{"title":"一例罕见的类固醇耐药肾病综合征并发韦尼克脑病。","authors":"Kazuya Yatani, Hiroshi Kaito, Yosuke Inaguma, Ryojiro Tanaka, Kazumoto Iijima","doi":"10.1007/s00467-025-06662-z","DOIUrl":null,"url":null,"abstract":"<p><p>Wernicke's encephalopathy (WE) is a severe neurological condition caused by the deficiency of thiamine, which is a vitamin B1 molecule. Herein, we present the case of a 3-year-old girl with steroid-resistant nephrotic syndrome (SRNS) who did not achieve remission despite steroid pulse therapy (MPT) and rituximab. She had frequent vomiting and decreased oral intake on the 61st day. High-calorie total parenteral nutrition was initiated on the 86th day, and levothyroxine was administered for suspected hypothyroidism. On the 88th day, she experienced altered consciousness with ocular deviation and abnormal electroencephalography findings, raising suspicion of WE. Following thiamine supplementation, recurrence of symptoms and neurological sequelae were not observed. Following 10 courses of MPT, rituximab, and mycophenolate mofetil, the patient achieved incomplete remission from SRNS. Monitoring levels of water-soluble vitamins in prolonged cases of nephrotic syndrome, such as SRNS, is critical to prevent WE and irreversible neurological damage.</p>","PeriodicalId":19735,"journal":{"name":"Pediatric Nephrology","volume":" ","pages":""},"PeriodicalIF":2.6000,"publicationDate":"2025-01-28","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"","citationCount":"0","resultStr":"{\"title\":\"A rare case of steroid-resistant nephrotic syndrome complicated by Wernicke's encephalopathy.\",\"authors\":\"Kazuya Yatani, Hiroshi Kaito, Yosuke Inaguma, Ryojiro Tanaka, Kazumoto Iijima\",\"doi\":\"10.1007/s00467-025-06662-z\",\"DOIUrl\":null,\"url\":null,\"abstract\":\"<p><p>Wernicke's encephalopathy (WE) is a severe neurological condition caused by the deficiency of thiamine, which is a vitamin B1 molecule. Herein, we present the case of a 3-year-old girl with steroid-resistant nephrotic syndrome (SRNS) who did not achieve remission despite steroid pulse therapy (MPT) and rituximab. She had frequent vomiting and decreased oral intake on the 61st day. High-calorie total parenteral nutrition was initiated on the 86th day, and levothyroxine was administered for suspected hypothyroidism. On the 88th day, she experienced altered consciousness with ocular deviation and abnormal electroencephalography findings, raising suspicion of WE. Following thiamine supplementation, recurrence of symptoms and neurological sequelae were not observed. Following 10 courses of MPT, rituximab, and mycophenolate mofetil, the patient achieved incomplete remission from SRNS. Monitoring levels of water-soluble vitamins in prolonged cases of nephrotic syndrome, such as SRNS, is critical to prevent WE and irreversible neurological damage.</p>\",\"PeriodicalId\":19735,\"journal\":{\"name\":\"Pediatric Nephrology\",\"volume\":\" \",\"pages\":\"\"},\"PeriodicalIF\":2.6000,\"publicationDate\":\"2025-01-28\",\"publicationTypes\":\"Journal Article\",\"fieldsOfStudy\":null,\"isOpenAccess\":false,\"openAccessPdf\":\"\",\"citationCount\":\"0\",\"resultStr\":null,\"platform\":\"Semanticscholar\",\"paperid\":null,\"PeriodicalName\":\"Pediatric Nephrology\",\"FirstCategoryId\":\"3\",\"ListUrlMain\":\"https://doi.org/10.1007/s00467-025-06662-z\",\"RegionNum\":3,\"RegionCategory\":\"医学\",\"ArticlePicture\":[],\"TitleCN\":null,\"AbstractTextCN\":null,\"PMCID\":null,\"EPubDate\":\"\",\"PubModel\":\"\",\"JCR\":\"Q1\",\"JCRName\":\"PEDIATRICS\",\"Score\":null,\"Total\":0}","platform":"Semanticscholar","paperid":null,"PeriodicalName":"Pediatric Nephrology","FirstCategoryId":"3","ListUrlMain":"https://doi.org/10.1007/s00467-025-06662-z","RegionNum":3,"RegionCategory":"医学","ArticlePicture":[],"TitleCN":null,"AbstractTextCN":null,"PMCID":null,"EPubDate":"","PubModel":"","JCR":"Q1","JCRName":"PEDIATRICS","Score":null,"Total":0}
引用次数: 0
摘要
韦尼克脑病(Wernicke's encephalopathy,WE)是一种严重的神经系统疾病,由缺乏维生素 B1 分子硫胺素引起。在此,我们介绍了一例患有类固醇耐药肾病综合征(SRNS)的 3 岁女孩的病例,尽管她接受了类固醇脉冲疗法(MPT)和利妥昔单抗治疗,但病情仍未缓解。第 61 天,她频繁呕吐,口服量减少。第86天开始使用高热量全肠外营养,并因怀疑甲状腺功能减退而使用左甲状腺素。第 88 天,她出现意识改变,眼球偏转,脑电图检查结果异常,这引起了对 WE 的怀疑。补充硫胺素后,症状和神经系统后遗症没有复发。经过10个疗程的MPT、利妥昔单抗和霉酚酸酯治疗后,患者的SRNS症状得到完全缓解。在肾病综合征(如 SRNS)病程较长的病例中,监测水溶性维生素的水平对于预防 WE 和不可逆的神经损伤至关重要。
A rare case of steroid-resistant nephrotic syndrome complicated by Wernicke's encephalopathy.
Wernicke's encephalopathy (WE) is a severe neurological condition caused by the deficiency of thiamine, which is a vitamin B1 molecule. Herein, we present the case of a 3-year-old girl with steroid-resistant nephrotic syndrome (SRNS) who did not achieve remission despite steroid pulse therapy (MPT) and rituximab. She had frequent vomiting and decreased oral intake on the 61st day. High-calorie total parenteral nutrition was initiated on the 86th day, and levothyroxine was administered for suspected hypothyroidism. On the 88th day, she experienced altered consciousness with ocular deviation and abnormal electroencephalography findings, raising suspicion of WE. Following thiamine supplementation, recurrence of symptoms and neurological sequelae were not observed. Following 10 courses of MPT, rituximab, and mycophenolate mofetil, the patient achieved incomplete remission from SRNS. Monitoring levels of water-soluble vitamins in prolonged cases of nephrotic syndrome, such as SRNS, is critical to prevent WE and irreversible neurological damage.
期刊介绍:
International Pediatric Nephrology Association
Pediatric Nephrology publishes original clinical research related to acute and chronic diseases that affect renal function, blood pressure, and fluid and electrolyte disorders in children. Studies may involve medical, surgical, nutritional, physiologic, biochemical, genetic, pathologic or immunologic aspects of disease, imaging techniques or consequences of acute or chronic kidney disease. There are 12 issues per year that contain Editorial Commentaries, Reviews, Educational Reviews, Original Articles, Brief Reports, Rapid Communications, Clinical Quizzes, and Letters to the Editors.