ArticleProceedings of the National Academy of Sciences of the United States of America2002
The Caenorhabditis elegans mucolipin-like gene cup-5 is essential for viability and regulates lysosomes in multiple cell types.
Article in Proceedings of the National Academy of Sciences of the United States of America, 2002. The graph could read no effect estimate from its abstract, so it casts no vote on the map. Cited by 54 papers, 2 of them syntheses that pooled it.
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Who cites it
54 citing papers in PubMed, 2 syntheses or guidelines pooled it, 107 citations in OpenAlex.
- Guidelines for the use and interpretation of assays for monitoring autophagy (4th edition)Autophagy · 2021Guideline
- Guidelines for the use and interpretation of assays for monitoring autophagy (3rd edition).Autophagy · 2016Guideline
- Microbial tryptophan metabolism activates host lysosomal activity to facilitate lipid breakdown.PLoS biology · 2026Article
- Filamin FLN-2 promotes MVB biogenesis by mediating vesicle docking on the actin cytoskeleton.The Journal of cell biology · 2022Article
- Identification and Characterization Analysis of Transient Receptor Potential Mucolipin Protein ofInsects · 2021Article
- Probing lysosomal activityBiophysics reports · 2021Article
- Evolutionary Aspects of TRPMLs and TPCs.International journal of molecular sciences · 2020Review
- Lysosome activity is modulated by multiple longevity pathways and is important for lifespan extension ineLife · 2020Article
- An ABCG Transporter Functions in Rab Localization and Lysosome-Related Organelle Biogenesis inGenetics · 2020Article
- An autism-causing calcium channel variant functions with selective autophagy to alter axon targeting and behavior.PLoS genetics · 2019Article
- MCOLN1 Promotes Proliferation and Predicts Poor Survival of Patients with Pancreatic Ductal Adenocarcinoma.Disease markers · 2019Article
- Function and regulation of the Caenorhabditis elegans Rab32 family member GLO-1 in lysosome-related organelle biogenesis.PLoS genetics · 2018Article
- Measurement of Lysosomal Size and Lysosomal Marker Intensities in AdultBio-protocol · 2018Article
- Spatiotemporal regulation of autophagy duringeLife · 2017Article
- A Non-Cell-Autonomous Role of BEC-1/BECN1/Beclin1 in Coordinating Cell-Cycle Progression and Stem Cell Proliferation during Germline Development.Current biology : CB · 2017Article
- Regulators of Lysosome Function and Dynamics inG3 (Bethesda, Md.) · 2017Article
- The C. elegans model in toxicity testing.Journal of applied toxicology : JAT · 2017Review
- Intracellular trafficking pathways in silver nanoparticle uptake and toxicity in Caenorhabditis elegans.Nanotoxicology · 2016Article
- ESCRT-Dependent Cell Death in a Caenorhabditis elegans Model of the Lysosomal Storage Disorder Mucolipidosis Type IV.Genetics · 2016Article
- The role of TRPMLs in endolysosomal trafficking and function.Cell calcium · 2015Review
Corrections and comments
PubMed lists nothing against this paper. Absence here is not a guarantee, only a check that was made.
Authors and funding
3 authors at 2 institutions in 1 country.
Funding
No grant is acknowledged in the PubMed record.
Abstract
The misregulation of programmed cell death, or apoptosis, contributes to the pathogenesis of many diseases. We used Nomarski microscopy to screen for mutants containing refractile cell corpses in a C. elegans strain in which all programmed cell death is blocked and such corpses are absent. We isolated a mutant strain that accumulates refractile bodies resembling irregular cell corpses. We rescued this mutant phenotype with the C. elegans mucolipidosis type IV (ML-IV) homolog, the recently identified cup-5 (coelomocyte-uptake defective) gene. ML-IV is a human autosomal recessive lysosomal storage disease characterized by psychomotor retardation and ophthalmological abnormalities. Our null mutations in cup-5 cause maternal-effect lethality. In addition, cup-5 mutants contain excess lysosomes in many and possibly all cell types and contain lamellar structures similar to those observed in ML-IV cell lines. The human ML-IV gene is capable of rescuing both the maternal-effect lethality and the lysosome-accumulation abnormality of cup-5 mutants. cup-5 mutants seem to contain excess apoptotic cells as detected by staining with terminal deoxynucleotidyltransferase-mediated dUTP nick end labeling. We suggest that the increased apoptosis seen in cup-5 mutants is a secondary consequence of the lysosomal defect, and that abnormalities in apoptosis may be associated with human lysosomal storage disorders.
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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.