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与罗萨伊-多夫曼病相关的黄斑囊样水肿:一例报告

Cystoid macular oedema associated with Rosai-Dorfman disease: a case report.

作者信息

Siqueira Rubens Camargo, E Cruz Antonio Augusto Velasco

机构信息

Department of Ophthalmology, Otorhinolaryngology and Head and Neck Surgery, Ribeirao Preto School of Medicine, University of Sao Paulo, Sao Paulo, Brazil.

Rubens Siqueira Research Centre, Rua Saldanha Marinho 2815 Sala 42, Sao Jose do Rio Preto, Brazil.

出版信息

BMC Ophthalmol. 2017 Aug 14;17(1):141. doi: 10.1186/s12886-017-0538-8.

Abstract

BACKGROUND

The aim of this study is to report a case of cystoid macular oedema (CME) associated with Rosai-Dorfman Disease (RDD).

CASE PRESENTATION

A 52-year-old male initially presented with a two-month history of a congested left eye. At presentation, visual acuity was 20/20 in the right eye and 20/80 in the left eye. Biomicroscopy showed conjunctival hyperaemia in the left eye with a slight elevation, suggesting a subconjunctival mass. Retinal fluorescein angiography and optical coherence tomography (OCT) revealed the presence of CME in the left eye. A clinical examination revealed nodular lesions in the ears and a lump in the subcutaneous tissue of the left arm. A biopsy of the subcutaneous lesion showed histological and immunohistochemical characteristics of RDD. The patient was treated with intravitreal triamcinolone (0.1 mL/4 mg). One month after treatment, there was complete regression of the oedema with a significant improvement in visual acuity to 20/20.

CONCLUSIONS

This is the first reported case of RDD associated with cystoid macular oedema. Macular oedema responded to intravitreal treatment with triamcinolone.

摘要

背景

本研究旨在报告一例与罗萨伊-多夫曼病(RDD)相关的黄斑囊样水肿(CME)病例。

病例介绍

一名52岁男性最初因左眼充血两个月前来就诊。就诊时,右眼视力为20/20,左眼视力为20/80。生物显微镜检查显示左眼结膜充血并稍有隆起,提示结膜下肿物。视网膜荧光血管造影和光学相干断层扫描(OCT)显示左眼存在CME。临床检查发现耳部有结节性病变,左臂皮下有一肿块。皮下病变活检显示具有RDD的组织学和免疫组化特征。患者接受了玻璃体内注射曲安奈德(0.1 mL/4 mg)治疗。治疗一个月后,水肿完全消退,视力显著提高至20/20。

结论

这是首例报道的与黄斑囊样水肿相关的RDD病例。黄斑水肿对玻璃体内注射曲安奈德治疗有反应。

https://cdn.ncbi.nlm.nih.gov/pmc/blobs/59ed/5557076/0af8689d3f02/12886_2017_538_Fig1_HTML.jpg

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