ArticlePloS one2017
Drosophila Vps13 Is Required for Protein Homeostasis in the Brain.
Article in PloS one, 2017. The graph could read no effect estimate from its abstract, so it casts no vote on the map. Cited by 26 papers.
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Who cites it
26 citing papers in PubMed.
- Parkinson's disease-associatedeLife · 2026Article
- Leave-One-Chromosome-Out Association Testing Decodes Uncapping Behaviour in Honeybees During Varroa destructor Infestation.Veterinary medicine and science · 2026Article
- Four subtypes of disease-causing missense mutations underlie pathogenic protein interactions in neurodegenerative VPS13A disease.The Journal of clinical investigation · 2026Article
- Impaired hematopoiesis and embryonic lethality at midgestation of mice lacking both lipid transfer proteins VPS13A and VPS13C.PLoS biology · 2025Article
- Neurodegenerative and Neurodevelopmental Roles for Bulk Lipid Transporters VPS13A and BLTP2.Movement disorders : official journal of the Movement Disorder Society · 2025Article
- Preserved VPS13A distribution and expression in Huntington's disease: divergent mechanisms of action for similar movement disorders?Frontiers in neuroscience · 2024Article
- Exploring the pathophysiological mechanisms and wet biomarkers ofFrontiers in neurology · 2024Review
- An Autopsy Series of Seven Cases of VPS13A Disease (Chorea-Acanthocytosis).Movement disorders : official journal of the Movement Disorder Society · 2023Article
- Effect of rapamycin on lysosomal accumulation in a CRISPR/Cas9-based cellular model of VPS13A deficiency.Journal of cellular and molecular medicine · 2023Article
- Emerging Epidemiological Data on Rare Intellectual Disability Syndromes from Analyzing the Data of a Large Iranian Cohort.Archives of Iranian medicine · 2023Article
- Inter-Species Rescue of Mutant Phenotype-The Standard for Genetic Analysis of Human Genetic Disorders inInternational journal of molecular sciences · 2022Review
- Unraveling the Spatiotemporal Distribution of VPS13A in the Mouse Brain.International journal of molecular sciences · 2021Article
- HIV infection of astrocytes compromises inter-organelle interactions and inositol phosphate metabolism: A potential mechanism of bystander damage and viral reservoir survival.Progress in neurobiology · 2021Article
- Insights into VPS13 properties and function reveal a new mechanism of eukaryotic lipid transport.Biochimica et biophysica acta. Molecular and cell biology of lipids · 2021Review
- Mitochondrial fission, integrity and completion of mitophagy require separable functions of Vps13D in Drosophila neurons.PLoS genetics · 2021Article
- The Vps13 Family of Lipid Transporters and Its Role at Membrane Contact Sites.International journal of molecular sciences · 2021Review
- Role of VPS13, a protein with similarity to ATG2, in physiology and disease.Current opinion in genetics & development · 2020Review
- Identification of two compound heterozygous VPS13A large deletions in chorea-acanthocytosis only by protein and quantitative DNA analysis.Molecular genetics & genomic medicine · 2020Article
- Neuronal lysosomes.Neuroscience letters · 2019Review
- VPS13A is closely associated with mitochondria and is required for efficient lysosomal degradation.Disease models & mechanisms · 2019Article
Corrections and comments
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Authors and funding
12 authors.
Funding
Abstract
Chorea-Acanthocytosis is a rare, neurodegenerative disorder characterized by progressive loss of locomotor and cognitive function. It is caused by loss of function mutations in the Vacuolar Protein Sorting 13A (VPS13A) gene, which is conserved from yeast to human. The consequences of VPS13A dysfunction in the nervous system are still largely unspecified. In order to study the consequences of VPS13A protein dysfunction in the ageing central nervous system we characterized a Drosophila melanogaster Vps13 mutant line. The Drosophila Vps13 gene encoded a protein of similar size as human VPS13A. Our data suggest that Vps13 is a peripheral membrane protein located to endosomal membranes and enriched in the fly head. Vps13 mutant flies showed a shortened life span and age associated neurodegeneration. Vps13 mutant flies were sensitive to proteotoxic stress and accumulated ubiquitylated proteins. Levels of Ref(2)P, the Drosophila orthologue of p62, were increased and protein aggregates accumulated in the central nervous system. Overexpression of the human Vps13A protein in the mutant flies partly rescued apparent phenotypes. This suggests a functional conservation of human VPS13A and Drosophila Vps13. Our results demonstrate that Vps13 is essential to maintain protein homeostasis in the larval and adult Drosophila brain. Drosophila Vps13 mutants are suitable to investigate the function of Vps13 in the brain, to identify genetic enhancers and suppressors and to screen for potential therapeutic targets for Chorea-Acanthocytosis.
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