{"status":"ok","message-type":"work","message-version":"1.0.0","message":{"indexed":{"date-parts":[[2026,9,29]],"date-time":"2026-09-29T17:53:52Z","timestamp":1790704432617,"version":"4.1.0"},"reference-count":45,"publisher":"Elsevier BV","issue":"3","license":[{"start":{"date-parts":[[2021,9,1]],"date-time":"2021-09-01T00:00:00Z","timestamp":1630454400000},"content-version":"tdm","delay-in-days":0,"URL":"https:\/\/www.elsevier.com\/tdm\/userlicense\/1.0\/"},{"start":{"date-parts":[[2021,9,1]],"date-time":"2021-09-01T00:00:00Z","timestamp":1630454400000},"content-version":"tdm","delay-in-days":0,"URL":"https:\/\/www.elsevier.com\/legal\/tdmrep-license"},{"start":{"date-parts":[[2021,8,6]],"date-time":"2021-08-06T00:00:00Z","timestamp":1628208000000},"content-version":"vor","delay-in-days":0,"URL":"http:\/\/creativecommons.org\/licenses\/by\/4.0\/"}],"content-domain":{"domain":["jbc.org","elsevier.com","sciencedirect.com"],"crossmark-restriction":true},"short-container-title":["Journal of Biological Chemistry"],"published-print":{"date-parts":[[2021,9]]},"DOI":"10.1016\/j.jbc.2021.101064","type":"journal-article","created":{"date-parts":[[2021,8,8]],"date-time":"2021-08-08T05:58:10Z","timestamp":1628402290000},"page":"101064","update-policy":"https:\/\/doi.org\/10.1016\/elsevier_cm_policy","source":"Crossref","is-referenced-by-count":4,"title":["Deletion of fatty acid amide hydrolase reduces lyso-sulfatide levels but exacerbates metachromatic leukodystrophy in mice"],"prefix":"10.1016","volume":"297","author":[{"given":"Claudia","family":"Yaghootfam","sequence":"first","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Bernd","family":"Gehrig","sequence":"additional","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]},{"ORCID":"https:\/\/orcid.org\/0000-0002-4275-0200","authenticated-orcid":false,"given":"Marc","family":"Sylvester","sequence":"additional","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Volkmar","family":"Gieselmann","sequence":"additional","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]},{"ORCID":"https:\/\/orcid.org\/0000-0003-1231-3875","authenticated-orcid":false,"given":"Ulrich","family":"Matzner","sequence":"additional","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]}],"member":"78","reference":[{"key":"10.1016\/j.jbc.2021.101064_bib1","doi-asserted-by":"crossref","first-page":"799","DOI":"10.1016\/j.bbalip.2013.10.015","article-title":"Sphingolipids and lysosomal pathologies","volume":"1841","author":"Schulze","year":"2014","journal-title":"Biochim. Biophys. Acta"},{"key":"10.1016\/j.jbc.2021.101064_bib2","doi-asserted-by":"crossref","first-page":"461","DOI":"10.1146\/annurev-biochem-013118-111518","article-title":"Lysosomal glycosphingolipid storage diseases","volume":"88","author":"Breiden","year":"2019","journal-title":"Annu. Rev. Biochem."},{"key":"10.1016\/j.jbc.2021.101064_bib3","series-title":"The Metabolic and Molecular Bases of Inherited Disease","first-page":"3695","article-title":"Metachromatic leukodystrophy","author":"von Figura","year":"2001"},{"key":"10.1016\/j.jbc.2021.101064_bib4","doi-asserted-by":"crossref","first-page":"251","DOI":"10.1023\/A:1022436928925","article-title":"Twenty five years of the \u201cpsychosine hypothesis\u201d: A personal perspective of its history and present status","volume":"23","author":"Suzuki","year":"1998","journal-title":"Neurochem. Res."},{"key":"10.1016\/j.jbc.2021.101064_bib5","doi-asserted-by":"crossref","first-page":"670","DOI":"10.1126\/science.3101176","article-title":"Lysosphingolipids inhibit protein kinase C: Implications for the sphingolipidoses","volume":"235","author":"Hannun","year":"1987","journal-title":"Science"},{"key":"10.1016\/j.jbc.2021.101064_bib6","doi-asserted-by":"crossref","first-page":"605","DOI":"10.1016\/0006-291X(89)90037-5","article-title":"Accumulation of lysosulfatide (sulfogalactosylsphingosine) in tissues of a boy with metachromatic leukodystrophy","volume":"159","author":"Toda","year":"1989","journal-title":"Biochem. Biophys. Res. Commun."},{"key":"10.1016\/j.jbc.2021.101064_bib7","doi-asserted-by":"crossref","first-page":"508","DOI":"10.1007\/s11064-008-9813-7","article-title":"Lysosulfatide regulates the motility of a neural precursor cell line via calcium-mediated process collapse","volume":"34","author":"Hans","year":"2009","journal-title":"Neurochem. Res."},{"key":"10.1016\/j.jbc.2021.101064_bib8","doi-asserted-by":"crossref","first-page":"565","DOI":"10.1042\/EBC20190090","article-title":"Lyso-glycosphingolipids: Presence and consequences","volume":"64","author":"van Eijk","year":"2020","journal-title":"Essays Biochem."},{"key":"10.1016\/j.jbc.2021.101064_bib9","doi-asserted-by":"crossref","first-page":"831","DOI":"10.1016\/0020-711X(82)90105-7","article-title":"Cerebroside sulfotransferase in rat gastric mucosa","volume":"14","author":"Lin","year":"1982","journal-title":"Int. J. Biochem."},{"key":"10.1016\/j.jbc.2021.101064_bib10","doi-asserted-by":"crossref","first-page":"479","DOI":"10.1042\/bj2970479","article-title":"Functional compartments of sulphatide metabolism in cultured living cells: Evidence for the involvement of a novel sulphatide-degrading pathway","volume":"297","author":"Tempesta","year":"1994","journal-title":"Biochem. J."},{"key":"10.1016\/j.jbc.2021.101064_bib11","doi-asserted-by":"crossref","first-page":"10187","DOI":"10.1074\/jbc.270.17.10187","article-title":"A\u00a0neutral galactocerebroside sulfate sulfatidase from mouse brain","volume":"270","author":"Sundaram","year":"1995","journal-title":"J.\u00a0Biol. Chem."},{"key":"10.1016\/j.jbc.2021.101064_bib12","doi-asserted-by":"crossref","first-page":"135","DOI":"10.1007\/978-981-10-4567-7_10","article-title":"Discovery and roles of ER-endolysosomal contact sites in disease","volume":"997","author":"Henne","year":"2017","journal-title":"Adv. Exp. Med. Biol."},{"key":"10.1016\/j.jbc.2021.101064_bib13","doi-asserted-by":"crossref","first-page":"69","DOI":"10.1002\/nrc.1028","article-title":"Sulfatide does not induce apoptosis in arylsulfatase A-deficient cells","volume":"29","author":"Habetha","year":"2001","journal-title":"Neurosci. Res. Commun."},{"key":"10.1016\/j.jbc.2021.101064_bib14","doi-asserted-by":"crossref","first-page":"323","DOI":"10.1007\/978-1-4939-8552-4_16","article-title":"Synthesis of fluorescent gangliosides","volume":"1804","author":"Schwarzmann","year":"2018","journal-title":"Methods Mol. Biol."},{"key":"10.1016\/j.jbc.2021.101064_bib15","doi-asserted-by":"crossref","first-page":"259","DOI":"10.1042\/bj20011880","article-title":"Transport of phosphatidylserine via MDR1 (multidrug resistance 1) P-glycoprotein in a human gastric carcinoma cell line","volume":"365","author":"Pohl","year":"2002","journal-title":"Biochem. J."},{"key":"10.1016\/j.jbc.2021.101064_bib16","doi-asserted-by":"crossref","first-page":"18","DOI":"10.1056\/NEJM199101033240104","article-title":"Molecular basis of different forms of metachromatic leukodystrophy","volume":"324","author":"Polten","year":"1991","journal-title":"N.\u00a0Engl. J. Med."},{"key":"10.1016\/j.jbc.2021.101064_bib17","doi-asserted-by":"crossref","first-page":"625","DOI":"10.1016\/j.bbamcr.2008.09.018","article-title":"Proteomics of the lysosome","volume":"1793","author":"L\u00fcbke","year":"2009","journal-title":"Biochim. Biophys. Acta"},{"key":"10.1016\/j.jbc.2021.101064_bib18","doi-asserted-by":"crossref","first-page":"17300","DOI":"10.1074\/jbc.M110688200","article-title":"Molecular cloning and characterization of sphingolipid ceramide N-deacylase from a marine bacterium, Shewanella alga G8","volume":"277","author":"Furusato","year":"2002","journal-title":"J.\u00a0Biol. Chem."},{"key":"10.1016\/j.jbc.2021.101064_bib19","doi-asserted-by":"crossref","first-page":"9482","DOI":"10.1523\/JNEUROSCI.2287-07.2007","article-title":"Increasing sulfatide synthesis in myelin-forming cells of arylsulfatase A-deficient mice causes demyelination and neurological symptoms reminiscent of human metachromatic leukodystrophy","volume":"27","author":"Ramakrishnan","year":"2007","journal-title":"J.\u00a0Neurosci."},{"key":"10.1016\/j.jbc.2021.101064_bib20","doi-asserted-by":"crossref","first-page":"2599","DOI":"10.1093\/hmg\/dds086","article-title":"Efficacy of enzyme replacement therapy in an aggravated mouse model of metachromatic leukodystrophy declines with age","volume":"21","author":"Matthes","year":"2012","journal-title":"Hum. Mol. Genet."},{"key":"10.1016\/j.jbc.2021.101064_bib21","doi-asserted-by":"crossref","first-page":"516","DOI":"10.1016\/j.biopsych.2015.07.028","article-title":"An introduction to the endogenous cannabinoid system","volume":"79","author":"Lu","year":"2016","journal-title":"Biol. Psychiatry"},{"key":"10.1016\/j.jbc.2021.101064_bib22","doi-asserted-by":"crossref","first-page":"1160","DOI":"10.1038\/mt.2015.69","article-title":"Anti-inflammatory therapy with simvastatin improves neuroinflammation and CNS function in a mouse model of metachromatic leukodystrophy","volume":"23","author":"Stein","year":"2015","journal-title":"Mol. Ther."},{"key":"10.1016\/j.jbc.2021.101064_bib23","doi-asserted-by":"crossref","first-page":"896","DOI":"10.1021\/jm980461j","article-title":"Substrate specificity and stereoselectivity of rat brain microsomal anandamide amidohydrolase","volume":"42","author":"Lang","year":"1999","journal-title":"J.\u00a0Med. Chem."},{"key":"10.1016\/j.jbc.2021.101064_bib24","doi-asserted-by":"crossref","first-page":"32332","DOI":"10.1074\/jbc.273.48.32332","article-title":"Anandamide hydrolysis by human cells in culture and brain","volume":"273","author":"Maccarrone","year":"1998","journal-title":"J.\u00a0Biol. Chem."},{"key":"10.1016\/j.jbc.2021.101064_bib25","doi-asserted-by":"crossref","first-page":"258","DOI":"10.1046\/j.1432-1327.1999.00631.x","article-title":"Biosynthesis and inactivation of the endocannabinoid 2-arachidonoylglycerol in circulating and tumoral macrophages","volume":"264","author":"Di Marzo","year":"1999","journal-title":"Eur. J. Biochem."},{"key":"10.1016\/j.jbc.2021.101064_bib26","doi-asserted-by":"crossref","first-page":"491","DOI":"10.3390\/cells8050491","article-title":"Anti-Inflammatory effects by pharmacological inhibition or knockdown of fatty acid amide hydrolase in BV2 microglial cells","volume":"8","author":"Tanaka","year":"2019","journal-title":"Cells"},{"key":"10.1016\/j.jbc.2021.101064_bib27","doi-asserted-by":"crossref","first-page":"244","DOI":"10.1016\/j.neuropharm.2008.07.037","article-title":"Endocannabinoid signaling in microglial cells","volume":"56","author":"Stella","year":"2009","journal-title":"Neuropharmacology"},{"key":"10.1016\/j.jbc.2021.101064_bib28","doi-asserted-by":"crossref","first-page":"9260","DOI":"10.1021\/bi700014q","article-title":"Cerebroside sulfotransferase forms homodimers in living cells","volume":"46","author":"Yaghootfam","year":"2007","journal-title":"Biochemistry"},{"key":"10.1016\/j.jbc.2021.101064_bib29","doi-asserted-by":"crossref","first-page":"707","DOI":"10.1007\/s10545-006-0411-z","article-title":"Glucosylceramide transfer from lysosomes--the missing link in molecular pathology of glucosylceramidase deficiency: A hypothesis based on existing data","volume":"29","author":"Elleder","year":"2006","journal-title":"J.\u00a0Inherit. Metab. Dis."},{"key":"10.1016\/j.jbc.2021.101064_bib30","doi-asserted-by":"crossref","first-page":"411","DOI":"10.1146\/annurev.biochem.74.082803.133450","article-title":"Structure and function of fatty acid amide hydrolase","volume":"74","author":"McKinney","year":"2005","journal-title":"Annu. Rev. Biochem."},{"key":"10.1016\/j.jbc.2021.101064_bib31","doi-asserted-by":"crossref","first-page":"1263","DOI":"10.1038\/s41401-020-0385-7","article-title":"Potential application of endocannabinoid system agents in neuropsychiatric and neurodegenerative diseases-focusing on FAAH\/MAGL inhibitors","volume":"41","author":"Ren","year":"2020","journal-title":"Acta Pharmacol. Sin."},{"key":"10.1016\/j.jbc.2021.101064_bib32","doi-asserted-by":"crossref","DOI":"10.15252\/emmm.201911776","article-title":"Inhibition of fatty acid amide hydrolase prevents pathology in neurovisceral acid sphingomyelinase deficiency by rescuing defective endocannabinoid signaling","volume":"12","author":"Bartoll","year":"2020","journal-title":"EMBO Mol. Med."},{"key":"10.1016\/j.jbc.2021.101064_bib33","doi-asserted-by":"crossref","first-page":"107","DOI":"10.1016\/0165-5728(94)90152-X","article-title":"Anandamide, an endogenous cannabinoid receptor agonist inhibits lymphocyte proliferation and induces apoptosis","volume":"55","author":"Schwarz","year":"1994","journal-title":"J.\u00a0Neuroimmunol."},{"key":"10.1016\/j.jbc.2021.101064_bib34","doi-asserted-by":"crossref","first-page":"31938","DOI":"10.1074\/jbc.M005722200","article-title":"Anandamide induces apoptosis in human cells via vanilloid receptors. Evidence for a protective role of cannabinoid receptors","volume":"275","author":"Maccarrone","year":"2000","journal-title":"J.\u00a0Biol. Chem."},{"key":"10.1016\/j.jbc.2021.101064_bib35","doi-asserted-by":"crossref","first-page":"3807","DOI":"10.1093\/hmg\/ddaa277","article-title":"Brain cell type-specific endocytosis of arylsulfatase A identifies limitations of enzyme-based therapies formetachromatic leukodystrophy","volume":"29","author":"Kaminski","year":"2021","journal-title":"Hum. Mol. Genet."},{"key":"10.1016\/j.jbc.2021.101064_bib36","doi-asserted-by":"crossref","DOI":"10.1002\/0471142735.im1401s83","article-title":"The isolation and characterization of murine macrophages","author":"Zhang","year":"2008","journal-title":"Curr. Protoc. Immunol."},{"key":"10.1016\/j.jbc.2021.101064_bib37","doi-asserted-by":"crossref","first-page":"80","DOI":"10.1016\/0005-2760(92)90129-J","article-title":"Uptake and metabolism of a fluorescent sulfatide analogue in cultured skin fibroblasts","volume":"1124","author":"Monti","year":"1992","journal-title":"Biochim. Biophys. Acta"},{"key":"10.1016\/j.jbc.2021.101064_bib38","doi-asserted-by":"crossref","first-page":"69","DOI":"10.1016\/S0022-2275(20)30188-7","article-title":"A\u00a0synthetic low density lipoprotein particle capable of supporting U937 proliferation in\u00a0vitro","volume":"43","author":"Baillie","year":"2002","journal-title":"J.\u00a0Lipid Res."},{"key":"10.1016\/j.jbc.2021.101064_bib39","doi-asserted-by":"crossref","first-page":"936","DOI":"10.1194\/jlr.M057232","article-title":"Quantification of sulfatides and lysosulfatides in tissues and body fluids by liquid chromatography-tandem mass spectrometry","volume":"56","author":"Mirzaian","year":"2015","journal-title":"J.\u00a0Lipid Res."},{"key":"10.1016\/j.jbc.2021.101064_bib40","doi-asserted-by":"crossref","first-page":"1139","DOI":"10.1093\/hmg\/ddi126","article-title":"Enzyme replacement improves nervous system pathology and function in a mouse model for metachromatic leukodystrophy","volume":"14","author":"Matzner","year":"2005","journal-title":"Hum. Mol. Genet."},{"key":"10.1016\/j.jbc.2021.101064_bib41","doi-asserted-by":"crossref","first-page":"1","DOI":"10.1365\/s10337-008-0841-x","article-title":"Simultaneous quantification of anandamide and other endocannabinoids in dorsal vagal complex of rat brainstem by LC-MS","volume":"69","author":"Chen","year":"2009","journal-title":"Chromatographia"},{"key":"10.1016\/j.jbc.2021.101064_bib42","doi-asserted-by":"crossref","first-page":"229","DOI":"10.1002\/mas.21540","article-title":"The Skyline ecosystem: Informatics for quantitative mass spectrometry proteomics","volume":"39","author":"Pino","year":"2020","journal-title":"Mass Spectrom. Rev."},{"key":"10.1016\/j.jbc.2021.101064_bib43","doi-asserted-by":"crossref","first-page":"143","DOI":"10.1016\/j.ab.2005.04.032","article-title":"A\u00a0fluorescence-based assay for fatty acid amide hydrolase compatible with high-throughput screening","volume":"343","author":"Ramarao","year":"2005","journal-title":"Anal. Biochem."},{"key":"10.1016\/j.jbc.2021.101064_bib44","doi-asserted-by":"crossref","first-page":"714","DOI":"10.1186\/1756-0500-7-714","article-title":"Harvesting the maximum length of sciatic nerve from adult mice: A step-by-step approach","volume":"7","author":"Bala","year":"2014","journal-title":"BMC Res. Notes"},{"key":"10.1016\/j.jbc.2021.101064_bib45","doi-asserted-by":"crossref","first-page":"31","DOI":"10.1515\/hsz-2019-0270","article-title":"Emerging mechanisms of drug-induced phospholipidosis","volume":"401","author":"Breiden","year":"2019","journal-title":"Biol. Chem."}],"container-title":["Journal of Biological Chemistry"],"original-title":[],"language":"en","link":[{"URL":"https:\/\/api.elsevier.com\/content\/article\/PII:S002192582100867X?httpAccept=text\/xml","content-type":"text\/xml","content-version":"vor","intended-application":"text-mining"},{"URL":"https:\/\/api.elsevier.com\/content\/article\/PII:S002192582100867X?httpAccept=text\/plain","content-type":"text\/plain","content-version":"vor","intended-application":"text-mining"}],"deposited":{"date-parts":[[2025,9,19]],"date-time":"2025-09-19T00:12:02Z","timestamp":1758240722000},"score":1,"resource":{"primary":{"URL":"https:\/\/linkinghub.elsevier.com\/retrieve\/pii\/S002192582100867X"}},"subtitle":[],"short-title":[],"issued":{"date-parts":[[2021,9]]},"references-count":45,"journal-issue":{"issue":"3","published-print":{"date-parts":[[2021,9]]}},"alternative-id":["S002192582100867X"],"URL":"https:\/\/doi.org\/10.1016\/j.jbc.2021.101064","relation":{},"ISSN":["0021-9258"],"issn-type":[{"value":"0021-9258","type":"print"}],"subject":[],"published":{"date-parts":[[2021,9]]},"assertion":[{"value":"Elsevier","name":"publisher","label":"This article is maintained by"},{"value":"Deletion of fatty acid amide hydrolase reduces lyso-sulfatide levels but exacerbates metachromatic leukodystrophy in mice","name":"articletitle","label":"Article Title"},{"value":"Journal of Biological Chemistry","name":"journaltitle","label":"Journal Title"},{"value":"https:\/\/doi.org\/10.1016\/j.jbc.2021.101064","name":"articlelink","label":"CrossRef DOI link to publisher maintained version"},{"value":"article","name":"content_type","label":"Content Type"},{"value":"\u00a9 2021 The Authors. Published by Elsevier Inc on behalf of American Society for Biochemistry and Molecular Biology.","name":"copyright","label":"Copyright"}],"article-number":"101064"}}