{"title":"一例抗 Ro/SSA 抗体升高的表层蚕豆病病例","authors":"Shoji Watanabe, Maulidina Amalia Putri, Hitoshi Yamahata, Ryosuke Hanaya","doi":"10.2176/jns-nmc.2023-0214","DOIUrl":null,"url":null,"abstract":"<p><p>Superficial siderosis (SS) of the central nervous system is a rare disorder that is caused by chronic or recurrent hemorrhage in the subarachnoid space via a dural defect at the spinal level. The most common clinical features of SS include slow-progressive sensorineural deafness, cerebellar symptoms, and pyramidal tract signs. Considering that SS can present with broad clinical manifestations, for precise diagnosis, this disease must be understood. Anti-Ro/SSA antibodies are commonly detected in patients with Sjögren's syndrome and are utilized as markers for autoimmune diseases. In this report, we present a unique pathological condition in which SS coincided with a positive anti-Ro/SSA antibody test result. During the diagnosis of gait disturbance, an elevation in anti-Ro/SSA antibody was detected, and steroid pulse therapy was initiated as the initial treatment for autoimmune diseases. Head magnetic resonance imaging (MRI) revealed extensive hypointensity as a dark band that surrounded the intracranial basal structures and cerebellar hemispheres. Spinal MRI indicated ventral longitudinal intraspinal fluid collection extending from C7 to T5 as well as a defect in the ventral T2-3 dura mater. Intraoperative visualization revealed that the intradural venous plexus was the source of bleeding that caused the SS. To our knowledge, this report is the first to discuss the presence of anti-Ro/SSA antibodies in patients with SS. The role of anti-Ro/SSA antibodies in the pathophysiology of SS remains unclear; therefore, to confirm a possible association, further research and accumulation of cases are required.</p>","PeriodicalId":101331,"journal":{"name":"NMC case report journal","volume":"11 ","pages":"151-155"},"PeriodicalIF":0.0000,"publicationDate":"2024-05-28","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"https://www.ncbi.nlm.nih.gov/pmc/articles/PMC11190658/pdf/","citationCount":"0","resultStr":"{\"title\":\"A Case of Superficial Siderosis with Elevated Anti-Ro/SSA Antibody.\",\"authors\":\"Shoji Watanabe, Maulidina Amalia Putri, Hitoshi Yamahata, Ryosuke Hanaya\",\"doi\":\"10.2176/jns-nmc.2023-0214\",\"DOIUrl\":null,\"url\":null,\"abstract\":\"<p><p>Superficial siderosis (SS) of the central nervous system is a rare disorder that is caused by chronic or recurrent hemorrhage in the subarachnoid space via a dural defect at the spinal level. The most common clinical features of SS include slow-progressive sensorineural deafness, cerebellar symptoms, and pyramidal tract signs. Considering that SS can present with broad clinical manifestations, for precise diagnosis, this disease must be understood. Anti-Ro/SSA antibodies are commonly detected in patients with Sjögren's syndrome and are utilized as markers for autoimmune diseases. In this report, we present a unique pathological condition in which SS coincided with a positive anti-Ro/SSA antibody test result. During the diagnosis of gait disturbance, an elevation in anti-Ro/SSA antibody was detected, and steroid pulse therapy was initiated as the initial treatment for autoimmune diseases. Head magnetic resonance imaging (MRI) revealed extensive hypointensity as a dark band that surrounded the intracranial basal structures and cerebellar hemispheres. Spinal MRI indicated ventral longitudinal intraspinal fluid collection extending from C7 to T5 as well as a defect in the ventral T2-3 dura mater. Intraoperative visualization revealed that the intradural venous plexus was the source of bleeding that caused the SS. To our knowledge, this report is the first to discuss the presence of anti-Ro/SSA antibodies in patients with SS. The role of anti-Ro/SSA antibodies in the pathophysiology of SS remains unclear; therefore, to confirm a possible association, further research and accumulation of cases are required.</p>\",\"PeriodicalId\":101331,\"journal\":{\"name\":\"NMC case report journal\",\"volume\":\"11 \",\"pages\":\"151-155\"},\"PeriodicalIF\":0.0000,\"publicationDate\":\"2024-05-28\",\"publicationTypes\":\"Journal Article\",\"fieldsOfStudy\":null,\"isOpenAccess\":false,\"openAccessPdf\":\"https://www.ncbi.nlm.nih.gov/pmc/articles/PMC11190658/pdf/\",\"citationCount\":\"0\",\"resultStr\":null,\"platform\":\"Semanticscholar\",\"paperid\":null,\"PeriodicalName\":\"NMC case report journal\",\"FirstCategoryId\":\"1085\",\"ListUrlMain\":\"https://doi.org/10.2176/jns-nmc.2023-0214\",\"RegionNum\":0,\"RegionCategory\":null,\"ArticlePicture\":[],\"TitleCN\":null,\"AbstractTextCN\":null,\"PMCID\":null,\"EPubDate\":\"2024/1/1 0:00:00\",\"PubModel\":\"eCollection\",\"JCR\":\"\",\"JCRName\":\"\",\"Score\":null,\"Total\":0}","platform":"Semanticscholar","paperid":null,"PeriodicalName":"NMC case report journal","FirstCategoryId":"1085","ListUrlMain":"https://doi.org/10.2176/jns-nmc.2023-0214","RegionNum":0,"RegionCategory":null,"ArticlePicture":[],"TitleCN":null,"AbstractTextCN":null,"PMCID":null,"EPubDate":"2024/1/1 0:00:00","PubModel":"eCollection","JCR":"","JCRName":"","Score":null,"Total":0}
引用次数: 0
摘要
中枢神经系统表层蛛网膜下腔出血(SS)是一种罕见的疾病,由脊柱硬膜缺损导致的蛛网膜下腔慢性或复发性出血引起。SS 最常见的临床特征包括缓慢进展的感音神经性耳聋、小脑症状和锥体束征。考虑到 SS 可有广泛的临床表现,要准确诊断这种疾病,必须对其有所了解。抗 Ro/SSA 抗体通常在斯约格伦综合征患者中检测到,并被用作自身免疫性疾病的标志物。在本报告中,我们介绍了一种独特的病理情况,即斯氏综合征与抗Ro/SSA抗体阳性检测结果同时出现。在诊断步态障碍期间,检测到抗 Ro/SSA 抗体升高,并启动了类固醇脉冲疗法作为自身免疫性疾病的初始治疗。头部磁共振成像(MRI)显示,颅内基底结构和小脑半球周围有广泛的低密度暗带。脊柱磁共振成像显示,腹侧纵向椎管内积液从C7延伸至T5,腹侧T2-3硬脑膜也有缺损。术中观察发现,硬膜内静脉丛是导致 SS 的出血源。据我们所知,该报告首次讨论了SS患者体内存在抗Ro/SSA抗体的问题。抗Ro/SSA抗体在SS病理生理学中的作用仍不清楚;因此,要证实可能的关联性,还需要进一步的研究和病例的积累。
A Case of Superficial Siderosis with Elevated Anti-Ro/SSA Antibody.
Superficial siderosis (SS) of the central nervous system is a rare disorder that is caused by chronic or recurrent hemorrhage in the subarachnoid space via a dural defect at the spinal level. The most common clinical features of SS include slow-progressive sensorineural deafness, cerebellar symptoms, and pyramidal tract signs. Considering that SS can present with broad clinical manifestations, for precise diagnosis, this disease must be understood. Anti-Ro/SSA antibodies are commonly detected in patients with Sjögren's syndrome and are utilized as markers for autoimmune diseases. In this report, we present a unique pathological condition in which SS coincided with a positive anti-Ro/SSA antibody test result. During the diagnosis of gait disturbance, an elevation in anti-Ro/SSA antibody was detected, and steroid pulse therapy was initiated as the initial treatment for autoimmune diseases. Head magnetic resonance imaging (MRI) revealed extensive hypointensity as a dark band that surrounded the intracranial basal structures and cerebellar hemispheres. Spinal MRI indicated ventral longitudinal intraspinal fluid collection extending from C7 to T5 as well as a defect in the ventral T2-3 dura mater. Intraoperative visualization revealed that the intradural venous plexus was the source of bleeding that caused the SS. To our knowledge, this report is the first to discuss the presence of anti-Ro/SSA antibodies in patients with SS. The role of anti-Ro/SSA antibodies in the pathophysiology of SS remains unclear; therefore, to confirm a possible association, further research and accumulation of cases are required.