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单侧眼黏膜类天疱疮罕见病例。

A rare unilateral case of ocular mucous membrane pemphigoid.

机构信息

The Save Sight Institute, The University of Sydney and The Sydney Eye Hospital, Sydney, Australia.

出版信息

Cornea. 2010 Dec;29(12):1462-4. doi: 10.1097/ICO.0b013e3181da580a.

Abstract

PURPOSE

Ocular mucous membrane pemphigoid (MMP) is known to be rapidly progressive in younger patients and affects both eyes. We are aware of only one other reported case of unilateral ocular MMP. The case presented in this study is unique in that it demonstrates genuinely unilateral ocular MMP in a younger patient.

METHODS

We report a case of a 50-year-old man who presented with a 3-month history of left eye redness, irritation, and mild discharge. He also suffered from mouth ulcers, skin lesions, and recurrent nose bleeds secondary to nasal mucosal lesions.

RESULTS

Examination revealed unilateral lid granulomas and cicatricial conjunctivitis in his left eye. There were no abnormal findings in his right eye. Biopsy of the lesions showed nonspecific inflammation with positive immunofluorescence for immunoglobulin G and C3 on epithelial basement membranes of conjunctiva, buccal mucosa, and skin, which were consistent with MMP. He was treated with immunosuppression and had no disease progression at 12-month follow-up.

CONCLUSIONS

This represents a rare case of unilateral nonprogressive ocular MMP in a younger patient. Histological analysis and immunofluorescence testing excluded a neoplastic process and confirmed the diagnosis. This case demonstrates that the presentation of unilateral eyelid pyogenic granulomas should include ocular MMP in the differential diagnosis once a neoplastic process has been excluded.

摘要

目的

眼黏膜类天疱疮(MMP)在年轻患者中进展迅速,且累及双眼。我们仅知晓另一例单侧眼 MMP 的报告病例。本研究中报告的病例较为独特,其在年轻患者中表现为真正的单侧眼 MMP。

方法

我们报告了 1 例 50 岁男性病例,其左眼出现 3 个月的眼红、刺激和轻度分泌物。他还患有口腔溃疡、皮肤病变和因鼻黏膜病变导致的复发性鼻出血。

结果

检查发现左眼有单侧的眼睑肉芽肿和瘢痕性结膜炎。右眼无异常发现。病变活检显示非特异性炎症,结膜、口腔颊黏膜和皮肤的上皮基底膜免疫荧光 IgG 和 C3 阳性,符合 MMP。他接受了免疫抑制治疗,在 12 个月的随访中无疾病进展。

结论

这是年轻患者中罕见的单侧非进展性眼 MMP 病例。组织学分析和免疫荧光检测排除了肿瘤过程,确诊为 MMP。该病例表明,单侧眼睑化脓性肉芽肿的表现应在排除肿瘤过程后将眼 MMP 纳入鉴别诊断。

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