Long-Term Efficacy of Infliximab Therapy in Idiopathic Retinal vasculitis, Aneurysms and Neuroretinitis (IRVAN) Syndrome.

IF 2.6 4区 医学 Q2 OPHTHALMOLOGY
Rahaf Abdullah Mandura, Ghislain Opdenakker, Ahmed Abu El-Asrar
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Abstract

Purpose: Idiopathic retinal vasculitis, aneurysms, and neuroretinitis (IRVAN) syndrome is a rare inflammatory vision-threatening retinal disease of unknown origin. If untreated, it can lead to severe vision loss from exudative maculopathy and neovascular sequelae of retinal ischaemia. Detailed IRVAN case reporting may help to provide new insights.

Methods: Successful treatment of a pediatric patient with infliximab.

Results: A 12-year-old girl with IRVAN syndrome received immunosuppressive and laser photocoagulation therapy without improvements. A shift to treatment with infliximab not only reversed disease symptoms on short term, but also led to a complete and long-term cure of the disease. The patient was followed-up for a period of 3 years and infliximab resulted in complete resolution of retinal and optic nerve head aneurysmal dilations, retinal vasculitis and optic disc swelling, and complete resorption of retinal exudation.

Conclusion: Infliximab is safe and effective and should be considered as a primary first-line therapy for IRVAN syndrome.

英夫利昔单抗治疗特发性视网膜血管炎、动脉瘤和神经视网膜炎(IRVAN)综合征的长期疗效。
目的:特发性视网膜血管炎、动脉瘤和神经视网膜炎(IRVAN)综合征是一种罕见的炎症性视网膜疾病,病因不明。如果不治疗,它会导致渗出性黄斑病变和视网膜缺血的新血管后遗症导致严重的视力丧失。详细的IRVAN病例报告可能有助于提供新的见解。方法:英夫利昔单抗成功治疗1例儿科患者。结果:1例12岁IRVAN综合征女孩接受免疫抑制和激光光凝治疗,无明显改善。改用英夫利昔单抗治疗不仅在短期内逆转了疾病症状,而且还导致了疾病的完全和长期治愈。患者随访3年,英夫利昔单抗使视网膜和视神经头动脉瘤扩张、视网膜血管炎和视盘肿胀完全消退,视网膜渗出物完全吸收。结论:英夫利昔单抗安全有效,可作为治疗IRVAN综合征的一线首选药物。
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
6.20
自引率
15.20%
发文量
285
审稿时长
6-12 weeks
期刊介绍: Ocular Immunology & Inflammation ranks 18 out of 59 in the Ophthalmology Category.Ocular Immunology and Inflammation is a peer-reviewed, scientific publication that welcomes the submission of original, previously unpublished manuscripts directed to ophthalmologists and vision scientists. Published bimonthly, the journal provides an international medium for basic and clinical research reports on the ocular inflammatory response and its control by the immune system. The journal publishes original research papers, case reports, reviews, letters to the editor, meeting abstracts, and invited editorials.
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