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伴有泪腺受累的眼眶肉芽肿性多血管炎的管理:在一个具有挑战性的病例中治疗随后出现的周边溃疡性角膜炎、前葡萄膜炎和渗出性视网膜脱离

Management of Orbital Granulomatous Polyangiitis Presenting with Lacrimal Gland Involvement: Treatment of Subsequent Peripheral Ulcerative Keratitis, Anterior Uveitis, and Exudative Retinal Detachment in a Challenging Case.

作者信息

Oklar Murat, Sarı Elif, Kanar Hatice Selen, Süslü Hikmet, Çiftçi Tufan, Tezcan Mehmet Engin

机构信息

University of Health Sciences Türkiye, Kartal Dr. Lütfi Kırdar City Hospital, Clinic of Ophthalmology, İstanbul, Türkiye.

University of Health Sciences Türkiye, Kartal Dr. Lütfi Kırdar City Hospital, Clinic of Neurosurgery, İstanbul, Türkiye.

出版信息

Turk J Ophthalmol. 2024 Dec 31;54(6):358-363. doi: 10.4274/tjo.galenos.2024.72317.

DOI:10.4274/tjo.galenos.2024.72317
PMID:39743940
原文链接:https://pmc.ncbi.nlm.nih.gov/articles/PMC11707461/
Abstract

This case report discusses a case of granulomatosis with polyangiitis (GPA) initially presenting with lacrimal gland involvement and subsequently developing additional features. GPA is a disease known for inflammation in the respiratory tract and kidneys. A 63-year-old male patient presented with a mass, swelling, and ptosis in the right upper eyelid. The patient was referred to us when his symptoms had persisted and worsened despite topical and oral antibiotic therapy administered at another center. Based on clinical and laboratory findings from evaluation and consultations, GPA was diagnosed and confirmed by biopsy. The patient initially presented with necrotizing scleritis and later developed peripheral ulcerative keratitis and anterior uveitis, which regressed under cyclophosphamide treatment but progressed to exudative retinal detachment due to orbital involvement. Clinical remission was achieved after adding rituximab therapy. Presentation with lacrimal gland involvement may serve as an initial manifestation of locally aggressive orbital and adnexal GPA, which can exhibit variable clinical features. Rapid diagnosis and aggressive treatment are critical for preserving vision and preventing complications in patients with GPA.

摘要

本病例报告讨论了一例肉芽肿性多血管炎(GPA),最初表现为泪腺受累,随后出现其他症状。GPA是一种以呼吸道和肾脏炎症为特征的疾病。一名63岁男性患者出现右上眼睑肿物、肿胀和上睑下垂。尽管在另一家中心接受了局部和口服抗生素治疗,但其症状持续且加重,随后转诊至我院。根据评估和会诊的临床及实验室检查结果,诊断为GPA,并经活检确诊。患者最初表现为坏死性巩膜炎,随后发展为周边溃疡性角膜炎和前葡萄膜炎,在环磷酰胺治疗下病情缓解,但由于眼眶受累进展为渗出性视网膜脱离。加用利妥昔单抗治疗后实现了临床缓解。泪腺受累可能是局部侵袭性眼眶及附件GPA的初始表现,其临床特征可能多样。快速诊断和积极治疗对于保护GPA患者的视力和预防并发症至关重要。

https://cdn.ncbi.nlm.nih.gov/pmc/blobs/c5f3/11707461/77f628489382/TurkJOphthalmol-54-358-figure-5.jpg
https://cdn.ncbi.nlm.nih.gov/pmc/blobs/c5f3/11707461/5e4799478a44/TurkJOphthalmol-54-358-figure-1.jpg
https://cdn.ncbi.nlm.nih.gov/pmc/blobs/c5f3/11707461/13afe2a65a7b/TurkJOphthalmol-54-358-figure-2.jpg
https://cdn.ncbi.nlm.nih.gov/pmc/blobs/c5f3/11707461/b3d2da1bd86b/TurkJOphthalmol-54-358-figure-3.jpg
https://cdn.ncbi.nlm.nih.gov/pmc/blobs/c5f3/11707461/a61a1639d4a4/TurkJOphthalmol-54-358-figure-4.jpg
https://cdn.ncbi.nlm.nih.gov/pmc/blobs/c5f3/11707461/77f628489382/TurkJOphthalmol-54-358-figure-5.jpg
https://cdn.ncbi.nlm.nih.gov/pmc/blobs/c5f3/11707461/5e4799478a44/TurkJOphthalmol-54-358-figure-1.jpg
https://cdn.ncbi.nlm.nih.gov/pmc/blobs/c5f3/11707461/13afe2a65a7b/TurkJOphthalmol-54-358-figure-2.jpg
https://cdn.ncbi.nlm.nih.gov/pmc/blobs/c5f3/11707461/b3d2da1bd86b/TurkJOphthalmol-54-358-figure-3.jpg
https://cdn.ncbi.nlm.nih.gov/pmc/blobs/c5f3/11707461/a61a1639d4a4/TurkJOphthalmol-54-358-figure-4.jpg
https://cdn.ncbi.nlm.nih.gov/pmc/blobs/c5f3/11707461/77f628489382/TurkJOphthalmol-54-358-figure-5.jpg

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Management of Orbital Granulomatous Polyangiitis Presenting with Lacrimal Gland Involvement: Treatment of Subsequent Peripheral Ulcerative Keratitis, Anterior Uveitis, and Exudative Retinal Detachment in a Challenging Case.伴有泪腺受累的眼眶肉芽肿性多血管炎的管理:在一个具有挑战性的病例中治疗随后出现的周边溃疡性角膜炎、前葡萄膜炎和渗出性视网膜脱离
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本文引用的文献

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Intralesional or intraorbital rituximab injection for the management of biopsy-proven idiopathic orbital inflammation involving the lacrimal gland.经活检证实的累及泪腺的特发性眼眶炎症的病灶内或眶内利妥昔单抗注射治疗。
Can J Ophthalmol. 2024 Oct;59(5):e496-e500. doi: 10.1016/j.jcjo.2023.10.016. Epub 2023 Nov 20.
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Granulomatosis with Polyangiitis Complicated by Severe Exudative Retinal Detachment and Orbital Granuloma Successfully Controlled with Rituximab: A Case Report.利妥昔单抗成功控制伴严重渗出性视网膜脱离和眼眶肉芽肿的肉芽肿性多血管炎:一例报告
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Peripheral Ulcerative Keratitis: A Review.
周边溃疡性角膜炎:综述
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