toll样受体4突变的成人双侧顺序性单纯疱疹2型全眼炎

IF 2.6 4区 医学 Q2 OPHTHALMOLOGY
Shiao W Wong, Sasa Pockar, Laura R Steeples, Felipe Dhawahir-Scala, Salma Babiker, Nicholas P Jones
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引用次数: 0

摘要

目的:报道首例双侧顺序性单纯疱疹病毒2型(HSV-2)全眼炎,导致一名toll样受体4 (TLR-4)突变的年轻成人严重视力丧失,并对先前报道的HSV-2全眼炎病例进行回顾。方法:回顾性病例报告。结果:一名29岁男性表现为严重疼痛,左眼肿胀和视力丧失。双眼视力均无光感。6年前,他患右眼全炎,经水液(AH)取样聚合酶链反应(PCR)证实病因为单纯疱疹病毒2型(HSV-2),预后不佳。目前,AH和玻璃体样本的PCR再次证实HSV-2感染。对高剂量静脉抗病毒药物的不良初始反应导致增加高剂量全身皮质类固醇。随后的晶状体切除术使视力部分恢复。基因检测发现TLR-4突变为rs4986790 A/G。结论:单纯疱疹病毒2型全眼炎是一种罕见且具有视觉破坏性的疾病,我们报告了首例双侧疾病。我们假设TLR-4突变可能导致过度炎症和严重的眼部损伤,而之前没有报道TLR-4突变与HSV眼部炎症相关。我们建议对所有严重病毒性眼内炎症患者进行全面的免疫学评估。及时诊断PCR和立即高剂量抗病毒治疗是至关重要的,免疫异常可能需要改进管理。
本文章由计算机程序翻译,如有差异,请以英文原文为准。
Bilateral Sequential Herpes Simplex Type 2 Panophthalmitis in an Adult with a Toll-Like Receptor 4 Mutation.

Purpose: To present the first reported case of bilateral sequential Herpes Simplex Virus Type 2 (HSV-2) panophthalmitis causing severe vision loss in a young adult with a toll-like receptor 4 (TLR-4) mutation, with a review of previously reported cases of HSV-2 panophthalmitis.

Methods: Retrospective case report.

Results: A 29-year-old man presented with a severely painful, swollen left eye and vision loss. Visual acuity was no light perception in both eyes. Six years earlier, he had developed right panophthalmitis, the cause confirmed as Herpes Simplex Virus Type 2 (HSV-2) by polymerase chain reaction (PCR) on aqueous humour (AH) sampling, with a poor outcome. At current presentation, PCR of AH and vitreous samples again confirmed HSV-2 infection. A poor initial response to high-dose intravenous antiviral led to the addition of high-dose systemic corticosteroid. Subsequent phacovitrectomy enabled partial visual recovery. Genetic testing revealed TLR-4 mutation rs4986790 A/G.

Conclusion: HSV-2 panophthalmitis is rare and visually destructive, and we present the first reported case of bilateral disease. We hypothesise that TLR-4 mutation, not previously reported in association with HSV ocular inflammation, may have contributed to excessive inflammation and severe ocular damage. We suggest comprehensive immunological evaluation in all patients with severe viral intraocular inflammation. Prompt diagnosis with PCR and immediate high-dose antiviral therapy is critical and immune abnormality may require amended management.

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来源期刊
CiteScore
6.20
自引率
15.20%
发文量
285
审稿时长
6-12 weeks
期刊介绍: Ocular Immunology & Inflammation ranks 18 out of 59 in the Ophthalmology Category.Ocular Immunology and Inflammation is a peer-reviewed, scientific publication that welcomes the submission of original, previously unpublished manuscripts directed to ophthalmologists and vision scientists. Published bimonthly, the journal provides an international medium for basic and clinical research reports on the ocular inflammatory response and its control by the immune system. The journal publishes original research papers, case reports, reviews, letters to the editor, meeting abstracts, and invited editorials.
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