{"status":"ok","message-type":"work","message-version":"1.0.0","message":{"indexed":{"date-parts":[[2026,5,9]],"date-time":"2026-05-09T05:39:21Z","timestamp":1778305161226,"version":"3.51.4"},"reference-count":40,"publisher":"Wiley","issue":"6","license":[{"start":{"date-parts":[[2025,5,7]],"date-time":"2025-05-07T00:00:00Z","timestamp":1746576000000},"content-version":"vor","delay-in-days":0,"URL":"http:\/\/creativecommons.org\/licenses\/by\/4.0\/"}],"content-domain":{"domain":["onlinelibrary.wiley.com"],"crossmark-restriction":true},"short-container-title":["Clinical Exper Ophthalmology"],"published-print":{"date-parts":[[2025,8]]},"abstract":"<jats:title>ABSTRACT<\/jats:title><jats:sec><jats:title>Background<\/jats:title><jats:p>Dominant Optic Atrophy (DOA) causes slowly progressive visual decline usually beginning in childhood. As new therapies come to clinical trial, the choice of biomarkers to be used as clinical trial endpoints has become a critical question to be addressed.<\/jats:p><\/jats:sec><jats:sec><jats:title>Methods<\/jats:title><jats:p>We undertook a systematic review and meta\u2010analysis of studies reporting longitudinal data of any biomarker in DOA patients.<\/jats:p><\/jats:sec><jats:sec><jats:title>Results<\/jats:title><jats:p>In total, seven studies were included in the systematic review, and four presented paired results compatible with meta\u2010analysis. Visual acuity was the only biomarker found with reported longitudinal data. Of the included studies in the meta\u2010analysis, the rate of yearly visual acuity decline (0.022 LogMAR\/year., 95% CI: \u22120.008 to 0.052) was not significantly different from zero (<jats:italic>Z<\/jats:italic>\u2009=\u20091.4, <jats:italic>p<\/jats:italic>\u2009=\u20090.155).<\/jats:p><\/jats:sec><jats:sec><jats:title>Conclusion<\/jats:title><jats:p>Quantifying this slow rate of visual decline has implications for future study design and suggests that further natural history studies examining alternative biomarkers in DOA are warranted.<\/jats:p><\/jats:sec>","DOI":"10.1111\/ceo.14543","type":"journal-article","created":{"date-parts":[[2025,5,7]],"date-time":"2025-05-07T05:04:44Z","timestamp":1746594284000},"page":"652-659","update-policy":"https:\/\/doi.org\/10.1002\/crossmark_policy","source":"Crossref","is-referenced-by-count":5,"title":["Longitudinal Visual Biomarkers in Dominant Optic Atrophy: A Systematic Review and Meta\u2010Analysis"],"prefix":"10.1111","volume":"53","author":[{"ORCID":"https:\/\/orcid.org\/0000-0001-8653-6073","authenticated-orcid":false,"given":"Christopher A.","family":"Ovens","sequence":"first","affiliation":[{"name":"Save Sight Institute, Faculty of Medicine and Health The University of Sydney, Sydney Eye Hospital Campus  Sydney Australia"}],"role":[{"role":"author","vocabulary":"crossref"}]},{"ORCID":"https:\/\/orcid.org\/0000-0002-6763-8119","authenticated-orcid":false,"given":"John R.","family":"Grigg","sequence":"additional","affiliation":[{"name":"Save Sight Institute, Faculty of Medicine and Health The University of Sydney, Sydney Eye Hospital Campus  Sydney Australia"},{"name":"Eye Genetics Research Unit, The Children's Hospital at Westmead, Save Sight Institute, Children's Medical Research Institute The University of Sydney  Sydney Australia"},{"name":"Sydney Eye Hospital  Sydney Australia"}],"role":[{"role":"author","vocabulary":"crossref"}]},{"ORCID":"https:\/\/orcid.org\/0000-0002-0379-7743","authenticated-orcid":false,"given":"Clare L.","family":"Fraser","sequence":"additional","affiliation":[{"name":"Save Sight Institute, Faculty of Medicine and Health The University of Sydney, Sydney Eye Hospital Campus  Sydney Australia"},{"name":"Sydney Eye Hospital  Sydney Australia"}],"role":[{"role":"author","vocabulary":"crossref"}]}],"member":"311","published-online":{"date-parts":[[2025,5,7]]},"reference":[{"key":"e_1_2_9_2_1","doi-asserted-by":"publisher","DOI":"10.1038\/79936"},{"key":"e_1_2_9_3_1","doi-asserted-by":"publisher","DOI":"10.1097\/APO.0b013e31824a65b1"},{"issue":"7","key":"e_1_2_9_4_1","first-page":"88","article-title":"OSPREY: An Open\u2010Label Study to Investigate Safety, Tolerability, and Exposure of the Antisense Oligonucleotide (ASO) STK\u2010002 in Patients With Autosomal Dominant Optic Atrophy (ADOA)","volume":"65","author":"Votruba M.","year":"2024","journal-title":"Investigative Ophthalmology & Visual Science"},{"key":"e_1_2_9_5_1","doi-asserted-by":"publisher","DOI":"10.1016\/j.ophtha.2009.12.038"},{"key":"e_1_2_9_6_1","doi-asserted-by":"publisher","DOI":"10.1186\/1750\u20101172\u20107\u201046"},{"key":"e_1_2_9_7_1","doi-asserted-by":"publisher","DOI":"10.1101\/gr.108696.110"},{"key":"e_1_2_9_8_1","doi-asserted-by":"publisher","DOI":"10.1097\/WNO.0000000000001367"},{"key":"e_1_2_9_9_1","doi-asserted-by":"publisher","DOI":"10.1002\/acn3.51026"},{"issue":"7","key":"e_1_2_9_10_1","first-page":"5305","article-title":"PYC\u2010001: A Promising RNA Therapeutic for Autosomal Dominant Optic Atrophy","volume":"65","author":"Grainok J.","year":"2024","journal-title":"Investigative Ophthalmology and Visual Science"},{"key":"e_1_2_9_11_1","doi-asserted-by":"publisher","DOI":"10.1186\/s13023-017-0597-1"},{"key":"e_1_2_9_12_1","doi-asserted-by":"publisher","DOI":"10.1136\/bmj.n71"},{"key":"e_1_2_9_13_1","unstructured":"G. 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