{"status":"ok","message-type":"work","message-version":"1.0.0","message":{"indexed":{"date-parts":[[2026,9,27]],"date-time":"2026-09-27T01:16:29Z","timestamp":1790471789478,"version":"4.1.0"},"reference-count":20,"publisher":"Elsevier BV","issue":"10","license":[{"start":{"date-parts":[[2019,10,1]],"date-time":"2019-10-01T00:00:00Z","timestamp":1569888000000},"content-version":"tdm","delay-in-days":0,"URL":"https:\/\/www.elsevier.com\/tdm\/userlicense\/1.0\/"},{"start":{"date-parts":[[2019,10,1]],"date-time":"2019-10-01T00:00:00Z","timestamp":1569888000000},"content-version":"tdm","delay-in-days":0,"URL":"https:\/\/www.elsevier.com\/legal\/tdmrep-license"},{"start":{"date-parts":[[2019,7,22]],"date-time":"2019-07-22T00:00:00Z","timestamp":1563753600000},"content-version":"vor","delay-in-days":0,"URL":"http:\/\/creativecommons.org\/licenses\/by\/4.0\/"}],"funder":[{"DOI":"10.13039\/100000901","name":"Juvenile Diabetes Research Foundation International","doi-asserted-by":"publisher","id":[{"id":"10.13039\/100000901","id-type":"DOI","asserted-by":"publisher"}]},{"DOI":"10.13039\/100000002","name":"National Institutes of Health","doi-asserted-by":"publisher","id":[{"id":"10.13039\/100000002","id-type":"DOI","asserted-by":"publisher"}]}],"content-domain":{"domain":["bmn.com","cell.com","clinicalkey.com","clinicalkey.com.au","clinicalkey.es","clinicalkey.fr","clinicalkey.jp","elsevier.com","sciencedirect.com"],"crossmark-restriction":true},"short-container-title":["Trends in Pharmacological Sciences"],"published-print":{"date-parts":[[2019,10]]},"DOI":"10.1016\/j.tips.2019.07.011","type":"journal-article","created":{"date-parts":[[2019,8,13]],"date-time":"2019-08-13T00:15:20Z","timestamp":1565655320000},"page":"711-714","update-policy":"https:\/\/doi.org\/10.1016\/elsevier_cm_policy","source":"Crossref","is-referenced-by-count":61,"title":["Current Landscape of Treatments for Wolfram Syndrome"],"prefix":"10.1016","volume":"40","author":[{"given":"Damien","family":"Abreu","sequence":"first","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Fumihiko","family":"Urano","sequence":"additional","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]}],"member":"78","reference":[{"key":"10.1016\/j.tips.2019.07.011_bb0005","doi-asserted-by":"crossref","first-page":"6","DOI":"10.1007\/s11892-015-0702-6","article-title":"Wolfram syndrome: diagnosis, management, and treatment","volume":"16","author":"Urano","year":"2016","journal-title":"Curr. Diab. Rep."},{"key":"10.1016\/j.tips.2019.07.011_bb0010","doi-asserted-by":"crossref","first-page":"1458","DOI":"10.1016\/S0140-6736(95)92473-6","article-title":"Neurodegeneration and diabetes: UK nationwide study of Wolfram (DIDMOAD) syndrome","volume":"346","author":"Barrett","year":"1995","journal-title":"Lancet"},{"key":"10.1016\/j.tips.2019.07.011_bb0015","doi-asserted-by":"crossref","DOI":"10.1371\/journal.pone.0040604","article-title":"Early brain vulnerability in Wolfram syndrome","volume":"7","author":"Hershey","year":"2012","journal-title":"PLoS One"},{"key":"10.1016\/j.tips.2019.07.011_bb0020","doi-asserted-by":"crossref","first-page":"64","DOI":"10.1186\/1750-1172-8-64","article-title":"Phenotypic characteristics of early Wolfram syndrome","volume":"8","author":"Marshall","year":"2013","journal-title":"Orphanet J. Rare Dis."},{"key":"10.1016\/j.tips.2019.07.011_bb0025","doi-asserted-by":"crossref","first-page":"411","DOI":"10.1001\/archotol.129.4.411","article-title":"Mutations in the Wolfram syndrome type 1 gene (WFS1) define a clinical entity of dominant low-frequency sensorineural hearing loss","volume":"129","author":"Lesperance","year":"2003","journal-title":"Arch. Otolaryngol. Head Neck Surg."},{"key":"10.1016\/j.tips.2019.07.011_bb0030","doi-asserted-by":"crossref","first-page":"3943","DOI":"10.2337\/db13-0571","article-title":"Autosomal dominant diabetes arising from a wolfram syndrome 1 mutation","volume":"62","author":"Bonnycastle","year":"2013","journal-title":"Diabetes"},{"key":"10.1016\/j.tips.2019.07.011_bb0035","doi-asserted-by":"crossref","first-page":"1298","DOI":"10.1002\/ajmg.a.33970","article-title":"Identification of p.A684V missense mutation in the WFS1 gene as a frequent cause of autosomal dominant optic atrophy and hearing impairment","volume":"155A","author":"Rendtorff","year":"2011","journal-title":"Am. J. Med. Genet. A"},{"key":"10.1016\/j.tips.2019.07.011_bb0040","doi-asserted-by":"crossref","first-page":"1356","DOI":"10.1038\/ejhg.2013.52","article-title":"Wolfram gene (WFS1) mutation causes autosomal dominant congenital nuclear cataract in humans","volume":"21","author":"Berry","year":"2013","journal-title":"Eur. J. Hum. Genet."},{"key":"10.1016\/j.tips.2019.07.011_bb0045","doi-asserted-by":"crossref","first-page":"2044","DOI":"10.2337\/db16-1296","article-title":"Dominant ER stress-inducing WFS1 mutations underlie a genetic syndrome of neonatal\/infancy-onset diabetes, congenital sensorineural deafness, and congenital cataracts","volume":"66","author":"De Franco","year":"2017","journal-title":"Diabetes"},{"key":"10.1016\/j.tips.2019.07.011_bb0050","doi-asserted-by":"crossref","first-page":"744","DOI":"10.1172\/JCI39678","article-title":"Wolfram syndrome 1 gene negatively regulates ER stress signaling in rodent and human cells","volume":"120","author":"Fonseca","year":"2010","journal-title":"J.\u00a0Clin. Invest."},{"key":"10.1016\/j.tips.2019.07.011_bb0055","doi-asserted-by":"crossref","DOI":"10.1371\/journal.pbio.1002511","article-title":"Role of\u00a0mitochondrial dynamics in neuronal\u00a0development: mechanism for Wolfram syndrome","volume":"14","author":"Cagalinec","year":"2016","journal-title":"PLoS Biol."},{"key":"10.1016\/j.tips.2019.07.011_bb0060","doi-asserted-by":"crossref","first-page":"923","DOI":"10.2337\/db13-0717","article-title":"\u03b2-Cell dysfunction due to increased ER stress in a stem cell model of Wolfram syndrome","volume":"63","author":"Shang","year":"2014","journal-title":"Diabetes"},{"key":"10.1016\/j.tips.2019.07.011_bb0065","doi-asserted-by":"crossref","first-page":"E5292","DOI":"10.1073\/pnas.1421055111","article-title":"A calcium-dependent protease as a potential therapeutic target for Wolfram syndrome","volume":"111","author":"Lu","year":"2014","journal-title":"Proc. Natl. Acad. Sci. U. S. A."},{"key":"10.1016\/j.tips.2019.07.011_bb0070","doi-asserted-by":"crossref","first-page":"203","DOI":"10.1093\/hmg\/dds400","article-title":"Vacuolar-type H+-ATPase V1A subunit is a molecular partner of Wolfram syndrome 1 (WFS1) protein, which regulates its expression and stability","volume":"22","author":"Gharanei","year":"2013","journal-title":"Hum. Mol. Genet."},{"key":"10.1016\/j.tips.2019.07.011_bb0075","doi-asserted-by":"crossref","DOI":"10.1371\/journal.pone.0004134","article-title":"Valproate, a mood stabilizer, induces WFS1 expression and modulates its interaction with ER stress protein GRP94","volume":"4","author":"Kakiuchi","year":"2009","journal-title":"PLoS One"},{"key":"10.1016\/j.tips.2019.07.011_bb0080","doi-asserted-by":"crossref","DOI":"10.1038\/s41598-018-28314-z","article-title":"Preventive treatment with liraglutide protects against development of glucose intolerance in a rat model of Wolfram syndrome","volume":"8","author":"Toots","year":"2018","journal-title":"Sci. Rep."},{"key":"10.1016\/j.tips.2019.07.011_bb0085","doi-asserted-by":"crossref","first-page":"2189","DOI":"10.1007\/s00125-018-4679-y","article-title":"Activation of GLP-1 receptor signalling alleviates cellular stresses and improves beta cell function in a mouse model of Wolfram syndrome","volume":"61","author":"Kondo","year":"2018","journal-title":"Diabetologia"},{"key":"10.1016\/j.tips.2019.07.011_bb0090","doi-asserted-by":"crossref","DOI":"10.1126\/scisignal.aaq1380","article-title":"ER-mitochondria cross-talk is regulated by the Ca(2+) sensor NCS1 and is impaired in Wolfram syndrome","author":"Angebault","year":"2018","journal-title":"Sci. Signal."},{"key":"10.1016\/j.tips.2019.07.011_bb0095","doi-asserted-by":"crossref","first-page":"844","DOI":"10.2337\/db13-1809","article-title":"Wolfram syndrome iPS cells: the first human cell model of endoplasmic reticulum disease","volume":"63","author":"Urano","year":"2014","journal-title":"Diabetes"},{"key":"10.1016\/j.tips.2019.07.011_bb0100","doi-asserted-by":"crossref","first-page":"366","DOI":"10.1016\/j.celrep.2014.03.023","article-title":"MANF is indispensable for the proliferation and survival of pancreatic beta cells","volume":"7","author":"Lindahl","year":"2014","journal-title":"Cell Rep."}],"container-title":["Trends in Pharmacological Sciences"],"original-title":[],"language":"en","link":[{"URL":"https:\/\/api.elsevier.com\/content\/article\/PII:S0165614719301634?httpAccept=text\/xml","content-type":"text\/xml","content-version":"vor","intended-application":"text-mining"},{"URL":"https:\/\/api.elsevier.com\/content\/article\/PII:S0165614719301634?httpAccept=text\/plain","content-type":"text\/plain","content-version":"vor","intended-application":"text-mining"}],"deposited":{"date-parts":[[2025,9,28]],"date-time":"2025-09-28T09:07:43Z","timestamp":1759050463000},"score":1,"resource":{"primary":{"URL":"https:\/\/linkinghub.elsevier.com\/retrieve\/pii\/S0165614719301634"}},"subtitle":[],"short-title":[],"issued":{"date-parts":[[2019,10]]},"references-count":20,"journal-issue":{"issue":"10","published-print":{"date-parts":[[2019,10]]}},"alternative-id":["S0165614719301634"],"URL":"https:\/\/doi.org\/10.1016\/j.tips.2019.07.011","relation":{},"ISSN":["0165-6147"],"issn-type":[{"value":"0165-6147","type":"print"}],"subject":[],"published":{"date-parts":[[2019,10]]},"assertion":[{"value":"Elsevier","name":"publisher","label":"This article is maintained by"},{"value":"Current Landscape of Treatments for Wolfram Syndrome","name":"articletitle","label":"Article Title"},{"value":"Trends in Pharmacological Sciences","name":"journaltitle","label":"Journal Title"},{"value":"https:\/\/doi.org\/10.1016\/j.tips.2019.07.011","name":"articlelink","label":"CrossRef DOI link to publisher maintained version"},{"value":"article","name":"content_type","label":"Content Type"},{"value":"\u00a9 2019 The Author(s). Published by Elsevier Ltd.","name":"copyright","label":"Copyright"}]}}