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亚急性硬化性全脑炎合并单侧坏死性视网膜炎病例的光学相干断层扫描血管造影随访

Optical Coherence Tomography Angiographic Follow-Up in a Case of Subacute Sclerosing Panencephalitis and Unilateral Necrotising Retinitis.

作者信息

Köksaldı Seher, Ala Rahmi Tumay, Oztura Ibrahim, Emirbayer Emre, Akdal Gulden, Emre Sinan, Tugal-Tutkun Ilknur, Saatci Ali Osman

机构信息

Department of Ophthalmology, Dokuz Eylul University, Izmir, Turkey.

Department of Neurology, Dokuz Eylul University, Izmir, Turkey.

出版信息

Neuroophthalmology. 2023 Nov 22;48(2):142-151. doi: 10.1080/01658107.2023.2284914. eCollection 2024.

Abstract

We present a 20-year-old woman who was diagnosed with subacute sclerosing panencephalitis (SSPE) 20 months after presenting with unilateral retinitis. At presentation, the patient had two inferotemporal macular lesions in her left eye. Corresponding to these areas, optical coherence tomography (OCT) showed hyporeflective spaces with loss of nearly all of the retinal layers. OCT-angiography (OCTA) demonstrated some flow deficit areas with a reduction in the vessel density. Her serum measles antibody titre was high (IgG >5000.0 mIU/ml). Twenty months later the macular lesions had diminished in size, and there was some focal retinal thinning with interruption of the ellipsoid zone. OCTA showed that the flow deficit areas were diminished in size together with the relatively improved perfusion density. Neurological examination disclosed myoclonic jerks. Neuropsychological assessment demonstrated impaired executive function, attention, and narrowed lexical fluency. Measles IgG antibody was high in the cerebrospinal fluid (>230.0 U/ml). Brain magnetic resonance imaging demonstrated bilateral, non-specific, small foci of T2 hyperintensity in the frontoparietal subcortical white matter and centrum semiovale. The present case is the first where OCTA findings of SSPE-related retinal lesions have been described.

摘要

我们报告一名20岁女性,她在出现单侧视网膜炎20个月后被诊断为亚急性硬化性全脑炎(SSPE)。就诊时,患者左眼颞下黄斑区有两处病变。对应这些区域,光学相干断层扫描(OCT)显示低反射区,几乎所有视网膜层均缺失。光学相干断层扫描血管造影(OCTA)显示一些血流缺损区域,血管密度降低。她的血清麻疹抗体滴度很高(IgG>5000.0 mIU/ml)。20个月后,黄斑病变大小减小,出现一些局灶性视网膜变薄,椭圆体带中断。OCTA显示血流缺损区域大小减小,灌注密度相对改善。神经系统检查发现有肌阵挛性抽搐。神经心理学评估显示执行功能、注意力受损,词汇流畅性受限。脑脊液中麻疹IgG抗体很高(>230.0 U/ml)。脑磁共振成像显示双侧额顶叶皮质下白质和半卵圆中心有非特异性小T2高信号灶。本病例是首次描述SSPE相关视网膜病变的OCTA表现。

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本文引用的文献

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