[烟雾病与RNF213基因]。

Q4 Medicine
Satoru Miyawaki
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引用次数: 0

摘要

随着RNF213作为烟雾病的易感基因被发现,烟雾病的遗传学研究取得了重大进展。大约80%的日本烟雾病患者携带RNF213 p.a g4810lys变异,该变异与临床表型的变化日益相关,包括疾病进展、发病方式和术后结果。值得注意的是,这种变异也与颅内动脉狭窄相关,不符合烟雾病的诊断标准,也与全身血管状况相关,如非心源性缺血性中风、冠状动脉疾病和肺动脉高压。尽管p.Arg4810Lys变异与该病有很强的相关性,但它在大约2%的普通人群中存在,这表明不完全外显性和其他致病因素的参与。与此同时,研究人员已经检查了除p.a g4810lys外的RNF213变体的临床相关性,并确定了可能导致疾病严重程度的罕见突变。此外,大规模遗传分析已经确定了其他易感基因,如DIAPH1和ANO1,其在烟雾病发病机制中的作用仍在研究中。尽管在阐明烟雾病的遗传结构方面取得了实质性进展,但疾病发病的确切机制仍然难以捉摸,这是正在进行的研究的一个重要领域。
本文章由计算机程序翻译,如有差异,请以英文原文为准。
[Moyamoya Disease and the RNF213 Gene].

Genetic research on moyamoya disease(MMD) has advanced significantly following the identification of RNF213 as a susceptibility gene. Approximately 80% of Japanese patients with MMD harbor the RNF213 p.Arg4810Lys variant, which has been increasingly linked to variations in clinical phenotypes, including disease progression, mode of onset, and postoperative outcomes. Of note, this variant is also associated with intracranial arterial stenosis that does not meet the diagnostic criteria for MMD, as well as with systemic vascular conditions such as non-cardioembolic ischemic stroke, coronary artery disease, and pulmonary hypertension. Despite its strong association with the disease, the p.Arg4810Lys variant is present in approximately 2% of the general population, suggesting incomplete penetrance and the involvement of additional pathogenic factors. In parallel, researchers have examined the clinical relevance of RNF213 variants other than p.Arg4810Lys and have identified rare mutations that may contribute to disease severity. Moreover, large-scale genetic analyses have identified additional susceptibility genes, such as DIAPH1 and ANO1, whose roles in MMD pathogenesis remain under investigation. Although substantial advances have been made in elucidating the genetic architecture of MMD, the precise mechanisms underlying disease onset remain elusive and represent an important area of ongoing research.

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来源期刊
Neurological Surgery
Neurological Surgery Medicine-Medicine (all)
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