ArticleProceedings of the National Academy of Sciences of the United States of America2004
Caenorhabditis elegans functional orthologue of human protein h-mucolipin-1 is required for lysosome biogenesis.
Article in Proceedings of the National Academy of Sciences of the United States of America, 2004. The graph could read no effect estimate from its abstract, so it casts no vote on the map. Cited by 144 papers.
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144 citing papers in PubMed, 232 citations in OpenAlex.
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- The neurotrophic factor MANF regulates autophagy and lysosome function to promote proteostasis inProceedings of the National Academy of Sciences of the United States of America · 2024Article
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- Neurotrophic factor MANF regulates autophagy and lysosome function to promote proteostasis inbioRxiv : the preprint server for biology · 2024Article
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- Hologenome analysis reveals independent evolution to chemosymbiosis by deep-sea bivalves.BMC biology · 2023Article
- Filamin FLN-2 promotes MVB biogenesis by mediating vesicle docking on the actin cytoskeleton.The Journal of cell biology · 2022Article
- M05B5.4 (lysosomal phospholipase A2) promotes disintegration of autophagic vesicles to maintainAutophagy · 2022Article
84 more citing papers are in PubMed but not listed here.
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6 authors at 3 institutions in 1 country.
Funding
Abstract
Mucolipidosis type IV (MLIV) is an autosomal recessive lysosomal storage disease characterized by severe psychomotor retardation, achlorhydria, and ophthalmological abnormalities. Cells from several tissues in MLIV patients accumulate large vacuoles that are presumed to be lysosomes, but whose exact nature remains to be determined. Other defects include the deterioration of neuronal integrity in the retina and the cerebellum. MCOLN1, the gene mutated in MLIV patients, encodes a protein called h-mucolipin-1 that has six predicted transmembrane domains and functions as a Ca(2+)-permeable channel that is modulated by changes in Ca2+ concentration. CUP-5 is the Caenorhabditis elegans functional orthologue of h-mucolipin-1. Mutations in cup-5 result in the accumulation of large vacuoles in several cells, in increased cell death, and in embryonic lethality. We demonstrate here that CUP-5 functions in the biogenesis of lysosomes originating from hybrid organelles. We also show that at least two h-mucolipin family members rescue cup-5 mutant endocytic defects, indicating that there may be functional redundancy among the human proteins. Finally, we propose a model that relates the lysosome biogenesis defect in the absence of CUP-5/h-mucolipin-1 to cellular phenotypes in worms and in humans.
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