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与线状硬皮病相关的泪腺炎:一例报告并文献复习

Dacryoadenitis associated with linear scleroderma : A case report and review of literature.

作者信息

Oustabassidis Eva, Keren Shay, David Joel, Norris Jonathan H

机构信息

Oxford Eye Hospital John Radcliffe Hospital Oxford UK.

Rheumatology Department Nuffield Orthopaedic Centre Oxford UK.

出版信息

Skin Health Dis. 2024 Apr 22;4(4):e391. doi: 10.1002/ski2.391. eCollection 2024 Aug.

Abstract

Dacryoadenitis in the setting of linear scleroderma (LScs) is an association that has not previously been described in the scientific literature. The purpose of this case report is to describe the co-existence of LScs and chronic dacryoadenitis and how it was managed. We report the case of a 42-year-old woman who presented with a 4-month history of left upper eyelid swelling with radiological enlargement of the left lacrimal gland on orbital CT and MRI imaging. Clinical examination revealed a left erythematous, swollen upper eyelid with lateral conjunctival injection and a palpable left lacrimal gland. An ipsilateral band-like cutaneous depression in the fronto-temporal region was also noted, which extended to the ipsilateral upper eyelid and brow. Serology revealed nucleolar antinuclear antibodies. A further incisional biopsy of the lacrimal gland confirmed chronic inflammatory changes and fibrous tissue. Based on both the histological and clinical findings, a diagnosis of dacryoadenitis in association with LScs was made. Oral methotrexate was commenced. The patient responded well with less frequent episodes of eyelid swelling and reduced periocular pain. This case describes for the first time, the ophthalmological manifestation of chronic dacryoadenitis in association with linear scleroderma .

摘要

线状硬皮病(LScs)合并泪腺炎是一种此前科学文献中未曾描述过的关联情况。本病例报告的目的是描述LScs与慢性泪腺炎的共存情况以及处理方式。我们报告一例42岁女性,有4个月的左上眼睑肿胀病史,眼眶CT和MRI成像显示左侧泪腺放射性增大。临床检查发现左侧上眼睑红肿,外侧结膜充血,可触及左侧泪腺。还注意到同侧额颞部有条带状皮肤凹陷,延伸至同侧上眼睑和眉毛。血清学检查显示核仁抗核抗体。对泪腺进行的进一步切开活检证实存在慢性炎症改变和纤维组织。基于组织学和临床发现,诊断为与LScs相关的泪腺炎。开始口服甲氨蝶呤。患者反应良好,眼睑肿胀发作频率降低,眼周疼痛减轻。本病例首次描述了与线状硬皮病相关的慢性泪腺炎的眼科表现。

https://cdn.ncbi.nlm.nih.gov/pmc/blobs/1292/11297447/0e8efd63b2fd/SKI2-4-e391-g002.jpg

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