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Best-corrected visual acuity (BCVA) is a common primary endpoint in clinical trials for retinal diseases, but the natural history of BCVA loss remains unclear because of the heterogeneity of manifestations in BCD patients.\n                    <\/jats:p>\n                  <\/jats:sec>\n                  <jats:sec>\n                    <jats:title>Methods<\/jats:title>\n                    <jats:p>Based on the individual data of untreated BCD patients, a disease progression model was established using the change in BCVA from baseline as an index, and covariates including age of onset, age, duration of disease, baseline BCVA, gender, race (East Asian\/non-East Asian), genotype, and family history. Then, based on the final model, the natural disease progression characteristics of BCD were simulated.<\/jats:p>\n                  <\/jats:sec>\n                  <jats:sec>\n                    <jats:title>Result<\/jats:title>\n                    <jats:p>A total of 14 studies met the inclusion criteria, with a total sample size of 117 cases, including 6 studies (N=80) with East Asian populations and 9 studies (N=37) with non-East Asian populations. The change of BCVA from baseline increased linearly with time, and the disease progression model of BCD was successfully established. BCVA increased by 0.06 logarithm of the minimum angle of resolution (LogMAR) per year in BCD patients. BCVA increased by 0.09 LogMAR per year in patients with BCVA\u22650.5LogMAR and disease duration more than 10 years.<\/jats:p>\n                  <\/jats:sec>\n                  <jats:sec>\n                    <jats:title>Conclusions<\/jats:title>\n                    <jats:p>For the first time, we successfully established a BCD disease progression model based on the change in BCVA from baseline. The mean visual acuity loss increased linearly with the progression of the disease. A sharper loss of BCVA may be expected in patients with BCVA\u22650.5LogMAR and disease duration \u226510 years.<\/jats:p>\n                  <\/jats:sec>","DOI":"10.1136\/bmjophth-2024-001908","type":"journal-article","created":{"date-parts":[[2025,4,12]],"date-time":"2025-04-12T17:55:16Z","timestamp":1744480516000},"page":"e001908","update-policy":"https:\/\/doi.org\/10.1136\/crossmarkpolicy","source":"Crossref","is-referenced-by-count":2,"title":["Natural history of progressive vision loss in Bietti crystalline dystrophy: a model-based meta-analysis"],"prefix":"10.1136","volume":"10","author":[{"given":"Haihan","family":"Zhang","sequence":"first","affiliation":[{"name":"Capital Medical University, Beijing, China"}],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"Shiyi","family":"Yin","sequence":"additional","affiliation":[{"name":"Capital Medical University, Beijing, China"}],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"Ning","family":"Guan","sequence":"additional","affiliation":[{"name":"Shanghai Vitalgen BioPharma Corporation Limited, Shanghai, China"}],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"Jinyuan","family":"Wang","sequence":"additional","affiliation":[{"name":"Capital Medical University, Beijing, China"},{"name":"Tsinghua University, Beijing, China"}],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"Qingqing","family":"Cheng","sequence":"additional","affiliation":[{"name":"Shanghai BioGuider Medical Technology, Shanghai, China"}],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"Lingxiao","family":"Zhang","sequence":"additional","affiliation":[{"name":"Shanghai BioGuider Medical Technology, Shanghai, China"}],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"Qingshan","family":"Zheng","sequence":"additional","affiliation":[{"name":"Shanghai University of Traditional Chinese Medicine, Shanghai, China"}],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"Hua","family":"Lv","sequence":"additional","affiliation":[{"name":"Shanghai Vitalgen BioPharma Corporation Limited, Shanghai, China"}],"role":[{"role":"author","vocabulary":"crossref"}]},{"ORCID":"https:\/\/orcid.org\/0000-0003-2386-0989","authenticated-orcid":false,"given":"Wenbin","family":"Wei","sequence":"additional","affiliation":[{"name":"Capital Medical University, Beijing, China"}],"role":[{"role":"author","vocabulary":"crossref"}]}],"member":"239","published-online":{"date-parts":[[2025,4,12]]},"reference":[{"key":"2026010721203208000_10.1.e001908.1","unstructured":"Bietti G . Ueber familiares vorkommen von\" retinitis punkutata albescens\"(verbunden mit\" dystrophia marginalis cristallinea corneae. In: Glitzern des Glaskorpers und anderen degenerativen Augenveranderungen. Klin Monatsbl Augenheilkd. 1937."},{"key":"2026010721203208000_10.1.e001908.2","doi-asserted-by":"publisher","DOI":"10.1086\/383228"},{"key":"2026010721203208000_10.1.e001908.3","doi-asserted-by":"publisher","DOI":"10.2147\/OPTH.S185744"},{"key":"2026010721203208000_10.1.e001908.4","first-page":"738","article-title":"Novel mutations in the CYP4V2 gene associated with Bietti crystalline corneoretinal dystrophy","volume":"11","author":"Shan","year":"2005","journal-title":"Mol Vis"},{"key":"2026010721203208000_10.1.e001908.5","doi-asserted-by":"crossref","DOI":"10.1167\/tvst.12.2.27","article-title":"An In-Depth Single-Gene Worldwide Carrier Frequency and Genetic Prevalence Analysis of CYP4V2 as the Cause of Bietti Crystalline Dystrophy","volume":"12","author":"Hanany","year":"2023","journal-title":"Transl Vis Sci Technol"},{"key":"2026010721203208000_10.1.e001908.6","doi-asserted-by":"publisher","DOI":"10.1097\/00006982-200406000-00013"},{"key":"2026010721203208000_10.1.e001908.7","unstructured":"Vargas M , Mitchell A , Yang P , et al . 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Clinical study on the efficacy of repeated intravitreal injection of rat nerve growth factor in the treatment of bietti\u2019s crystalline dystrophy. Third Military Medical University, 2009."},{"key":"2026010721203208000_10.1.e001908.19","doi-asserted-by":"crossref","DOI":"10.1371\/journal.pone.0094960","article-title":"Molecular analysis and phenotypic study in 14 Chinese families with Bietti crystalline dystrophy","volume":"9","author":"Yin","year":"2014","journal-title":"PLoS ONE"},{"key":"2026010721203208000_10.1.e001908.20","first-page":"1970","article-title":"Molecular screening of the CYP4V2 gene in Bietti crystalline dystrophy that is associated with choroidal neovascularization","volume":"17","author":"Mamatha","year":"2011","journal-title":"Mol Vis"},{"key":"2026010721203208000_10.1.e001908.21","doi-asserted-by":"crossref","first-page":"189","DOI":"10.3341\/kjo.2014.28.2.189","article-title":"Acetazolamide for cystoid macular oedema in Bietti crystalline retinal dystrophy","volume":"28","author":"Broadhead","year":"2014","journal-title":"Korean J Ophthalmol"},{"key":"2026010721203208000_10.1.e001908.22","doi-asserted-by":"crossref","first-page":"212","DOI":"10.4274\/tjo.galenos.2022.86681","article-title":"Asymptomatic Unilateral Full-Thickness Macular Hole in a Patient with Bietti Crystalline Dystrophy During 13-Year Follow-up with Optical Coherence Tomography","volume":"52","author":"Saatci","year":"2022","journal-title":"Turk J Ophthalmol"},{"key":"2026010721203208000_10.1.e001908.23","first-page":"1182","article-title":"Clinical and molecular findings in three Lebanese families with Bietti crystalline dystrophy: report on a novel mutation","volume":"18","author":"Haddad","year":"2012","journal-title":"Mol Vis"},{"key":"2026010721203208000_10.1.e001908.24","doi-asserted-by":"publisher","DOI":"10.1038\/gim.2015.30"},{"key":"2026010721203208000_10.1.e001908.25","doi-asserted-by":"publisher","DOI":"10.1002\/sim.8086"},{"key":"2026010721203208000_10.1.e001908.26","first-page":"1806","article-title":"Identification of novel CYP4V2 gene mutations in 92 Chinese families with Bietti\u2019s crystalline corneoretinal dystrophy","volume":"20","author":"Meng","year":"2014","journal-title":"Mol Vis"},{"key":"2026010721203208000_10.1.e001908.27","doi-asserted-by":"crossref","first-page":"154","DOI":"10.1016\/j.exer.2016.03.007","article-title":"Identification of CYP4V2 mutation in 36 Chinese families with Bietti crystalline corneoretinal dystrophy","volume":"146","author":"Yin","year":"2016","journal-title":"Exp Eye Res"},{"key":"2026010721203208000_10.1.e001908.28","doi-asserted-by":"publisher","DOI":"10.1097\/00006982-199212010-00004"},{"key":"2026010721203208000_10.1.e001908.29","doi-asserted-by":"crossref","first-page":"562","DOI":"10.1055\/s-2001-17139","article-title":"Corneoretinal dystrophy (Bietti)-- Long-term course of one patient over a period of 30 years, and interindividual variability of clinical and electrophysiological findings in two patients","volume":"218","author":"Jurklies","year":"2001","journal-title":"Klin Monbl Augenheilkd"},{"key":"2026010721203208000_10.1.e001908.30","doi-asserted-by":"crossref","unstructured":"Mansour A , Uwaydat S , Chan C-C . 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