Tocilizumab for cystoid macular edema secondary to retinitis pigmentosa.

IF 2.9 Q1 OPHTHALMOLOGY
Stéphane Abramowicz, Prochore Kamgang Semeu, Isabelle Nubourgh, Laurence Postelmans, François Willermain
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Abstract

Purpose: To describe the effect of tocilizumab (TCZ) on cystoid macular edema (CME) and retinal vascular leakage (RVL) in retinitis pigmentosa (RP).

Methods: Retrospective case series.

Results: We present 2 cases of RP with marked inflammatory features in the form of CME and RVL. There was initial diagnostic uncertainty with posterior uveitis. Both patients were treated with corticosteroids, conventional disease-modifying antirheumatic drugs (cDMARDs), and biological DMARDs (bDMARDs) for the inflammatory features with partial and inconsistent treatment response. When treatment was switched to intravenous (IV) TCZ, dramatic reduction in CME and RVL were observed in both patients. Diagnosis of RP was eventually made based on findings of ancillary tests (macular spectral-domain optical coherence tomography, visual fields, full-field electroretinogram). Genetic testing led to a molecular diagnosis of EYS-related autosomal recessive RP in patient 1, while patient 2 had negative gene panel results.

Conclusions: IV TCZ can be an effective treatment option in RP-related CME and RVL. Whether this treatment strategy has an effect on prognosis remains to be established, but it is possible considering chronic CME-related retinal damage is a major driver of central vision loss in RP.

托西珠单抗治疗继发于视网膜色素变性的囊样黄斑水肿。
目的:描述托西珠单抗(TCZ)对视网膜色素变性(RP)患者囊样黄斑水肿(CME)和视网膜血管渗漏(RVL)的影响:方法:回顾性病例系列:结果:我们报告了两例具有明显炎症特征的视网膜色素变性病例,表现为CME和RVL。最初的诊断并不确定是后葡萄膜炎。两名患者均接受了皮质类固醇、传统的改善病情抗风湿药(cDMARDs)和生物DMARDs(bDMARDs)治疗,但治疗效果不佳且不一致。改用静脉注射(IV)TCZ治疗后,两名患者的CME和RVL均显著减少。根据辅助检查结果(黄斑光谱域光学相干断层扫描、视野、全场视网膜电图),最终确诊为 RP。基因检测结果显示,患者1的分子诊断为与EYS相关的常染色体隐性RP,而患者2的基因检测结果为阴性:结论:静脉滴注TCZ可以有效治疗RP相关的CME和RVL。这种治疗策略是否会对预后产生影响仍有待确定,但考虑到慢性CME相关视网膜损伤是RP中心视力丧失的主要原因,因此这种治疗策略是可能的。
本文章由计算机程序翻译,如有差异,请以英文原文为准。
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来源期刊
CiteScore
3.80
自引率
3.40%
发文量
39
审稿时长
13 weeks
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