ArticleDevelopment (Cambridge, England)2014
Ubpy controls the stability of the ESCRT-0 subunit Hrs in development.
Article in Development (Cambridge, England), 2014. The graph could read no effect estimate from its abstract, so it casts no vote on the map. Cited by 19 papers.
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Who cites it
19 citing papers in PubMed, 35 citations in OpenAlex.
- ESCRTing the RABs through conversion.Biochemical Society transactions · 2025Review
- Spatiotemporal recruitment of the ubiquitin-specific protease USP8 directs endosome maturation.eLife · 2024Article
- Ubiquitination of the ubiquitin-binding machinery: how early ESCRT components are controlled.Essays in biochemistry · 2022Review
- Usp8 promotes tumor cell migration through activating the JNK pathway.Cell death & disease · 2022Article
- O-GlcNAcylation regulates epidermal growth factor receptor intracellular trafficking and signaling.Proceedings of the National Academy of Sciences of the United States of America · 2022Article
- Dual functions of Rack1 in regulating Hedgehog pathway.Cell death and differentiation · 2020Article
- Roles for ER:endosome membrane contact sites in ligand-stimulated intraluminal vesicle formation.Biochemical Society transactions · 2018Review
- Review
- CHMP1B is a target of USP8/UBPY regulated by ubiquitin during endocytosis.PLoS genetics · 2018Article
- HRS plays an important role for TLR7 signaling to orchestrate inflammation and innate immunity upon EV71 infection.PLoS pathogens · 2017Article
- Integration of cellular ubiquitin and membrane traffic systems: focus on deubiquitylases.The FEBS journal · 2017Article
- A functional endosomal pathway is necessary for lysosome biogenesis in Drosophila.BMC cell biology · 2016Article
- Article
- Stuxnet Facilitates the Degradation of Polycomb Protein during Development.Developmental cell · 2016Article
- The emerging role of deubiquitinating enzymes in genomic integrity, diseases, and therapeutics.Cell & bioscience · 2016Review
- Smoothened regulation in response to Hedgehog stimulation.Frontiers in biology · 2015Article
- The Myopic-Ubpy-Hrs nexus enables endosomal recycling of Frizzled.Molecular biology of the cell · 2015Article
- Motor and Sensory Deficits in the teetering Mice Result from Mutation of the ESCRT Component HGS.PLoS genetics · 2015Article
- The Deubiquitinating Enzyme UBPY Is Required for Lysosomal Biogenesis and Productive Autophagy in Drosophila.PloS one · 2015Article
Corrections and comments
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Authors and funding
5 authors at 1 institution in 1 country.
Funding
Abstract
Ubiquitylated developmental membrane signaling proteins are often internalized for endocytic trafficking, through which endosomal sorting complexes required for transport (ESCRT) act sequentially to deliver internalized cargos to lysosomes. The ESCRT function in endocytic sorting is well established; however, it is not fully understood how the sorting machinery itself is regulated. Here, we show that Ubiquitin isopeptidase Y (Ubpy) plays a conserved role in vivo in the homeostasis of an essential ESCRT-0 complex component Hrs. We find that, in the absence of Drosophila Ubpy, multiple membrane proteins that are essential components of important signaling pathways accumulate in enlarged, aberrant endosomes. We further demonstrate that this phenotype results from endocytic pathway defects. We provide evidence that Ubpy interacts with and deubiquitylates Hrs. In Ubpy-null cells, Hrs becomes ubiquitylated and degraded in lysosomes, thus disrupting the integrity of ESCRT sorting machinery. Lastly, we find that signaling proteins are enriched in enlarged endosomes when Hrs activity is abolished. Together, our data support a model in which Ubpy plays a dual role in both cargo deubiquitylation and the ESCRT-0 stability during development.
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Registered trials
Read under generation 80e0d062 · epoch 390. Bibliography from PubMed, PubMed Central and OpenAlex; grants from NIH RePORTER; trial links from ClinicalTrials.gov; estimates, votes and beliefs from the OpenQuestion graph.