Nidha Shapoo, Abdul Rehman, Joseph Mattana, Wang Jen-Chin, Vladimir Gotlieb
Department of Medicine, New York Medical College, Metropolitan Hospital, Valhalla, USA.
Division of Hematology and Oncology, Brookdale University Hospital Medical Center, Brooklyn, NY, USA.
J Investig Med High Impact Case Rep. 2025 Jan-Dec;13:23247096251355151. doi: 10.1177/23247096251355151. Epub 2025 Jun 28.
Cemiplimab is a programmed death receptor-1 inhibitor that has been approved for the treatment of advanced cutaneous squamous cell carcinoma (CSCC). The immune-related adverse events most commonly reported with cemiplimab are thyroiditis, pneumonitis, and hepatitis. Ocular adverse events have been rarely reported. We present a rare case of Vogt-Koyanagi-Harada (VKH)-like syndrome in a patient with CSCC being treated with cemiplimab. A 53-year-old woman presented with bilateral progressive visual disturbances, eye pain, and headache for 4 weeks after 4 cycles of neo-adjuvant cemiplimab therapy for stage IV CSCC of the lower back. The detailed ophthalmologic evaluation established the diagnosis of VKH-like syndrome with panuveitis, subretinal fibrosis, and exudative retinal detachment. The patient received high-dose intravenous and topical steroids followed by a gradual taper of oral steroids over 6 months. Cemiplimab was discontinued. There was significant improvement in her symptoms with the resolution of subretinal fluid, choroidal elevations, and retinal detachment. VKH-like syndrome is an immune-mediated disorder that affects pigmented structures and is a significant cause of noninfectious uveitis. Drug-related uveitis has been seen more commonly with immune checkpoint inhibitors than with other drug classes and can occur anytime during the treatment. The mainstay of treatment of VKH-like syndrome is high-dose intravenous corticosteroids. In resistant cases, immunosuppressive therapies and biological agents are beneficial. This case highlights the importance of prompt recognition of VKH-like syndrome associated with cemiplimab, enabling timely intervention to prevent permanent vision loss.
西米普利单抗是一种程序性死亡受体-1抑制剂，已被批准用于治疗晚期皮肤鳞状细胞癌（CSCC）。西米普利单抗最常报告的免疫相关不良事件是甲状腺炎、肺炎和肝炎。眼部不良事件很少被报告。我们报告了一例在用西米普利单抗治疗的CSCC患者中出现的罕见的类Vogt-小柳原田（VKH）综合征病例。一名53岁女性在接受了4个周期的新辅助西米普利单抗治疗下背部IV期CSCC后4周，出现双侧进行性视力障碍、眼痛和头痛4周。详细的眼科评估确诊为类VKH综合征，伴有全葡萄膜炎、视网膜下纤维化和渗出性视网膜脱离。患者接受了大剂量静脉和局部类固醇治疗，随后在6个月内逐渐减少口服类固醇剂量。西米普利单抗停药。随着视网膜下液、脉络膜隆起和视网膜脱离的消退，她的症状有了显著改善。类VKH综合征是一种免疫介导的疾病，影响色素结构，是非感染性葡萄膜炎的重要原因。与免疫检查点抑制剂相比，药物相关性葡萄膜炎在其他药物类别中更为常见，并且可在治疗期间的任何时候发生。类VKH综合征的主要治疗方法是大剂量静脉注射皮质类固醇。在耐药病例中，免疫抑制疗法和生物制剂是有益的。本病例强调了及时识别与西米普利单抗相关的类VKH综合征的重要性，以便能够及时干预以防止永久性视力丧失。
