{"status":"ok","message-type":"work","message-version":"1.0.0","message":{"indexed":{"date-parts":[[2026,10,12]],"date-time":"2026-10-12T13:45:23Z","timestamp":1791812723754,"version":"4.3.4"},"reference-count":54,"publisher":"Rockefeller University Press","issue":"2","license":[{"start":{"date-parts":[[2025,12,16]],"date-time":"2025-12-16T00:00:00Z","timestamp":1765843200000},"content-version":"vor","delay-in-days":0,"URL":"https:\/\/creativecommons.org\/licenses\/by\/4.0\/"}],"funder":[{"DOI":"10.13039\/501100001809","name":"University of Namur","doi-asserted-by":"publisher","id":[{"id":"10.13039\/501100001809","id-type":"DOI","asserted-by":"publisher"}]},{"DOI":"10.13039\/501100002661","name":"Fonds De La Recherche Scientifique - FNRS","doi-asserted-by":"publisher","award":["CDR J.0012.19"],"award-info":[{"award-number":["CDR J.0012.19"]}],"id":[{"id":"10.13039\/501100002661","id-type":"DOI","asserted-by":"publisher"}]}],"content-domain":{"domain":["rupress.org"],"crossmark-restriction":true},"short-container-title":[],"published-print":{"date-parts":[[2026,2,2]]},"abstract":"<jats:p>Spastic paraplegia 21 is a neurodegenerative disease characterized by the degeneration of corticospinal axons. It is caused by mutations in the SPG21 gene, which encodes maspardin, a cytosolic protein of unknown function that associates with the late endosomal\/lysosomal membrane. Intriguingly, we found that the phosphorylation level of the transcription factor EB (TFEB), a master regulator of the CLEAR gene network, is decreased in SPG21 knockout cells, leading to TFEB nuclear translocation. Our investigations revealed that the Rag-mediated presentation of TFEB to the mTOR kinase and its subsequent phosphorylation is disturbed by a delocalization of the RAB7 GTPase, a maspardin-binding partner, from retromer-positive late endosomes to lysosomes. This redistribution decreases the interaction between RAB7 and its GTPase-activating protein (GAP), TBC1D5. Consequently, RAB7 remains primarily GTP-bound, recruiting more FYCO1 to lysosomes and promoting the anterograde movement of these organelles along microtubules. These findings identify maspardin as a newly discovered RAB7 effector and shed light on several consequences of its deficiency.<\/jats:p>","DOI":"10.1083\/jcb.202501135","type":"journal-article","created":{"date-parts":[[2025,10,30]],"date-time":"2025-10-30T06:21:09Z","timestamp":1761805269000},"update-policy":"https:\/\/doi.org\/10.1083\/jcb.crossmarkpolicy","source":"Crossref","is-referenced-by-count":2,"title":["Maspardin\/SPG21 controls lysosome motility and TFEB phosphorylation through RAB7 positioning"],"prefix":"10.1083","volume":"225","author":[{"ORCID":"https:\/\/orcid.org\/0009-0000-3116-3564","authenticated-orcid":true,"given":"Thomas","family":"Jacqmin","sequence":"first","affiliation":[{"id":[{"id":"https:\/\/ror.org\/03d1maw17","id-type":"ROR","asserted-by":"publisher"}],"name":"NARILIS, University of Namur 1 Laboratory of Intracellular Trafficking Biology, URPhyM, , Namur,","place":["Belgium"]}],"role":[{"vocabulary":"crossref","role":"author"}]},{"ORCID":"https:\/\/orcid.org\/0009-0008-3446-9575","authenticated-orcid":true,"given":"Florentine","family":"Gilis","sequence":"additional","affiliation":[{"id":[{"id":"https:\/\/ror.org\/03d1maw17","id-type":"ROR","asserted-by":"publisher"}],"name":"NARILIS, University of Namur 1 Laboratory of Intracellular Trafficking Biology, URPhyM, , Namur,","place":["Belgium"]},{"id":[{"id":"https:\/\/ror.org\/03d1maw17","id-type":"ROR","asserted-by":"publisher"}],"name":"NARILIS, University of Namur 2 Laboratory of Physiological Chemistry, URPhyM, , Namur,","place":["Belgium"]}],"role":[{"vocabulary":"crossref","role":"author"}]},{"ORCID":"https:\/\/orcid.org\/0009-0009-5526-3629","authenticated-orcid":true,"given":"Martine","family":"Albert","sequence":"additional","affiliation":[{"id":[{"id":"https:\/\/ror.org\/03d1maw17","id-type":"ROR","asserted-by":"publisher"}],"name":"NARILIS, University of Namur 1 Laboratory of Intracellular Trafficking Biology, URPhyM, , Namur,","place":["Belgium"]}],"role":[{"vocabulary":"crossref","role":"author"}]},{"ORCID":"https:\/\/orcid.org\/0009-0005-6148-033X","authenticated-orcid":true,"given":"Jean-Fran\u00e7ois","family":"Gaussin","sequence":"additional","affiliation":[{"id":[{"id":"https:\/\/ror.org\/03d1maw17","id-type":"ROR","asserted-by":"publisher"}],"name":"NARILIS, University of Namur 1 Laboratory of Intracellular Trafficking Biology, URPhyM, , Namur,","place":["Belgium"]}],"role":[{"vocabulary":"crossref","role":"author"}]},{"ORCID":"https:\/\/orcid.org\/0000-0002-7425-3006","authenticated-orcid":true,"given":"Michel","family":"Jadot","sequence":"additional","affiliation":[{"id":[{"id":"https:\/\/ror.org\/03d1maw17","id-type":"ROR","asserted-by":"publisher"}],"name":"NARILIS, University of Namur 2 Laboratory of Physiological Chemistry, URPhyM, , Namur,","place":["Belgium"]}],"role":[{"vocabulary":"crossref","role":"author"}]},{"ORCID":"https:\/\/orcid.org\/0000-0002-1884-7846","authenticated-orcid":true,"given":"Marielle","family":"Boonen","sequence":"additional","affiliation":[{"id":[{"id":"https:\/\/ror.org\/03d1maw17","id-type":"ROR","asserted-by":"publisher"}],"name":"NARILIS, University of Namur 1 Laboratory of Intracellular Trafficking Biology, URPhyM, , Namur,","place":["Belgium"]}],"role":[{"vocabulary":"crossref","role":"author"}]}],"member":"291","published-online":{"date-parts":[[2025,12,16]]},"reference":[{"key":"2025121609560400600_bib1","doi-asserted-by":"publisher","first-page":"4245","DOI":"10.1038\/s41467-021-24499-6","article-title":"TSC2 regulates lysosome biogenesis via a non-canonical RAGC and TFEB-dependent mechanism","volume":"12","author":"Alesi","year":"2021","journal-title":"Nat. Commun."},{"key":"2025121609560400600_bib2","doi-asserted-by":"publisher","first-page":"1337","DOI":"10.1083\/jcb.201609033","article-title":"Defects in ER-endosome contacts impact lysosome function in hereditary spastic paraplegia","volume":"216","author":"Allison","year":"2017","journal-title":"J. Cell Biol."},{"key":"2025121609560400600_bib3","doi-asserted-by":"publisher","first-page":"799953","DOI":"10.3389\/fneur.2021.799953","article-title":"Mast syndrome outside the Amish community: SPG21 in Europe","volume":"12","author":"Amprosi","year":"2021","journal-title":"Front. Neurol."},{"key":"2025121609560400600_bib4","doi-asserted-by":"publisher","first-page":"1135","DOI":"10.1074\/jbc.M112.417766","article-title":"Rab7 mutants associated with Charcot-Marie-Tooth disease cause delayed growth factor receptor transport and altered endosomal and nuclear signaling","volume":"288","author":"BasuRay","year":"2013","journal-title":"J. Biol. Chem."},{"key":"2025121609560400600_bib5","doi-asserted-by":"publisher","first-page":"380","DOI":"10.1038\/s42003-019-0615-z","article-title":"Loss of spatacsin impairs cholesterol trafficking and calcium homeostasis","volume":"2","author":"Boutry","year":"2019","journal-title":"Commun. Biol."},{"key":"2025121609560400600_bib6","doi-asserted-by":"publisher","first-page":"5249","DOI":"10.1172\/JCI77598","article-title":"Spastic paraplegia proteins spastizin and spatacsin mediate autophagic lysosome reformation","volume":"124","author":"Chang","year":"2014","journal-title":"J. Clin. Invest."},{"key":"2025121609560400600_bib7","doi-asserted-by":"publisher","first-page":"819","DOI":"10.1126\/science.1231143","article-title":"Multiplex genome engineering using CRISPR\/Cas systems","volume":"339","author":"Cong","year":"2013","journal-title":"Science"},{"key":"2025121609560400600_bib8","doi-asserted-by":"publisher","first-page":"260","DOI":"10.1159\/000443666","article-title":"Loss of maspardin attenuates the growth and maturation of mouse cortical neurons","volume":"16","author":"Davenport","year":"2016","journal-title":"Neurodegener. Dis."},{"key":"2025121609560400600_bib9","doi-asserted-by":"publisher","first-page":"283","DOI":"10.1016\/j.bbrc.2008.05.060","article-title":"Characterization of the Rab7K157N mutant protein associated with Charcot-Marie-Tooth type 2B","volume":"372","author":"De Luca","year":"2008","journal-title":"Biochem. Biophys. Res. Commun."},{"key":"2025121609560400600_bib10","doi-asserted-by":"publisher","first-page":"5243","DOI":"10.1242\/jcs.02633","article-title":"Distribution and dynamics of Lamp1-containing endocytic organelles in fibroblasts deficient in BLOC-3","volume":"118","author":"Falc\u00f3n-P\u00e9rez","year":"2005","journal-title":"J. Cell Sci."},{"key":"2025121609560400600_bib11","doi-asserted-by":"publisher","first-page":"3153","DOI":"10.3390\/cells11193153","article-title":"TFEB; beyond its role as an autophagy and lysosomes regulator","volume":"11","author":"Franco-Ju\u00e1rez","year":"2022","journal-title":"Cells"},{"key":"2025121609560400600_bib12","doi-asserted-by":"publisher","first-page":"309","DOI":"10.1111\/tra.12143","article-title":"Rab7 is functionally required for selective cargo sorting at the early endosome","volume":"15","author":"Girard","year":"2014","journal-title":"Traffic"},{"key":"2025121609560400600_bib13","doi-asserted-by":"publisher","first-page":"7950","DOI":"10.1523\/JNEUROSCI.16-24-07950.1996","article-title":"Endocytosis of activated TrkA: Evidence that nerve growth factor induces formation of signaling endosomes","volume":"16","author":"Grimes","year":"1996","journal-title":"J. Neurosci."},{"key":"2025121609560400600_bib14","doi-asserted-by":"publisher","first-page":"217","DOI":"10.1007\/s10048-009-0172-6","article-title":"Interaction of the SPG21 protein ACP33\/maspardin with the aldehyde dehydrogenase ALDH16A1","volume":"10","author":"Hanna","year":"2009","journal-title":"Neurogenetics"},{"key":"2025121609560400600_bib15","doi-asserted-by":"publisher","first-page":"3703","DOI":"10.1242\/jcs.071472","article-title":"The cargo-selective retromer complex is a recruiting hub for protein complexes that regulate endosomal tubule dynamics","volume":"123","author":"Harbour","year":"2010","journal-title":"J. Cell Sci."},{"key":"2025121609560400600_bib16","doi-asserted-by":"publisher","first-page":"1151","DOI":"10.1016\/s0140-6736(83)92879-9","article-title":"Classification of the hereditary ataxias and paraplegias","volume":"1","author":"Harding","year":"1983","journal-title":"Lancet"},{"key":"2025121609560400600_bib17","doi-asserted-by":"publisher","first-page":"103","DOI":"10.1016\/0006-8993(74)90536-8","article-title":"The retrograde axonal transport of nerve growth factor","volume":"68","author":"Hendry","year":"1974","journal-title":"Brain Res."},{"key":"2025121609560400600_bib18","doi-asserted-by":"publisher","first-page":"2558","DOI":"10.1091\/mbc.E13-03-0170","article-title":"Interaction between AP-5 and the hereditary spastic paraplegia proteins SPG11 and SPG15","volume":"24","author":"Hirst","year":"2013","journal-title":"Mol. Biol. Cell"},{"key":"2025121609560400600_bib19","doi-asserted-by":"publisher","first-page":"4984","DOI":"10.1093\/hmg\/ddv220","article-title":"Loss of AP-5 results in accumulation of aberrant endolysosomes: Defining a new type of lysosomal storage disease","volume":"24","author":"Hirst","year":"2015","journal-title":"Hum. Mol. Genet."},{"key":"2025121609560400600_bib20","doi-asserted-by":"publisher","first-page":"163","DOI":"10.1038\/jhg.2013.139","article-title":"Molecular epidemiology and clinical spectrum of hereditary spastic paraplegia in the Japanese population based on comprehensive mutational analyses","volume":"59","author":"Ishiura","year":"2014","journal-title":"J. Hum. Genet."},{"key":"2025121609560400600_bib21","doi-asserted-by":"publisher","first-page":"194","DOI":"10.1074\/mcp.M116.064527","article-title":"Accounting for protein subcellular localization: A compartmental map of the rat liver proteome","volume":"16","author":"Jadot","year":"2017","journal-title":"Mol. Cell. Proteomics"},{"key":"2025121609560400600_bib22","doi-asserted-by":"publisher","first-page":"235","DOI":"10.15252\/embj.201797128","article-title":"Control of RAB7 activity and localization through the retromer-TBC1D5 complex enables RAB7-dependent mitophagy","volume":"37","author":"Jimenez-Orgaz","year":"2018","journal-title":"EMBO J."},{"key":"2025121609560400600_bib23","doi-asserted-by":"publisher","first-page":"1680","DOI":"10.1016\/s0960-9822(01)00531-0","article-title":"The Rab7 effector protein RILP controls lysosomal transport by inducing the recruitment of dynein-dynactin motors","volume":"11","author":"Jordens","year":"2001","journal-title":"Curr. Biol."},{"key":"2025121609560400600_bib24","doi-asserted-by":"publisher","first-page":"419","DOI":"10.1016\/j.nbd.2019.03.026","article-title":"A mouse model for SPG48 reveals a block of autophagic flux upon disruption of adaptor protein complex five","volume":"127","author":"Khundadze","year":"2019","journal-title":"Neurobiol. Dis."},{"key":"2025121609560400600_bib25","doi-asserted-by":"publisher","first-page":"ar123","DOI":"10.1091\/mbc.E25-07-0346","article-title":"The hereditary spastic paraplegia type 21 (SPG21) protein is a RAB7A effector that promotes noncanonical mTORC1-catalyzed TFEB phosphorylation and cytoplasmic retention","volume":"36","author":"Kunselman","year":"2025","journal-title":"Mol. Biol. Cell"},{"key":"2025121609560400600_bib26","doi-asserted-by":"publisher","first-page":"3019","DOI":"10.1083\/jcb.201812110","article-title":"Retromer and TBC1D5 maintain late endosomal RAB7 domains to enable amino acid-induced mTORC1 signaling","volume":"218","author":"Kvainickas","year":"2019","journal-title":"J. Cell Biol."},{"key":"2025121609560400600_bib27","doi-asserted-by":"publisher","first-page":"15866","DOI":"10.1038\/ncomms15866","article-title":"Negative regulation of EGFR signalling by the human folliculin tumour suppressor protein","volume":"8","author":"Laviolette","year":"2017","journal-title":"Nat. Commun."},{"key":"2025121609560400600_bib28","doi-asserted-by":"publisher","DOI":"10.1371\/journal.pbio.3001594","article-title":"Folliculin promotes substrate-selective mTORC1 activity by activating RagC to recruit TFE3","volume":"20","author":"Li","year":"2022","journal-title":"PLoS Biol."},{"key":"2025121609560400600_bib29","doi-asserted-by":"publisher","first-page":"8346","DOI":"10.1128\/JVI.00100-12","article-title":"Transport to late endosomes is required for efficient reovirus infection","volume":"86","author":"Mainou","year":"2012","journal-title":"J. Virol."},{"key":"2025121609560400600_bib30","doi-asserted-by":"publisher","first-page":"ar102","DOI":"10.1091\/mbc.E21-09-0473","article-title":"AP-4 regulates neuronal lysosome composition, function, and transport via regulating export of critical lysosome receptor proteins at the trans-Golgi network","volume":"33","author":"Majumder","year":"2022","journal-title":"Mol. Biol. Cell"},{"key":"2025121609560400600_bib31","doi-asserted-by":"publisher","first-page":"1033","DOI":"10.1093\/hmg\/ddp567","article-title":"Disease mutations in Rab7 result in unregulated nucleotide exchange and inappropriate activation","volume":"19","author":"McCray","year":"2010","journal-title":"Hum. Mol. Genet."},{"key":"2025121609560400600_bib32","doi-asserted-by":"publisher","first-page":"288","DOI":"10.1038\/ncb3114","article-title":"Lysosomal calcium signalling regulates autophagy through calcineurin and TFEB","volume":"17","author":"Medina","year":"2015","journal-title":"Nat. Cell Biol."},{"key":"2025121609560400600_bib33","doi-asserted-by":"publisher","first-page":"311","DOI":"10.1111\/boc.202000158","article-title":"A comprehensive library of fluorescent constructs of SARS-CoV-2 proteins and their initial characterisation in different cell types","volume":"113","author":"Miserey-Lenkei","year":"2021","journal-title":"Biol. Cell"},{"key":"2025121609560400600_bib34","doi-asserted-by":"publisher","first-page":"1399","DOI":"10.3390\/biom13091399","article-title":"Regulation of endosomal trafficking by Rab7 and its effectors in neurons: Clues from Charcot-Marie-Tooth 2B disease","volume":"13","author":"Mulligan","year":"2023","journal-title":"Biomolecules"},{"key":"2025121609560400600_bib35","doi-asserted-by":"publisher","first-page":"920","DOI":"10.1016\/j.tcb.2022.04.012","article-title":"Non-canonical mTORC1 signaling at the lysosome","volume":"32","author":"Napolitano","year":"2022","journal-title":"Trends Cell Biol."},{"key":"2025121609560400600_bib36","doi-asserted-by":"publisher","first-page":"597","DOI":"10.1038\/s41586-020-2444-0","article-title":"A substrate-specific mTORC1 pathway underlies Birt-Hogg-Dub\u00e9 syndrome","volume":"585","author":"Napolitano","year":"2020","journal-title":"Nature"},{"key":"2025121609560400600_bib37","doi-asserted-by":"publisher","first-page":"253","DOI":"10.1083\/jcb.200907015","article-title":"FYCO1 is a Rab7 effector that binds to LC3 and PI3P to mediate microtubule plus end-directed vesicle transport","volume":"188","author":"Pankiv","year":"2010","journal-title":"J. Cell Biol."},{"key":"2025121609560400600_bib38","doi-asserted-by":"publisher","DOI":"10.1371\/journal.pbio.3002337","article-title":"Spatacsin regulates directionality of lysosome trafficking by promoting the degradation of its partner AP5Z1","volume":"21","author":"Pierga","year":"2023","journal-title":"PLoS Biol."},{"key":"2025121609560400600_bib39","doi-asserted-by":"publisher","first-page":"1666","DOI":"10.1016\/j.bbadis.2017.04.007","article-title":"Spastin regulates VAMP7-containing vesicles trafficking in cortical neurons","volume":"1863","author":"Plaud","year":"2017","journal-title":"Biochim. Biophys. Acta Mol. Basis Dis."},{"key":"2025121609560400600_bib40","doi-asserted-by":"publisher","first-page":"ra42","DOI":"10.1126\/scisignal.2002790","article-title":"The transcription factor TFEB links mTORC1 signaling to transcriptional control of lysosome homeostasis","volume":"5","author":"Roczniak-Ferguson","year":"2012","journal-title":"Sci. Signal."},{"key":"2025121609560400600_bib41","doi-asserted-by":"publisher","first-page":"2831","DOI":"10.1091\/mbc.e08-09-0911","article-title":"Rab7 activation by growth factor withdrawal contributes to the induction of apoptosis","volume":"20","author":"Romero Rosales","year":"2009","journal-title":"Mol. Biol. Cell"},{"key":"2025121609560400600_bib42","doi-asserted-by":"publisher","first-page":"473","DOI":"10.1126\/science.1174447","article-title":"A gene network regulating lysosomal biogenesis and function","volume":"325","author":"Sardiello","year":"2009","journal-title":"Science"},{"key":"2025121609560400600_bib43","doi-asserted-by":"publisher","first-page":"552","DOI":"10.1016\/j.molcel.2017.09.026","article-title":"Intersubunit crosstalk in the rag GTPase heterodimer enables mTORC1 to respond rapidly to amino acid availability","volume":"68","author":"Shen","year":"2017","journal-title":"Mol. Cell"},{"key":"2025121609560400600_bib54","doi-asserted-by":"publisher","first-page":"2371","DOI":"10.1242\/jcs.048686","article-title":"Membrane recruitment of the cargo-selective retromer subcomplex is catalysed by the small GTPase Rab7 and inhibited by the Rab-GAP TBC1D5.","volume":"122","author":"Seaman","year":"2009","journal-title":"J. Cell Sci"},{"key":"2025121609560400600_bib44","doi-asserted-by":"publisher","first-page":"9545","DOI":"10.1073\/pnas.1811727115","article-title":"Ragulator and SLC38A9 activate the rag GTPases through noncanonical GEF mechanisms","volume":"115","author":"Shen","year":"2018","journal-title":"Proc. Natl. Acad. Sci. USA"},{"key":"2025121609560400600_bib45","doi-asserted-by":"publisher","first-page":"1147","DOI":"10.1086\/379522","article-title":"Maspardin is mutated in mast syndrome, a complicated form of hereditary spastic paraplegia associated with dementia","volume":"73","author":"Simpson","year":"2003","journal-title":"Am. J. Hum. Genet."},{"key":"2025121609560400600_bib46","doi-asserted-by":"publisher","first-page":"369","DOI":"10.1007\/s10048-010-0252-7","article-title":"Targeted disruption of the Mast syndrome gene SPG21 in mice impairs hind limb function and alters axon branching in cultured cortical neurons","volume":"11","author":"Soderblom","year":"2010","journal-title":"Neurogenetics"},{"key":"2025121609560400600_bib47","doi-asserted-by":"publisher","first-page":"1640","DOI":"10.1523\/JNEUROSCI.3677-07.2008","article-title":"Functional characterization of Rab7 mutant proteins associated with Charcot-Marie-Tooth type 2B disease","volume":"28","author":"Spinosa","year":"2008","journal-title":"J. Neurosci."},{"key":"2025121609560400600_bib48","doi-asserted-by":"publisher","first-page":"495","DOI":"10.1016\/j.molcel.2013.09.016","article-title":"The folliculin tumor suppressor is a GAP for the RagC\/D GTPases that signal amino acid levels to mTORC1","volume":"52","author":"Tsun","year":"2013","journal-title":"Mol. Cell"},{"key":"2025121609560400600_bib49","doi-asserted-by":"publisher","first-page":"34","DOI":"10.1080\/15548627.2018.1507438","article-title":"ZFYVE26\/SPASTIZIN and SPG11\/SPATACSIN mutations in hereditary spastic paraplegia types AR-SPG15 and AR-SPG11 have different effects on autophagy and endocytosis","volume":"15","author":"Vantaggiato","year":"2019","journal-title":"Autophagy"},{"key":"2025121609560400600_bib50","doi-asserted-by":"publisher","first-page":"516","DOI":"10.1016\/j.cellsig.2010.09.012","article-title":"Rab7: Role of its protein interaction cascades in endo-lysosomal traffic","volume":"23","author":"Wang","year":"2011","journal-title":"Cell. Signal."},{"key":"2025121609560400600_bib51","doi-asserted-by":"publisher","first-page":"829","DOI":"10.1080\/15548627.2021.1960116","article-title":"C5orf51 is a component of the MON1-CCZ1 complex and controls RAB7A localization and stability during mitophagy","volume":"18","author":"Yan","year":"2022","journal-title":"Autophagy"},{"key":"2025121609560400600_bib52","doi-asserted-by":"publisher","first-page":"929","DOI":"10.1002\/stem.3359","article-title":"Maintenance of neural stem-progenitor cells by the lysosomal biosynthesis regulators TFEB and TFE3 in the embryonic mouse telencephalon","volume":"39","author":"Yuizumi","year":"2021","journal-title":"Stem Cells"},{"key":"2025121609560400600_bib53","doi-asserted-by":"publisher","first-page":"7451","DOI":"10.1523\/JNEUROSCI.4322-12.2013","article-title":"Defective axonal transport of Rab7 GTPase results in dysregulated trophic signaling","volume":"33","author":"Zhang","year":"2013","journal-title":"J. Neurosci."}],"container-title":["Journal of Cell Biology"],"original-title":[],"language":"en","link":[{"URL":"https:\/\/rupress.org\/jcb\/article-pdf\/doi\/10.1083\/jcb.202501135\/1955100\/jcb_202501135.pdf","content-type":"application\/pdf","content-version":"vor","intended-application":"syndication"},{"URL":"https:\/\/rupress.org\/jcb\/article-pdf\/doi\/10.1083\/jcb.202501135\/1955100\/jcb_202501135.pdf","content-type":"unspecified","content-version":"vor","intended-application":"similarity-checking"}],"deposited":{"date-parts":[[2025,12,16]],"date-time":"2025-12-16T14:56:24Z","timestamp":1765896984000},"score":1,"resource":{"primary":{"URL":"https:\/\/rupress.org\/jcb\/article\/225\/2\/e202501135\/278579\/Maspardin-SPG21-controls-lysosome-motility-and"}},"subtitle":[],"short-title":[],"issued":{"date-parts":[[2025,12,16]]},"references-count":54,"journal-issue":{"issue":"2","published-print":{"date-parts":[[2026,2,2]]}},"URL":"https:\/\/doi.org\/10.1083\/jcb.202501135","relation":{},"ISSN":["0021-9525","1540-8140"],"issn-type":[{"value":"0021-9525","type":"print"},{"value":"1540-8140","type":"electronic"}],"subject":[],"published":{"date-parts":[[2025,12,16]]},"article-number":"e202501135"}}