Search Engine Integrated into the MaxQuant Environment

(2011) Jürgen Cox et al.   JOURNAL OF PROTEOME RESEARCH

Mutations in the colony stimulating factor 1 receptor (CSF1R) gene cause hereditary diffuse leukoencephalopathy with spheroids

(2011) Rosa Rademakers et al.   NATURE GENETICS

Absence of Colony Stimulation Factor-1 Receptor Results in Loss of Microglia, Disrupted Brain Development and Olfactory Deficits

(2011) Bryna Erblich et al.   PLoS One

Synaptic Pruning by Microglia Is Necessary for Normal Brain Development

(2011) R. C. Paolicelli et al.   SCIENCE

mpeg1 promoter transgenes direct macrophage-lineage expression in zebrafish

(2010) F. Ellett et al.   BLOOD

Microglia in neurodegenerative disease

(2010) V. Hugh Perry et al.   Nature Reviews Neurology

Immunohistochemical characterization of microglia in Nasu-Hakola disease brains

(2010) Jun-ichi Satoh et al.   NEUROPATHOLOGY

Functions of astrocytes and their potential as therapeutic targets

(2010) Harold K. Kimelberg et al.   Neurotherapeutics

Microglial Interactions with Synapses Are Modulated by Visual Experience

(2010) Marie-Ève Tremblay et al.   PLOS BIOLOGY

edgeR: a Bioconductor package for differential expression analysis of digital gene expression data

(2009) M. D. Robinson et al.   BIOINFORMATICS

Mapping identifiers for the integration of genomic datasets with the R/Bioconductor package biomaRt

(2009) Steffen Durinck et al.   Nature Protocols

Live Imaging of Neuronal Degradation by Microglia Reveals a Role for v0-ATPase a1 in Phagosomal Fusion In Vivo

(2008) Francesca Peri et al.   CELL

Gap Junctions Couple Astrocytes and Oligodendrocytes

(2008) Jennifer L. Orthmann-Murphy et al.   JOURNAL OF MOLECULAR NEUROSCIENCE

A Transcriptome Database for Astrocytes, Neurons, and Oligodendrocytes: A New Resource for Understanding Brain Development and Function

(2008) J. D. Cahoy et al.   JOURNAL OF NEUROSCIENCE

High-resolution in situ hybridization to whole-mount zebrafish embryos

(2007) Christine Thisse et al.   Nature Protocols