{"status":"ok","message-type":"work","message-version":"1.0.0","message":{"indexed":{"date-parts":[[2026,9,5]],"date-time":"2026-09-05T17:43:31Z","timestamp":1788630211903,"version":"build-2803163510"},"reference-count":35,"publisher":"Elsevier BV","issue":"12","license":[{"start":{"date-parts":[[2022,12,1]],"date-time":"2022-12-01T00:00:00Z","timestamp":1669852800000},"content-version":"tdm","delay-in-days":0,"URL":"https:\/\/www.elsevier.com\/tdm\/userlicense\/1.0\/"},{"start":{"date-parts":[[2022,12,1]],"date-time":"2022-12-01T00:00:00Z","timestamp":1669852800000},"content-version":"tdm","delay-in-days":0,"URL":"https:\/\/www.elsevier.com\/legal\/tdmrep-license"},{"start":{"date-parts":[[2023,11,30]],"date-time":"2023-11-30T00:00:00Z","timestamp":1701302400000},"content-version":"am","delay-in-days":364,"URL":"http:\/\/www.elsevier.com\/open-access\/userlicense\/1.0\/"}],"funder":[{"DOI":"10.13039\/100001818","name":"Research to Prevent Blindness","doi-asserted-by":"publisher","id":[{"id":"10.13039\/100001818","id-type":"DOI","asserted-by":"publisher"}]},{"DOI":"10.13039\/100006668","name":"Oregon Health and Science University","doi-asserted-by":"publisher","id":[{"id":"10.13039\/100006668","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"}]},{"DOI":"10.13039\/100000053","name":"National Eye Institute","doi-asserted-by":"publisher","id":[{"id":"10.13039\/100000053","id-type":"DOI","asserted-by":"publisher"}]}],"content-domain":{"domain":["elsevier.com","sciencedirect.com"],"crossmark-restriction":true},"short-container-title":["Ophthalmology Retina"],"published-print":{"date-parts":[[2022,12]]},"DOI":"10.1016\/j.oret.2022.05.032","type":"journal-article","created":{"date-parts":[[2022,6,9]],"date-time":"2022-06-09T22:53:14Z","timestamp":1654815194000},"page":"1113-1121","update-policy":"https:\/\/doi.org\/10.1016\/elsevier_cm_policy","source":"Crossref","is-referenced-by-count":8,"title":["The Prevalence of Retinal Disease and Associated CNS Disease in Young Patients with Incontinentia Pigmenti"],"prefix":"10.1016","volume":"6","author":[{"given":"Ian D.","family":"Danford","sequence":"first","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Brittni A.","family":"Scruggs","sequence":"additional","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]},{"suffix":"Jr.","given":"Antonio","family":"Capone","sequence":"additional","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Michael T.","family":"Trese","sequence":"additional","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Kim A.","family":"Drenser","sequence":"additional","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Aristomenis","family":"Thanos","sequence":"additional","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Eric","family":"Nudleman","sequence":"additional","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Atchara","family":"Amphornphruet","sequence":"additional","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Boontip","family":"Tipsuriyaporn","sequence":"additional","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"G. Baker","family":"Hubbard","sequence":"additional","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Anna","family":"Ells","sequence":"additional","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]},{"suffix":"III","given":"C. Armitage","family":"Harper","sequence":"additional","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Jessica","family":"Goldstein","sequence":"additional","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Charles","family":"Calvo","sequence":"additional","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Chris","family":"Wallace-Carrete","sequence":"additional","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Duncan","family":"Berry","sequence":"additional","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Emmanuel","family":"Chang","sequence":"additional","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Lisa","family":"Leishman","sequence":"additional","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Michael","family":"Shapiro","sequence":"additional","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Michael","family":"Blair","sequence":"additional","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Mikel","family":"Mikhail","sequence":"additional","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Carol L.","family":"Shields","sequence":"additional","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Rachel","family":"Schwendeman","sequence":"additional","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Yoshihiro","family":"Yonekawa","sequence":"additional","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Mrinali P.","family":"Gupta","sequence":"additional","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Anton","family":"Orlin","sequence":"additional","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Supalert","family":"Prakhunhungsit","sequence":"additional","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Shizuo","family":"Mukai","sequence":"additional","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Audina","family":"Berrocal","sequence":"additional","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"M. Elizabeth","family":"Hartnett","sequence":"additional","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]},{"ORCID":"https:\/\/orcid.org\/0000-0001-7964-9475","authenticated-orcid":false,"given":"J. Peter","family":"Campbell","sequence":"additional","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]}],"member":"78","reference":[{"key":"10.1016\/j.oret.2022.05.032_bib1","doi-asserted-by":"crossref","first-page":"1415","DOI":"10.1111\/jdv.16403","article-title":"Multidisciplinary consensus recommendations from a European network for the diagnosis and practical management of patients with incontinentia pigmenti","volume":"34","author":"Bodemer","year":"2020","journal-title":"J\u00a0Eur Acad Dermatol Venereol"},{"key":"10.1016\/j.oret.2022.05.032_bib2","unstructured":"Wakap SN. Prevalence and incidence of rare diseases: bibliographic data. Vol. No. 1: Diseases Listed in Alphabetical Order. 2020. Ophanet Report Series: Rare Diseases Collection."},{"key":"10.1016\/j.oret.2022.05.032_bib3","doi-asserted-by":"crossref","first-page":"25","DOI":"10.1186\/1750-1172-8-25","article-title":"Systematic review of central nervous system anomalies in incontinentia pigmenti","volume":"8","author":"Mini\u0107","year":"2013","journal-title":"Orphanet J Rare Dis"},{"key":"10.1016\/j.oret.2022.05.032_bib4","doi-asserted-by":"crossref","first-page":"271","DOI":"10.1186\/s13023-019-1234-y","article-title":"Incontinentia pigmenti burden scale: designing a family burden questionnaire","volume":"14","author":"Taieb","year":"2019","journal-title":"Orphanet J Rare Dis"},{"key":"10.1016\/j.oret.2022.05.032_bib5","doi-asserted-by":"crossref","first-page":"650","DOI":"10.3928\/23258160-20150610-09","article-title":"Incontinentia pigmenti: a comprehensive review and update","volume":"46","author":"Swinney","year":"2015","journal-title":"Ophthalmic Surg Lasers Imaging Retina"},{"key":"10.1016\/j.oret.2022.05.032_bib6","doi-asserted-by":"crossref","first-page":"1645","DOI":"10.1016\/S0161-6420(93)31422-3","article-title":"Retinal and other manifestations of incontinentia pigmenti (Bloch-Sulzberger syndrome)","volume":"100","author":"Goldberg","year":"1993","journal-title":"Ophthalmology"},{"key":"10.1016\/j.oret.2022.05.032_bib7","series-title":"Pediatric Retina","article-title":"Incontinentia pigmenti","author":"Bara\u00f1ano","year":"2014"},{"key":"10.1016\/j.oret.2022.05.032_bib8","doi-asserted-by":"crossref","first-page":"535","DOI":"10.1001\/archderm.1976.01630280059017","article-title":"Incontinentia pigmenti. A world statistical analysis","volume":"112","author":"Carney","year":"1976","journal-title":"Arch Dermatol"},{"key":"10.1016\/j.oret.2022.05.032_bib9","doi-asserted-by":"crossref","first-page":"743","DOI":"10.1001\/archopht.1976.03910030353001","article-title":"Retinal vascular changes of incontinentia pigmenti","volume":"94","author":"Watzke","year":"1976","journal-title":"Arch Ophthalmol"},{"key":"10.1016\/j.oret.2022.05.032_bib10","doi-asserted-by":"crossref","first-page":"348","DOI":"10.1034\/j.1600-0420.2000.078003348.x","article-title":"Ocular manifestations of incontinentia pigmenti","volume":"78","author":"Holmstr\u00f6m","year":"2000","journal-title":"Acta Ophthalmol Scand"},{"key":"10.1016\/j.oret.2022.05.032_bib11","doi-asserted-by":"crossref","first-page":"1163","DOI":"10.1001\/archderm.139.9.1163","article-title":"Clinical study of 40 cases of incontinentia pigmenti","volume":"139","author":"Hadj-Rabia","year":"2003","journal-title":"Arch Dermatol"},{"key":"10.1016\/j.oret.2022.05.032_bib12","doi-asserted-by":"crossref","first-page":"408","DOI":"10.2298\/SARH1008408M","article-title":"Ocular anomalies in incontinentia pigmenti: literature review and meta-analysis","volume":"138","author":"Mini\u0107","year":"2010","journal-title":"Srp Arh Celok Lek"},{"key":"10.1016\/j.oret.2022.05.032_bib13","doi-asserted-by":"crossref","first-page":"2627","DOI":"10.1097\/IAE.0000000000000615","article-title":"Variable expression of retinopathy in a pedigree of patients with incontinentia pigmenti","volume":"35","author":"Chen","year":"2015","journal-title":"Retina"},{"key":"10.1016\/j.oret.2022.05.032_bib14","doi-asserted-by":"crossref","first-page":"200","DOI":"10.1007\/BF00273442","article-title":"Lyonization and the lines of Blaschko","volume":"70","author":"Happle","year":"1985","journal-title":"Hum Genet"},{"key":"10.1016\/j.oret.2022.05.032_bib15","doi-asserted-by":"crossref","first-page":"409","DOI":"10.47102\/annals-acadmedsg.V30N4p409","article-title":"A\u00a0retrospective study of incontinentia pigmenti seen at the National Skin Centre, Singapore over a 10-year period","volume":"30","author":"Chan","year":"2001","journal-title":"Ann Acad Med Singap"},{"key":"10.1016\/j.oret.2022.05.032_bib16","doi-asserted-by":"crossref","first-page":"e45","DOI":"10.1016\/j.pedhc.2017.07.003","article-title":"Incontinentia pigmenti: a summary review of this rare ectodermal dysplasia with neurologic manifestations, including treatment protocols","volume":"31","author":"Greene-Roethke","year":"2017","journal-title":"J\u00a0Pediatr Health Care"},{"key":"10.1016\/j.oret.2022.05.032_bib17","doi-asserted-by":"crossref","first-page":"1076","DOI":"10.1111\/dmcn.13140","article-title":"Clinical presentation and spectrum of neuroimaging findings in newborn infants with incontinentia pigmenti","volume":"58","author":"Soltirovska Salamon","year":"2016","journal-title":"Dev Med Child Neurol"},{"key":"10.1016\/j.oret.2022.05.032_bib18","doi-asserted-by":"crossref","first-page":"100","DOI":"10.1177\/0883073813517509","article-title":"High-dose glucocorticoid therapy in the management of seizures in neonatal incontinentia pigmenti: a case report","volume":"30","author":"Wolf","year":"2015","journal-title":"J\u00a0Child Neurol"},{"key":"10.1016\/j.oret.2022.05.032_bib19","doi-asserted-by":"crossref","first-page":"323","DOI":"10.1016\/j.ejmg.2012.04.007","article-title":"Neurological findings in incontinentia pigmenti: a review","volume":"55","author":"Meuwissen","year":"2012","journal-title":"Eur J Med Genet"},{"key":"10.1016\/j.oret.2022.05.032_bib20","doi-asserted-by":"crossref","first-page":"474","DOI":"10.1111\/j.1365-2230.2005.01848.x","article-title":"Incontinentia pigmenti case series: clinical spectrum of incontinentia pigmenti in 53 female patients and their relatives. Incontinentia Pigmenti Case Series","volume":"30","author":"Phan","year":"2005","journal-title":"Clin Exp Dermatol"},{"key":"10.1016\/j.oret.2022.05.032_bib21","doi-asserted-by":"crossref","first-page":"11","DOI":"10.1136\/bjo.2009.164434","article-title":"Incontinentia pigmenti\u2014ophthalmological observation of a series of cases and review of the literature","volume":"95","author":"O\u2019Doherty","year":"2011","journal-title":"Br J Ophthalmol"},{"key":"10.1016\/j.oret.2022.05.032_bib22","doi-asserted-by":"crossref","first-page":"223","DOI":"10.1186\/s13023-020-01509-2","article-title":"Early management of sight threatening retinopathy in incontinentia pigmenti","volume":"15","author":"Michel","year":"2020","journal-title":"Orphanet J Rare Dis"},{"key":"10.1016\/j.oret.2022.05.032_bib23","doi-asserted-by":"crossref","first-page":"708","DOI":"10.1097\/IAE.0b013e3181cd4942","article-title":"Regression of retinal neovascularization after laser photocoagulation in incontinentia pigmenti","volume":"30","author":"Ranchod","year":"2010","journal-title":"Retina"},{"key":"10.1016\/j.oret.2022.05.032_bib24","doi-asserted-by":"crossref","first-page":"542","DOI":"10.1001\/jamaophthalmol.2015.22","article-title":"Extended follow-up of treated and untreated retinopathy in incontinentia pigmenti: analysis of peripheral vascular changes and incidence of retinal detachment","volume":"133","author":"Chen","year":"2015","journal-title":"JAMA Ophthalmol"},{"key":"10.1016\/j.oret.2022.05.032_bib25","doi-asserted-by":"crossref","first-page":"536","DOI":"10.1111\/cge.12223","article-title":"Incontinentia pigmenti diagnostic criteria update","volume":"85","author":"Mini\u0107","year":"2014","journal-title":"Clin Genet"},{"key":"10.1016\/j.oret.2022.05.032_bib26","doi-asserted-by":"crossref","first-page":"262","DOI":"10.1016\/j.ophtha.2013.08.010","article-title":"High prevalence of peripheral retinal vascular anomalies in family members of patients with familial exudative vitreoretinopathy","volume":"121","author":"Kashani","year":"2014","journal-title":"Ophthalmology"},{"key":"10.1016\/j.oret.2022.05.032_bib27","doi-asserted-by":"crossref","first-page":"529","DOI":"10.4321\/S0365-66912009001000008","article-title":"[Fluorescein angiography with Retcam in incontinentia pigmenti: a case report]","volume":"84","author":"Escudero","year":"2009","journal-title":"Arch Soc Esp Oftalmol"},{"key":"10.1016\/j.oret.2022.05.032_bib28","doi-asserted-by":"crossref","first-page":"696","DOI":"10.1016\/S0002-9394(14)77070-9","article-title":"Incontinentia pigmenti","volume":"110","author":"Catalano","year":"1990","journal-title":"Am J Ophthalmol"},{"key":"10.1016\/j.oret.2022.05.032_bib29","doi-asserted-by":"crossref","first-page":"977","DOI":"10.1001\/jamaophthalmol.2017.2481","article-title":"Handheld optical coherence tomography angiography and ultra-wide-field optical coherence tomography in retinopathy of prematurity","volume":"135","author":"Campbell","year":"2017","journal-title":"JAMA Ophthalmol"},{"key":"10.1016\/j.oret.2022.05.032_bib30","doi-asserted-by":"crossref","first-page":"788","DOI":"10.4103\/ijo.IJO_1688_18","article-title":"Ultra-widefield fundus fluorescein angiography in pediatric retinal vascular diseases","volume":"67","author":"Temkar","year":"2019","journal-title":"Indian J Ophthalmol"},{"key":"10.1016\/j.oret.2022.05.032_bib31","doi-asserted-by":"crossref","first-page":"796","DOI":"10.1097\/01.anes.0000344728.34332.5d","article-title":"Early exposure to anesthesia and learning disabilities in a population-based birth cohort","volume":"110","author":"Wilder","year":"2009","journal-title":"Anesthesiology"},{"key":"10.1016\/j.oret.2022.05.032_bib32","doi-asserted-by":"crossref","first-page":"602","DOI":"10.1016\/j.oret.2019.12.015","article-title":"Late-onset retinal findings and complications in untreated retinopathy of prematurity","volume":"4","author":"Hamad","year":"2020","journal-title":"Ophthalmol Retina"},{"key":"10.1016\/j.oret.2022.05.032_bib33","doi-asserted-by":"crossref","first-page":"2171","DOI":"10.1093\/hmg\/10.19.2171","article-title":"A\u00a0recurrent deletion in the ubiquitously expressed NEMO (IKK-gamma) gene accounts for the vast majority of incontinentia pigmenti mutations","volume":"10","author":"Aradhya","year":"2001","journal-title":"Hum Mol Genet"},{"key":"10.1016\/j.oret.2022.05.032_bib34","doi-asserted-by":"crossref","first-page":"73","DOI":"10.2147\/EB.S193026","article-title":"Optical coherence tomography angiography in neurodegenerative diseases: a review","volume":"12","author":"Tsokolas","year":"2020","journal-title":"Eye Brain"},{"key":"10.1016\/j.oret.2022.05.032_bib35","first-page":"1706","article-title":"Optical coherence tomography angiography in neurodegenerative disorders","volume":"9","author":"Pellegrini","year":"2020","journal-title":"J\u00a0Clin Med"}],"container-title":["Ophthalmology Retina"],"original-title":[],"language":"en","link":[{"URL":"https:\/\/api.elsevier.com\/content\/article\/PII:S2468653022002913?httpAccept=text\/xml","content-type":"text\/xml","content-version":"vor","intended-application":"text-mining"},{"URL":"https:\/\/api.elsevier.com\/content\/article\/PII:S2468653022002913?httpAccept=text\/plain","content-type":"text\/plain","content-version":"vor","intended-application":"text-mining"}],"deposited":{"date-parts":[[2025,11,11]],"date-time":"2025-11-11T09:31:27Z","timestamp":1762853487000},"score":1,"resource":{"primary":{"URL":"https:\/\/linkinghub.elsevier.com\/retrieve\/pii\/S2468653022002913"}},"subtitle":[],"short-title":[],"issued":{"date-parts":[[2022,12]]},"references-count":35,"journal-issue":{"issue":"12","published-print":{"date-parts":[[2022,12]]}},"alternative-id":["S2468653022002913"],"URL":"https:\/\/doi.org\/10.1016\/j.oret.2022.05.032","relation":{},"ISSN":["2468-6530"],"issn-type":[{"value":"2468-6530","type":"print"}],"subject":[],"published":{"date-parts":[[2022,12]]},"assertion":[{"value":"Elsevier","name":"publisher","label":"This article is maintained by"},{"value":"The Prevalence of Retinal Disease and Associated CNS Disease in Young Patients with Incontinentia Pigmenti","name":"articletitle","label":"Article Title"},{"value":"Ophthalmology Retina","name":"journaltitle","label":"Journal Title"},{"value":"https:\/\/doi.org\/10.1016\/j.oret.2022.05.032","name":"articlelink","label":"CrossRef DOI link to publisher maintained version"},{"value":"article","name":"content_type","label":"Content Type"},{"value":"\u00a9 2022 by the American Academy of Ophthalmology","name":"copyright","label":"Copyright"}]}}