ArticleThe Journal of biological chemistry2008
Niemann-Pick C1 functions in regulating lysosomal amine content.
Article in The Journal of biological chemistry, 2008. The graph could read no effect estimate from its abstract, so it casts no vote on the map. Cited by 31 papers.
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Who cites it
31 citing papers in PubMed, 53 citations in OpenAlex.
- Human AKR1C3 binds agonists of GPR84 and participates in an expanded polyamine pathway.Cell chemical biology · 2025Article
- Connexin43 promotes exocytosis of damaged lysosomes through actin remodelling.The EMBO journal · 2024Article
- Endosomal escape: A bottleneck for LNP-mediated therapeutics.Proceedings of the National Academy of Sciences of the United States of America · 2024Review
- Polyamine-Derived Aminoaldehydes and Acrolein: Cytotoxicity, Reactivity and Analysis of the Induced Protein Modifications.Molecules (Basel, Switzerland) · 2023Review
- Maintenance and loss of endocytic organelle integrity: mechanisms and implications for antigen cross-presentation.Open biology · 2021Review
- Article
- Repurposing the Ebola and Marburg Virus Inhibitors Tilorone, Quinacrine, and Pyronaridine:ACS omega · 2021Article
- Human iNSC-derived brain organoid model of lysosomal storage disorder in Niemann-Pick disease type C.Cell death & disease · 2020Article
- Review
- Repurposing the Ebola and Marburg Virus Inhibitors Tilorone, Quinacrine and Pyronaridine: In vitro Activity Against SARS-CoV-2 and Potential Mechanisms.bioRxiv : the preprint server for biology · 2020Article
- Article
- Boosting Intracellular Delivery of Lipid Nanoparticle-Encapsulated mRNA.Nano letters · 2017Article
- Identifying the structure-activity relationship of leelamine necessary for inhibiting intracellular cholesterol transport.Oncotarget · 2017Article
- Monitoring of Dual CRISPR/Cas9-Mediated Steroidogenic Acute Regulatory Protein Gene Deletion and Cholesterol Accumulation Using High-Resolution FluorescenceFrontiers in endocrinology · 2017Article
- Endolysosomes Are the Principal Intracellular Sites of Acid Hydrolase Activity.Current biology : CB · 2016Article
- Acidic CaMessenger (Los Angeles, Calif. : Print) · 2016Article
- Article
- Human Ebola virus infection in West Africa: a review of available therapeutic agents that target different steps of the life cycle of Ebola virus.Infectious diseases of poverty · 2014Article
- Cationic amphiphilic drugs cause a marked expansion of apparent lysosomal volume: implications for an intracellular distribution-based drug interaction.Molecular pharmaceutics · 2012Article
- A novel role for α-tocopherol transfer protein (α-TTP) in protecting against chloroquine toxicity.The Journal of biological chemistry · 2012Article
Corrections and comments
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Authors and funding
2 authors at 1 institution in 1 country.
Funding
Abstract
Mutations in the late endosomal/lysosomal membrane protein Niemann-Pick C1 (NPC1) are known to cause a generalized block in retrograde vesicle-mediated transport, resulting in the hyper-accumulation of multiple lysosomal cargos. An important, yet often overlooked, category of lysosomal cargo includes the vast array of small molecular weight amine-containing molecules that are substrates for ion trapping in the highly acidic organelle lumen. We show here that the introduction of amine-containing molecules in lysosomes can significantly stimulate NPC1-mediated late endosome/lysosome fusion, and subsequently the secretion of lysosomal cargo. To illustrate the physiological importance of this NPC1-mediated transport pathway, we show that NPC1-deficient cells are more susceptible to the toxic effects of a lysosomotropic polyamine metabolite 3-aminopropanal. Moreover, NPC fibroblasts are shown to have higher levels of polyamine oxidase, an enzyme involved in the formation of 3-aminopropanal. Collectively, these findings provide strong support for a novel functional role for NPC1 and may also provide clues toward understanding NPC disease progression.
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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.