{"status":"ok","message-type":"work","message-version":"1.0.0","message":{"indexed":{"date-parts":[[2026,10,6]],"date-time":"2026-10-06T06:57:00Z","timestamp":1791269820475,"version":"4.1.0"},"reference-count":37,"publisher":"Elsevier BV","issue":"5","license":[{"start":{"date-parts":[[2008,11,1]],"date-time":"2008-11-01T00:00:00Z","timestamp":1225497600000},"content-version":"tdm","delay-in-days":0,"URL":"https:\/\/www.elsevier.com\/tdm\/userlicense\/1.0\/"},{"start":{"date-parts":[[2008,11,1]],"date-time":"2008-11-01T00:00:00Z","timestamp":1225497600000},"content-version":"tdm","delay-in-days":0,"URL":"https:\/\/www.elsevier.com\/legal\/tdmrep-license"}],"funder":[{"DOI":"10.13039\/501100001700","name":"Ministry of Education, Culture, Sports, Science and Technology","doi-asserted-by":"publisher","award":["18390310"],"award-info":[{"award-number":["18390310"]}],"id":[{"id":"10.13039\/501100001700","id-type":"DOI","asserted-by":"publisher"}]},{"DOI":"10.13039\/501100001700","name":"Ministry of Education, Culture, Sports, Science and Technology","doi-asserted-by":"publisher","award":["20390304"],"award-info":[{"award-number":["20390304"]}],"id":[{"id":"10.13039\/501100001700","id-type":"DOI","asserted-by":"publisher"}]}],"content-domain":{"domain":["ajp.amjpathol.org","clinicalkey.com","clinicalkey.com.au","clinicalkey.es","clinicalkey.fr","clinicalkey.jp","elsevier.com","sciencedirect.com"],"crossmark-restriction":true},"short-container-title":["The American Journal of Pathology"],"published-print":{"date-parts":[[2008,11]]},"DOI":"10.2353\/ajpath.2008.080005","type":"journal-article","created":{"date-parts":[[2008,10,2]],"date-time":"2008-10-02T20:43:55Z","timestamp":1222980235000},"page":"1349-1360","update-policy":"https:\/\/doi.org\/10.1016\/elsevier_cm_policy","source":"Crossref","is-referenced-by-count":33,"title":["CGI-58 Is an \u03b1\/\u03b2-Hydrolase within Lipid Transporting Lamellar Granules of Differentiated Keratinocytes"],"prefix":"10.1016","volume":"173","author":[{"given":"Masashi","family":"Akiyama","sequence":"first","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Kaori","family":"Sakai","sequence":"additional","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Chitoshi","family":"Takayama","sequence":"additional","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Teruki","family":"Yanagi","sequence":"additional","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Yasuko","family":"Yamanaka","sequence":"additional","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"James R.","family":"McMillan","sequence":"additional","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Hiroshi","family":"Shimizu","sequence":"additional","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]}],"member":"78","reference":[{"key":"10.2353\/ajpath.2008.080005_bib1","doi-asserted-by":"crossref","first-page":"703","DOI":"10.1101\/gr.10.5.703","article-title":"Identification of novel human genes evolutionarily conserved in Caenorhabditis elegans by comparative proteomics","volume":"10","author":"Lai","year":"2000","journal-title":"Genome Res"},{"key":"10.2353\/ajpath.2008.080005_bib2","doi-asserted-by":"crossref","first-page":"309","DOI":"10.1016\/j.cmet.2006.03.005","article-title":"Adipose triglyceride lipase-mediated lipolysis of cellular fat stores is activated by CGI-58 and defective in Chanarin-Dorfman syndrome","volume":"3","author":"Lass","year":"2006","journal-title":"Cell Metab"},{"key":"10.2353\/ajpath.2008.080005_bib3","doi-asserted-by":"crossref","first-page":"1002","DOI":"10.1086\/324121","article-title":"Mutations in CGI-58, the gene encoding a new protein of the esterase\/lipase\/thioesterase subfamily, in Chanarin-Dorfman syndrome","volume":"69","author":"Lef\u00e8vre","year":"2001","journal-title":"Am J Hum Genet"},{"key":"10.2353\/ajpath.2008.080005_bib4","doi-asserted-by":"crossref","first-page":"1029","DOI":"10.1046\/j.1523-1747.2003.12520.x","article-title":"Truncation of CGI-58 protein causes malformation of lamellar granules resulting in ichthyosis in Dorfman-Chanarin syndrome","volume":"121","author":"Akiyama","year":"2003","journal-title":"J Invest Dermatol"},{"key":"10.2353\/ajpath.2008.080005_bib5","doi-asserted-by":"crossref","first-page":"214","DOI":"10.1002\/ajmg.a.30228","article-title":"CGI-58\/ABHD5 gene is mutated in Dorfman-Chanarin syndrome","volume":"129","author":"Caux","year":"2004","journal-title":"Am J Med Genet A"},{"key":"10.2353\/ajpath.2008.080005_bib6","doi-asserted-by":"crossref","first-page":"798","DOI":"10.1001\/archderm.141.6.798","article-title":"Two new mutations of the ABHD5 gene in a new adult case of Chanarin Dorfman syndrome: an uncommon lipid storage disease","volume":"141","author":"Schleinitz","year":"2005","journal-title":"Arch Dermatol"},{"key":"10.2353\/ajpath.2008.080005_bib7","doi-asserted-by":"crossref","first-page":"838","DOI":"10.1111\/j.1365-2133.2005.06828.x","article-title":"Erythrokeratoderma variabilis-like ichthyosis in Chanarin-Dorfman syndrome","volume":"153","author":"Pujol","year":"2005","journal-title":"Br J Dermatol"},{"key":"10.2353\/ajpath.2008.080005_bib8","doi-asserted-by":"crossref","first-page":"261","DOI":"10.1001\/archderm.1974.01630080059017","article-title":"Ichthyosiform dermatosis with systemic lipidosis","volume":"110","author":"Dorfman","year":"1974","journal-title":"Arch Dermatol"},{"key":"10.2353\/ajpath.2008.080005_bib9","doi-asserted-by":"crossref","first-page":"553","DOI":"10.1136\/bmj.1.5957.553","article-title":"Neutral-lipid storage disease: a new disorder of lipid metabolism","volume":"1","author":"Chanarin","year":"1975","journal-title":"Br Med J"},{"key":"10.2353\/ajpath.2008.080005_bib10","series-title":"Principles and Practice of Dermatology","first-page":"379","article-title":"Generalized disorders of cornification: The ichthyoses","author":"Williams","year":"1996"},{"key":"10.2353\/ajpath.2008.080005_bib11","doi-asserted-by":"crossref","first-page":"430","DOI":"10.1046\/j.1365-2133.2001.04051.x","article-title":"Dorfman-Chanarin syndrome (neutral lipid storage disease): new clinical features","volume":"144","author":"Pena-Penabad","year":"2001","journal-title":"Br J Dermatol"},{"key":"10.2353\/ajpath.2008.080005_bib12","doi-asserted-by":"crossref","first-page":"183","DOI":"10.1111\/j.0022-202X.2005.23668.x","article-title":"Stratum corneum defensive functions: an integrated view","volume":"125","author":"Elias","year":"2005","journal-title":"J Invest Dermatol"},{"key":"10.2353\/ajpath.2008.080005_bib13","doi-asserted-by":"crossref","first-page":"1777","DOI":"10.1172\/JCI24834","article-title":"Mutations in lipid transporter ABCA12 in harlequin ichthyosis and functional recovery by corrective gene transfer","volume":"115","author":"Akiyama","year":"2005","journal-title":"J Clin Invest"},{"key":"10.2353\/ajpath.2008.080005_bib14","doi-asserted-by":"crossref","first-page":"857","DOI":"10.1038\/sj.gt.3302472","article-title":"Beta defensin-3 engineered epidermis shows highly protective effect for bacterial infection","volume":"12","author":"Sawamura","year":"2005","journal-title":"Gene Ther"},{"key":"10.2353\/ajpath.2008.080005_bib15","doi-asserted-by":"crossref","first-page":"903","DOI":"10.1046\/j.1523-1747.1999.00592.x","article-title":"Periderm cells form cornified cell envelope in their regression process during human epidermal development","volume":"112","author":"Akiyama","year":"1999","journal-title":"J Invest Dermatol"},{"key":"10.2353\/ajpath.2008.080005_bib16","doi-asserted-by":"crossref","first-page":"131","DOI":"10.1007\/BF01799862","article-title":"Neutral lipid storage disease with ichthyosis: lipid content and metabolism of fibroblasts","volume":"11","author":"Williams","year":"1988","journal-title":"J Inherit Metab Dis"},{"key":"10.2353\/ajpath.2008.080005_bib17","doi-asserted-by":"crossref","first-page":"27027","DOI":"10.1074\/jbc.270.45.27027","article-title":"The turnover of cytoplasmic triacylglycerols in human fibroblasts involves two separate acyl chian length-dependent degradation pathways","volume":"270","author":"Hilaire","year":"1995","journal-title":"J Biol Chem"},{"key":"10.2353\/ajpath.2008.080005_bib18","doi-asserted-by":"crossref","first-page":"162","DOI":"10.1016\/0925-4439(91)90055-E","article-title":"Neutral lipid storage disease: a possible functional defect in phospholipid-linked triacylglycerol metabolism","volume":"1096","author":"Williams","year":"1991","journal-title":"Biochim Biophys Acta"},{"key":"10.2353\/ajpath.2008.080005_bib19","doi-asserted-by":"crossref","first-page":"467","DOI":"10.1042\/bj2970467","article-title":"Cellular uptake and catabolism of high-density-lipoprotein triacylglycerol in human cultured fibroblasts: degradation block in neural lipid storage disease","volume":"297","author":"Hilaire","year":"1994","journal-title":"Biochem J"},{"key":"10.2353\/ajpath.2008.080005_bib20","doi-asserted-by":"crossref","first-page":"16644","DOI":"10.1074\/jbc.271.28.16644","article-title":"Acylglycerol recycling from triacylglycerol to phospholipid, not lipase activity, is defective in neutral lipid storage disease fibroblasts","volume":"271","author":"Igal","year":"1996","journal-title":"J Biol Chem"},{"key":"10.2353\/ajpath.2008.080005_bib21","doi-asserted-by":"crossref","first-page":"31","DOI":"10.1016\/S0022-2275(20)34200-0","article-title":"Neutral lipid storage disease: a genetic disorder with abnormalities in the regulation of phospholipid metabolism","volume":"39","author":"Igal","year":"1998","journal-title":"J Lipid Res"},{"key":"10.2353\/ajpath.2008.080005_bib22","doi-asserted-by":"crossref","first-page":"197","DOI":"10.1093\/protein\/5.3.197","article-title":"The alpha\/beta hydrolase fold","volume":"5","author":"Ollis","year":"1992","journal-title":"Protein Eng"},{"key":"10.2353\/ajpath.2008.080005_bib23","first-page":"R141","article-title":"Of barn owls and bankers: a lush variety of alpha\/beta hydrolases","author":"Heikinheimo","year":"1997","journal-title":"Struct Fold Des"},{"key":"10.2353\/ajpath.2008.080005_bib24","doi-asserted-by":"crossref","first-page":"535","DOI":"10.1016\/S1359-0278(98)00069-8","article-title":"Functional analysis of the Escherichia coli genome for members of the alpha\/beta hydrolase family","volume":"3","author":"Zhang","year":"1998","journal-title":"Fold Des"},{"key":"10.2353\/ajpath.2008.080005_bib25","doi-asserted-by":"crossref","first-page":"732","DOI":"10.1016\/S0959-440X(99)00037-8","article-title":"alpha\/beta Hydrolase fold enzymes: the family keeps growing","volume":"9","author":"Nardini","year":"1999","journal-title":"Curr Opin Struct Biol"},{"key":"10.2353\/ajpath.2008.080005_bib26","doi-asserted-by":"crossref","first-page":"30490","DOI":"10.1074\/jbc.M403920200","article-title":"CGI-58 interacts with perilipin and is localized to lipid droplets. Possible involvement of CGI-58 mislocalization in Chanarin-Dorfman syndrome","volume":"279","author":"Yamaguchi","year":"2004","journal-title":"J Biol Chem"},{"key":"10.2353\/ajpath.2008.080005_bib27","doi-asserted-by":"crossref","first-page":"489","DOI":"10.1054\/tice.1999.0050","article-title":"Coordinate assembly of lipids and enzyme proteins into epidermal lamellar bodies","volume":"31","author":"Rassner","year":"1999","journal-title":"Tissue Cell"},{"key":"10.2353\/ajpath.2008.080005_bib28","doi-asserted-by":"crossref","first-page":"1000","DOI":"10.1001\/archderm.1985.01660080054014","article-title":"Neutral lipid storage disease with ichthyosis. Defective lamellar body contents and intracellular dispersion","volume":"121","author":"Elias","year":"1985","journal-title":"Arch Dermatol"},{"key":"10.2353\/ajpath.2008.080005_bib29","doi-asserted-by":"crossref","first-page":"2032","DOI":"10.1038\/sj.jid.5700332","article-title":"Barrier dysfunction and pathogenesis of neutral lipid storage disease with ichthyosis (Chanarin-Dorfman syndrome)","volume":"126","author":"Demerjian","year":"2006","journal-title":"J Invest Dermatol"},{"key":"10.2353\/ajpath.2008.080005_bib30","doi-asserted-by":"crossref","first-page":"6","DOI":"10.1111\/1523-1747.ep12873301","article-title":"Heterogeneity in harlequin ichthyosis, an inborn error of epidermal keratinization: variable morphology and structural protein expression and a defect in lamellar granules","volume":"94","author":"Dale","year":"1990","journal-title":"J Invest Dermatol"},{"key":"10.2353\/ajpath.2008.080005_bib31","doi-asserted-by":"crossref","first-page":"824","DOI":"10.1111\/1523-1747.ep12614791","article-title":"Abnormal lamellar granules in harlequin ichthyosis","volume":"99","author":"Milner","year":"1992","journal-title":"J Invest Dermatol"},{"key":"10.2353\/ajpath.2008.080005_bib32","doi-asserted-by":"crossref","first-page":"210","DOI":"10.1111\/1523-1747.ep12371764","article-title":"Characteristic morphological abnormality of harlequin ichthyosis detected in amniotic fluid cells","volume":"102","author":"Akiyama","year":"1994","journal-title":"J Invest Dermatol"},{"key":"10.2353\/ajpath.2008.080005_bib33","doi-asserted-by":"crossref","first-page":"10","DOI":"10.1111\/1523-1747.ep12277335","article-title":"Murine keratinocyte cultures grown at the air\/medium interface synthesize stratum corneum lipids and \u201crecycle\u201d linoleate during differentiation","volume":"93","author":"Madison","year":"1989","journal-title":"J Invest Dermatol"},{"key":"10.2353\/ajpath.2008.080005_bib34","doi-asserted-by":"crossref","first-page":"1839","DOI":"10.1016\/S0022-2275(20)42327-2","article-title":"Metabolism of linoleic acid in porcine epidermis","volume":"31","author":"Wertz","year":"1990","journal-title":"J Lipid Res"},{"key":"10.2353\/ajpath.2008.080005_bib35","doi-asserted-by":"crossref","first-page":"289","DOI":"10.1111\/1523-1747.ep12276826","article-title":"Lamellar body-enriched fractions from neonatal mice: preparative techniques and partial characterization","volume":"85","author":"Grayson","year":"1985","journal-title":"J Invest Dermatol"},{"key":"10.2353\/ajpath.2008.080005_bib36","doi-asserted-by":"crossref","first-page":"223","DOI":"10.1046\/j.1365-2133.2000.03288.x","article-title":"Expression of transglutaminase activity in developing human epidermis","volume":"142","author":"Akiyama","year":"2000","journal-title":"Br J Dermatol"},{"key":"10.2353\/ajpath.2008.080005_bib37","doi-asserted-by":"crossref","first-page":"43","DOI":"10.2353\/ajpath.2007.061207","article-title":"Expression of the keratinocyte lipid transporter ABCA12 in developing and reconstituted human epidermis","volume":"171","author":"Yamanaka","year":"2007","journal-title":"Am J Pathol"}],"container-title":["The American Journal of Pathology"],"original-title":[],"language":"en","link":[{"URL":"https:\/\/api.elsevier.com\/content\/article\/PII:S0002944010615210?httpAccept=text\/xml","content-type":"text\/xml","content-version":"vor","intended-application":"text-mining"},{"URL":"https:\/\/api.elsevier.com\/content\/article\/PII:S0002944010615210?httpAccept=text\/plain","content-type":"text\/plain","content-version":"vor","intended-application":"text-mining"}],"deposited":{"date-parts":[[2025,9,11]],"date-time":"2025-09-11T14:10:07Z","timestamp":1757599807000},"score":1,"resource":{"primary":{"URL":"https:\/\/linkinghub.elsevier.com\/retrieve\/pii\/S0002944010615210"}},"subtitle":[],"short-title":[],"issued":{"date-parts":[[2008,11]]},"references-count":37,"journal-issue":{"issue":"5","published-print":{"date-parts":[[2008,11]]}},"alternative-id":["S0002944010615210"],"URL":"https:\/\/doi.org\/10.2353\/ajpath.2008.080005","relation":{},"ISSN":["0002-9440"],"issn-type":[{"value":"0002-9440","type":"print"}],"subject":[],"published":{"date-parts":[[2008,11]]},"assertion":[{"value":"Elsevier","name":"publisher","label":"This article is maintained by"},{"value":"CGI-58 Is an \u03b1\/\u03b2-Hydrolase within Lipid Transporting Lamellar Granules of Differentiated Keratinocytes","name":"articletitle","label":"Article Title"},{"value":"The American Journal of Pathology","name":"journaltitle","label":"Journal Title"},{"value":"https:\/\/doi.org\/10.2353\/ajpath.2008.080005","name":"articlelink","label":"CrossRef DOI link to publisher maintained version"},{"value":"article","name":"content_type","label":"Content Type"},{"value":"Copyright \u00a9 2008 American Society for Investigative Pathology. Published by Elsevier Inc. All rights reserved.","name":"copyright","label":"Copyright"}]}}