Mauricio Arango-Hurtado, Sara Turizo-Mejía, Luis M Jaramillo-Mayo, Karol Quintero-Lizcano
{"title":"先前未报道的女性x连锁视网膜色素变性患者的RPGR基因变异。","authors":"Mauricio Arango-Hurtado, Sara Turizo-Mejía, Luis M Jaramillo-Mayo, Karol Quintero-Lizcano","doi":"10.5693/djo.02.2024.06.004","DOIUrl":null,"url":null,"abstract":"<p><p>We present the case of a 40-year-old woman with a history of high myopia and nyctalopia. Her best-corrected visual acuity was 20/80 in the right eye and 20/100 in the left eye. Fundus examination revealed generalized vascular attenuation, optic nerve pallor, and bone spicule pigmentation. Fundus autofluorescence in both eyes showed a Robson-Holder ring in the macula and multiple hypoautofluorescent lesions in the peripheral retina. Macular optical coherence tomography scans revealed a generalized thinning of retinal layers, with atrophy of the outer retinal layers. 10-2 visual fields revealed a small island of central vision in both eyes, and full field electroretinogram showed absence of scotopic and photopic responses. Genetic studies documented a rare variant in the <i>RPGR</i> gene (c.1991C>G p.(Ser664*)). Findings compatible with retinitis pigmentosa in our patient suggests that this mutation is pathogenic. Further study is required to confirm this hypothesis.</p>","PeriodicalId":38112,"journal":{"name":"Digital journal of ophthalmology : DJO","volume":"31 1","pages":"20-24"},"PeriodicalIF":0.0000,"publicationDate":"2025-02-22","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"https://www.ncbi.nlm.nih.gov/pmc/articles/PMC11977291/pdf/","citationCount":"0","resultStr":"{\"title\":\"A previously unreported <i>RPGR</i> gene variant in a female patient with X-linked retinitis pigmentosa.\",\"authors\":\"Mauricio Arango-Hurtado, Sara Turizo-Mejía, Luis M Jaramillo-Mayo, Karol Quintero-Lizcano\",\"doi\":\"10.5693/djo.02.2024.06.004\",\"DOIUrl\":null,\"url\":null,\"abstract\":\"<p><p>We present the case of a 40-year-old woman with a history of high myopia and nyctalopia. Her best-corrected visual acuity was 20/80 in the right eye and 20/100 in the left eye. Fundus examination revealed generalized vascular attenuation, optic nerve pallor, and bone spicule pigmentation. Fundus autofluorescence in both eyes showed a Robson-Holder ring in the macula and multiple hypoautofluorescent lesions in the peripheral retina. Macular optical coherence tomography scans revealed a generalized thinning of retinal layers, with atrophy of the outer retinal layers. 10-2 visual fields revealed a small island of central vision in both eyes, and full field electroretinogram showed absence of scotopic and photopic responses. Genetic studies documented a rare variant in the <i>RPGR</i> gene (c.1991C>G p.(Ser664*)). Findings compatible with retinitis pigmentosa in our patient suggests that this mutation is pathogenic. Further study is required to confirm this hypothesis.</p>\",\"PeriodicalId\":38112,\"journal\":{\"name\":\"Digital journal of ophthalmology : DJO\",\"volume\":\"31 1\",\"pages\":\"20-24\"},\"PeriodicalIF\":0.0000,\"publicationDate\":\"2025-02-22\",\"publicationTypes\":\"Journal Article\",\"fieldsOfStudy\":null,\"isOpenAccess\":false,\"openAccessPdf\":\"https://www.ncbi.nlm.nih.gov/pmc/articles/PMC11977291/pdf/\",\"citationCount\":\"0\",\"resultStr\":null,\"platform\":\"Semanticscholar\",\"paperid\":null,\"PeriodicalName\":\"Digital journal of ophthalmology : DJO\",\"FirstCategoryId\":\"1085\",\"ListUrlMain\":\"https://doi.org/10.5693/djo.02.2024.06.004\",\"RegionNum\":0,\"RegionCategory\":null,\"ArticlePicture\":[],\"TitleCN\":null,\"AbstractTextCN\":null,\"PMCID\":null,\"EPubDate\":\"2025/1/1 0:00:00\",\"PubModel\":\"eCollection\",\"JCR\":\"Q3\",\"JCRName\":\"Medicine\",\"Score\":null,\"Total\":0}","platform":"Semanticscholar","paperid":null,"PeriodicalName":"Digital journal of ophthalmology : DJO","FirstCategoryId":"1085","ListUrlMain":"https://doi.org/10.5693/djo.02.2024.06.004","RegionNum":0,"RegionCategory":null,"ArticlePicture":[],"TitleCN":null,"AbstractTextCN":null,"PMCID":null,"EPubDate":"2025/1/1 0:00:00","PubModel":"eCollection","JCR":"Q3","JCRName":"Medicine","Score":null,"Total":0}
A previously unreported RPGR gene variant in a female patient with X-linked retinitis pigmentosa.
We present the case of a 40-year-old woman with a history of high myopia and nyctalopia. Her best-corrected visual acuity was 20/80 in the right eye and 20/100 in the left eye. Fundus examination revealed generalized vascular attenuation, optic nerve pallor, and bone spicule pigmentation. Fundus autofluorescence in both eyes showed a Robson-Holder ring in the macula and multiple hypoautofluorescent lesions in the peripheral retina. Macular optical coherence tomography scans revealed a generalized thinning of retinal layers, with atrophy of the outer retinal layers. 10-2 visual fields revealed a small island of central vision in both eyes, and full field electroretinogram showed absence of scotopic and photopic responses. Genetic studies documented a rare variant in the RPGR gene (c.1991C>G p.(Ser664*)). Findings compatible with retinitis pigmentosa in our patient suggests that this mutation is pathogenic. Further study is required to confirm this hypothesis.