{"status":"ok","message-type":"work","message-version":"1.0.0","message":{"indexed":{"date-parts":[[2026,8,23]],"date-time":"2026-08-23T18:06:01Z","timestamp":1787508361747,"version":"build-2736575974"},"reference-count":33,"publisher":"Wiley","issue":"5","license":[{"start":{"date-parts":[[2023,12,22]],"date-time":"2023-12-22T00:00:00Z","timestamp":1703203200000},"content-version":"vor","delay-in-days":0,"URL":"http:\/\/creativecommons.org\/licenses\/by-nc-nd\/4.0\/"}],"content-domain":{"domain":["onlinelibrary.wiley.com"],"crossmark-restriction":true},"short-container-title":["Acta Ophthalmologica"],"published-print":{"date-parts":[[2024,8]]},"abstract":"Abstract<\/jats:title>\n \n Purpose<\/jats:title>\n Congenital aniridia is a serious eye disease characterized by absence of iris to various degrees. The aims of this study were to investigate health\u2010related quality of life (HRQoL) in adults with aniridia and assess the relationships between HRQoL, psychological status, ocular health and obesity.<\/jats:p>\n <\/jats:sec>\n \n Methods<\/jats:title>\n Twenty\u2010nine adults with congenital aniridia (48% male, aged 18\u201379\u2009years) participated. HRQoL was measured with SF\u201036 and the EQ visual analogue scale (VAS). The physical (PCS) and mental (MCS) component summaries of the SF\u201036 were calculated with higher scores indicating better HRQoL. Symptoms of anxiety and depression were measured using the Hospital Anxiety and Depression Scale (HADS). Obesity was assessed with the Patient\u2010Reported Outcomes in Obesity (PROS). Sociodemographic characteristics, genetic variants and ocular and medical health variables were also analysed.<\/jats:p>\n <\/jats:sec>\n \n Results<\/jats:title>\n \n The participants scored significantly lower in the general health domain of the SF\u201036 than the general population (65.2 vs. 75.3,\n p<\/jats:italic>\n \u2009=\u20090.017). The EQ VAS score was also lower in the aniridia group (64.9 vs. 77.9,\n p<\/jats:italic>\n \u2009=\u20090.021). Low PCS score was correlated with presence of ocular pain (\n p<\/jats:italic>\n \u2009=\u20090.019), high HADS score (\n p<\/jats:italic>\n \u2009=\u20090.017) and high PROS score (\n p<\/jats:italic>\n \u2009=\u20090.009). Low MCS score was related to higher educational level (\n p<\/jats:italic>\n \u2009=\u20090.038) and high HADS score (\n p<\/jats:italic>\n \u2009<\u20090.001). High HADS and PROS scores were both related to low EQ VAS scores.\n <\/jats:p>\n <\/jats:sec>\n \n Conclusion<\/jats:title>\n Adults with congenital aniridia scored worse on certain measures of HRQoL than the general population. Poorer HRQoL was associated with increased symptoms of anxiety, depression and obesity and with presence of ocular pain.<\/jats:p>\n <\/jats:sec>","DOI":"10.1111\/aos.16615","type":"journal-article","created":{"date-parts":[[2023,12,22]],"date-time":"2023-12-22T04:59:26Z","timestamp":1703221166000},"page":"590-599","update-policy":"https:\/\/doi.org\/10.1002\/crossmark_policy","source":"Crossref","is-referenced-by-count":2,"title":["Relationship between physical and psychological functioning and\n health\u2010related<\/scp>\n quality of life in congenital Aniridia"],"prefix":"10.1111","volume":"102","author":[{"ORCID":"https:\/\/orcid.org\/0000-0002-9789-1932","authenticated-orcid":false,"given":"Erlend Christoffer Sommer","family":"Landsend","sequence":"first","affiliation":[{"name":"Department of Ophthalmology Oslo University Hospital Oslo Norway"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Charlotte","family":"von der Lippe","sequence":"additional","affiliation":[{"name":"Department of Medical Genetics Telemark Hospital Trust Skien Norway"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Line","family":"Medi\u00e5","sequence":"additional","affiliation":[{"name":"Centre for Rare Disorders Oslo University Hospital Oslo Norway"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Jeanette Ullmann","family":"Miller","sequence":"additional","affiliation":[{"name":"Centre for Rare Disorders Oslo University Hospital Oslo Norway"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Knut Erik","family":"Berge","sequence":"additional","affiliation":[{"name":"Department of Medical Genetics Oslo University Hospital Oslo Norway"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Solrun","family":"Sigurdardottir","sequence":"additional","affiliation":[{"name":"Centre for Rare Disorders Oslo University Hospital Oslo Norway"}],"role":[{"vocabulary":"crossref","role":"author"}]}],"member":"311","published-online":{"date-parts":[[2023,12,22]]},"reference":[{"key":"e_1_2_8_2_1","first-page":"1045","article-title":"Patient\u2010Reported Quality of Life with Obesity \u2013 Development of a New Measurement Scale","volume":"139","author":"Aasprang A.","year":"2019","journal-title":"Tidsskrift for Den norske legeforeni"},{"key":"e_1_2_8_3_1","doi-asserted-by":"publisher","DOI":"10.12740\/PP\/OnlineFirst\/85408"},{"key":"e_1_2_8_4_1","doi-asserted-by":"publisher","DOI":"10.1016\/S0022-3999(01)00296-3"},{"key":"e_1_2_8_5_1","doi-asserted-by":"publisher","DOI":"10.1186\/s13023-017-0730-1"},{"key":"e_1_2_8_6_1","doi-asserted-by":"publisher","DOI":"10.1186\/s12889-022-13077-0"},{"key":"e_1_2_8_7_1","doi-asserted-by":"publisher","DOI":"10.1167\/iovs.03-0661"},{"key":"e_1_2_8_8_1","doi-asserted-by":"publisher","DOI":"10.3109\/09286586.2016.1168851"},{"key":"e_1_2_8_9_1","doi-asserted-by":"publisher","DOI":"10.1016\/j.preteyeres.2022.101133"},{"key":"e_1_2_8_10_1","unstructured":"EuroQol Research Foundation. (n.d.)(1): EQ\u20105D\u20105L User Guide \u2013 Basic information on how to use the EQ\u20105D\u20105L instrument. Available from:https:\/\/euroqol.org\/publications\/user\u2010guides\/[Accessed 8th March 2023]."},{"key":"e_1_2_8_11_1","doi-asserted-by":"publisher","DOI":"10.1007\/s11136-021-02938-7"},{"key":"e_1_2_8_12_1","doi-asserted-by":"publisher","DOI":"10.1186\/s12955-017-0625-9"},{"key":"e_1_2_8_13_1","doi-asserted-by":"publisher","DOI":"10.1016\/S0925-4773(01)00646-3"},{"key":"e_1_2_8_14_1","doi-asserted-by":"publisher","DOI":"10.1053\/apmr.2002.36837"},{"key":"e_1_2_8_15_1","first-page":"1576","article-title":"Quality of life survey using NEI VFQ\u201025 in patients with aniridia","volume":"61","author":"Inokuchi C.","year":"2020","journal-title":"Investigative Ophthalmology & Visual Science"},{"key":"e_1_2_8_16_1","doi-asserted-by":"publisher","DOI":"10.1007\/s11136-017-1684-4"},{"key":"e_1_2_8_17_1","doi-asserted-by":"publisher","DOI":"10.1016\/j.survophthal.2021.02.011"},{"key":"e_1_2_8_18_1","doi-asserted-by":"publisher","DOI":"10.1016\/j.survophthal.2017.09.004"},{"key":"e_1_2_8_19_1","doi-asserted-by":"publisher","DOI":"10.1080\/09286580601139212"},{"key":"e_1_2_8_20_1","doi-asserted-by":"publisher","DOI":"10.1007\/s00417-023-06050-z"},{"key":"e_1_2_8_21_1","doi-asserted-by":"publisher","DOI":"10.1167\/iovs.11-9094"},{"key":"e_1_2_8_22_1","doi-asserted-by":"publisher","DOI":"10.1177\/14034948980260040401"},{"key":"e_1_2_8_23_1","doi-asserted-by":"publisher","DOI":"10.1001\/archopht.119.7.1050"},{"key":"e_1_2_8_24_1","unstructured":"National Organization for Rare Disorders. (2021)NORD \u2013 national organization for rare disorders."},{"key":"e_1_2_8_25_1","doi-asserted-by":"publisher","DOI":"10.1016\/j.jaapos.2011.07.009"},{"key":"e_1_2_8_26_1","unstructured":"Orphanet. (2022)Orphanet the portal for rare diseases and orphan drugs. Orphanet."},{"key":"e_1_2_8_27_1","doi-asserted-by":"publisher","DOI":"10.1186\/s13023-018-0953-9"},{"key":"e_1_2_8_28_1","unstructured":"Statistics Norway. (2020)Educational level in the Norwegian population. Statistics Norway Web Page. Statistics Norway."},{"key":"e_1_2_8_29_1","doi-asserted-by":"crossref","unstructured":"Statistics Norway. (2022)Unemployed in the Norwegian population aged 18\u201367\u2009years in 2021. Statistics Norway Web Page. Statistics Norway.","DOI":"10.1787\/4e56073a-en"},{"key":"e_1_2_8_30_1","doi-asserted-by":"publisher","DOI":"10.1016\/j.ophtha.2006.04.028"},{"key":"e_1_2_8_31_1","article-title":"Comparison of methods for the scoring and statistical analysis of SF\u201036 health profile and summary measures: summary of results from the medical outcomes study","volume":"33","author":"Ware J.E.","year":"1995","journal-title":"Medical Care"},{"key":"e_1_2_8_32_1","unstructured":"World Health Organization. (2021)Obesity and overweight. World Health Organization."},{"key":"e_1_2_8_33_1","doi-asserted-by":"publisher","DOI":"10.3390\/ijerph13101024"},{"key":"e_1_2_8_34_1","doi-asserted-by":"publisher","DOI":"10.1111\/j.1600-0447.1983.tb09716.x"}],"container-title":["Acta Ophthalmologica"],"original-title":[],"language":"en","link":[{"URL":"https:\/\/onlinelibrary.wiley.com\/doi\/pdf\/10.1111\/aos.16615","content-type":"unspecified","content-version":"vor","intended-application":"similarity-checking"}],"deposited":{"date-parts":[[2024,7,8]],"date-time":"2024-07-08T07:41:46Z","timestamp":1720424506000},"score":1,"resource":{"primary":{"URL":"https:\/\/onlinelibrary.wiley.com\/doi\/10.1111\/aos.16615"}},"subtitle":[],"short-title":[],"issued":{"date-parts":[[2023,12,22]]},"references-count":33,"journal-issue":{"issue":"5","published-print":{"date-parts":[[2024,8]]}},"alternative-id":["10.1111\/aos.16615"],"URL":"https:\/\/doi.org\/10.1111\/aos.16615","archive":["Portico"],"relation":{},"ISSN":["1755-375X","1755-3768"],"issn-type":[{"value":"1755-375X","type":"print"},{"value":"1755-3768","type":"electronic"}],"subject":[],"published":{"date-parts":[[2023,12,22]]},"assertion":[{"value":"2023-10-11","order":0,"name":"received","label":"Received","group":{"name":"publication_history","label":"Publication History"}},{"value":"2023-12-08","order":1,"name":"accepted","label":"Accepted","group":{"name":"publication_history","label":"Publication History"}},{"value":"2023-12-22","order":2,"name":"published","label":"Published","group":{"name":"publication_history","label":"Publication History"}}]}}