Idiopathic Multicentric Castleman's Disease Presenting with Chorioretinal Undulation: A Case Report.

IF 2 4区医学 Q2 OPHTHALMOLOGY Ocular Immunology and Inflammation Pub Date : 2025-07-01 Epub Date: 2025-03-18 DOI:10.1080/09273948.2025.2453883

Suowang Zhou, Liang Wang, Xin Li, Xiaona Zuo, Aifeng Wu, Xiaoyan Peng

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Abstract

Purpose: To describe a case of idiopathic multicentric Castleman's disease (iMCD) with initial ocular involvement, presenting with chorioretinal undulation.

Methods: Observational case report of iMCD patient with ocular manifestation on fundus photography, auto-fluorescein and swept-source optical coherence tomography (SS-OCT), initially and after treatment.

Results: A 75-year-old patient with painless vision loss for 2 years right eye (RE) and 3-month left eye (LE). Fundus examination revealed several yellow-white lesions along with the inferior arcade, with extensive pigment disturbance of the right eye and exudative retinal detachment of the left eye. SS-OCT showed choroid and retinal pigment epithelium (RPE) undulations, choroid thickening and hyperreflective dots at the choroidal level. Interleukin-6 (IL-6) in the anterior chamber was very high (1595 pg/ml RE and 749.3 pg/ml LE). After cervical lymph node biopsy, the patient was finally diagnosed with iMCD. Siltuximab, an interleukin-6 inhibitor, was administered. After two cycles of therapy, the patient's ocular manifestations improved significantly, the choroid and RPE undulation and the ERD resolved without local treatment.

Conclusion: Chorioretinal undulation and elevated IL-6 in intraocular fluid could be ocular manifestations of MCD.

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表现为脉络膜视网膜起伏的特发性多中心卡斯特曼病：病例报告

目的：报告1例特发性多中心性Castleman病（iMCD），首发眼部受累，表现为脉络膜视网膜波动。方法：对治疗初期和治疗后眼底摄影、自动荧光素和扫源光学相干断层扫描（SS-OCT）有眼部表现的iMCD患者进行病例观察。结果：患者75岁，右眼无痛性视力丧失2年，左眼无痛性视力丧失3个月。眼底检查显示沿下拱廊有几个黄白色病变，右眼有广泛的色素紊乱，左眼有渗出性视网膜脱离。SS-OCT显示脉络膜和视网膜色素上皮（RPE）波动，脉络膜增厚和脉络膜水平的高反射点。前房的白细胞介素-6 （IL-6）非常高（RE为1595 pg/ml， LE为749.3 pg/ml）。经颈部淋巴结活检，最终确诊为iMCD。给予白细胞介素-6抑制剂西妥昔单抗。经过两个周期的治疗，患者的眼部表现明显改善，脉络膜和RPE波动，ERD消除，无需局部治疗。结论：视网膜脉络膜波动和眼内液IL-6升高可能是MCD的眼部表现。

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来源期刊

Ocular Immunology and Inflammation 医学-眼科学

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.