{"status":"ok","message-type":"work","message-version":"1.0.0","message":{"indexed":{"date-parts":[[2026,3,31]],"date-time":"2026-03-31T08:00:52Z","timestamp":1774944052576,"version":"3.50.1"},"reference-count":14,"publisher":"Wiley","issue":"11","license":[{"start":{"date-parts":[[2011,11,1]],"date-time":"2011-11-01T00:00:00Z","timestamp":1320105600000},"content-version":"vor","delay-in-days":0,"URL":"http:\/\/onlinelibrary.wiley.com\/termsAndConditions#vor"},{"start":{"date-parts":[[2011,11,1]],"date-time":"2011-11-01T00:00:00Z","timestamp":1320105600000},"content-version":"tdm","delay-in-days":0,"URL":"http:\/\/doi.wiley.com\/10.1002\/tdm_license_1.1"}],"content-domain":{"domain":[],"crossmark-restriction":false},"short-container-title":["OVS"],"published-print":{"date-parts":[[2011,11]]},"abstract":"<jats:title>ABSTRACT<\/jats:title>\n                  <jats:sec>\n                    <jats:label\/>\n                    <jats:p>Wolfram Syndrome (WS) is a rare, autosomal recessive disorder that causes non\u2010autoimmune type 1 diabetes. The etiology involves a single gene mutation of the wolframin protein inducing endoplasmic reticulum stress and apoptosis in selected cell types with resultant diabetes insipidus, diabetes mellitus, optic atrophy, and sensory\u2010neural deafness. Symptoms are initially absent and signs within the posterior segment of the eye are usually the earliest indicator of WS.<\/jats:p>\n                    <jats:p>These cases characterize unusual and poorly described findings of pigmentary maculopathy in WS and illustrate the importance of collaboration between diabetes and eye care providers; especially in cases of non\u2010autoimmune type 1 diabetes exhibiting atypical human leukocyte\u2010associated antigen haplotypes.<\/jats:p>\n                  <\/jats:sec>","DOI":"10.1097\/opx.0b013e31822f4d8f","type":"journal-article","created":{"date-parts":[[2011,10,4]],"date-time":"2011-10-04T14:22:38Z","timestamp":1317738158000},"source":"Crossref","is-referenced-by-count":10,"title":["Wolfram Syndrome: A Rare Optic Neuropathy in Youth with Type 1 Diabetes"],"prefix":"10.1002","volume":"88","author":[{"given":"Brian C.","family":"Bucca","sequence":"first","affiliation":[{"name":"The Barbara Davis Center for Childhood Diabetes University of Colorado Denver Aurora Colorado"},{"name":"Departments of Pediatrics and Ophthalmology University of Colorado Denver Aurora Colorado"}],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"Georgeanna","family":"Klingensmith","sequence":"additional","affiliation":[{"name":"The Barbara Davis Center for Childhood Diabetes University of Colorado Denver Aurora Colorado"},{"name":"Department of Pediatrics University of Colorado Denver Aurora Colorado"}],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"Jeffrey L.","family":"Bennett","sequence":"additional","affiliation":[{"name":"Departments of Neurology and Ophthalmology University of Colorado Denver Aurora Colorado"}],"role":[{"role":"author","vocabulary":"crossref"}]}],"member":"311","published-online":{"date-parts":[[2011,11,1]]},"reference":[{"key":"e_1_2_2_2_2","doi-asserted-by":"crossref","first-page":"715","DOI":"10.1016\/S0025-6196(26)04711-7","article-title":"Diabetes Mellitus and Simple Optic Atrophy Among Siblings: Report of Four Cases. 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Staff Meet","volume":"13","author":"Wolfram D","year":"1938","journal-title":"Mayo Clin"},{"key":"e_1_2_2_3_2","doi-asserted-by":"crossref","first-page":"28","DOI":"10.1111\/j.1399-5448.2009.00518.x","article-title":"Wolfram syndrome: important implications for pediatricians and pediatric endocrinologists","volume":"11","author":"Kumar S","year":"2010","journal-title":"Pediatr Diabetes"},{"key":"e_1_2_2_4_2","doi-asserted-by":"crossref","first-page":"517","DOI":"10.1016\/j.ajo.2005.03.017","article-title":"Hereditary optic neuropathies: from the mitochondria to the optic nerve","volume":"140","author":"Newman NJ","year":"2005","journal-title":"Am J Ophthalmol"},{"key":"e_1_2_2_5_2","doi-asserted-by":"crossref","first-page":"729","DOI":"10.1007\/s002340050832","article-title":"MRI of Wolfram syndrome (DIDMOAD)","volume":"41","author":"Galluzzi P","year":"1999","journal-title":"Neuroradiology"},{"key":"e_1_2_2_6_2","doi-asserted-by":"crossref","first-page":"38","DOI":"10.1016\/S0008-4182(06)80064-5","article-title":"Pigmentary maculopathy in a patient with Wolfram syndrome","volume":"41","author":"Dhalla MS","year":"2006","journal-title":"Can J Ophthalmol"},{"key":"e_1_2_2_7_2","doi-asserted-by":"crossref","first-page":"77","DOI":"10.3109\/13816819409098867","article-title":"Wolfram syndrome. A report of four cases and review of the literature","volume":"15","author":"Bitoun P","year":"1994","journal-title":"Ophthalmic Genet"},{"key":"e_1_2_2_8_2","doi-asserted-by":"crossref","first-page":"1","DOI":"10.1111\/j.1651-2227.1977.tb15069.x","article-title":"Juvenile diabetes mellitus, optic atrophy, hearing loss, diabetes insipidus, atonia of the urinary tract and bladder, and other abnormalities (Wolfram syndrome). A review of 88 cases from the literature with personal observations on 3 new patients","volume":"264","author":"Cremers CW","year":"1977","journal-title":"Acta Paediatr Scand Suppl"},{"key":"e_1_2_2_9_2","doi-asserted-by":"crossref","first-page":"565","DOI":"10.1016\/S0022-3476(76)80387-3","article-title":"Juvenile diabetes mellitus, optic atrophy, sensory nerve deafness, and diabetes insipidus\u2014a syndrome","volume":"89","author":"Gunn T","year":"1976","journal-title":"J Pediatr"},{"key":"e_1_2_2_10_2","doi-asserted-by":"crossref","first-page":"9355","DOI":"10.1186\/1757-1626-2-9355","article-title":"Wolfram Syndrome presenting with optic atrophy and diabetes mellitus: two case reports","volume":"2","author":"Manaviat MR","year":"2009","journal-title":"Cases J"},{"key":"e_1_2_2_11_2","doi-asserted-by":"crossref","first-page":"2327","DOI":"10.2337\/dc07-0380","article-title":"Microvascular diabetes complications in Wolfram syndrome (diabetes insipidus, diabetes mellitus, optic atrophy, and deafness [DIDMOAD]): an age\u2010 and duration\u2010matched comparison with common type 1 diabetes","volume":"30","author":"Cano A","year":"2007","journal-title":"Diabetes Care"},{"key":"e_1_2_2_12_2","doi-asserted-by":"crossref","first-page":"415","DOI":"10.1007\/s00401-009-0546-8","article-title":"Wolfram syndrome: a clinicopathologic correlation","volume":"118","author":"Hilson JB","year":"2009","journal-title":"Acta Neuropathol"},{"key":"e_1_2_2_13_2","doi-asserted-by":"crossref","first-page":"145","DOI":"10.1136\/jmg.2007.054270","article-title":"Inherited mitochondrial optic neuropathies","volume":"46","author":"Yu\u2010Wai\u2010Man P","year":"2009","journal-title":"J Med Genet"},{"key":"e_1_2_2_14_2","volume-title":"American Academy of Ophthalmology Basic and Clinical Sciences Course: Section 12, Retina and Vitreous","author":"Regillo C","year":"2009"},{"key":"e_1_2_2_15_2","doi-asserted-by":"crossref","first-page":"24","DOI":"10.1097\/WCO.0b013e3283347b27","article-title":"Genetically determined optic neuropathies","volume":"23","author":"Milea D","year":"2010","journal-title":"Curr Opin Neurol"}],"container-title":["Optometry and Vision Science"],"original-title":[],"language":"en","link":[{"URL":"https:\/\/onlinelibrary.wiley.com\/doi\/pdf\/10.1097\/OPX.0b013e31822f4d8f","content-type":"application\/pdf","content-version":"vor","intended-application":"text-mining"},{"URL":"https:\/\/onlinelibrary.wiley.com\/doi\/full-xml\/10.1097\/OPX.0b013e31822f4d8f","content-type":"application\/xml","content-version":"vor","intended-application":"text-mining"},{"URL":"https:\/\/onlinelibrary.wiley.com\/doi\/pdf\/10.1097\/OPX.0b013e31822f4d8f","content-type":"unspecified","content-version":"vor","intended-application":"similarity-checking"}],"deposited":{"date-parts":[[2026,3,31]],"date-time":"2026-03-31T06:29:46Z","timestamp":1774938586000},"score":1,"resource":{"primary":{"URL":"https:\/\/onlinelibrary.wiley.com\/doi\/10.1097\/OPX.0b013e31822f4d8f"}},"subtitle":[],"short-title":[],"issued":{"date-parts":[[2011,11]]},"references-count":14,"journal-issue":{"issue":"11","published-print":{"date-parts":[[2011,11]]}},"alternative-id":["10.1097\/OPX.0b013e31822f4d8f"],"URL":"https:\/\/doi.org\/10.1097\/opx.0b013e31822f4d8f","archive":["Portico"],"relation":{},"ISSN":["1040-5488","1538-9235"],"issn-type":[{"value":"1040-5488","type":"print"},{"value":"1538-9235","type":"electronic"}],"subject":[],"published":{"date-parts":[[2011,11]]}}}