Chiba-Mayumi Miyako, Hirakata Toshiaki, Yamaguchi Masahiro, Murakami Akira
Department of Ophthalmology, Juntendo University Faculty of Medicine, Tokyo, Japan.
Am J Ophthalmol Case Rep. 2021 Dec 14;25:101244. doi: 10.1016/j.ajoc.2021.101244. eCollection 2022 Mar.
To report the efficacy of anti-tumor necrosis factor α (anti-TNFα) on autoimmune-mediated macular cone dysfunction in a Japanese woman with ulcerative colitis (UC).
A 41-year-old woman presented with bilateral visual acuity loss and photophobia. She suffered from UC, and had been treated with prednisolone and 5-aminosalicylate since age 37. Although fundus photographs and optic coherence tomography images were unremarkable, electroretinograms (ERGs) were abnormal. A full-field electroretinogram (full-field ERG) revealed mildly decreased cone responses and oscillatory potential responses bilaterally. Importantly, focal-macular ERG (fmERG) and a multifocal electroretinogram (mfERG) revealed severe macular cone dysfunction in both eyes. Infliximab, a chimeric monoclonal anti-TNFα antibody, was administrated to treat recurrent abdominal symptoms and continued at 8-week intervals. Almost 6 months after infliximab therapy, the mfERG response (especially in the central retina), the fmERG response, and visual acuity recovered bilaterally. Abdominal symptoms also improved after infliximab therapy.
Bilateral cone dysfunction with normal fundus were observed in a UC patient, resulting in loss of visual acuity and photophobia. This retinopathy may have been caused by an autoimmune mechanism, such as an autoimmune retinopathy or acute zonal occult outer retinopathy, which is most identifiable by ERG changes. This is the first report demonstrating the efficacy of infliximab in autoimmune retinal dysfunction.
报告抗肿瘤坏死因子α（抗TNFα）对一名患有溃疡性结肠炎（UC）的日本女性自身免疫介导的黄斑视锥细胞功能障碍的疗效。
一名41岁女性出现双侧视力丧失和畏光症状。她患有UC，自37岁起接受泼尼松龙和5-氨基水杨酸治疗。尽管眼底照片和光学相干断层扫描图像无明显异常，但视网膜电图（ERG）异常。全视野视网膜电图（全视野ERG）显示双侧视锥细胞反应和振荡电位反应轻度降低。重要的是，黄斑局部视网膜电图（fmERG）和多焦视网膜电图（mfERG）显示双眼存在严重的黄斑视锥细胞功能障碍。英夫利昔单抗，一种嵌合单克隆抗TNFα抗体，用于治疗复发性腹部症状，并以8周的间隔持续给药。英夫利昔单抗治疗近6个月后，双侧mfERG反应（尤其是在视网膜中央）、fmERG反应和视力均恢复。英夫利昔单抗治疗后腹部症状也有所改善。
在一名UC患者中观察到双侧视锥细胞功能障碍且眼底正常，导致视力丧失和畏光。这种视网膜病变可能由自身免疫机制引起，如自身免疫性视网膜病变或急性区域性隐匿性外层视网膜病变，通过ERG变化最易识别。这是首次报告证明英夫利昔单抗在自身免疫性视网膜功能障碍中的疗效。
