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血清阳性眼肌型重症肌无力患者接受依氟鸟氨酸治疗后出现卡波西水痘样疹和疱疹性结膜炎:病例报告及文献复习。

Efgartigimod-associated Kaposi's varicelliform eruption and herpetic conjunctivitis in a patient with seropositive ocular myasthenia gravis: a case report and review.

机构信息

Department of Dermatology, The Second Affiliated Hospital of Shandong First Medical University, Tai'an, China.

Department of Graduate Studies, Shandong First Medical University & Shandong Academy of Medical Sciences, Jinan, China.

出版信息

Front Immunol. 2024 Aug 1;15:1409480. doi: 10.3389/fimmu.2024.1409480. eCollection 2024.

DOI:10.3389/fimmu.2024.1409480
PMID:39148733
原文链接:https://pmc.ncbi.nlm.nih.gov/articles/PMC11324432/
Abstract

BACKGROUND

Efgartigimod (Efgartigimod alpha fcab, Vyvgart™) is a pioneering neonatal Fc receptor (FcRn) antagonist for the treatment of severe autoimmune diseases mediated by pathogenic immunoglobulin G (IgG) autoantibodies, including myasthenia gravis (MG). It is a well-tolerated drug with minor side effects, such as headache and upper respiratory (lung) and urinary tract infections. Here, we present a case of Kaposi's varicelliform eruption (KVE) and herpetic conjunctivitis related to efgartigimod in a 60-year-old patient with ocular MG (OMG).

CASE DESCRIPTION

A 60-year-old Chinese male suffered from acetylcholine receptor antibody positive (AChR Ab+) OMG for 8 years. During this period, he underwent first-line treatment with systemic corticosteroids, cyclosporine, cyclophosphamide, and so on, but had poor symptom improvement. On the recommendation of his attending neurologist, he received one cycle of intravenous efgartigimod (10mg/kg, once weekly for 4 weeks). The patient experienced fever, widespread painful blisters, and edema on the face on the third day after his last intravenous infusion. The patient also complained of increased secretions and a foreign body sensation in both eyes. Laboratory tests confirmed infection with herpes simplex virus (HSV). A diagnosis of efgartigimod-associated KVE and herpetic conjunctivitis was made. After intravenous administration (5mg/kg, 3 times a day, every 8 hours) for 10 days, the patient was cured without residual complications.

CONCLUSIONS

This case is the first report of a patient with KVE and herpetic conjunctivitis related to efgartigimod in PubMed. This is rare and unusual. Clinicians should be alert to the rare symptoms related to efgartigimod.

摘要

背景

依非格司亭(依非格司亭 alpha fcab,Vyvgart™)是一种开创性的新生儿 Fc 受体(FcRn)拮抗剂,用于治疗致病性免疫球蛋白 G(IgG)自身抗体介导的严重自身免疫性疾病,包括重症肌无力(MG)。它是一种耐受性良好的药物,副作用较小,如头痛、上呼吸道(肺部)和尿路感染。在这里,我们报告了一例与依非格司亭相关的卡波西水痘样疹(KVE)和疱疹性结膜炎,患者为 60 岁男性,患有眼肌型 MG(OMG)。

病例描述

一名 60 岁的中国男性患有乙酰胆碱受体抗体阳性(AChR Ab+)OMG 8 年。在此期间,他接受了一线治疗,包括全身皮质类固醇、环孢素、环磷酰胺等,但症状改善不佳。根据他的主治神经科医生的建议,他接受了一个周期的依非格司亭静脉输注(10mg/kg,每周一次,共 4 周)。患者在最后一次静脉输注后第 3 天出现发热、广泛疼痛性水疱和面部水肿。患者还主诉双眼分泌物增多和异物感。实验室检查证实感染单纯疱疹病毒(HSV)。诊断为依非格司亭相关 KVE 和疱疹性结膜炎。静脉输注(5mg/kg,每日 3 次,每 8 小时 1 次)治疗 10 天后,患者痊愈,无残留并发症。

结论

这是 PubMed 上首例报告的与依非格司亭相关的 KVE 和疱疹性结膜炎病例。这种情况罕见且不常见。临床医生应警惕与依非格司亭相关的罕见症状。

https://cdn.ncbi.nlm.nih.gov/pmc/blobs/8ac3/11324432/92c95913f0ad/fimmu-15-1409480-g002.jpg
https://cdn.ncbi.nlm.nih.gov/pmc/blobs/8ac3/11324432/b59412546ec9/fimmu-15-1409480-g001.jpg
https://cdn.ncbi.nlm.nih.gov/pmc/blobs/8ac3/11324432/92c95913f0ad/fimmu-15-1409480-g002.jpg
https://cdn.ncbi.nlm.nih.gov/pmc/blobs/8ac3/11324432/b59412546ec9/fimmu-15-1409480-g001.jpg
https://cdn.ncbi.nlm.nih.gov/pmc/blobs/8ac3/11324432/92c95913f0ad/fimmu-15-1409480-g002.jpg

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Effect of FcRn antagonism on protective antibodies and to vaccines in IgG-mediated autoimmune diseases pemphigus and generalised myasthenia gravis.FcRn 拮抗作用对 IgG 介导的自身免疫性疾病天疱疮和重症肌无力中保护性抗体和疫苗的影响。
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