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烟雾病在一名携带此前未报道的RNF213错义变异(p.Thr554Ile)的印度女性中表现为短暂性非局灶性神经发作。

Moyamoya disease presenting with transient nonfocal neurological attacks in an Indian woman carrying a previously unreported RNF213 missense variant (p.Thr554Ile).

作者信息

Mondal Ritwick, Deb Shramana, Ray Nirmalya, Purakayastha Sukalyan, Tiwari Mona, Benito-León Julián, Roy Jayanta

机构信息

Department of Stroke Medicine, Institute of Neurosciences, Kolkata, India.

Department of Neuroradiology, Manipal Group of Hospitals, Kolkata, India.

出版信息

Neurogenetics. 2025 Jul 19;26(1):54. doi: 10.1007/s10048-025-00836-5.

Abstract

Moyamoya disease is a rare cerebrovascular disorder characterized by progressive internal carotid artery stenosis and compensatory collateral vessel formation, producing a characteristic "puff of smoke" angiographic appearance. Genetic predisposition, particularly involving the RNF213 gene, plays a central role. We report a 48-year-old Indian woman with type 2 diabetes, arterial hypertension, and a prior transient ischemic attack who presented with intermittent bilateral upper limb paresthesia. Imaging revealed bilateral supraclinoid internal carotid artery stenosis (Suzuki stage III). Genetic testing identified a heterozygous RNF213 missense variant (Thr554Ile, rs766831703), which is extremely rare in global databases and predicted to be deleterious by multiple in silico tools. This variant has not been previously described in association with Moyamoya disease. The patient underwent bilateral superficial temporal artery to middle cerebral artery bypass, achieving sustained clinical improvement without recurrent events over two years.

摘要

烟雾病是一种罕见的脑血管疾病,其特征是颈内动脉进行性狭窄和代偿性侧支血管形成,产生特征性的“烟雾”血管造影表现。遗传易感性,特别是涉及RNF213基因,起着核心作用。我们报告一名48岁的印度女性,患有2型糖尿病、动脉高血压,既往有短暂性脑缺血发作,出现间歇性双侧上肢感觉异常。影像学检查显示双侧鞍上颈内动脉狭窄(铃木分期III期)。基因检测发现一个杂合的RNF213错义变异(Thr554Ile,rs766831703),在全球数据库中极为罕见,多种计算机模拟工具预测该变异有害。此前尚未有该变异与烟雾病相关的描述。该患者接受了双侧颞浅动脉至大脑中动脉搭桥手术,术后两年临床持续改善,无复发事件。

https://cdn.ncbi.nlm.nih.gov/pmc/blobs/5100/12276106/396a1d6dc88b/10048_2025_836_Fig1_HTML.jpg

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