{"status":"ok","message-type":"work","message-version":"1.0.0","message":{"indexed":{"date-parts":[[2026,10,21]],"date-time":"2026-10-21T07:39:44Z","timestamp":1792568384016,"version":"4.5.0"},"reference-count":101,"publisher":"Elsevier BV","license":[{"start":{"date-parts":[[2026,12,1]],"date-time":"2026-12-01T00:00:00Z","timestamp":1796083200000},"content-version":"tdm","delay-in-days":0,"URL":"https:\/\/www.elsevier.com\/tdm\/userlicense\/1.0\/"},{"start":{"date-parts":[[2026,12,1]],"date-time":"2026-12-01T00:00:00Z","timestamp":1796083200000},"content-version":"tdm","delay-in-days":0,"URL":"https:\/\/www.elsevier.com\/legal\/tdmrep-license"},{"start":{"date-parts":[[2026,10,6]],"date-time":"2026-10-06T00:00:00Z","timestamp":1791244800000},"content-version":"vor","delay-in-days":0,"URL":"http:\/\/creativecommons.org\/licenses\/by\/4.0\/"}],"funder":[{"DOI":"10.13039\/501100001826","name":"ZonMw","doi-asserted-by":"publisher","id":[{"id":"10.13039\/501100001826","id-type":"DOI","asserted-by":"publisher"}]},{"DOI":"10.13039\/501100001659","name":"Deutsche Forschungsgemeinschaft","doi-asserted-by":"publisher","id":[{"id":"10.13039\/501100001659","id-type":"DOI","asserted-by":"publisher"}]},{"DOI":"10.13039\/501100013956","name":"NWO ENW","doi-asserted-by":"publisher","id":[{"id":"10.13039\/501100013956","id-type":"DOI","asserted-by":"publisher"}]},{"DOI":"10.13039\/501100002341","name":"Research Council of Finland","doi-asserted-by":"publisher","id":[{"id":"10.13039\/501100002341","id-type":"DOI","asserted-by":"publisher"}]},{"DOI":"10.13039\/100010665","name":"Horizon 2020 Marie Sk\u0142odowska-Curie Actions","doi-asserted-by":"publisher","id":[{"id":"10.13039\/100010665","id-type":"DOI","asserted-by":"publisher"}]}],"content-domain":{"domain":["clinicalkey.com","clinicalkey.com.au","clinicalkey.es","clinicalkey.fr","clinicalkey.jp","elsevier.com","sciencedirect.com"],"crossmark-restriction":true},"short-container-title":["Molecular Metabolism"],"published-print":{"date-parts":[[2026,12]]},"DOI":"10.1016\/j.molmet.2026.102456","type":"journal-article","created":{"date-parts":[[2026,10,11]],"date-time":"2026-10-11T05:56:54Z","timestamp":1791698214000},"page":"102456","update-policy":"https:\/\/doi.org\/10.1016\/elsevier_cm_policy","source":"Crossref","is-referenced-by-count":0,"special_numbering":"C","title":["Hepatic retromer is essential for systemic cholesterol homeostasis by regulating lysosomal cholesterol handling"],"prefix":"10.1016","volume":"114","author":[{"ORCID":"https:\/\/orcid.org\/0000-0002-8359-0370","authenticated-orcid":false,"given":"Cristy R.C.","family":"Verzijl","sequence":"first","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Markus G.","family":"Barbosa","sequence":"additional","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]},{"ORCID":"https:\/\/orcid.org\/0000-0003-3505-0422","authenticated-orcid":false,"given":"Dyonne Y.","family":"Vos","sequence":"additional","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]},{"ORCID":"https:\/\/orcid.org\/0000-0002-1316-9513","authenticated-orcid":false,"given":"Andries H.","family":"Heida","sequence":"additional","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]},{"ORCID":"https:\/\/orcid.org\/0000-0001-6409-2336","authenticated-orcid":false,"given":"Amandine","family":"Caillaud","sequence":"additional","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]},{"ORCID":"https:\/\/orcid.org\/0000-0002-7902-0009","authenticated-orcid":false,"given":"Lauri","family":"Vanharanta","sequence":"additional","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Xiaoke","family":"Ge","sequence":"additional","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]},{"ORCID":"https:\/\/orcid.org\/0000-0003-0335-128X","authenticated-orcid":false,"given":"Jo\u00ebl J.","family":"Tissink","sequence":"additional","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]},{"ORCID":"https:\/\/orcid.org\/0000-0001-6312-3321","authenticated-orcid":false,"given":"Mirjam H.","family":"Koster","sequence":"additional","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Marieke","family":"Smit","sequence":"additional","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]},{"ORCID":"https:\/\/orcid.org\/0009-0001-8221-3178","authenticated-orcid":false,"given":"Nicolette","family":"Huijkman","sequence":"additional","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]},{"ORCID":"https:\/\/orcid.org\/0009-0000-5500-3282","authenticated-orcid":false,"given":"Niels J.","family":"Kloosterhuis","sequence":"additional","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Aurore","family":"Girardeau","sequence":"additional","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]},{"ORCID":"https:\/\/orcid.org\/0009-0000-1019-8848","authenticated-orcid":false,"given":"Zo\u00e9","family":"Begu\u00e9-Racap\u00e9","sequence":"additional","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Vincent W.","family":"Bloks","sequence":"additional","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Rick","family":"Havinga","sequence":"additional","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Marceline M.","family":"Fuh","sequence":"additional","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Fulvio","family":"Reggiori","sequence":"additional","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Muriel","family":"Mari","sequence":"additional","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Ludger","family":"Scheja","sequence":"additional","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]},{"ORCID":"https:\/\/orcid.org\/0000-0003-2518-737X","authenticated-orcid":false,"given":"Folkert","family":"Kuipers","sequence":"additional","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]},{"ORCID":"https:\/\/orcid.org\/0000-0002-7988-8983","authenticated-orcid":false,"given":"Santiago","family":"Sol\u00e9-Dom\u00e8nech","sequence":"additional","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]},{"ORCID":"https:\/\/orcid.org\/0000-0002-0014-5571","authenticated-orcid":false,"given":"Sander","family":"Kooijman","sequence":"additional","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]},{"ORCID":"https:\/\/orcid.org\/0000-0002-5647-1034","authenticated-orcid":false,"given":"Joerg","family":"Heeren","sequence":"additional","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]},{"ORCID":"https:\/\/orcid.org\/0000-0001-6759-3291","authenticated-orcid":false,"given":"Jan Freark","family":"de Boer","sequence":"additional","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Patrick C.N.","family":"Rensen","sequence":"additional","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Elina","family":"Ikonen","sequence":"additional","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Antoine","family":"Rimbert","sequence":"additional","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]},{"ORCID":"https:\/\/orcid.org\/0000-0003-0066-3720","authenticated-orcid":false,"given":"Justina C.","family":"Wolters","sequence":"additional","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Jan Albert","family":"Kuivenhoven","sequence":"additional","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]},{"ORCID":"https:\/\/orcid.org\/0000-0003-3039-4365","authenticated-orcid":false,"given":"Bart","family":"van de Sluis","sequence":"additional","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]}],"member":"78","reference":[{"issue":"7","key":"10.1016\/j.molmet.2026.102456_bib1","article-title":"Function of the endolysosomal network in cholesterol homeostasis and metabolic-associated fatty liver disease (MAFLD)","volume":"158","author":"Vos","year":"2021","journal-title":"Mol Metabol"},{"issue":"4223","key":"10.1016\/j.molmet.2026.102456_bib2","doi-asserted-by":"crossref","first-page":"150","DOI":"10.1126\/science.174194","article-title":"Receptor-mediated control of cholesterol metabolism","volume":"191","author":"Brown","year":"1976","journal-title":"Science"},{"issue":"2","key":"10.1016\/j.molmet.2026.102456_bib3","doi-asserted-by":"crossref","first-page":"125","DOI":"10.1038\/nrm2336","article-title":"Cellular cholesterol trafficking and compartmentalization","volume":"9","author":"Ikonen","year":"2008","journal-title":"Nat Rev Mol Cell Biol"},{"issue":"21","key":"10.1016\/j.molmet.2026.102456_bib4","doi-asserted-by":"crossref","first-page":"8487","DOI":"10.1016\/S0021-9258(19)40786-2","article-title":"Role of lysosomal acid lipase in the metabolism of plasma low density lipoprotein. Observations in cultured fibroblasts from a patient with cholesteryl ester storage disease","volume":"250","author":"Goldstein","year":"1975","journal-title":"J Biol Chem"},{"issue":"40","key":"10.1016\/j.molmet.2026.102456_bib5","doi-asserted-by":"crossref","first-page":"15287","DOI":"10.1073\/pnas.0807328105","article-title":"NPC2 facilitates bidirectional transfer of cholesterol between NPC1 and lipid bilayers, a step in cholesterol egress from lysosomes","volume":"105","author":"Infante","year":"2008","journal-title":"Proc Natl Acad Sci"},{"issue":"1","key":"10.1016\/j.molmet.2026.102456_bib6","doi-asserted-by":"crossref","first-page":"3521","DOI":"10.1038\/s41467-019-11425-0","article-title":"Lysosomal integral membrane protein-2 (LIMP-2\/SCARB2) is involved in lysosomal cholesterol export","volume":"10","author":"Heybrock","year":"2019","journal-title":"Nat Commun"},{"issue":"2","key":"10.1016\/j.molmet.2026.102456_bib7","doi-asserted-by":"crossref","first-page":"329","DOI":"10.1016\/S0022-2275(20)42093-0","article-title":"Disposition of intracellular cholesterol in human fibroblasts","volume":"32","author":"Lange","year":"1991","journal-title":"JLR (J Lipid Res)"},{"issue":"3","key":"10.1016\/j.molmet.2026.102456_bib8","doi-asserted-by":"crossref","first-page":"331","DOI":"10.1016\/S0092-8674(00)80213-5","article-title":"The SREBP pathway: regulation of cholesterol metabolism by proteolysis of a membrane-bound transcription factor","volume":"89","author":"Brown","year":"1997","journal-title":"Cell"},{"issue":"12","key":"10.1016\/j.molmet.2026.102456_bib9","doi-asserted-by":"crossref","first-page":"1648","DOI":"10.1161\/CIRCRESAHA.117.312004","article-title":"The COMMD family regulates plasma LDL levels and attenuates atherosclerosis through stabilizing the CCC complex in endosomal LDLR trafficking","volume":"122","author":"Fedoseienko","year":"2018","journal-title":"Circ Res"},{"key":"10.1016\/j.molmet.2026.102456_bib10","doi-asserted-by":"crossref","DOI":"10.1038\/ncomms10961","article-title":"CCC- and WASH-mediated endosomal sorting of LDLR is required for normal clearance of circulating LDL","volume":"7","author":"Bartuzi","year":"2016","journal-title":"Nat Commun"},{"issue":"11","key":"10.1016\/j.molmet.2026.102456_bib11","doi-asserted-by":"crossref","DOI":"10.1172\/jci.insight.126462","article-title":"The hepatic WASH complex is required for efficient plasma LDL and HDL cholesterol clearance","volume":"4","author":"Wijers","year":"2019","journal-title":"JCI Insight"},{"issue":"1","key":"10.1016\/j.molmet.2026.102456_bib12","doi-asserted-by":"crossref","first-page":"16","DOI":"10.1186\/1750-1172-5-16","article-title":"Niemann-Pick disease type C","volume":"5","author":"Vanier","year":"2010","journal-title":"Orphanet J Rare Dis"},{"issue":"1","key":"10.1016\/j.molmet.2026.102456_bib13","doi-asserted-by":"crossref","first-page":"21","DOI":"10.1016\/j.atherosclerosis.2014.04.003","article-title":"Lysosomal acid lipase deficiency \u2013 an under-recognized cause of dyslipidaemia and liver dysfunction","volume":"235","author":"Reiner","year":"2014","journal-title":"Atherosclerosis"},{"issue":"9","key":"10.1016\/j.molmet.2026.102456_bib14","doi-asserted-by":"crossref","first-page":"670","DOI":"10.1016\/S2468-1253(17)30052-3","article-title":"Wolman's disease and cholesteryl ester storage disorder: the phenotypic spectrum of lysosomal acid lipase deficiency","volume":"2","author":"Pericleous","year":"2017","journal-title":"Lancet Gastroenterol Hepatol"},{"issue":"2","key":"10.1016\/j.molmet.2026.102456_bib15","doi-asserted-by":"crossref","first-page":"242","DOI":"10.1016\/S0022-3476(05)81695-6","article-title":"Niemann-Pick disease type C: diagnosis and outcome in children, with particular reference to liver disease","volume":"123","author":"Kelly","year":"1993","journal-title":"J Pediatr"},{"issue":"12","key":"10.1016\/j.molmet.2026.102456_bib16","doi-asserted-by":"crossref","first-page":"4105","DOI":"10.1091\/mbc.11.12.4105","article-title":"Human orthologs of yeast vacuolar protein sorting proteins Vps26, 29, and 35: assembly into multimeric complexes","volume":"11","author":"Haft","year":"2000","journal-title":"Mol Biol Cell"},{"issue":"2","key":"10.1016\/j.molmet.2026.102456_bib17","doi-asserted-by":"crossref","DOI":"10.1101\/cshperspect.a016774","article-title":"Retromer: a master conductor of endosome sorting","volume":"6","author":"Burd","year":"2014","journal-title":"Cold Spring Harbor Perspect Biol"},{"issue":"8","key":"10.1016\/j.molmet.2026.102456_bib18","doi-asserted-by":"crossref","first-page":"578","DOI":"10.1111\/tra.12574","article-title":"Endosomal receptor trafficking: retromer and beyond","volume":"19","author":"Wang","year":"2018","journal-title":"Traffic"},{"issue":"7","key":"10.1016\/j.molmet.2026.102456_bib19","doi-asserted-by":"crossref","first-page":"608","DOI":"10.1016\/j.tibs.2020.12.009","article-title":"The retromer complex: from genesis to revelations","volume":"46","author":"Seaman","year":"2021","journal-title":"Trends Biochem Sci"},{"issue":"1","key":"10.1016\/j.molmet.2026.102456_bib20","doi-asserted-by":"crossref","DOI":"10.1038\/ncomms11919","article-title":"Arf6 controls retromer traffic and intracellular cholesterol distribution via a phosphoinositide-based mechanism","volume":"7","author":"Marquer","year":"2016","journal-title":"Nat Commun"},{"issue":"1","key":"10.1016\/j.molmet.2026.102456_bib21","doi-asserted-by":"crossref","first-page":"123","DOI":"10.1083\/jcb.200312055","article-title":"Role of the mammalian retromer in sorting of the cation-independent mannose 6-phosphate receptor","volume":"165","author":"Arighi","year":"2004","journal-title":"J Cell Biol"},{"issue":"1","key":"10.1016\/j.molmet.2026.102456_bib22","doi-asserted-by":"crossref","first-page":"111","DOI":"10.1083\/jcb.200312034","article-title":"Cargo-selective endosomal sorting for retrieval to the Golgi requires retromer","volume":"165","author":"Seaman","year":"2004","journal-title":"J Cell Biol"},{"issue":"24","key":"10.1016\/j.molmet.2026.102456_bib23","doi-asserted-by":"crossref","DOI":"10.3390\/ijms222413256","article-title":"Impaired retromer function in niemann-pick type C disease is dependent on intracellular cholesterol accumulation","volume":"22","author":"Dominko","year":"2021","journal-title":"Int J Mol Sci"},{"issue":"5","key":"10.1016\/j.molmet.2026.102456_bib24","doi-asserted-by":"crossref","first-page":"216","DOI":"10.1111\/tra.12884","article-title":"SWIP mediates retromer-independent membrane recruitment of the WASH complex","volume":"24","author":"Dost\u00e1l","year":"2023","journal-title":"Traffic"},{"issue":"11","key":"10.1016\/j.molmet.2026.102456_bib25","doi-asserted-by":"crossref","first-page":"522","DOI":"10.1016\/j.tcb.2013.04.010","article-title":"Retromer-mediated endosomal protein sorting: all WASHed up","volume":"23","author":"Seaman","year":"2013","journal-title":"Trends Cell Biol"},{"issue":"2","key":"10.1016\/j.molmet.2026.102456_bib26","doi-asserted-by":"crossref","first-page":"615","DOI":"10.1083\/jcb.201806153","article-title":"Retromer has a selective function in cargo sorting via endosome transport carriers","volume":"218","author":"Cui","year":"2019","journal-title":"JCB (J Cell Biol)"},{"issue":"9","key":"10.1016\/j.molmet.2026.102456_bib27","doi-asserted-by":"crossref","first-page":"2217","DOI":"10.1080\/15548627.2020.1821545","article-title":"Retromer regulates the lysosomal clearance of MAPT\/tau","volume":"17","author":"Carosi","year":"2021","journal-title":"Autophagy"},{"issue":"10","key":"10.1016\/j.molmet.2026.102456_bib28","doi-asserted-by":"crossref","first-page":"1214","DOI":"10.1038\/ncb3610","article-title":"Retriever is a multiprotein complex for retromer-independent endosomal cargo recycling","volume":"19","author":"McNally","year":"2017","journal-title":"Nat Cell Biol"},{"issue":"1","key":"10.1016\/j.molmet.2026.102456_bib29","first-page":"e29","article-title":"Cargo-specific role for retriever subunit VPS26C in hepatocyte lipoprotein receptor recycling to control postprandial triglyceride-rich lipoproteins","volume":"43","author":"Vos","year":"2022","journal-title":"Arterioscler Thromb Vasc Biol"},{"issue":"7929","key":"10.1016\/j.molmet.2026.102456_bib30","doi-asserted-by":"crossref","first-page":"1005","DOI":"10.1038\/s41586-022-05221-y","article-title":"CLN3 is required for the clearance of glycerophosphodiesters from lysosomes","volume":"609","author":"Laqtom","year":"2022","journal-title":"Nature"},{"issue":"3","key":"10.1016\/j.molmet.2026.102456_bib31","doi-asserted-by":"crossref","first-page":"689","DOI":"10.1172\/JCI1240","article-title":"Inducible inactivation of hepatic LRP gene by cre-mediated recombination confirms role of LRP in clearance of chylomicron remnants","volume":"101","author":"Rohlmann","year":"1998","journal-title":"J Clin Investig"},{"issue":"4","key":"10.1016\/j.molmet.2026.102456_bib32","doi-asserted-by":"crossref","first-page":"408","DOI":"10.1002\/cpmo.16","article-title":"Measurement of intestinal and peripheral cholesterol fluxes by a dual-tracer balance method","volume":"6","author":"Ronda","year":"2016","journal-title":"Curr Protoc Mol Biol"},{"issue":"1","key":"10.1016\/j.molmet.2026.102456_bib33","doi-asserted-by":"crossref","first-page":"E136","DOI":"10.1152\/ajpendo.1993.264.1.E136","article-title":"Measurement of endogenous synthesis of plasma cholesterol in rats and humans using MIDA","volume":"264","author":"Neese","year":"1993","journal-title":"Am J Physiol Endocrinol Metabol"},{"issue":"29","key":"10.1016\/j.molmet.2026.102456_bib34","doi-asserted-by":"crossref","first-page":"19211","DOI":"10.1074\/jbc.M109.014860","article-title":"Activation of the liver X receptor stimulates Trans-intestinal excretion of plasma cholesterol","volume":"284","author":"Veen","year":"2009","journal-title":"J Biol Chem"},{"issue":"8","key":"10.1016\/j.molmet.2026.102456_bib35","doi-asserted-by":"crossref","first-page":"G836","DOI":"10.1152\/ajpgi.00243.2014","article-title":"Hepatic entrapment of esterified cholesterol drives continual expansion of whole body sterol pool in lysosomal acid lipase-deficient mice","volume":"307","author":"Aqul","year":"2014","journal-title":"Am J Physiol Gastrointest Liver Physiol"},{"issue":"1","key":"10.1016\/j.molmet.2026.102456_bib36","doi-asserted-by":"crossref","first-page":"3828","DOI":"10.1038\/ncomms4828","article-title":"Mutation in VPS35 associated with Parkinson's disease impairs WASH complex association and inhibits autophagy","volume":"5","author":"Zavodszky","year":"2014","journal-title":"Nat Commun"},{"issue":"10","key":"10.1016\/j.molmet.2026.102456_bib37","doi-asserted-by":"crossref","first-page":"1088","DOI":"10.1111\/tra.12309","article-title":"Retromer ensures the degradation of autophagic cargo by maintaining lysosome function in drosophila","volume":"16","author":"Maruzs","year":"2015","journal-title":"Traffic"},{"issue":"1","key":"10.1016\/j.molmet.2026.102456_bib38","doi-asserted-by":"crossref","first-page":"3086","DOI":"10.1038\/s41467-023-38719-8","article-title":"Multi-omic approach characterises the neuroprotective role of retromer in regulating lysosomal health","volume":"14","author":"Daly","year":"2023","journal-title":"Nat Commun"},{"issue":"10","key":"10.1016\/j.molmet.2026.102456_bib39","article-title":"Real-time pH imaging of macrophage lysosomes using the pH-sensitive probe ApHID","volume":"5","author":"Sol\u00e9-Dom\u00e8nech","year":"2025","journal-title":"Cell Rep Methods"},{"issue":"13","key":"10.1016\/j.molmet.2026.102456_bib40","doi-asserted-by":"crossref","first-page":"8229","DOI":"10.1074\/jbc.M708735200","article-title":"Niemann-Pick type C1 I1061T mutant encodes a functional protein that is selected for endoplasmic Reticulum-associated degradation due to protein misfolding","volume":"283","author":"Gelsthorpe","year":"2008","journal-title":"J Biol Chem"},{"issue":"1","key":"10.1016\/j.molmet.2026.102456_bib41","doi-asserted-by":"crossref","first-page":"3671","DOI":"10.1038\/s41467-018-06115-2","article-title":"Coordinate regulation of mutant NPC1 degradation by selective ER autophagy and MARCH6-dependent ERAD","volume":"9","author":"Schultz","year":"2018","journal-title":"Nat Commun"},{"issue":"1","key":"10.1016\/j.molmet.2026.102456_bib42","doi-asserted-by":"crossref","first-page":"149","DOI":"10.1007\/s00018-011-0743-1","article-title":"The copper-transporting capacity of ATP7A mutants associated with Menkes disease is ameliorated by COMMD1 as a result of improved protein expression","volume":"69","author":"Vonk","year":"2011","journal-title":"Cell Mol Life Sci : CMLS"},{"key":"10.1016\/j.molmet.2026.102456_bib43","doi-asserted-by":"crossref","first-page":"181","DOI":"10.1016\/j.brainres.2016.03.035","article-title":"Lysosome and endoplasmic reticulum quality control pathways in Niemann-Pick type C disease","author":"Schultz","year":"2016","journal-title":"Brain Research 1649(Pt B)"},{"issue":"7","key":"10.1016\/j.molmet.2026.102456_bib44","doi-asserted-by":"crossref","first-page":"2576","DOI":"10.3390\/ijms21072576","article-title":"Lysosomal exocytosis, exosome release and secretory autophagy: the Autophagic- and endo-lysosomal systems Go extracellular","volume":"21","author":"Buratta","year":"2020","journal-title":"Int J Mol Sci"},{"issue":"34","key":"10.1016\/j.molmet.2026.102456_bib45","doi-asserted-by":"crossref","first-page":"26279","DOI":"10.1074\/jbc.M110.134775","article-title":"Exosome secretion ameliorates lysosomal storage of cholesterol in niemann-pick type C disease","volume":"285","author":"Strauss","year":"2010","journal-title":"J Biol Chem"},{"issue":"6","key":"10.1016\/j.molmet.2026.102456_bib46","doi-asserted-by":"crossref","first-page":"995","DOI":"10.1194\/jlr.R046896","article-title":"Lysosomal exocytosis and lipid storage disorders","volume":"55","author":"Samie","year":"2014","journal-title":"JLR (J Lipid Res)"},{"issue":"1","key":"10.1016\/j.molmet.2026.102456_bib47","doi-asserted-by":"crossref","DOI":"10.1016\/j.bbamcr.2020.118857","article-title":"IKK\u03b2 activation promotes amphisome formation and extracellular vesicle secretion in tumor cells","volume":"1868","author":"Peng","year":"2021","journal-title":"Biochim Biophys Acta Mol Cell Res"},{"key":"10.1016\/j.molmet.2026.102456_bib48","doi-asserted-by":"crossref","first-page":"65","DOI":"10.1016\/j.arr.2016.05.001","article-title":"Impact of lysosome status on extracellular vesicle content and release","volume":"32","author":"Eitan","year":"2016","journal-title":"Ageing Res Rev"},{"issue":"11","key":"10.1016\/j.molmet.2026.102456_bib49","doi-asserted-by":"crossref","first-page":"1833","DOI":"10.1038\/s12276-022-00898-7","article-title":"Dissecting exosome inhibitors: therapeutic insights into small-molecule chemicals against cancer","volume":"54","author":"Kim","year":"2022","journal-title":"Exp Mol Med"},{"key":"10.1016\/j.molmet.2026.102456_bib50","doi-asserted-by":"crossref","DOI":"10.3389\/fcell.2021.722560","article-title":"SPAAC Pulse-Chase: a novel click chemistry-based method to determine the half-life of cellular proteins","volume":"9","author":"Morey","year":"2021","journal-title":"Front Cell Dev Biol"},{"issue":"1","key":"10.1016\/j.molmet.2026.102456_bib51","doi-asserted-by":"crossref","first-page":"168","DOI":"10.1016\/j.ajhg.2011.06.008","article-title":"A mutation in VPS35, encoding a subunit of the retromer complex, causes late-onset parkinson disease","volume":"89","author":"Zimprich","year":"2011","journal-title":"Am J Hum Genet"},{"issue":"1","key":"10.1016\/j.molmet.2026.102456_bib52","doi-asserted-by":"crossref","first-page":"162","DOI":"10.1016\/j.ajhg.2011.06.001","article-title":"VPS35 mutations in parkinson disease","volume":"89","author":"Vilari\u00f1o-G\u00fcell","year":"2011","journal-title":"Am J Hum Genet"},{"issue":"6","key":"10.1016\/j.molmet.2026.102456_bib53","doi-asserted-by":"crossref","first-page":"909","DOI":"10.1002\/ana.20667","article-title":"Model-guided microarray implicates the retromer complex in Alzheimer's disease. - PubMed - NCBI","volume":"58","author":"Small","year":"2005","journal-title":"Ann Neurol"},{"issue":"1","key":"10.1016\/j.molmet.2026.102456_bib54","doi-asserted-by":"crossref","first-page":"91","DOI":"10.1091\/mbc.e14-06-1073","article-title":"COMMD1 is linked to the WASH complex and regulates endosomal trafficking of the copper transporter ATP7A","volume":"26","author":"Phillips-Krawczak","year":"2015","journal-title":"Mol Biol Cell"},{"issue":"10","key":"10.1016\/j.molmet.2026.102456_bib55","doi-asserted-by":"crossref","first-page":"807","DOI":"10.1016\/j.tcb.2018.06.005","article-title":"Endosomal retrieval of cargo: Retromer is not alone","volume":"28","author":"McNally","year":"2018","journal-title":"Trends Cell Biol"},{"issue":"18","key":"10.1016\/j.molmet.2026.102456_bib56","doi-asserted-by":"crossref","first-page":"3783","DOI":"10.1016\/j.bpj.2023.08.005","article-title":"The LDL receptor is regulated by membrane cholesterol as revealed by fluorescence fluctuation analysis","volume":"122","author":"Morales","year":"2023","journal-title":"Biophys J"},{"key":"10.1016\/j.molmet.2026.102456_bib57","doi-asserted-by":"crossref","DOI":"10.3389\/fcell.2022.826379","article-title":"Low-density lipoprotein internalization, degradation and receptor recycling along membrane contact sites","volume":"10","author":"Islam","year":"2022","journal-title":"Front Cell Dev Biol"},{"key":"10.1016\/j.molmet.2026.102456_bib58","doi-asserted-by":"crossref","first-page":"88","DOI":"10.1016\/j.molmet.2018.07.010","article-title":"The adaptor protein PID1 regulates receptor-dependent endocytosis of postprandial triglyceride-rich lipoproteins","volume":"16","author":"Fischer","year":"2018","journal-title":"Mol Metabol"},{"issue":"6","key":"10.1016\/j.molmet.2026.102456_bib59","doi-asserted-by":"crossref","DOI":"10.1371\/journal.pone.0038330","article-title":"Impaired LDL receptor-related protein 1 translocation correlates with improved dyslipidemia and atherosclerosis in apoE-Deficient mice","volume":"7","author":"Gordts","year":"2012","journal-title":"PLoS One"},{"issue":"28","key":"10.1016\/j.molmet.2026.102456_bib60","doi-asserted-by":"crossref","first-page":"25517","DOI":"10.1074\/jbc.M302588200","article-title":"NPC1 and NPC2 regulate cellular cholesterol homeostasis through generation of low density lipoprotein cholesterol-derived oxysterols","volume":"278","author":"Frolov","year":"2003","journal-title":"J Biol Chem"},{"issue":"35","key":"10.1016\/j.molmet.2026.102456_bib61","doi-asserted-by":"crossref","first-page":"17002","DOI":"10.1016\/S0021-9258(18)45483-X","article-title":"Low density lipoprotein (LDL)-mediated suppression of cholesterol synthesis and LDL uptake is defective in Niemann-Pick type C fibroblasts","volume":"262","author":"Liscum","year":"1987","journal-title":"J Biol Chem"},{"issue":"7","key":"10.1016\/j.molmet.2026.102456_bib62","doi-asserted-by":"crossref","first-page":"2377","DOI":"10.1073\/pnas.0810895106","article-title":"Reversal of defective lysosomal transport in NPC disease ameliorates liver dysfunction and neurodegeneration in the npc1\u2212\/\u2212 mouse","volume":"106","author":"Liu","year":"2009","journal-title":"Proc Natl Acad Sci"},{"issue":"14","key":"10.1016\/j.molmet.2026.102456_bib63","doi-asserted-by":"crossref","first-page":"2017","DOI":"10.1161\/01.CIR.0000143163.76212.0B","article-title":"ACAT2 is localized to hepatocytes and is the major cholesterol-esterifying enzyme in human liver","volume":"110","author":"Parini","year":"2004","journal-title":"Circulation"},{"issue":"1","key":"10.1016\/j.molmet.2026.102456_bib64","doi-asserted-by":"crossref","first-page":"2573","DOI":"10.1038\/s41467-023-38165-6","article-title":"Restoration of lysosomal acidification rescues autophagy and metabolic dysfunction in non-alcoholic fatty liver disease","volume":"14","author":"Zeng","year":"2023","journal-title":"Nat Commun"},{"issue":"6","key":"10.1016\/j.molmet.2026.102456_bib65","doi-asserted-by":"crossref","DOI":"10.1371\/journal.pgen.1010800","article-title":"The chloride antiporter CLCN7 is a modifier of lysosome dysfunction in FIG 4 and VAC14 mutants","volume":"19","author":"Cao","year":"2023","journal-title":"PLoS Genet"},{"issue":"6","key":"10.1016\/j.molmet.2026.102456_bib66","doi-asserted-by":"crossref","first-page":"1127","DOI":"10.1016\/j.ajhg.2019.04.008","article-title":"Lysosomal Storage and Albinism Due to Effects of a De Novo CLCN7 Variant on Lysosomal Acidification","volume":"104","author":"Nicoli","year":"2019","journal-title":"Am J Hum Genet"},{"issue":"3","key":"10.1016\/j.molmet.2026.102456_bib67","doi-asserted-by":"crossref","first-page":"601","DOI":"10.1091\/mbc.12.3.601","article-title":"Dynamic movements of organelles containing Niemann-Pick C1 protein: NPC1 involvement in late endocytic events","volume":"12","author":"Ko","year":"2001","journal-title":"Mol Biol Cell"},{"issue":"3","key":"10.1016\/j.molmet.2026.102456_bib68","doi-asserted-by":"crossref","first-page":"438","DOI":"10.1074\/mcp.M116.063636","article-title":"Quantitative proteome analysis of mouse liver lysosomes provides evidence for mannose 6-phosphate-independent targeting mechanisms of acid hydrolases in mucolipidosis II","volume":"16","author":"Markmann","year":"2017","journal-title":"Mol Cell Proteomics"},{"issue":"3","key":"10.1016\/j.molmet.2026.102456_bib69","doi-asserted-by":"crossref","first-page":"202","DOI":"10.1038\/nrm1050","article-title":"Mannose 6-phosphate receptors: new twists in the tale","volume":"4","author":"Ghosh","year":"2003","journal-title":"Nat Rev Mol Cell Biol"},{"issue":"2","key":"10.1016\/j.molmet.2026.102456_bib70","doi-asserted-by":"crossref","first-page":"230","DOI":"10.1111\/tra.12136","article-title":"The Vps35 D620N mutation linked to Parkinson's disease disrupts the cargo sorting function of retromer","volume":"15","author":"Follett","year":"2014","journal-title":"Traffic"},{"issue":"12","key":"10.1016\/j.molmet.2026.102456_bib71","doi-asserted-by":"crossref","first-page":"1759","DOI":"10.1111\/j.1600-0854.2011.01284.x","article-title":"Vps26A and Vps26B subunits define distinct retromer complexes","volume":"12","author":"Bugarcic","year":"2011","journal-title":"Traffic"},{"issue":"3","key":"10.1016\/j.molmet.2026.102456_bib72","doi-asserted-by":"crossref","first-page":"513","DOI":"10.1083\/jcb.200804048","article-title":"Regulation of retromer recruitment to endosomes by sequential action of Rab5 and Rab7","volume":"183","author":"Rojas","year":"2008","journal-title":"J Cell Biol"},{"key":"10.1016\/j.molmet.2026.102456_bib73","first-page":"1","article-title":"Oxidation of retromer complex controls mitochondrial translation","author":"Zhang","year":"2025","journal-title":"Nature"},{"issue":"1","key":"10.1016\/j.molmet.2026.102456_bib74","first-page":"1","article-title":"A trimeric Rab7 GEF controls NPC1-dependent lysosomal cholesterol export","volume":"11","author":"Boomen","year":"2020","journal-title":"Nat Commun"},{"issue":"4","key":"10.1016\/j.molmet.2026.102456_bib75","doi-asserted-by":"crossref","first-page":"1009","DOI":"10.1007\/s11095-026-04058-8","article-title":"Small molecules to elevate Rab7-GTPase activity and lower cholesterol accumulation in Niemann-Pick type C disease","volume":"43","author":"Nguyen","year":"2026","journal-title":"Pharm Res"},{"issue":"14","key":"10.1016\/j.molmet.2026.102456_bib76","doi-asserted-by":"crossref","first-page":"2839","DOI":"10.1007\/s00018-019-03330-y","article-title":"Annexin A6 modulates TBC1D15\/Rab7\/StARD3 axis to control endosomal cholesterol export in NPC1 cells","volume":"77","author":"Meneses-Salas","year":"2020","journal-title":"Cell Mol Life Sci"},{"issue":"12","key":"10.1016\/j.molmet.2026.102456_bib77","doi-asserted-by":"crossref","first-page":"1541","DOI":"10.1172\/JCI0215420","article-title":"Rab proteins mediate Golgi transport of caveola-internalized glycosphingolipids and correct lipid trafficking in Niemann-Pick C cells","volume":"109","author":"Choudhury","year":"2002","journal-title":"J Clin Investig"},{"issue":"2","key":"10.1016\/j.molmet.2026.102456_bib78","doi-asserted-by":"crossref","first-page":"474","DOI":"10.1002\/hep.22363","article-title":"Dietary cholesterol, rather than liver steatosis, leads to hepatic inflammation in hyperlipidemic mouse models of nonalcoholic steatohepatitis","volume":"48","author":"Wouters","year":"2008","journal-title":"Hepatology"},{"issue":"1","key":"10.1016\/j.molmet.2026.102456_bib79","doi-asserted-by":"crossref","DOI":"10.1038\/srep24631","article-title":"Perturbed rhythmic activation of signaling pathways in mice deficient for sterol carrier protein 2-dependent diurnal lipid transport and metabolism","volume":"6","author":"Jouffe","year":"2016","journal-title":"Sci Rep"},{"issue":"8","key":"10.1016\/j.molmet.2026.102456_bib80","doi-asserted-by":"crossref","DOI":"10.1016\/j.jcmgh.2026.101788","article-title":"Vacuolar protein sorting 35 controls hepatocellular proliferation through SRC signaling and promotes diethyl nitrosamine\u2013induced tumor initiation","volume":"20","author":"Barbosa","year":"2026","journal-title":"Cell Mol Gastroenterol Hepatol"},{"issue":"10","key":"10.1016\/j.molmet.2026.102456_bib81","doi-asserted-by":"crossref","DOI":"10.1371\/journal.pone.0076971","article-title":"Retromer dependent recycling of the Wnt secretion factor Wls is dispensable for stem cell maintenance in the mammalian intestinal epithelium. - PubMed - NCBI","volume":"8","author":"Groot","year":"2013","journal-title":"PLoS One"},{"issue":"2","key":"10.1016\/j.molmet.2026.102456_bib82","doi-asserted-by":"crossref","first-page":"440","DOI":"10.1016\/j.cell.2014.09.014","article-title":"CRISPR-Cas9 Knockin mice for genome editing and cancer modeling","volume":"159","author":"Platt","year":"2014","journal-title":"Cell"},{"issue":"1","key":"10.1016\/j.molmet.2026.102456_bib83","doi-asserted-by":"crossref","first-page":"2475","DOI":"10.1038\/s41467-020-16287-5","article-title":"Large-scale electron microscopy database for human type 1 diabetes","volume":"11","author":"Boer","year":"2020","journal-title":"Nat Commun"},{"issue":"4","key":"10.1016\/j.molmet.2026.102456_bib84","doi-asserted-by":"crossref","first-page":"271","DOI":"10.1007\/s00418-020-01846-0","article-title":"Neodymium as an alternative contrast for uranium in electron microscopy","volume":"153","author":"Kuipers","year":"2020","journal-title":"Histochem Cell Biol"},{"issue":"8","key":"10.1016\/j.molmet.2026.102456_bib85","doi-asserted-by":"crossref","first-page":"911","DOI":"10.1139\/o59-099","article-title":"A rapid method of total lipid extraction and purification","volume":"37","author":"Bligh","year":"1959","journal-title":"Can J Biochem Physiol"},{"issue":"10","key":"10.1016\/j.molmet.2026.102456_bib86","doi-asserted-by":"crossref","first-page":"2656","DOI":"10.3390\/cells10102656","article-title":"Oxysterol 7-\u03b1 hydroxylase (CYP7B1) attenuates metabolic-associated fatty liver disease in mice at thermoneutrality","volume":"10","author":"Evangelakos","year":"2021","journal-title":"Cells"},{"issue":"6364","key":"10.1016\/j.molmet.2026.102456_bib87","doi-asserted-by":"crossref","first-page":"807","DOI":"10.1126\/science.aan6298","article-title":"Lysosomal metabolomics reveals V-ATPase- and mTOR-dependent regulation of amino acid efflux from lysosomes","volume":"358","author":"Abu-Remaileh","year":"2017","journal-title":"Science"},{"key":"10.1016\/j.molmet.2026.102456_bib88","first-page":"1","article-title":"Haploid genetic screens identify SPRING\/C12ORF49 as a determinant of SREBP signaling and cholesterol metabolism","author":"Loregger","year":"2020","journal-title":"Nat Commun"},{"issue":"9","key":"10.1016\/j.molmet.2026.102456_bib89","doi-asserted-by":"crossref","first-page":"1540","DOI":"10.1038\/nprot.2010.111","article-title":"Preparation and incubation of precision-cut liver and intestinal slices for application in drug metabolism and toxicity studies","volume":"5","author":"Graaf","year":"2010","journal-title":"Nat Protoc"},{"issue":"4","key":"10.1016\/j.molmet.2026.102456_bib90","doi-asserted-by":"crossref","DOI":"10.1002\/btm2.10659","article-title":"Human induced pluripotent stem cells-derived liver organoids grown on a Biomimesys\u00ae hyaluronic acid-based hydroscaffold as a new model for studying human lipoprotein metabolism","volume":"9","author":"Roudaut","year":"2024","journal-title":"Bioeng Transl Med"},{"key":"10.1016\/j.molmet.2026.102456_bib91","doi-asserted-by":"crossref","DOI":"10.1016\/j.scr.2022.102721","article-title":"Generation of a GPR146 knockout human induced pluripotent stem cell line (ITXi001-A-1)","volume":"60","author":"Bray","year":"2022","journal-title":"Stem Cell Res"},{"issue":"4","key":"10.1016\/j.molmet.2026.102456_bib92","doi-asserted-by":"crossref","DOI":"10.1016\/j.xpro.2022.101680","article-title":"FACS-assisted CRISPR-Cas9 genome editing of human induced pluripotent stem cells","volume":"3","author":"Caillaud","year":"2022","journal-title":"STAR Protoc"},{"issue":"9","key":"10.1016\/j.molmet.2026.102456_bib93","doi-asserted-by":"crossref","first-page":"2005","DOI":"10.1038\/s12276-023-01074-1","article-title":"Scalable production of tissue-like vascularized liver organoids from human PSCs","volume":"55","author":"Harrison","year":"2023","journal-title":"Exp Mol Med"},{"issue":"4","key":"10.1016\/j.molmet.2026.102456_bib94","doi-asserted-by":"crossref","first-page":"291","DOI":"10.1038\/nmeth745","article-title":"In vivo protein biotinylation for identification of organ-specific antigens accessible from the vasculature","volume":"2","author":"Rybak","year":"2005","journal-title":"Nat Methods"},{"issue":"26","key":"10.1016\/j.molmet.2026.102456_bib95","doi-asserted-by":"crossref","DOI":"10.1002\/adhm.202202670","article-title":"Targeting ligand independent tropism of siRNA-LNP by small molecules for directed therapy of liver or Myeloid immune cells","volume":"13","author":"Lin","year":"2023","journal-title":"Adv Healthcare Mater"},{"issue":"1","key":"10.1016\/j.molmet.2026.102456_bib96","doi-asserted-by":"crossref","DOI":"10.15252\/msb.20199111","article-title":"Automated sample preparation with SP3 for low-input clinical proteomics","volume":"16","author":"M\u00fcller","year":"2020","journal-title":"Mol Syst Biol"},{"issue":"1","key":"10.1016\/j.molmet.2026.102456_bib97","first-page":"265","article-title":"Accumulation of cholesterol with age in human Bruch's membrane","volume":"42","author":"Curcio","year":"2001","journal-title":"Investig Ophthalmol Vis Sci"},{"issue":"1","key":"10.1016\/j.molmet.2026.102456_bib98","doi-asserted-by":"crossref","first-page":"915","DOI":"10.1038\/s41467-023-36375-6","article-title":"Cholesterol esters form supercooled lipid droplets whose nucleation is facilitated by triacylglycerols","volume":"14","author":"Dumesnil","year":"2023","journal-title":"Nat Commun"},{"key":"10.1016\/j.molmet.2026.102456_bib99","doi-asserted-by":"crossref","first-page":"417","DOI":"10.1016\/j.neuroimage.2017.11.060","article-title":"Developing 3D microscopy with CLARITY on human brain tissue: towards a tool for informing and validating MRI-based histology","volume":"182","author":"Morawski","year":"2018","journal-title":"Neuroimage"},{"issue":"10","key":"10.1016\/j.molmet.2026.102456_bib100","doi-asserted-by":"crossref","DOI":"10.1186\/gb-2006-7-10-r100","article-title":"CellProfiler: image analysis software for identifying and quantifying cell phenotypes","volume":"7","author":"Carpenter","year":"2006","journal-title":"Genome Biol"},{"issue":"1","key":"10.1016\/j.molmet.2026.102456_bib101","doi-asserted-by":"crossref","first-page":"100","DOI":"10.1038\/s41592-020-01018-x","article-title":"Cellpose: a generalist algorithm for cellular segmentation","volume":"18","author":"Stringer","year":"2021","journal-title":"Nat Methods"}],"container-title":["Molecular Metabolism"],"original-title":[],"language":"en","link":[{"URL":"https:\/\/api.elsevier.com\/content\/article\/PII:S2212877826001407?httpAccept=text\/xml","content-type":"text\/xml","content-version":"vor","intended-application":"text-mining"},{"URL":"https:\/\/api.elsevier.com\/content\/article\/PII:S2212877826001407?httpAccept=text\/plain","content-type":"text\/plain","content-version":"vor","intended-application":"text-mining"}],"deposited":{"date-parts":[[2026,10,21]],"date-time":"2026-10-21T06:52:47Z","timestamp":1792565567000},"score":1,"resource":{"primary":{"URL":"https:\/\/linkinghub.elsevier.com\/retrieve\/pii\/S2212877826001407"}},"subtitle":[],"short-title":[],"issued":{"date-parts":[[2026,12]]},"references-count":101,"alternative-id":["S2212877826001407"],"URL":"https:\/\/doi.org\/10.1016\/j.molmet.2026.102456","relation":{},"ISSN":["2212-8778"],"issn-type":[{"value":"2212-8778","type":"print"}],"subject":[],"published":{"date-parts":[[2026,12]]},"assertion":[{"value":"Elsevier","name":"publisher","label":"This article is maintained by"},{"value":"Hepatic retromer is essential for systemic cholesterol homeostasis by regulating lysosomal cholesterol handling","name":"articletitle","label":"Article Title"},{"value":"Molecular Metabolism","name":"journaltitle","label":"Journal Title"},{"value":"https:\/\/doi.org\/10.1016\/j.molmet.2026.102456","name":"articlelink","label":"CrossRef DOI link to publisher maintained version"},{"value":"article","name":"content_type","label":"Content Type"},{"value":"\u00a9 2026 The Author(s). Published by Elsevier GmbH.","name":"copyright","label":"Copyright"}],"article-number":"102456"}}