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厚皮性骨膜病伴严重泡性角结膜炎继发视力丧失

Pachydermoperiostosis Presenting With Vision Loss Secondary to Severe Phlyctenular Keratoconjunctivitis.

作者信息

Cheng Shuk Kei, Michael Anika, Rizzuti Allison E

机构信息

Department of Ophthalmology, SUNY Downstate Medical Center, Brooklyn, NY.

出版信息

Cornea. 2022 Jan 1;41(1):113-115. doi: 10.1097/ICO.0000000000002700.

Abstract

PURPOSE

To report a case of severe phlyctenular keratoconjunctivitis in a patient with pachydermoperiostosis (PDP).

METHODS

A 29-year-old Hispanic man presented with a 1-year history of vision loss in the right eye and redness, photophobia, and pain in both eyes. Associated symptoms included enlargement of his hands and feet for 3 years, acne, and joint pain. Examination was notable for severe meibomian gland dysfunction, corneal and limbal phlyctenules in both eyes, and central stromal scarring of the right cornea. He had cystic acne of the face and a coarse, wrinkled forehead and scalp. Examination of his hands and feet revealed clubbing of the digits.

RESULTS

The patient had an extensive laboratory workup that was significant for elevated erythrocyte sediment rate and C-reactive protein. X-ray of his hands and feet revealed diffuse periosteal hyperostosis with diffuse bone expansion. The patient was diagnosed with PDP with severe meibomian gland dysfunction, phlyctenular keratoconjunctivitis, and corneal scarring. His pain and photophobia resolved with medical management of the phlyctenular keratoconjunctivitis but decreased vision in the right eye persisted because of neovascularization and scarring of the cornea.

CONCLUSIONS

We report a rare case of phlyctenular keratoconjunctivitis associated with PDP. To our knowledge, this is the first case of PDP to initially present with vision loss.

摘要

目的

报告1例厚皮性骨膜病(PDP)患者发生的严重泡性角结膜炎。

方法

一名29岁的西班牙裔男性,右眼视力下降1年,双眼发红、畏光及疼痛。相关症状包括手脚增大3年、痤疮和关节疼痛。检查发现严重睑板腺功能障碍、双眼角膜及角膜缘泡疹,以及右眼角膜中央基质瘢痕形成。他面部有囊肿性痤疮,前额和头皮粗糙、有皱纹。检查其手足发现手指杵状指。

结果

患者进行了全面的实验室检查,结果显示红细胞沉降率和C反应蛋白升高。其手足X线检查显示弥漫性骨膜增生伴弥漫性骨质膨胀。该患者被诊断为PDP,伴有严重睑板腺功能障碍、泡性角结膜炎和角膜瘢痕形成。经泡性角结膜炎的药物治疗,其疼痛和畏光症状缓解,但由于角膜新生血管形成和瘢痕,右眼视力仍持续下降。

结论

我们报告了1例与PDP相关的罕见泡性角结膜炎病例。据我们所知,这是首例以视力下降为首发表现的PDP病例。

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