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一名患有胸部软组织肉瘤的少女中PD-1抑制剂诱发全葡萄膜炎的诊断挑战——病例报告

Diagnostic Challenges in PD-1 Inhibitor Induced Panuveitis in a Teenage Girl with Chest Soft Tissue Sarcoma - A Case Report.

作者信息

Shen Yinchen, Gong Yuanyuan, Su Li

机构信息

Department of Ophthalmology, Shanghai General Hospital, School of Medicine, Shanghai Jiao Tong University, Shanghai, China.

出版信息

Ocul Immunol Inflamm. 2023 Dec;31(10):2065-2068. doi: 10.1080/09273948.2023.2233015. Epub 2023 Jul 13.

DOI:10.1080/09273948.2023.2233015
PMID:37442377
Abstract

PURPOSE

To report a case of programmed cell death receptor-1 (PD-1) inhibitor induced panuveitis.

METHOD

Observational case report of a 13-year-old Chinese girl presented as panuveitis. The clinical course, imaging performance, laboratory examination, differential diagnosis, treatment and prognosis were described.

RESULT

Patient presented with bilateral anterior granulomatous uveitis, vitritis, papillitis, and various creamy yellow nodular lesions in the mid-peripheral fundus. She had a history of biopsy proven alveolar soft tissue sarcoma on the chest wall and pulmonary metastasis, and a PD-1 inhibitor (sintilimab) was intravenously administered. Blood tests, magnetic resonance imaging of the cranium and the orbit, aqueous humor assay of inflammatory cytokines and microbial DNA were performed to distinguish infectious and non-infectious uveitis, choroidal metastases, and intravenous injection-related endophthalmitis. The oncologist evaluated that the sarcoma was stable and terminated sintilimab dosage. After sintilimab withdrawal, the blurred vision improved. Then, the patient received oral corticosteroids, resulted in resolution of the panuveitis. A diagnosis of PD-1 inhibitor induced panuveitis was made.

CONCLUSION

For patients taking PD-1 inhibitors, the major diagnostic challenge is to identify whether the cause of the uveitis is due to the antitumor treatment or not. It is suggested to be screened by eye care specialist and timely referral to uveitis specialist with any suspicion of intraocular inflammation for these patients.

摘要

目的

报告1例程序性细胞死亡受体1(PD-1)抑制剂诱发的全葡萄膜炎病例。

方法

对1例表现为全葡萄膜炎的13岁中国女孩进行观察性病例报告。描述其临床病程、影像学表现、实验室检查、鉴别诊断、治疗及预后。

结果

患者表现为双侧前部肉芽肿性葡萄膜炎、玻璃体炎、视乳头炎,以及眼底中周部多个奶油黄色结节状病变。她有胸壁活检证实的肺泡软组织肉瘤病史且伴有肺转移,曾静脉注射PD-1抑制剂(信迪利单抗)。进行了血液检查、头颅和眼眶磁共振成像、房水炎性细胞因子及微生物DNA检测,以鉴别感染性和非感染性葡萄膜炎、脉络膜转移瘤及静脉注射相关的眼内炎。肿瘤学家评估肉瘤病情稳定并停用信迪利单抗。停用信迪利单抗后,视力模糊症状改善。随后,患者接受口服糖皮质激素治疗,全葡萄膜炎消退。诊断为PD-1抑制剂诱发的全葡萄膜炎。

结论

对于服用PD-1抑制剂的患者,主要的诊断挑战在于确定葡萄膜炎的病因是否为抗肿瘤治疗所致。建议由眼科专科医生进行筛查,对于这些患者,一旦怀疑有眼内炎症应及时转诊至葡萄膜炎专科医生处。

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