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两名兄弟姐妹同时出现双侧虹膜急性色素脱失。

Bilateral acute depigmentation of the iris in two siblings simultaneously.

作者信息

Amin Rana, Nabih Amena, Khater Noha

机构信息

Department of Ophthalmology, Faculty of Medicine, Kasr El Aini Hospital, Cairo University, 1 Kasr El Aini Street, Al Manial, Cairo, Egypt.

Al Mouneer Center for Diabetic Eye Care, 92 El Tharir Street, 4th Floor, Giza, Egypt.

出版信息

Am J Ophthalmol Case Rep. 2018 Mar 16;10:257-260. doi: 10.1016/j.ajoc.2018.03.016. eCollection 2018 Jun.

Abstract

PURPOSE

To report the first simultaneous onset of bilateral acute depigmentation of the iris (BADI) in two siblings.

OBSERVATIONS

Two sisters presented with bilateral ocular pain, redness and light sensitivity. Examination revealed bilateral circulating pigment in the anterior chamber with pigment dusting on backs of the corneas, patchy iris depigmentation and heavy pigment deposition in the angle. Both patients had recently suffered from upper respiratory tract infections. Bilateral visual acuities were preserved and no transillumination defects were observed. The patients were diagnosed with BADI. Both cases were successfully controlled with topical corticosteroids and anti-glaucoma drops as well as topical glanciclovir gel.

CONCLUSIONS AND IMPORTANCE

To date, there had been no published reports of BADI in the Middle East and Africa. This is the first observation of this entity in these regions. Moreover it is the first occurrence of BADI in two immediate siblings simultaneously. We also report the rare asymmetrical presentation with BADI in one of our patients. These observations point to the possibility of genetic factors underlying BADI as well as an infectious cause behind the etiology.

摘要

目的

报告首例两姐妹同时出现双侧急性虹膜色素脱失(BADI)的病例。

观察结果

两名姐妹均出现双侧眼痛、眼红和畏光症状。检查发现前房有双侧循环色素,角膜后有色素沉着,虹膜呈片状色素脱失,房角有大量色素沉着。两名患者近期均患过上呼吸道感染。双侧视力均得以保留,未观察到透照缺损。两名患者均被诊断为BADI。两例均通过局部使用皮质类固醇、抗青光眼滴眼液以及局部使用更昔洛韦凝胶成功得到控制。

结论与意义

迄今为止,中东和非洲地区尚无关于BADI的 published reports。这是在这些地区首次观察到该病症。此外,这也是首次在两名亲姐妹中同时出现BADI。我们还报告了其中一名患者出现的罕见的BADI不对称表现。这些观察结果表明BADI可能存在遗传因素以及病因背后的感染原因。

https://cdn.ncbi.nlm.nih.gov/pmc/blobs/81af/5956750/bf59f28b0067/gr1.jpg

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