儿童Blau综合征眼内手术后全葡萄膜炎的表现。

IF 0.5 Q4 OPHTHALMOLOGY
Middle East African Journal of Ophthalmology Pub Date : 2021-12-31 eCollection Date: 2021-07-01 DOI:10.4103/meajo.meajo_15_21
Moustafa S Magliyah, Abdullah M Khan, Mohammed AlShamrani, Patrik Schatz, Hassan A Dhibi
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引用次数: 1

摘要

Blau综合征(BS)是一种罕见的常染色体显性遗传肉芽肿性疾病。它的特点是皮炎、关节炎和复发性葡萄膜炎。本病例在眼内手术后出现BS的全葡萄膜炎。一名10岁男孩在接受早发性白内障手术6年后因左眼视网膜脱离就诊。双侧全膜炎发生于手术修复后3周,导致左眼完全失明,右眼持续常规治疗。基因检测显示NOD2基因突变。在治疗方案中加入阿达木单抗可长期控制葡萄膜炎并维持右眼20/70视力。我们认为,眼内手术可激活nod2介导的炎症级联反应,从而导致BS的出现。
本文章由计算机程序翻译,如有差异,请以英文原文为准。

Manifestation of Panuveitis after Intraocular Surgery in a Child with Blau Syndrome.

Manifestation of Panuveitis after Intraocular Surgery in a Child with Blau Syndrome.

Manifestation of Panuveitis after Intraocular Surgery in a Child with Blau Syndrome.

Blau syndrome (BS) is a rare granulomatous disease with autosomal dominant inheritance. It is characterized by a triad of dermatitis, arthritis, and recurrent uveitis. This case presents the onset of panuveitis in BS after intraocular surgery. A 10-year-old boy presented to the outpatient clinic with retinal detachment in the left eye after 6 years following early-onset cataract surgery. Bilateral panuveitis occurred 3 weeks after surgical repair and resulted in a total visual loss in the left eye and was persistent to conventional treatment in the right eye. Genetic testing revealed a mutation in NOD2 gene. The addition of adalimumab to the treatment regimen resulted in long-term uveitis control and maintenance of 20/70 vision in the right eye. We propose that NOD2-mediated inflammatory cascade can be activated by intraocular surgery and results in the manifestation of BS.

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来源期刊
CiteScore
1.40
自引率
0.00%
发文量
1
期刊介绍: The Middle East African Journal of Ophthalmology (MEAJO), published four times per year in print and online, is an official journal of the Middle East African Council of Ophthalmology (MEACO). It is an international, peer-reviewed journal whose mission includes publication of original research of interest to ophthalmologists in the Middle East and Africa, and to provide readers with high quality educational review articles from world-renown experts. MEAJO, previously known as Middle East Journal of Ophthalmology (MEJO) was founded by Dr Akef El Maghraby in 1993.
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