Murai Yasuo, Matano Fumihiro, Kubota Asami, Nounaka Yohei, Ishisaka Eitaro, Shirokane Kazutaka, Koketsu Kenta, Nakae Ryuta, Tamaki Tomonori
Department of Neurological Surgery, Nippon Medical School Hospital.
Department of Neurological Surgery, Nippon Medical School Musashi Kosugi Hospital.
J Nippon Med Sch. 2024;91(2):140-145. doi: 10.1272/jnms.JNMS.2024_91-215.
Moyamoya disease (MMD) is a cerebrovascular disorder that is predominantly observed in women of East Asian descent, and is characterized by progressive stenosis of the internal carotid artery, beginning in early childhood, and a distinctive network of collateral vessels known as "moyamoya vessels" in the basal ganglia. Additionally, a prevalent genetic variant found in most MMD cases is the p.R4810K polymorphism of RNF213 on chromosome 17q25.3. Recent studies have revealed that RNF213 mutations are associated not only with MMD, but also with other systemic vascular disorders, including intracranial atherosclerosis and systemic vascular abnormalities such as pulmonary artery stenosis and coronary artery diseases. Therefore, the concept of "RNF213-related vasculopathy" has been proposed. This review focuses on polymorphisms in the RNF213 gene and describes a wide range of clinical and genetic phenotypes associated with RNF213-related vasculopathy. The RNF213 gene has been suggested to play an important role in the pathogenesis of vascular diseases and developing new therapies. Therefore, further research and knowledge sharing through collaboration between clinicians and researchers are required.
烟雾病(MMD)是一种主要在东亚裔女性中观察到的脑血管疾病,其特征是颈内动脉自儿童早期开始进行性狭窄,以及基底节区出现独特的侧支血管网络,即“烟雾血管”。此外,在大多数烟雾病病例中发现的一种常见基因变异是17q25.3染色体上RNF213的p.R4810K多态性。最近的研究表明,RNF213突变不仅与烟雾病有关,还与其他全身性血管疾病有关,包括颅内动脉粥样硬化和全身性血管异常,如肺动脉狭窄和冠状动脉疾病。因此,有人提出了“RNF213相关血管病”的概念。本综述重点关注RNF213基因的多态性,并描述与RNF213相关血管病相关的广泛临床和遗传表型。RNF213基因被认为在血管疾病的发病机制和开发新疗法中起重要作用。因此,临床医生和研究人员之间需要通过合作进行进一步的研究和知识共享。