Choroidal osteoma: Spontaneous Resolution of Subretinal Detachment Unresponsive to Anti-VEGF Agent and multimodal imaging.

Hyuk Jun Lee, Se Joon Woo
{"title":"Choroidal osteoma: Spontaneous Resolution of Subretinal Detachment Unresponsive to Anti-VEGF Agent and multimodal imaging.","authors":"Hyuk Jun Lee, Se Joon Woo","doi":"10.3341/kjo.2022.0113","DOIUrl":null,"url":null,"abstract":"Dear Editor, Choroidal osteoma (CO) is a rare benign ossifying choroidal tumor that typically affects young female patients [1]. While there is no standard treatment for CO, patients with complications, such as choroidal neovascularization (CNV) and serous retinal detachment (SRD), require treatment [2]. Several reports have discussed the role of antivascular endothelial growth factor (anti-VEGF) agents in the treatment of SRD with or without CNV, associated with CO [2,3]. We describe the spontaneous resolution of SRD in a patient with CO, that was unresponsive to intravitreal anti-VEGF treatment. The patient provided informed consent for publication of the research details and clinical images. A healthy 9-year-old female patient presented with blurred vision in the left eye. The best-corrected visual acuity (BCVA) was 20 / 25 in the left eye. Funduscopic examination of the left eye revealed a juxtafoveal yellow-orange subretinal lesion with distinct borders (Fig. 1A). Fundus f luorescein angiography (FFA) revealed an early patchy hyperf luorescent choroidal filling pattern, dye staining in the area of retinal pigment epithelium (RPE) decompensation overlying the CO, pooling hyperfluorescence in the late phase, and no evidence of CNV (Fig. 1B, 1C). En face optical coherence tomography (OCT) showed the tumor border (Fig 1D, 1E). OCT angiography demonstrated the paucivascular tumor at the level of choroid, and absence of CNV (Fig. 1F). OCT showed SRD, a disrupted photoreceptor layer, and a hyperreflective mass posterior to the RPE (Fig. 1G). Therefore, the patient was diagnosed with CO with subretinal fluid in the absence of CNV. To resolve the SRD, an intravitreal injection of bevacizumab (1.25 mg/0.05 mL, Avastin, Genentech) was administered to her left eye. One month after bevacizumab treatment, her BCVA decreased to 20 / 100, and OCT showed an increased SRD and intraretinal fluid (Fig. 1H). Because the lesion was unresponsive to bevacizumab, the patient was closely monitored without additional treatment. After 4 months, the SRD decreased spontaneously, leading to an improved BCVA of 20 / 50. After 1 year, complete resolution of SRD and intraretinal fluid was confirmed on OCT (Fig. 1I). However, disruption of the photoreceptor layer was detected, and the final BCVA remained unchanged at 20 / 50. SRD, associated with CO, frequently occurs in the absence of CNV. It possibly resulted from multiple pinpoint RPE leakages over the osteoma noted on FFA [4]. Alternatively, the gradual atrophy of the RPE and Bruch’s membrane likely decreased the capacity of the RPE to remove subretinal fluid from the disrupted outer blood-retinal barrier [3]. While it has been established to treat SRD with CNV, the significance of anti-VEGF in treating SRD without CNV remains controversial. Song et al. [3] reported that 83% of patients with CO and SRD did not present with signs of CNV. Following bevacizumab treatment, all patients showed a substantial decrease in SRD based on OCT, and 80% of the patients experienced improved visual acuity. Korean J Ophthalmol 2023;37(1):82-84 https://doi.org/10.3341/kjo.2022.0113","PeriodicalId":17883,"journal":{"name":"Korean Journal of Ophthalmology : KJO","volume":" ","pages":""},"PeriodicalIF":0.0000,"publicationDate":"2022-10-25","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"","citationCount":"1","resultStr":null,"platform":"Semanticscholar","paperid":null,"PeriodicalName":"Korean Journal of Ophthalmology : KJO","FirstCategoryId":"1085","ListUrlMain":"https://doi.org/10.3341/kjo.2022.0113","RegionNum":0,"RegionCategory":null,"ArticlePicture":[],"TitleCN":null,"AbstractTextCN":null,"PMCID":null,"EPubDate":"","PubModel":"","JCR":"Q3","JCRName":"Medicine","Score":null,"Total":0}
引用次数: 1

Abstract

Dear Editor, Choroidal osteoma (CO) is a rare benign ossifying choroidal tumor that typically affects young female patients [1]. While there is no standard treatment for CO, patients with complications, such as choroidal neovascularization (CNV) and serous retinal detachment (SRD), require treatment [2]. Several reports have discussed the role of antivascular endothelial growth factor (anti-VEGF) agents in the treatment of SRD with or without CNV, associated with CO [2,3]. We describe the spontaneous resolution of SRD in a patient with CO, that was unresponsive to intravitreal anti-VEGF treatment. The patient provided informed consent for publication of the research details and clinical images. A healthy 9-year-old female patient presented with blurred vision in the left eye. The best-corrected visual acuity (BCVA) was 20 / 25 in the left eye. Funduscopic examination of the left eye revealed a juxtafoveal yellow-orange subretinal lesion with distinct borders (Fig. 1A). Fundus f luorescein angiography (FFA) revealed an early patchy hyperf luorescent choroidal filling pattern, dye staining in the area of retinal pigment epithelium (RPE) decompensation overlying the CO, pooling hyperfluorescence in the late phase, and no evidence of CNV (Fig. 1B, 1C). En face optical coherence tomography (OCT) showed the tumor border (Fig 1D, 1E). OCT angiography demonstrated the paucivascular tumor at the level of choroid, and absence of CNV (Fig. 1F). OCT showed SRD, a disrupted photoreceptor layer, and a hyperreflective mass posterior to the RPE (Fig. 1G). Therefore, the patient was diagnosed with CO with subretinal fluid in the absence of CNV. To resolve the SRD, an intravitreal injection of bevacizumab (1.25 mg/0.05 mL, Avastin, Genentech) was administered to her left eye. One month after bevacizumab treatment, her BCVA decreased to 20 / 100, and OCT showed an increased SRD and intraretinal fluid (Fig. 1H). Because the lesion was unresponsive to bevacizumab, the patient was closely monitored without additional treatment. After 4 months, the SRD decreased spontaneously, leading to an improved BCVA of 20 / 50. After 1 year, complete resolution of SRD and intraretinal fluid was confirmed on OCT (Fig. 1I). However, disruption of the photoreceptor layer was detected, and the final BCVA remained unchanged at 20 / 50. SRD, associated with CO, frequently occurs in the absence of CNV. It possibly resulted from multiple pinpoint RPE leakages over the osteoma noted on FFA [4]. Alternatively, the gradual atrophy of the RPE and Bruch’s membrane likely decreased the capacity of the RPE to remove subretinal fluid from the disrupted outer blood-retinal barrier [3]. While it has been established to treat SRD with CNV, the significance of anti-VEGF in treating SRD without CNV remains controversial. Song et al. [3] reported that 83% of patients with CO and SRD did not present with signs of CNV. Following bevacizumab treatment, all patients showed a substantial decrease in SRD based on OCT, and 80% of the patients experienced improved visual acuity. Korean J Ophthalmol 2023;37(1):82-84 https://doi.org/10.3341/kjo.2022.0113
查看原文
分享 分享
微信好友 朋友圈 QQ好友 复制链接
本刊更多论文
脉络膜骨瘤:对抗vegf药物无反应的视网膜下脱离的自发消退和多模态成像。
本文章由计算机程序翻译,如有差异,请以英文原文为准。
求助全文
约1分钟内获得全文 去求助
来源期刊
Korean Journal of Ophthalmology : KJO
Korean Journal of Ophthalmology : KJO Medicine-Ophthalmology
CiteScore
2.40
自引率
0.00%
发文量
84
期刊最新文献
Restoration of Corneal Transparency in a Patient with Corneal Scarring Using Mesenchyme Stem Cells: A Case Report. One-Year Outcomes of Ab Externo XEN45 Gel Stent Implantation with an Open Conjunctiva Approach in Patients with Open-Angle Glaucoma. Comparison of Accuracy of Six Modern Intraocular Lens Power Calculation Formulas. Clinical Outcomes of Descemet Membrane Endothelial Keratoplasty Using a Preloaded Imported Graft. Short-term Efficacy and Safety of Intravitreal Brolucizumab Injection for Treatment-Naive Exudate Age-related Macular Degeneration: A Multicenter Study.
×
引用
GB/T 7714-2015
复制
MLA
复制
APA
复制
导出至
BibTeX EndNote RefMan NoteFirst NoteExpress
×
×
提示
您的信息不完整,为了账户安全,请先补充。
现在去补充
×
提示
您因"违规操作"
具体请查看互助需知
我知道了
×
提示
现在去查看 取消
×
提示
确定
0
微信
客服QQ
Book学术公众号 扫码关注我们
反馈
×
意见反馈
请填写您的意见或建议
请填写您的手机或邮箱
已复制链接
已复制链接
快去分享给好友吧!
我知道了
×
扫码分享
扫码分享
Book学术官方微信
Book学术文献互助
Book学术文献互助群
群 号:481959085
Book学术
文献互助 智能选刊 最新文献 互助须知 联系我们:info@booksci.cn
Book学术提供免费学术资源搜索服务,方便国内外学者检索中英文文献。致力于提供最便捷和优质的服务体验。
Copyright © 2023 Book学术 All rights reserved.
ghs 京公网安备 11010802042870号 京ICP备2023020795号-1