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本文引用的文献

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ISCEV extended protocol for the photopic On-Off ERG.国际临床视觉电生理学会(ISCEV)明视开-关视网膜电图扩展协议。
Doc Ophthalmol. 2018 Jun;136(3):199-206. doi: 10.1007/s10633-018-9645-y. Epub 2018 Jun 22.
2
Seeing is believing: A review of apheresis therapy in the treatment of ophthalmologic disease.眼见为实:单采疗法治疗眼科疾病的综述
J Clin Apher. 2018 Jun;33(3):380-392. doi: 10.1002/jca.21607. Epub 2017 Nov 18.
3
ISCEV Standard for full-field clinical electroretinography (2015 update).国际临床视觉电生理学会全视野临床视网膜电图标准(2015年更新版)
Doc Ophthalmol. 2015 Feb;130(1):1-12. doi: 10.1007/s10633-014-9473-7. Epub 2014 Dec 14.
4
Congenital stationary night blindness: an analysis and update of genotype-phenotype correlations and pathogenic mechanisms.先天性静止性夜盲症:基因型-表型相关性和发病机制的分析与更新。
Prog Retin Eye Res. 2015 Mar;45:58-110. doi: 10.1016/j.preteyeres.2014.09.001. Epub 2014 Oct 13.
5
Local therapies for inflammatory eye disease in translation: past, present and future.炎症性眼病的局部治疗转化:过去、现在和未来。
BMC Ophthalmol. 2013 Aug 6;13(1):39. doi: 10.1186/1471-2415-13-39.
6
Delayed presentation of melanoma-associated retinopathy and subsequent resolution with cytoreduction surgery.黑色素瘤相关性视网膜病变的延迟表现及随后通过减瘤手术缓解
Doc Ophthalmol. 2013 Oct;127(2):165-71. doi: 10.1007/s10633-013-9398-6. Epub 2013 Jun 22.
7
ISCEV standard for clinical pattern electroretinography (PERG): 2012 update.国际临床视觉电生理学会(ISCEV)临床图形视网膜电图(PERG)标准:2012年更新版
Doc Ophthalmol. 2013 Feb;126(1):1-7. doi: 10.1007/s10633-012-9353-y. Epub 2012 Oct 17.
8
Melanoma associated retinopathy and how to understand the electroretinogram.黑色素瘤相关性视网膜病变以及如何解读视网膜电图
Pract Neurol. 2011 Aug;11(4):234-9. doi: 10.1136/practneurol-2011-000061.
9
Autoantibodies in melanoma-associated retinopathy target TRPM1 cation channels of retinal ON bipolar cells.黑色素瘤相关性视网膜病变中的自身抗体靶向视网膜 ON 双极细胞的 TRPM1 阳离子通道。
J Neurosci. 2011 Mar 16;31(11):3962-7. doi: 10.1523/JNEUROSCI.6007-10.2011.
10
Improvement in visual fields in a patient with melanoma-associated retinopathy treated with intravenous immunoglobulin.接受静脉注射免疫球蛋白治疗的黑色素瘤相关性视网膜病变患者视野的改善
J Neuroophthalmol. 2008 Mar;28(1):23-6. doi: 10.1097/WNO.0b013e31816754c4.

在未进行全身免疫抑制治疗的情况下,用于治疗黑色素瘤相关性视网膜病变的缓释型眼内氟轻松植入物的相关结果。

Outcomes Associated With Sustained-Release Intraocular Fluocinolone Implants in a Case of Melanoma-Associated Retinopathy Treated Without Systemic Immunosuppression.

机构信息

Royal Surrey County Hospital, Guildford, United Kingdom.

Moorfields Eye Hospital, London, United Kingdom.

出版信息

JAMA Ophthalmol. 2019 May 1;137(5):564-567. doi: 10.1001/jamaophthalmol.2019.0284.

DOI:10.1001/jamaophthalmol.2019.0284
PMID:30896772
原文链接:https://pmc.ncbi.nlm.nih.gov/articles/PMC6512254/
Abstract

IMPORTANCE

Melanoma-associated retinopathy (MAR) is a paraneoplastic syndrome in which antiretinal antibodies crossreact with retinal ON-bipolar cells, resulting in night blindness and progressive visual field loss. Current therapeutic options include cytoreductive surgery in combination with immunoglobulin, corticosteroids, or plasmapheresis, but their effectiveness is limited and may be contraindicated, given the possible protective role of circulating autoantibodies against metastatic spread. We report 3-year follow-up of the first case (to our knowledge) of MAR treated with intravitreal long-acting steroid implants.

OBJECTIVE

To report on a patient with MAR who was treated with intravitreal fluocinolone acetonide implants in the absence of systemic immunosuppression.

DESIGN, SETTING, AND PARTICIPANTS: This is a 3-year follow-up of a 73-year-old woman with a history of surgical excision of a malignant melanoma of the left pinna who presented with visual symptoms of shimmering and nyctalopia. Fundus examination, fundus autofluorescence, and optical coherence tomography were normal, with no evidence of cystoid macular edema. Automated perimetry showed a reduction in visual field and full-field electroretinography (ERG) demonstrated findings consistent with generalized ON-bipolar cell dysfunction, typical of MAR. The patient was treated with bilateral fluocinolone acetonide intravitreal implants.

MAIN OUTCOMES AND MEASURES

Visual acuity, visual field, and electroretinography testing for 3 years after treatment.

RESULTS

Visual fields improved in this 73-year-old patient from 20/30 (Snellen measured as 6/9) OD and 20/16 (6/5) OS at baseline to 20/20 OU within 1 week of treatment. Detailed electroretinography monitoring indicated characteristic abnormalities that partly resolved after treatment, consistent with improved inner retinal ON-bipolar cell function. Bilateral cataracts developed approximately 2 years after injection; cataract surgery was performed uneventfully. At 3 years posttreatment, the patient remained visually stable and in systemic disease remission, with best-corrected visual acuity remaining at 20/20 OU.

CONCLUSIONS AND RELEVANCE

We report what is, to our knowledge, the first case of MAR treated with intravitreal slow-release corticosteroid implants, which shows improvements in visual symptoms, visual fields, and retinal function. Sustained-release intraocular steroid implants may offer an effective and safe alternative to systemic immunosuppression in MAR, although results from 1 case should be generalized with abundant caution.

摘要

重要性

黑色素瘤相关视网膜病变(MAR)是一种副肿瘤综合征,其中抗视网膜抗体与视网膜 ON-双极细胞发生交叉反应,导致夜盲和进行性视野丧失。目前的治疗选择包括细胞减少手术联合免疫球蛋白、皮质类固醇或血浆置换,但由于循环自身抗体可能对转移性扩散有保护作用,其疗效有限,且可能存在禁忌。我们报告首例(据我们所知)MAR 患者接受玻璃体内长效类固醇植入物治疗的 3 年随访结果。

目的

报告一例未经全身免疫抑制治疗的 MAR 患者接受玻璃体内氟轻松醋酸酯植入物治疗的情况。

设计、地点和参与者:这是一名 73 岁女性的 3 年随访结果,该女性曾因左耳恶性黑色素瘤接受手术切除,出现闪烁和夜盲等视觉症状。眼底检查、眼底自发荧光和光学相干断层扫描均正常,无黄斑囊样水肿证据。自动视野检查显示视野缩小,全视野视网膜电图(ERG)显示符合广义 ON-双极细胞功能障碍的结果,这是 MAR 的典型表现。该患者接受了双侧氟轻松醋酸酯玻璃体内植入物治疗。

主要结局和测量指标

治疗后 3 年的视力、视野和视网膜电图检查结果。

结果

这名 73 岁患者的视野从基线时的右眼 20/30(Snellen 测量值为 6/9)和左眼 20/16(6/5)改善到治疗后 1 周内的双眼 20/20。详细的视网膜电图监测显示出特征性异常,在治疗后部分得到解决,这与改善的内视网膜 ON-双极细胞功能一致。大约在注射后 2 年出现双侧白内障;白内障手术顺利进行。治疗后 3 年,患者视力稳定且全身疾病处于缓解期,最佳矫正视力仍为双眼 20/20。

结论和相关性

我们报告了首例接受玻璃体内缓慢释放皮质类固醇植入物治疗的 MAR 病例,该病例显示视觉症状、视野和视网膜功能均有所改善。缓释眼内类固醇植入物可能为 MAR 提供一种有效且安全的替代全身免疫抑制的方法,尽管来自 1 例的结果应谨慎推广。