Retinal Vasculopathy and Choroiditis after Pegcetacoplan Injection: Clinicopathologic Support for a Drug Hypersensitivity Reaction.

IF 4.4 Q1 OPHTHALMOLOGY Ophthalmology. Retina Pub Date : 2024-10-16 DOI:10.1016/j.oret.2024.10.005
Ankur Nahar, Dean Eliott, Robert L Avery, Jose Pulido, Ralph C Eagle, Jacqueline R Carrasco, Courtney Crawford, Tatyana Milman, Anna M Stagner
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Abstract

Purpose: To report the detailed histopathology of 2 enucleated eyes from 2 patients who developed severe visual loss associated with retinal hemorrhages, vessel sheathing, and vascular nonperfusion after administration of an initial dose of intravitreal pegcetacoplan, and propose, with supportive histopathology, the pathogenesis of the clinical syndrome previously termed hemorrhagic occlusive retinal vasculitis.

Design: Case series.

Subjects: Two enucleated eyes from 2 patients treated with intravitreal pegcetacoplan.

Methods: Retrospective, multicenter, consecutive clinical-pathologic analysis.

Main outcome measures: Histopathologic review and immunophenotypic characterization.

Results: Both patients presented with inflammation and significant vision loss 9 days after the initial injection of pegcetacoplan with no subsequent improvement and underwent enucleation for pain control. Histologic examination of the enucleated eyes (patient 1 at 4 months postinjection and patient 2 at 40 days) revealed extensive vascular thrombosis, retinal hemorrhages and necrosis, and a dense inflammatory infiltrate in the uvea and, variably, the optic nerve, episclera, and muscle tendons composed of predominantly of T cells, macrophages, and eosinophils. Notably, the inflammatory infiltrate was absent from the retina. In addition, 1 eye demonstrated multiple foci of glomerular-like vascular proliferations in the uveal tract and thrombosis with focal recanalization of vessels in the optic nerve.

Conclusions: Drug-induced, immune-mediated, retinal vasculopathy and choroiditis (DIRVAC) is a rare complication after pegcetacoplan injection. Although some limitations arise in interpretation of histopathologic findings because of compensatory changes in the eyes over time (before enucleation), the authors propose that the combined clinical, histopathologic, and immunohistochemical findings suggest a mixed-type, delayed hypersensitivity reaction as the mechanism of initial injury.

Financial disclosure(s): Proprietary or commercial disclosure may be found in the Footnotes and Disclosures at the end of this article.

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注射培高氯普兰后出现视网膜血管病变和脉络膜炎:临床病理支持药物超敏反应。
目的:报告两名患者的两只去核眼球的详细组织病理学,这两名患者在使用初始剂量的玻璃体内培加氯普兰后出现了与视网膜出血、血管鞘化及血管非灌注相关的严重视力丧失,并通过支持性组织病理学提出了以前被称为出血性闭塞性视网膜血管炎(HORV)的临床综合征的发病机制:设计:病例系列:方法:回顾性多中心临床试验:回顾性、多中心连续临床病理分析:组织病理学检查和免疫表型鉴定:结果:两名患者均在首次注射培加氯普兰九天后出现炎症和视力明显下降,随后情况无改善,为控制疼痛接受了去核手术。眼球摘除术后的组织学检查(一号患者在注射后 4 个月,二号患者在注射后 40 天)发现了广泛的血管栓塞、视网膜出血和坏死,葡萄膜和视神经、巩膜和肌腱处也有不同程度的致密炎症浸润,主要由 T 细胞、巨噬细胞和嗜酸性粒细胞组成。值得注意的是,视网膜上没有炎症浸润。此外,一只眼睛的葡萄膜道出现多个肾小球样血管增生灶,视神经血管出现血栓形成和灶性再通:结论:药物诱导、免疫介导的视网膜血管病变和脉络膜炎(DIRVAC)是注射培加氯普兰后的一种罕见并发症。虽然由于眼球随着时间的推移(去核前)发生代偿性变化,对组织病理学结果的解释存在一定的局限性,但作者认为,综合临床、组织病理学和免疫组化结果,最初的损伤机制是混合型迟发性超敏反应。
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来源期刊
Ophthalmology. Retina
Ophthalmology. Retina Medicine-Ophthalmology
CiteScore
7.80
自引率
6.70%
发文量
274
审稿时长
33 days
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