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Eales 病伴继发性“似外套”样反应:一例报告。

Eales' disease with secondary coats'-like reaction: a case report.

机构信息

Department of Ophthalmology, Faculty of Medicine, 1UM Eye Research Centre (UMERC), Universiti Malaya, Kuala Lumpur, Malaysia.

2Department of Ophthalmology, Faculty of Medicine, Universiti Teknologi MARA, Sungai Buloh Campus, Selangor, Malaysia.

出版信息

BMC Ophthalmol. 2024 Sep 27;24(1):420. doi: 10.1186/s12886-024-03668-3.

DOI:10.1186/s12886-024-03668-3
PMID:39333939
原文链接:https://pmc.ncbi.nlm.nih.gov/articles/PMC11437731/
Abstract

BACKGROUND

Eales' disease is an idiopathic, inflammatory condition characterized by peripheral retinal phlebitis, distal non-perfusion, and neovascularization. Coats' disease, on the other hand, is an idiopathic, retinal vasculopathy characterized by telangiectasia and yellowish exudation. Both diseases commonly affect males. Here, we report an atypical case of unilateral Eales' disease with co-existing, secondary Coats'-like reaction in a 25-year-old male exhibiting unilateral retinal telangiectasia accompanied by subtle macular edema, extensive peripheral vascular sheathing, and distal non-perfusion in the absence of clinically visible exudation.

METHODS

Case report.

RESULTS

A 25-year-old male presented with painless blurring of vision in the right eye for two months. Best-corrected Snellen visual acuity was 3/60 in the affected eye, with relative afferent pupillary defect observed, and 6/9 contralaterally. Anterior segment examination was unremarkable. Right eye fundoscopy showed aneurysmal telangiectasia at the macula, 360 degrees perivascular sheathing and telangiectasia of peripheral retinal vessels. There was no exudation, neovascularization, tractional membranes, vitritis, retinitis, and choroiditis. Left eye fundus was unremarkable. Optical coherence tomography of the macula revealed disorganization and thinning of the inner retinal layers with minimal intraretinal fluid. Fundus fluorescein angiography showed leakage from the aneurysms but not from the sheathed vessels, and an enlarged foveolar avascular zone. A well-defined zone of non-perfusion and retinal vascular telangiectasia with shunts was observed. Blood investigations ruled out metabolic, infectious, haematological, and autoimmune causes. Carotid Doppler showed no stenosis, and CT angiography of the brain did not show any vascular abnormalities. In view that no cause was identified, a diagnosis of Eales' disease with secondary Coats'-like reaction was made. Treatment consisted of FFA-guided laser photocoagulation to the areas of non-perfusion. Intavitreal anti-vascular endothelial growth factor injection was not done in view of poor visual prognosis. The condition and visual acuity remained stable during 18 months of follow-up.

CONCLUSION

This report highlights an atypical case of Eales' disease with co-existing, secondary Coats'-like reaction.

摘要

背景

Eales 病是一种特发性、炎症性疾病,其特征为周边视网膜静脉炎、远端无灌注和新生血管形成。另一方面,Coats 病是一种特发性视网膜血管病变,其特征为毛细血管扩张和黄色渗出。这两种疾病通常都影响男性。在这里,我们报告了一例 25 岁男性单侧 Eales 病伴共存、继发性类似 Coats 病反应的不典型病例,该患者表现为单侧视网膜毛细血管扩张,伴有轻微的黄斑水肿、广泛的周边血管鞘和远端无灌注,但无临床可见的渗出。

方法

病例报告。

结果

一名 25 岁男性因右眼无痛性视力模糊两个月就诊。受影响眼的最佳矫正视力为 3/60,伴有相对传入性瞳孔缺陷,而对侧眼为 6/9。眼前段检查未见异常。右眼眼底检查显示黄斑处有动脉瘤性毛细血管扩张,周边视网膜血管呈 360 度血管鞘和毛细血管扩张。无渗出、新生血管、牵引膜、玻璃体炎症、视网膜炎症和脉络膜炎。左眼眼底无异常。黄斑光学相干断层扫描显示内层视网膜组织排列紊乱和变薄,仅有微量视网膜内液。眼底荧光素血管造影显示动脉瘤渗漏,但血管鞘无渗漏,且中心凹无血管区扩大。观察到一个明确的无灌注区和伴有分流的视网膜血管扩张。血液检查排除了代谢、感染、血液和自身免疫性原因。颈动脉多普勒显示无狭窄,脑部 CT 血管造影未显示任何血管异常。由于未发现任何原因,诊断为 Eales 病伴继发性类似 Coats 病反应。治疗包括根据荧光素血管造影引导对无灌注区进行激光光凝。由于视力预后不佳,未行玻璃体腔内抗血管内皮生长因子注射。在 18 个月的随访期间,病情和视力保持稳定。

结论

本报告强调了一例伴有共存、继发性类似 Coats 病反应的不典型 Eales 病病例。

https://cdn.ncbi.nlm.nih.gov/pmc/blobs/e88f/11437731/4806c66de45d/12886_2024_3668_Fig3_HTML.jpg
https://cdn.ncbi.nlm.nih.gov/pmc/blobs/e88f/11437731/9b4667bbcf32/12886_2024_3668_Fig1_HTML.jpg
https://cdn.ncbi.nlm.nih.gov/pmc/blobs/e88f/11437731/503f89dbae1d/12886_2024_3668_Fig2_HTML.jpg
https://cdn.ncbi.nlm.nih.gov/pmc/blobs/e88f/11437731/4806c66de45d/12886_2024_3668_Fig3_HTML.jpg
https://cdn.ncbi.nlm.nih.gov/pmc/blobs/e88f/11437731/9b4667bbcf32/12886_2024_3668_Fig1_HTML.jpg
https://cdn.ncbi.nlm.nih.gov/pmc/blobs/e88f/11437731/503f89dbae1d/12886_2024_3668_Fig2_HTML.jpg
https://cdn.ncbi.nlm.nih.gov/pmc/blobs/e88f/11437731/4806c66de45d/12886_2024_3668_Fig3_HTML.jpg

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本文引用的文献

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An atypical form of retinitis pigmentosa: A case report.一种非典型性视网膜色素变性:病例报告。
Clin Case Rep. 2023 Nov 15;11(11):e8221. doi: 10.1002/ccr3.8221. eCollection 2023 Nov.
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Coats-like Vasculopathy in Inherited Retinal Disease: Prevalence, Characteristics, Genetics, and Management.遗传性视网膜疾病中的 Coats 样血管病变:患病率、特征、遗传学及治疗。
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成人特发性周边视网膜毛细血管扩张症:病例系列及文献综述
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Indian J Ophthalmol. 2019 Jun;67(6):763-771. doi: 10.4103/ijo.IJO_841_19.
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Secondary Coats' response to retinal vasculitis managed with bevacizumab.继发 Coats 反应对贝伐单抗治疗视网膜血管炎的作用。
Indian J Ophthalmol. 2018 Sep;66(9):1355-1357. doi: 10.4103/ijo.IJO_279_18.
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