{"title":"Levofloxacin-associated panuveitis with chorioretinal lesions.","authors":"Nicholas J Butler, Eric B Suhler","doi":"10.1001/archophthalmol.2012.2260","DOIUrl":null,"url":null,"abstract":"Comment. The first report of unilateral RPE dysgenesis was published in 2002. In this report, 3 young men and 1 woman aged 16 to 34 years were noted to have a round leopard-spot lesion contiguous with the optic nerve. In 2009, the typical characteristics of unilateral RPE dysgenesis were described in a set of 9 affected patients, 6 males and 3 females aged 14 to 42 years. The margin of the lesion is pathognomonic, with a scalloped reticular fringe of mild fibrosis and atrophy with inverted FAF imaging relative to fluorescein angiography, and is identical to the lesion in our patient. The differential diagnosis of this lesion includes acute zonal occult outer retinopathy. However, acute zonal occult outer retinopathy typically has a smooth curvilinear border on autofluorescence and the electroretinographic findings are abnormal. Alternative differential diagnoses include traumatic retinal pigment epitheliopathy and combined hamartoma of the retina and RPE. This case is unique in that it shows bilateral loss of the RPE in a multifocal, scattered, stellate fashion, best appreciated with FAF imaging. To our knowledge, this is the first reported case of unilateral RPE dysgenesis with abnormal findings on FAF imaging in the fellow eye. In the previously reported series of 9 patients by Cohen et al, only 3 cases were evaluated with FAF imaging and no evidence of bilateral disease was seen. However, as most patients in the case series did not have FAF imaging performed, some of these unilateral cases may have had subclinical bilateral disease. The etiology of this condition remains elusive. It may be an RPE dysgenesis or dystrophy, or it could reflect previous inflammatory, infectious, or autoimmune insult to the RPE. It is intuitive that a dysgenesis should ultimately be bilateral, and this is the first report to our knowledge illustrating this for RPE dysgenesis, which perhaps should no longer be termed unilateral.","PeriodicalId":8303,"journal":{"name":"Archives of ophthalmology","volume":null,"pages":null},"PeriodicalIF":0.0000,"publicationDate":"2012-10-01","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"https://sci-hub-pdf.com/10.1001/archophthalmol.2012.2260","citationCount":"9","resultStr":null,"platform":"Semanticscholar","paperid":null,"PeriodicalName":"Archives of ophthalmology","FirstCategoryId":"1085","ListUrlMain":"https://doi.org/10.1001/archophthalmol.2012.2260","RegionNum":0,"RegionCategory":null,"ArticlePicture":[],"TitleCN":null,"AbstractTextCN":null,"PMCID":null,"EPubDate":"","PubModel":"","JCR":"","JCRName":"","Score":null,"Total":0}
引用次数: 9
Abstract
Comment. The first report of unilateral RPE dysgenesis was published in 2002. In this report, 3 young men and 1 woman aged 16 to 34 years were noted to have a round leopard-spot lesion contiguous with the optic nerve. In 2009, the typical characteristics of unilateral RPE dysgenesis were described in a set of 9 affected patients, 6 males and 3 females aged 14 to 42 years. The margin of the lesion is pathognomonic, with a scalloped reticular fringe of mild fibrosis and atrophy with inverted FAF imaging relative to fluorescein angiography, and is identical to the lesion in our patient. The differential diagnosis of this lesion includes acute zonal occult outer retinopathy. However, acute zonal occult outer retinopathy typically has a smooth curvilinear border on autofluorescence and the electroretinographic findings are abnormal. Alternative differential diagnoses include traumatic retinal pigment epitheliopathy and combined hamartoma of the retina and RPE. This case is unique in that it shows bilateral loss of the RPE in a multifocal, scattered, stellate fashion, best appreciated with FAF imaging. To our knowledge, this is the first reported case of unilateral RPE dysgenesis with abnormal findings on FAF imaging in the fellow eye. In the previously reported series of 9 patients by Cohen et al, only 3 cases were evaluated with FAF imaging and no evidence of bilateral disease was seen. However, as most patients in the case series did not have FAF imaging performed, some of these unilateral cases may have had subclinical bilateral disease. The etiology of this condition remains elusive. It may be an RPE dysgenesis or dystrophy, or it could reflect previous inflammatory, infectious, or autoimmune insult to the RPE. It is intuitive that a dysgenesis should ultimately be bilateral, and this is the first report to our knowledge illustrating this for RPE dysgenesis, which perhaps should no longer be termed unilateral.