Rao Arthi, Hwang Jodi, Wen Joyce, Edminster Sarah, Sibug-Saber Maria, Rao Narsing, Toy Brian C
Roski Eye Institute, Keck School of Medicine, University of Southern California, Los Angeles, CA, USA.
Am J Ophthalmol Case Rep. 2024 Nov 5;36:102220. doi: 10.1016/j.ajoc.2024.102220. eCollection 2024 Dec.
This case describes the unique course and management of a patient with progressive, refractory multi-system sarcoidosis that initially presented with ocular and dermatologic findings.
A 47-year-old male presented with acute anterior uveitis and was found to have simultaneous inflammation of his skin at a tattoo site. Diagnosis of ocular sarcoidosis was confirmed through skin biopsy. Treatment with prednisone and adalimumab was initiated. Despite systemic immunosuppression and control of systemic inflammation, the patient had refractory ocular disease and developed bilateral disc edema and peripapillary choroidal neovascular membranes with subretinal fluid, so oral methotrexate and as-needed intravitreal bevacizumab injections were added. However, the patient manifested hepatic and worsening pulmonary involvement, prompting discontinuation of both methotrexate and adalimumab. Biopsy of the liver later confirmed hepatic sarcoidosis over drug-induced liver injury. Out of precaution for methotrexate-associated hepatic toxicity, mycophenolate mofetil was initiated instead, which then led to resolution of the subretinal fluid and disease quiescence.
This case offers insight into the varying presentation and recalcitrant course of sarcoidosis. It documents a clinicopathologic case with unique initial presentation with biopsies of multiple organs, escalation of therapy with several immunomodulatory agents, and multidisciplinary collaboration to achieve ocular and systemic quiescence.
本病例描述了一名进行性、难治性多系统结节病患者的独特病程及治疗情况,该患者最初表现为眼部和皮肤症状。
一名47岁男性因急性前葡萄膜炎就诊,同时发现其纹身部位皮肤有炎症。通过皮肤活检确诊为眼部结节病。开始使用泼尼松和阿达木单抗进行治疗。尽管进行了全身免疫抑制并控制了全身炎症,但患者的眼部疾病仍难以治愈,并出现双侧视盘水肿和视乳头周围脉络膜新生血管膜伴视网膜下液,因此加用了口服甲氨蝶呤和必要时的玻璃体内注射贝伐单抗。然而,患者出现肝脏受累且肺部病情恶化,促使停用甲氨蝶呤和阿达木单抗。后来肝脏活检证实为结节病而非药物性肝损伤。出于对甲氨蝶呤相关肝毒性的预防,改用霉酚酸酯,随后视网膜下液消退,病情静止。
本病例有助于深入了解结节病的不同表现和顽固病程。它记录了一个临床病理病例,具有独特的初始表现,包括多个器官的活检、多种免疫调节药物治疗的升级以及多学科协作以实现眼部和全身病情静止。