Neuronal endolysosomal transport and lysosomal functionality in maintaining axonostasis

Roney et al. review recent advances in how axonal endolysosomal trafficking, distribution, and functionality maintain distal degradation capacity in neuronal health and become disrupted in several neurodegenerative diseases.

• Publication year

  2022

• Venue

  Journal of Cell Biology

• Publication date

  2022-02-10

• Fields of study

  Biology, Medicine

• Identifiers
  DOI 10.1083/jcb.202111077PMID 35142819PMCID 8932522
• External record

  Open on Semantic Scholar

• Source metadata

  Semantic Scholar, PubMed

• No claims are published for this paper.

• No concepts are published for this paper.

• Lysosome biogenesis: Regulation and functions

  2021cited by this paper
• Post-Golgi carriers, not lysosomes, confer lysosomal properties to pre-degradative organelles in normal and dystrophic axons.

  2021cited by this paper
• Lipid-mediated motor-adaptor sequestration impairs axonal lysosome delivery leading to autophagic stress and dystrophy in Niemann-Pick type C.

  2021influential reference
• Synaptic activity controls autophagic vacuole motility and function in dendrites

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• Quantitative correlative microscopy reveals the ultrastructural distribution of endogenous endosomal proteins

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• The rapidly evolving view of lysosomal storage diseases

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• Lysosomal TPCN (two pore segment channel) inhibition ameliorates beta-amyloid pathology and mitigates memory impairment in Alzheimer disease

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• Increased LRRK2 kinase activity alters neuronal autophagy by disrupting the axonal transport of autophagosomes.

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• Specific KIF1A–adaptor interactions control selective cargo recognition

  2021cited by this paper
• Axon terminals control endolysosome diffusion to support synaptic remodelling

  2021cited by this paper
• The Rab7 effector WDR91 promotes autophagy-lysosome degradation in neurons by regulating lysosome fusion

  2021cited by this paper
• A human iPSC-derived inducible neuronal model of Niemann-Pick disease, type C1

  2021cited by this paper
• Sequential dynein effectors regulate axonal autophagosome motility in a maturation-dependent pathway

  2020cited by this paper
• How Lysosomes Sense, Integrate, and Cope with Stress.

  2020cited by this paper
• Synaptic Vesicle Precursors and Lysosomes Are Transported by Different Mechanisms in the Axon of Mammalian Neurons

  2020cited by this paper
• JIP3 links lysosome transport to regulation of multiple components of the axonal cytoskeleton

  2020cited by this paper
• ER – lysosome contacts at a pre-axonal region regulate axonal lysosome availability

  2020cited by this paper
• Axonal precursor miRNAs hitchhike on endosomes and locally regulate the development of neural circuits

  2020cited by this paper
• The FTLD Risk Factor TMEM106B Regulates the Transport of Lysosomes at the Axon Initial Segment of Motoneurons.

  2020cited by this paper
• The Dynein Adaptor RILP Controls Neuronal Autophagosome Biogenesis, Transport, and Clearance.

  2020cited by this paper
• Rab2 drives axonal transport of dense core vesicles and lysosomal organelles

  2020cited by this paper
• Overlapping roles of JIP3 and JIP4 in promoting axonal transport of lysosomes in human iPSC-derived neurons

  2020cited by this paper
• ARL8 Relieves SKIP Autoinhibition to Enable Coupling of Lysosomes to Kinesin-1.

  2020cited by this paper
• Sensing of nutrients by CPT1C regulates late endosome/lysosome anterograde transport and axon growth

  2019cited by this paper
• Maturation and Clearance of Autophagosomes in Neurons Depends on a Specific Cysteine Protease Isoform, ATG-4.2.

  2019cited by this paper
• Neuronal Soma-Derived Degradative Lysosomes Are Continuously Delivered to Distal Axons to Maintain Local Degradation Capacity

  2019cited by this paper
• Lysosomal storage disorders – challenges, concepts and avenues for therapy: beyond rare diseases

  2019cited by this paper
• Lysosome Fission: Planning for an Exit.

  2019cited by this paper
• Neuronal lysosomes.

  2019cited by this paper
• F‐actin patches associated with glutamatergic synapses control positioning of dendritic lysosomes

  2019cited by this paper
• Activity-Dependent Secretion of Synaptic Organizer Cbln1 from Lysosomes in Granule Cell Axons.

  2019cited by this paper
• Lysosome trafficking and signaling in health and neurodegenerative diseases.

  2019cited by this paper
• Late Endosomes Act as mRNA Translation Platforms and Sustain Mitochondria in Axons

  2019cited by this paper
• RNA Granules Hitchhike on Lysosomes for Long-Distance Transport, Using Annexin A11 as a Molecular Tether

  2019cited by this paper
• Retrograde signaling via axonal transport through signaling endosomes.

  2019cited by this paper
• Lysosomal Dysfunction at the Centre of Parkinson's Disease and Frontotemporal Dementia/Amyotrophic Lateral Sclerosis.

  2019cited by this paper
• Lysosomes as dynamic regulators of cell and organismal homeostasis

  2019cited by this paper
• A Mutation in the Borcs7 Subunit of the Lysosome Regulatory BORC Complex Results in Motor Deficits and Dystrophic Axonopathy in Mice.

  2018cited by this paper
• Endo-lysosomal dysfunction: a converging mechanism in neurodegenerative diseases.

  2018cited by this paper
• Live Observation of Two Parallel Membrane Degradation Pathways at Axon Terminals

  2018cited by this paper
• Axonal transport and maturation of lysosomes.

  2018cited by this paper
• The cytoplasmic dynein transport machinery and its many cargoes

  2018cited by this paper
• Signaling

  2018cited by this paper
• Lysosomes and Brain Health.

  2018cited by this paper
• The Endolysosomal System and Proteostasis: From Development to Degeneration

  2018cited by this paper
• Lysosomal storage diseases

  2018cited by this paper
• Presynaptic Biogenesis Requires Axonal Transport of Lysosome-Related Vesicles.

  2018cited by this paper
• Characterization of LAMP1-labeled nondegradative lysosomal and endocytic compartments in neurons

  2018cited by this paper
• Degradation of dendritic cargos requires Rab7-dependent transport to somatic lysosomes

  2018influential reference
• Impaired axonal retrograde trafficking of the retromer complex augments lysosomal deficits in Alzheimer’s disease neurons

  2017cited by this paper
• Impaired retrograde transport of axonal autophagosomes contributes to autophagic stress in Alzheimer’s disease neurons

  2017cited by this paper
• WDR91 is a Rab7 effector required for neuronal development

  2017cited by this paper
• Activity-Dependent Exocytosis of Lysosomes Regulates the Structural Plasticity of Dendritic Spines

  2017cited by this paper
• Moving and positioning the endolysosomal system.

  2017cited by this paper
• Activity-dependent trafficking of lysosomes in dendrites and dendritic spines

  2017cited by this paper
• Impaired JIP3-dependent axonal lysosome transport promotes amyloid plaque pathology

  2017cited by this paper
• BORC Regulates the Axonal Transport of Synaptic Vesicle Precursors by Activating ARL-8.

  2017cited by this paper
• TFEB regulates lysosomal positioning by modulating TMEM55B expression and JIP4 recruitment to lysosomes

  2017cited by this paper
• Autophagy and Neurodegeneration: Pathogenic Mechanisms and Therapeutic Opportunities.

  2017cited by this paper
• Loss of TMEM106B Ameliorates Lysosomal and Frontotemporal Dementia-Related Phenotypes in Progranulin-Deficient Mice.

  2017cited by this paper
• Age-Dependent Dopaminergic Neurodegeneration and Impairment of the Autophagy-Lysosomal Pathway in LRRK-Deficient Mice.

  2017cited by this paper
• Altered lysosome distribution is an early neuropathological event in neurological forms of Gaucher disease

  2017cited by this paper
• Emerging Roles for the Lysosome in Lipid Metabolism.

  2017cited by this paper
• JIP1 and JIP3 cooperate to mediate TrkB anterograde axonal transport by activating kinesin-1

  2017cited by this paper
• Frontotemporal Dementia.

  2017cited by this paper
• BORC/kinesin-1 ensemble drives polarized transport of lysosomes into the axon

  2017cited by this paper
• The neurotrophin receptor signaling endosome: Where trafficking meets signaling

  2017cited by this paper
• BORC coordinates encounter and fusion of lysosomes with autophagosomes

  2017cited by this paper
• Kinesin 1 Drives Autolysosome Tubulation.

  2016cited by this paper
• Fluorogenic Probes for Multicolor Imaging in Living Cells.

  2016cited by this paper
• BORC Functions Upstream of Kinesins 1 and 3 to Coordinate Regional Movement of Lysosomes along Different Microtubule Tracks.

  2016cited by this paper
• Endolysosomes Are the Principal Intracellular Sites of Acid Hydrolase Activity

  2016cited by this paper
• Neurotrophin signaling endosomes: biogenesis, regulation, and functions.

  2016cited by this paper
• Phosphoinositides in Control of Membrane Dynamics.

  2016cited by this paper
• The Lysosome as a Regulatory Hub.

  2016cited by this paper
• Mechanisms and functions of lysosome positioning

  2016cited by this paper
• The lysosome as a command-and-control center for cellular metabolism

  2016cited by this paper
• Negative regulation of phosphatidylinositol 3-phosphate levels in early-to-late endosome conversion

  2016cited by this paper
• BORC, a multisubunit complex that regulates lysosome positioning.

  2015cited by this paper
• Massive accumulation of luminal protease-deficient axonal lysosomes at Alzheimer’s disease amyloid plaques

  2015influential reference
• Axonal autophagosomes recruit dynein for retrograde transport through fusion with late endosomes

  2015influential reference
• Bridging the age spectrum of neurodegenerative storage diseases.

  2015cited by this paper
• Repeated ER–endosome contacts promote endosome translocation and neurite outgrowth

  2015cited by this paper
• Endolysosomal Deficits Augment Mitochondria Pathology in Spinal Motor Neurons of Asymptomatic fALS Mice.

  2015influential reference
• Regulation of synaptic activity by snapin‐mediated endolysosomal transport and sorting

  2015cited by this paper
• Presenilin 1 Maintains Lysosomal Ca(2+) Homeostasis via TRPML1 by Regulating vATPase-Mediated Lysosome Acidification.

  2015cited by this paper
• The Vesicle Protein SAM-4 Regulates the Processivity of Synaptic Vesicle Transport

  2014cited by this paper
• Axonal transport: cargo-specific mechanisms of motility and regulation.

  2014cited by this paper
• Integrated regulation of motor-driven organelle transport by scaffolding proteins.

  2014cited by this paper
• The Regulation of Autophagosome Dynamics by Huntingtin and HAP1 Is Disrupted by Expression of Mutant Huntingtin, Leading to Defective Cargo Degradation

  2014influential reference
• LC3 binding to the scaffolding protein JIP1 regulates processive dynein-driven transport of autophagosomes.

  2014cited by this paper
• Signals from the lysosome: a control centre for cellular clearance and energy metabolism

  2013cited by this paper
• The FTLD risk factor TMEM106B and MAP6 control dendritic trafficking of lysosomes

  2013cited by this paper
• The balance between capture and dissociation of presynaptic proteins controls the spatial distribution of synapses.

  2013cited by this paper
• Late endosomal transport and tethering are coupled processes controlled by RILP and the cholesterol sensor ORP1L

  2013cited by this paper
• JNK-Interacting Protein 3 Mediates the Retrograde Transport of Activated c-Jun N-Terminal Kinase and Lysosomes

  2013cited by this paper
• The cell biology of disease: lysosomal storage disorders: the cellular impact of lysosomal dysfunction.

  2012cited by this paper
• Lysosomal calcium homeostasis defects, not proton pump defects, cause endo-lysosomal dysfunction in PSEN-deficient cells

  2012cited by this paper
• Autophagosomes initiate distally and mature during transport toward the cell soma in primary neurons

  2012cited by this paper
• Alterations in axonal transport motor proteins in sporadic and experimental Parkinson's disease.

  2012cited by this paper

• Cerebrospinal Fluid Proteome Reveals Dysregulation of Lysosomal and Axonal Proteins in Neurosyphilis

  2026cites this paper
• Lysosome trafficking across intracellular and intercellular networks in the brain

  2026cites this paper
• Cholinergic Phenotypes of Acetyl-CoA with ATP-Citrate Lyase Link

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• Distribution and Levels of Insulin-like Growth Factor 2 Receptor Across Mouse Brain Cell Types.

  2025cites this paper
• CK1δ-Dependent SNAPIN Dysregulation Drives Lysosomal Failure in HIV-1 Vpr–Exposed Neurons: A Targetable Mechanism in HAND

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• Pharmacologic reversion of Merkel cell carcinoma via CBP/p300 inhibition.

  2025cites this paper
• Maintenance and Decline of Neuronal Lysosomal Function in Aging

  2025cites this paper
• Non-canonical roles of lysosomes in neurons.

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• Fluoxetine promotes lysosome biogenesis via regulation of a RIOK2-TFEB axis

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• PKA orchestrates long-range lysosomal vesicle transport during synaptic maintenance

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• MEG3 Enhances Survival of Developing Human Neurons with CLCN4-Linked Autophagy Impairment

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• Rab27b Promotes Lysosomal Function and Alpha-Synuclein Clearance in Neurons

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• Multi-organelle-mediated mRNA localization in neurons and links to disease.

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• RNF13 variants L311S and L312P associated with developmental epileptic encephalopathy alter dendritic organization in hippocampal neurons

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• Endosomal protein DENND10 promotes developmental competence of neurite extension

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• Glia phagocytose neuronal sphingolipids to infiltrate developing synapses

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• Autophagy dysfunction and neurodegeneration: Where does it go wrong?

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• Commissureless acts as a substrate adapter in a conserved Nedd4 E3 ubiquitin ligase pathway to promote axon growth across the midline

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• Macroautophagy at the service of synapses.

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• Multifactorial etiology of progressive supranuclear palsy (PSP): the genetic component

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• Seeing the Unseen: Structured Illumination Microscopy for Organelles Dynamics

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• The Influence of Schwann Cell Metabolism and Dysfunction on Axon Maintenance

  2025cites this paper
• White matter injury after brain ischemia.

  2025cites this paper
• Integrated bioinformatics and experimental validation identify lysosome and immune infiltration-related genes as therapeutic targets in late-onset major depressive disorder

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• CK1δ-dependent SNAPIN dysregulation drives lysosomal failure in HIV-1 Vpr-exposed neurons: A targetable mechanism in HAND

  2025cites this paper
• Spatial Regulation of Lysosomal Vesicle Acidification Along the Axon via mRAVE-Dependent v-ATPase Assembly

  2025influential citation
• 3-Nitrotyrosine shortens axons of non-dopaminergic neurons by inhibiting mitochondrial motility.

  2024cites this paper
• Endolysosomal vesicles at the center of B cell activation

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• VPS13C regulates phospho-Rab10-mediated lysosomal function in human dopaminergic neurons

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• Atlastin-1 regulates endosomal tubulation and lysosomal proteolysis in human cortical neurons

  2024cites this paper
• Efficient axonal transport of endolysosomes relies on the balanced ratio of microtubule tyrosination and detyrosination

  2024cites this paper
• Association of TMEM106B with Cortical APOE Gene Expression in Neurodegenerative Conditions

  2024cites this paper
• Presynaptic perspective: Axonal transport defects in neurodevelopmental disorders

  2024cites this paper
• Axonal organelle buildup from loss of AP-4 complex function causes exacerbation of amyloid plaque pathology and gliosis in Alzheimer’s disease mouse model

  2024cites this paper
• Messenger RNA transport on lysosomal vesicles maintains axonal mitochondrial homeostasis and prevents axonal degeneration

  2024cites this paper
• Mucopolysaccharidosis type II zebrafish model exhibits early impaired proteasomal-mediated degradation of the axon guidance receptor Dcc

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• Autophagy initiation triggers p150Glued–AP-2β interaction on the lysosomes and facilitates their transport

  2024cites this paper
• The amyloid precursor protein and its derived fragments concomitantly contribute to the alterations of mitochondrial transport machinery in Alzheimer’s disease

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• Pathological Functions of Lysosomal Ion Channels in the Central Nervous System

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• Disruptions in axonal lysosome transport and its contribution to neurological disease

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• Rab27b promotes lysosomal function and alpha-synuclein clearance in neurons

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• Genes for endosomal pH regulators NHE6 and NHE9 are dysregulated in the substantia nigra in parkinson's disease.

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• PKA Activity-Driven Modulation of Bidirectional Long-Distance transport of Lysosomal vesicles During Synapse Maintenance

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• A decrease in Fkbp52 alters autophagosome maturation and A152T-tau clearance in vivo

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• SNX32 Regulates Sorting and Trafficking of Activated EGFR to the Lysosomal Degradation Pathway

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• Axonal Organelle Buildup from Loss of AP-4 Complex Function Causes Exacerbation of Amyloid Plaque Pathology and Gliosis in Alzheimer's Disease Mouse Model

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• Emerging tools for studying receptor endocytosis and signaling.

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• Spatacsin regulates directionality of lysosome trafficking by promoting the degradation of its partner AP5Z1

  2023cites this paper
• Commissureless acts as a substrate adapter in a conserved Nedd4 E3 ubiquitin ligase pathway to promote axon growth across the midline

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• FMRP Long-Range Transport and Degradation Are Mediated by Dynlrb1 in Sensory Neurons

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• How neurons maintain their axons long-term: an integrated view of axon biology and pathology

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• SGIP1 binding to the α-helical H9 domain of cannabinoid receptor 1 promotes axonal surface expression

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• Connecting neurodevelopment to neurodegeneration: a spotlight on the role of kinesin superfamily protein 2A (KIF2A)

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• Spatacsin regulates directionality of lysosome trafficking

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• Spatial Regulation of Lysosomal Vesicle Acidification Along the Axon 2 via mRAVE-Dependent v-ATPase Assembly

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