{"status":"ok","message-type":"work","message-version":"1.0.0","message":{"indexed":{"date-parts":[[2026,8,24]],"date-time":"2026-08-24T04:18:41Z","timestamp":1787545121975,"version":"build-2736575974"},"reference-count":12,"publisher":"BMJ","issue":"8","content-domain":{"domain":["bmj.com"],"crossmark-restriction":true},"short-container-title":["BMJ Case Rep"],"accepted":{"date-parts":[[2026,8,2]]},"published-print":{"date-parts":[[2026,8]]},"abstract":"<jats:p>Voretigene neparvovec can improve visual function in children with RPE65-associated inherited retinal dystrophy. However, the effect of rapid asymmetric visual improvement on binocular development and amblyopia remains uncertain.<\/jats:p>\n                  <jats:p>A young child underwent sequential treatment 8\u2009months apart. After right-eye treatment, visual acuity improved, fixation switched and a manifest left esotropia developed. Left-eye visual acuity subsequently deteriorated. We describe this as reverse amblyopia in a novel context: reversal of the interocular amblyopic pattern following treatment-induced visual improvement without fellow-eye penalisation. Despite left-eye treatment, refractive correction and attempted occlusion, the marked visual asymmetry persisted. This case highlights a potential binocular effect of rapid unilateral visual improvement and the need for close monitoring between sequential treatments.<\/jats:p>","DOI":"10.1136\/bcr-2026-273165","type":"journal-article","created":{"date-parts":[[2026,8,24]],"date-time":"2026-08-24T03:22:06Z","timestamp":1787541726000},"page":"e273165","update-policy":"https:\/\/doi.org\/10.1136\/crossmarkpolicy","source":"Crossref","is-referenced-by-count":0,"title":["Reverse amblyopia following gene therapy-induced visual improvement in a child with retinal pigment epithelium-65 inherited retinal dystrophy"],"prefix":"10.1136","volume":"19","author":[{"ORCID":"https:\/\/orcid.org\/0000-0001-5317-1194","authenticated-orcid":false,"given":"Aimee","family":"Lloyd","sequence":"first","affiliation":[{"name":"Manchester Royal Eye Hospital, Manchester, UK"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Peter","family":"Kiraly","sequence":"additional","affiliation":[{"name":"Manchester Royal Eye Hospital, Manchester, UK"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Evita Evangelia","family":"Christou","sequence":"additional","affiliation":[{"name":"Manchester Royal Eye Hospital, Manchester, UK"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Jane","family":"Ashworth","sequence":"additional","affiliation":[{"name":"Manchester Royal Eye Hospital, Manchester, UK"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Assad","family":"Jalil","sequence":"additional","affiliation":[{"name":"Manchester Royal Eye Hospital, Manchester, UK"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Tsveta","family":"Ivanova","sequence":"additional","affiliation":[{"name":"Manchester Royal Eye Hospital, Manchester, UK"}],"role":[{"vocabulary":"crossref","role":"author"}]}],"member":"239","published-online":{"date-parts":[[2026,8,20]]},"reference":[{"key":"2026082006251432000_19.8.e273165.1","doi-asserted-by":"publisher","DOI":"10.1016\/S0140-6736(17)31868-8"},{"key":"2026082006251432000_19.8.e273165.2","doi-asserted-by":"publisher","DOI":"10.1016\/S0140-6736(06)68581-4"},{"key":"2026082006251432000_19.8.e273165.3","doi-asserted-by":"publisher","DOI":"10.1016\/j.preteyeres.2012.11.001"},{"key":"2026082006251432000_19.8.e273165.4","first-page":"25","article-title":"Reverse amblyopia with atropine treatment","volume":"24","author":"Hainline","year":"2009","journal-title":"Binocul Vis Strabismus Q"},{"key":"2026082006251432000_19.8.e273165.5","doi-asserted-by":"crossref","first-page":"52","DOI":"10.3928\/01913913-20050101-07","article-title":"Severe amblyopia of the sound eye resulting from atropine therapy combined with optical penalization","volume":"42","author":"Morrison","year":"2005","journal-title":"J Pediatr Ophthalmol Strabismus"},{"key":"2026082006251432000_19.8.e273165.6","doi-asserted-by":"crossref","first-page":"148","DOI":"10.4103\/0974-620X.71897","article-title":"Refractory reverse amblyopia with atropine penalization","volume":"3","author":"Patil","year":"2010","journal-title":"Oman J Ophthalmol"},{"key":"2026082006251432000_19.8.e273165.7","doi-asserted-by":"crossref","first-page":"106","DOI":"10.1016\/j.jaapos.2019.12.001","article-title":"Severe reverse amblyopia with atropine penalization","volume":"24","author":"Elhusseiny","year":"2020","journal-title":"J AAPOS"},{"key":"2026082006251432000_19.8.e273165.8","doi-asserted-by":"publisher","DOI":"10.1016\/j.ophtha.2022.11.003"},{"key":"2026082006251432000_19.8.e273165.9","unstructured":"Spark Therapeutics, Inc . LUXTURNA (voretigene neparvovec-rzyl) intraocular suspension for subretinal injection: US prescribing information. Available: https:\/\/www.gene.com\/download\/pdf\/luxturna_prescribing.pdf [accessed 8 Feb 2026]"},{"key":"2026082006251432000_19.8.e273165.10","doi-asserted-by":"crossref","DOI":"10.3389\/fimmu.2013.00261","article-title":"Immunology of AAV-Mediated Gene Transfer in the Eye","volume":"4","author":"Willett","year":"2013","journal-title":"Front Immunol"},{"key":"2026082006251432000_19.8.e273165.11","doi-asserted-by":"crossref","DOI":"10.3390\/biom14010122","article-title":"Real-World Safety and Effectiveness of Voretigene Neparvovec: Results up to 2 Years from the Prospective, Registry-Based PERCEIVE Study","volume":"14","author":"Fischer","year":"2024","journal-title":"Biomolecules"},{"key":"2026082006251432000_19.8.e273165.12","doi-asserted-by":"crossref","first-page":"156","DOI":"10.1001\/jamaophthalmol.2025.5505","article-title":"Vision Outcomes in Children Treated With Voretigene Neparvovec for RPE65-Associated Retinopathy","volume":"144","author":"Thompson","year":"2026","journal-title":"JAMA Ophthalmol"}],"container-title":["BMJ Case Reports"],"original-title":[],"language":"en","link":[{"URL":"https:\/\/syndication.highwire.org\/content\/doi\/10.1136\/bcr-2026-273165","content-type":"unspecified","content-version":"vor","intended-application":"similarity-checking"}],"deposited":{"date-parts":[[2026,8,24]],"date-time":"2026-08-24T03:22:12Z","timestamp":1787541732000},"score":1,"resource":{"primary":{"URL":"https:\/\/casereports.bmj.com\/lookup\/doi\/10.1136\/bcr-2026-273165"}},"subtitle":[],"short-title":[],"issued":{"date-parts":[[2026,8]]},"references-count":12,"journal-issue":{"issue":"8","published-online":{"date-parts":[[2026,8,20]]},"published-print":{"date-parts":[[2026,8]]}},"alternative-id":["10.1136\/bcr-2026-273165"],"URL":"https:\/\/doi.org\/10.1136\/bcr-2026-273165","relation":{},"ISSN":["1757-790X"],"issn-type":[{"value":"1757-790X","type":"electronic"}],"subject":[],"published":{"date-parts":[[2026,8]]}}}