Department of Ophthalmology, Miguel Servet University Hospital, Zaragoza, Spain.
Miguel Servet Ophthalmology Research Group (GIMSO), Aragon Health Research Institute (IIS Aragón), Zaragoza, Spain.
Retin Cases Brief Rep. 2024 May 1;18(3):387-392. doi: 10.1097/ICB.0000000000001389.
The aim of this report is to describe the resolution of refractory cystoid macular edema (CME) associated to retinitis pigmentosa (RP) with IV tocilizumab in three patients.
Retrospective study of a series of consecutive cases of patients treated with off-label IV tocilizumab (anti IL6) for CME refractory to acetazolamide 250 mg for 3 months. Patients were diagnosed with RP by fundus appearance, electrophysiology, visual fields, and genetic testing. A complete ophthalmic examination including spectral-domain optical coherence tomography was performed.
Three patients with RP and CME refractory to acetazolamide 250 mg for 3 months were treated with monthly IV tocilizumab for at least six months.All patients resolved CME and improved visual acuity after the third month of IV tocilizumab, resolving systemic and ocular adverse events related to previous treatments for CME. Tocilizumab was well tolerated with no other adverse events.
CME causes visual impairment in RP, but current treatments are usually deficient. Tocilizumab has been successfully used as treatment for refractory CME in uveitis, retinal dystrophies, and autoimmune retinopathies. This article reports, for the first time, the long-term resolution of refractory CME in RP with IV tocilizumab.
本报告旨在描述三例因对乙酰唑胺（250mg）治疗 3 个月仍无法缓解的难治性囊样黄斑水肿（CME）而使用 IV 托珠单抗治疗的色素性视网膜炎（RP）患者的 CME 缓解情况。
对使用 IV 托珠单抗（抗 IL6）治疗的难治性 CME 患者进行回顾性病例系列研究，这些患者之前接受了为期 3 个月的 250mg 乙酰唑胺治疗但未缓解。通过眼底表现、电生理学、视野和基因检测诊断为 RP。进行了全面的眼科检查，包括频域光相干断层扫描。
三例因对乙酰唑胺（250mg）治疗 3 个月仍无法缓解的难治性 CME 的 RP 患者，每月接受 IV 托珠单抗治疗至少 6 个月。所有患者在接受 IV 托珠单抗治疗第三个月后均缓解了 CME 并改善了视力，同时缓解了与之前治疗 CME 相关的全身和眼部不良事件。托珠单抗耐受性良好，无其他不良反应。
CME 会导致 RP 患者视力受损，但目前的治疗方法通常效果不佳。托珠单抗已成功用于治疗葡萄膜炎、视网膜营养不良和自身免疫性视网膜病变等疾病的难治性 CME。本文首次报道了使用 IV 托珠单抗治疗 RP 患者的难治性 CME 长期缓解情况。
