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眼睑脓性皮病-脓性口炎型增殖性天疱疮病例报告及文献复习

Pyodermatitis-pyostomatitis vegetans of the eyelids case report and review of the literature.

作者信息

Leibovitch Igal, Ooi Colin, Huilgol Shyamala C, Reid Cathy, James Craig L, Selva Dinesh

机构信息

Oculoplastic & Orbital Division, Department of Ophthalmology & Visual Sciences, Royal Adelaide Hospital, University of Adelaide, Adelaide, Australia.

出版信息

Ophthalmology. 2005 Oct;112(10):1809-13. doi: 10.1016/j.ophtha.2005.04.027.

Abstract

OBJECTIVES

To present the first reported case of eyelid involvement in pyodermatitis-pyostomatitis vegetans (PDPSV) leading to the diagnosis of ulcerative colitis, and to review the literature.

DESIGN

Interventional case report.

METHODS

A 29-year-old man presented with a 4-week history of severe bilateral upper and lower eyelid margin ulceration and pustules unresponsive to topical and systemic broad-spectrum antibiotic treatment. Further questioning revealed the existence of skin and oral lesions.

MAIN OUTCOME MEASURES

Clinical course, histological findings, and response to treatment.

RESULTS

The histological and immunofluorescence studies were suggestive of PDPSV. Colonoscopy showed significant chronic active ulcerative colitis. Treatment with systemic steroids and sulfasalazine resulted in complete resolution of eyelid, oral, and skin lesions. However, stopping the steroids resulted in recurrence of eyelid and oral lesions and required recommencement of treatment.

CONCLUSION

It is important to be familiar with this pustular skin condition, as correct diagnosis may lead to the diagnosis of inflammatory bowel disease. Although periocular involvement is probably rare, the combined typical skin and oral lesions and the characteristic histological and immunofluorescence tests should suggest the correct diagnosis.

摘要

目的

报告首例睑部受累的增殖性脓皮病-增殖性口炎(PDPSV)病例,该病例导致溃疡性结肠炎的诊断,并对相关文献进行综述。

设计

介入性病例报告。

方法

一名29岁男性,双侧上下睑缘严重溃疡和脓疱病史4周,局部和全身应用广谱抗生素治疗无效。进一步询问发现存在皮肤和口腔病变。

主要观察指标

临床病程、组织学表现及治疗反应。

结果

组织学和免疫荧光研究提示为PDPSV。结肠镜检查显示重度慢性活动性溃疡性结肠炎。全身应用类固醇和柳氮磺胺吡啶治疗后,眼睑、口腔和皮肤病变完全消退。然而,停用类固醇后,眼睑和口腔病变复发,需要重新开始治疗。

结论

熟悉这种脓疱性皮肤病很重要,因为正确诊断可能会引出炎症性肠病的诊断。虽然眼周受累可能罕见,但典型的皮肤和口腔病变组合以及特征性的组织学和免疫荧光检查应能提示正确诊断。

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