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儿童患者以双侧泪腺炎为首发表现的抗中性粒细胞胞质抗体相关性血管炎。

Bilateral dacryoadenitis as the initial presentation of ANCA-associated vasculitis in a pediatric patient.

机构信息

Department of Ophthalmology and Visual Science, Yale University School of Medicine, New Haven, Connecticut, USA.

Department of Pathology, Yale University School of Medicine, New Haven, Connecticut, USA.

出版信息

Orbit. 2024 Apr;43(2):231-235. doi: 10.1080/01676830.2022.2093920. Epub 2022 Jul 8.

DOI:10.1080/01676830.2022.2093920
PMID:35801829
Abstract

A 14-year-old female presented with 2 weeks of progressive right eye pain, erythema, and proptosis. Examination demonstrated an enlarged palpable mass along the right superior lateral orbit and bilateral conjunctival petechiae. Of note, she was asymptomatic on the left side, and the petechiae were present only on the superior bulbar conjunctiva with eyelid eversion. Imaging demonstrated bilateral lacrimal gland enhancement. Testing was significant for elevated inflammatory markers, but otherwise negative workup. Biopsy of the right lacrimal gland demonstrated acute-on-chronic inflammation without evidence of lymphoproliferative disease. On repeat testing, myeloperoxidase antibody levels (MPO/p-ANCA) were elevated, indicative of an underlying immune-mediated vasculitis. This case illustrates a rare presentation of ANCA-associated vasculitis in a pediatric patient. It further demonstrates the phenomenon of initial negative serology and subsequent auto-antibody seroconversion in a patient with localized granulomatosis with polyangiitis.

摘要

一位 14 岁女性因右眼疼痛、红斑和突出进行性加重 2 周就诊。检查发现右侧眶上外侧可触及增大的肿块,双侧球结膜瘀点。值得注意的是,左侧无症状,瘀点仅在上部球结膜翻转时出现。影像学显示双侧泪腺增强。检测结果显示炎症标志物升高,但其他检查均为阴性。右侧泪腺活检显示慢性炎症急性发作,无淋巴增生性疾病证据。重复检测显示髓过氧化物酶抗体(MPO/p-ANCA)水平升高,提示潜在的免疫介导性血管炎。本例罕见地表现为儿童抗中性粒细胞胞浆抗体相关性血管炎。它进一步表明在局部肉芽肿性多血管炎患者中,初始阴性血清学和随后自身抗体血清转化的现象。

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引用本文的文献

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Limited granulomatosis with polyangiitis in children with idiopathic orbital inflammation: a case series and literature review.儿童特发性眼眶炎症相关局限性肉芽肿伴多血管炎:病例系列研究及文献复习。
Rheumatol Int. 2024 Nov;44(11):2679-2690. doi: 10.1007/s00296-023-05366-7. Epub 2023 Jun 15.