{"title":"甲状腺自身抗体与环指蛋白213变异在烟雾病中的协同作用","authors":"Thiparpa Thamamongood, Shoko Hara, Hiroyuki Akagawa, Motoki Inaji, Yoji Tanaka, Tadashi Nariai, Taketoshi Maehara","doi":"10.2176/jns-nmc.2023-0169","DOIUrl":null,"url":null,"abstract":"<p><p>Recently, thyroid autoantibodies were found to be associated with moyamoya disease (MMD). The ring finger protein 213 (RNF213) p.R4810K variant represents the most important susceptibility genotype of this disease, but its relationship with thyroid autoantibodies remains to be elucidated. Thus, in this study, we aimed to evaluate the clinical relevance of thyroid autoantibodies in each RNF213 genotype in patients with MMD. Included in this study were patients with MMD without a thyroid disease history and in euthyroid status; they were then classified into the mutated or nonmutated based on the RNF213 p.R4810K genotype and positive or negative based on thyroid autoantibody (thyroperoxidase and thyroglobulin) levels. Clinical data of each group were thereafter evaluated. Among the 209 patients, the mutated RNF213 p.R4810K variant and positive thyroid autoantibodies were detected in 155 and 41 patients, respectively. Positive thyroid autoantibodies were found to be more common in the nonmutated patients than in the mutated patients (31.5% vs. 15.5%; P = 0.011). In the mutated patients, as compared to autoantibody-negative patients, autoantibody-positive patients were determined to be more likely to have advanced disease with posterior cerebral artery involvement (54.2% vs. 29.0%; P = 0.017), white matter infarction (58.3% vs. 37.6%; P = 0.046), and a higher modified Rankin Scale at last visit (16.7% vs. 3.1%; P = 0.021). These results suggest that thyroid autoantibodies can act as an immunity inducer in patients with MMD lacking the susceptibility gene RNF213 p.R4810K variant. Moreover, the simultaneous presence of thyroid autoantibodies and the variant seems to aggravate the disease, which indicates synergy between thyroid autoantibodies and the variant.</p>","PeriodicalId":19225,"journal":{"name":"Neurologia medico-chirurgica","volume":" ","pages":"43-49"},"PeriodicalIF":2.4000,"publicationDate":"2024-01-15","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"https://www.ncbi.nlm.nih.gov/pmc/articles/PMC10835577/pdf/","citationCount":"0","resultStr":"{\"title\":\"Synergistic Interaction of Thyroid Autoantibodies and Ring Finger Protein 213 Variant in Moyamoya Disease.\",\"authors\":\"Thiparpa Thamamongood, Shoko Hara, Hiroyuki Akagawa, Motoki Inaji, Yoji Tanaka, Tadashi Nariai, Taketoshi Maehara\",\"doi\":\"10.2176/jns-nmc.2023-0169\",\"DOIUrl\":null,\"url\":null,\"abstract\":\"<p><p>Recently, thyroid autoantibodies were found to be associated with moyamoya disease (MMD). The ring finger protein 213 (RNF213) p.R4810K variant represents the most important susceptibility genotype of this disease, but its relationship with thyroid autoantibodies remains to be elucidated. Thus, in this study, we aimed to evaluate the clinical relevance of thyroid autoantibodies in each RNF213 genotype in patients with MMD. Included in this study were patients with MMD without a thyroid disease history and in euthyroid status; they were then classified into the mutated or nonmutated based on the RNF213 p.R4810K genotype and positive or negative based on thyroid autoantibody (thyroperoxidase and thyroglobulin) levels. Clinical data of each group were thereafter evaluated. Among the 209 patients, the mutated RNF213 p.R4810K variant and positive thyroid autoantibodies were detected in 155 and 41 patients, respectively. Positive thyroid autoantibodies were found to be more common in the nonmutated patients than in the mutated patients (31.5% vs. 15.5%; P = 0.011). In the mutated patients, as compared to autoantibody-negative patients, autoantibody-positive patients were determined to be more likely to have advanced disease with posterior cerebral artery involvement (54.2% vs. 29.0%; P = 0.017), white matter infarction (58.3% vs. 37.6%; P = 0.046), and a higher modified Rankin Scale at last visit (16.7% vs. 3.1%; P = 0.021). These results suggest that thyroid autoantibodies can act as an immunity inducer in patients with MMD lacking the susceptibility gene RNF213 p.R4810K variant. Moreover, the simultaneous presence of thyroid autoantibodies and the variant seems to aggravate the disease, which indicates synergy between thyroid autoantibodies and the variant.</p>\",\"PeriodicalId\":19225,\"journal\":{\"name\":\"Neurologia medico-chirurgica\",\"volume\":\" \",\"pages\":\"43-49\"},\"PeriodicalIF\":2.4000,\"publicationDate\":\"2024-01-15\",\"publicationTypes\":\"Journal Article\",\"fieldsOfStudy\":null,\"isOpenAccess\":false,\"openAccessPdf\":\"https://www.ncbi.nlm.nih.gov/pmc/articles/PMC10835577/pdf/\",\"citationCount\":\"0\",\"resultStr\":null,\"platform\":\"Semanticscholar\",\"paperid\":null,\"PeriodicalName\":\"Neurologia medico-chirurgica\",\"FirstCategoryId\":\"3\",\"ListUrlMain\":\"https://doi.org/10.2176/jns-nmc.2023-0169\",\"RegionNum\":4,\"RegionCategory\":\"医学\",\"ArticlePicture\":[],\"TitleCN\":null,\"AbstractTextCN\":null,\"PMCID\":null,\"EPubDate\":\"2023/12/6 0:00:00\",\"PubModel\":\"Epub\",\"JCR\":\"Q2\",\"JCRName\":\"CLINICAL NEUROLOGY\",\"Score\":null,\"Total\":0}","platform":"Semanticscholar","paperid":null,"PeriodicalName":"Neurologia medico-chirurgica","FirstCategoryId":"3","ListUrlMain":"https://doi.org/10.2176/jns-nmc.2023-0169","RegionNum":4,"RegionCategory":"医学","ArticlePicture":[],"TitleCN":null,"AbstractTextCN":null,"PMCID":null,"EPubDate":"2023/12/6 0:00:00","PubModel":"Epub","JCR":"Q2","JCRName":"CLINICAL NEUROLOGY","Score":null,"Total":0}
引用次数: 0
摘要
近年来,甲状腺自身抗体被发现与烟雾病(MMD)有关。ring finger protein 213 (RNF213) p.R4810K变异是本病最重要的易感基因型,但其与甲状腺自身抗体的关系尚不清楚。因此,在本研究中,我们旨在评估每个RNF213基因型甲状腺自身抗体在烟雾病患者中的临床相关性。本研究包括无甲状腺病史且甲状腺功能正常的烟雾病患者;然后根据RNF213 p.R4810K基因型将他们分为突变或非突变,根据甲状腺自身抗体(甲状腺过氧化物酶和甲状腺球蛋白)水平将他们分为阳性或阴性。评估各组临床资料。在209例患者中,分别有155例和41例患者检测到RNF213 p.R4810K突变和甲状腺自身抗体阳性。甲状腺自身抗体阳性在非突变患者中比在突变患者中更常见(31.5%比15.5%;P = 0.011)。在突变患者中,与自身抗体阴性患者相比,自身抗体阳性患者更有可能出现晚期疾病,并累及大脑后动脉(54.2% vs 29.0%;P = 0.017),白质梗死(58.3% vs. 37.6%;P = 0.046),最后一次访问时修改后的Rankin量表更高(16.7% vs. 3.1%;P = 0.021)。这些结果表明,甲状腺自身抗体可以在缺乏易感基因RNF213 p.R4810K变体的烟雾病患者中发挥免疫诱导作用。此外,甲状腺自身抗体和变异同时存在似乎加重了疾病,这表明甲状腺自身抗体和变异之间存在协同作用。
Synergistic Interaction of Thyroid Autoantibodies and Ring Finger Protein 213 Variant in Moyamoya Disease.
Recently, thyroid autoantibodies were found to be associated with moyamoya disease (MMD). The ring finger protein 213 (RNF213) p.R4810K variant represents the most important susceptibility genotype of this disease, but its relationship with thyroid autoantibodies remains to be elucidated. Thus, in this study, we aimed to evaluate the clinical relevance of thyroid autoantibodies in each RNF213 genotype in patients with MMD. Included in this study were patients with MMD without a thyroid disease history and in euthyroid status; they were then classified into the mutated or nonmutated based on the RNF213 p.R4810K genotype and positive or negative based on thyroid autoantibody (thyroperoxidase and thyroglobulin) levels. Clinical data of each group were thereafter evaluated. Among the 209 patients, the mutated RNF213 p.R4810K variant and positive thyroid autoantibodies were detected in 155 and 41 patients, respectively. Positive thyroid autoantibodies were found to be more common in the nonmutated patients than in the mutated patients (31.5% vs. 15.5%; P = 0.011). In the mutated patients, as compared to autoantibody-negative patients, autoantibody-positive patients were determined to be more likely to have advanced disease with posterior cerebral artery involvement (54.2% vs. 29.0%; P = 0.017), white matter infarction (58.3% vs. 37.6%; P = 0.046), and a higher modified Rankin Scale at last visit (16.7% vs. 3.1%; P = 0.021). These results suggest that thyroid autoantibodies can act as an immunity inducer in patients with MMD lacking the susceptibility gene RNF213 p.R4810K variant. Moreover, the simultaneous presence of thyroid autoantibodies and the variant seems to aggravate the disease, which indicates synergy between thyroid autoantibodies and the variant.