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以眼眶壁破坏为表现的严重局限性肉芽肿性多血管炎的罕见病例:文献综述与病例报告

Rare Presentation of Severely Limited Granulomatosis With Polyangiitis Manifesting With Orbital Wall Destruction: Literature Review and Case Report.

作者信息

Havuz Erol, GÜdÜl Havuz Seda

机构信息

Department of Ophthalmology, University of Health Sciences, SUAM Samsun Hospital, Samsun, Turkey.

Department of Microbiology, Public Health Institution of Turkey, Samsun, Turkey.

出版信息

Arch Rheumatol. 2020 Feb 7;35(2):292-299. doi: 10.46497/ArchRheumatol.2020.7503. eCollection 2020 Jun.

Abstract

Necrosis resulting from mechanical local factors can be seen in patients with granulomatosis with polyangiitis (GPA) even in remission. GPA can cause serious morbidity even when limited. An ocular prosthesis that increases inflammation and damages local circulation should be used very carefully in such patients. In this article, we report a 68-year-old male patient who was diagnosed with localized GPA 11 years ago and referred to our clinic with the complaint of displacement of an ocular prosthesis inside the nose and epistaxis. Four years ago, the left eye was enucleated because of pain and vision loss. Two months after the enucleation, the patient began to use an ocular prosthesis. Orbital medial wall destruction developed while the patient was receiving maintenance therapy that consisted of cyclophosphamide (150 mg/day) plus prednisolone (32 mg). When the ocular prosthesis was displaced in the nasal cavity, the prosthesis was first removed and the patient was clinically stabilized. Later, orbital wall reconstruction was performed at another center.

摘要

即使在缓解期,显微镜下多血管炎(GPA)患者也可见由机械性局部因素导致的坏死。GPA即使病情局限也可导致严重发病。在这类患者中,应非常谨慎地使用会增加炎症反应并损害局部循环的眼假体。在本文中,我们报告了一名68岁男性患者,他11年前被诊断为局限性GPA,因鼻腔内眼假体移位和鼻出血的主诉转诊至我们诊所。4年前,左眼因疼痛和视力丧失而被摘除。摘除术后2个月,患者开始使用眼假体。在患者接受由环磷酰胺(150毫克/天)加泼尼松龙(32毫克)组成的维持治疗期间,眶内侧壁出现破坏。当眼假体在鼻腔内移位时,首先取出假体,患者临床症状稳定。后来,在另一家中心进行了眶壁重建。

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本文引用的文献

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Clinical features of different orbital manifestations of granulomatosis with polyangiitis.肉芽肿性多血管炎不同眼眶表现的临床特征。
Graefes Arch Clin Exp Ophthalmol. 2018 Sep;256(9):1751-1756. doi: 10.1007/s00417-018-4014-9. Epub 2018 May 30.
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