{"title":"髓鞘少突胶质细胞糖蛋白抗体相关性脑脊髓炎患儿隐匿性软骨脉络膜瘤","authors":"Sayaka Enomoto , Hiroki Tsuchiya , Yoshiyuki Ayada , Yoshiki Takai , Toshiki Takenouchi","doi":"10.1016/j.jneuroim.2025.578674","DOIUrl":null,"url":null,"abstract":"<div><div>Myelin oligodendrocyte glycoprotein antibody-associated disease is a central inflammatory demyelinating disorder that cause diverse neurological symptoms. Paraneoplastic neurologic syndromes occur in association with malignant neoplasms and manifest with a wide range of neuropsychiatric symptoms. Paraneoplastic neurological syndrome is rare in myelin oligodendrocyte glycoprotein antibody-associated disease and, when reported, is typically seen in adults. Herein, we report a 3-year-old girl who presented with lower limb paralysis, urinary retention, and seropositivity for myelin oligodendrocyte glycoprotein antibody. Her symptoms did not respond to various immunotherapies. She was incidentally found to have a cartilaginous choristoma in the neck, after surgical resection of which, she showed symptomatic recovery without recurrence. Immunohistochemical analysis of the resected tumor revealed expression of myelin oligodendrocyte glycoprotein within the cartilage tissue. Based on this case experience, children presenting with myelin oligodendrocyte glycoprotein antibody-associated disease who fail to respond to immunotherapies may need prompt screening for occult tumors.</div></div>","PeriodicalId":16671,"journal":{"name":"Journal of neuroimmunology","volume":"406 ","pages":"Article 578674"},"PeriodicalIF":2.5000,"publicationDate":"2025-06-19","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"","citationCount":"0","resultStr":"{\"title\":\"Occult cartilaginous choristoma in a child with myelin oligodendrocyte glycoprotein antibody-associated encephalomyelitis\",\"authors\":\"Sayaka Enomoto , Hiroki Tsuchiya , Yoshiyuki Ayada , Yoshiki Takai , Toshiki Takenouchi\",\"doi\":\"10.1016/j.jneuroim.2025.578674\",\"DOIUrl\":null,\"url\":null,\"abstract\":\"<div><div>Myelin oligodendrocyte glycoprotein antibody-associated disease is a central inflammatory demyelinating disorder that cause diverse neurological symptoms. Paraneoplastic neurologic syndromes occur in association with malignant neoplasms and manifest with a wide range of neuropsychiatric symptoms. Paraneoplastic neurological syndrome is rare in myelin oligodendrocyte glycoprotein antibody-associated disease and, when reported, is typically seen in adults. Herein, we report a 3-year-old girl who presented with lower limb paralysis, urinary retention, and seropositivity for myelin oligodendrocyte glycoprotein antibody. Her symptoms did not respond to various immunotherapies. She was incidentally found to have a cartilaginous choristoma in the neck, after surgical resection of which, she showed symptomatic recovery without recurrence. Immunohistochemical analysis of the resected tumor revealed expression of myelin oligodendrocyte glycoprotein within the cartilage tissue. Based on this case experience, children presenting with myelin oligodendrocyte glycoprotein antibody-associated disease who fail to respond to immunotherapies may need prompt screening for occult tumors.</div></div>\",\"PeriodicalId\":16671,\"journal\":{\"name\":\"Journal of neuroimmunology\",\"volume\":\"406 \",\"pages\":\"Article 578674\"},\"PeriodicalIF\":2.5000,\"publicationDate\":\"2025-06-19\",\"publicationTypes\":\"Journal Article\",\"fieldsOfStudy\":null,\"isOpenAccess\":false,\"openAccessPdf\":\"\",\"citationCount\":\"0\",\"resultStr\":null,\"platform\":\"Semanticscholar\",\"paperid\":null,\"PeriodicalName\":\"Journal of neuroimmunology\",\"FirstCategoryId\":\"3\",\"ListUrlMain\":\"https://www.sciencedirect.com/science/article/pii/S0165572825001559\",\"RegionNum\":4,\"RegionCategory\":\"医学\",\"ArticlePicture\":[],\"TitleCN\":null,\"AbstractTextCN\":null,\"PMCID\":null,\"EPubDate\":\"\",\"PubModel\":\"\",\"JCR\":\"Q3\",\"JCRName\":\"IMMUNOLOGY\",\"Score\":null,\"Total\":0}","platform":"Semanticscholar","paperid":null,"PeriodicalName":"Journal of neuroimmunology","FirstCategoryId":"3","ListUrlMain":"https://www.sciencedirect.com/science/article/pii/S0165572825001559","RegionNum":4,"RegionCategory":"医学","ArticlePicture":[],"TitleCN":null,"AbstractTextCN":null,"PMCID":null,"EPubDate":"","PubModel":"","JCR":"Q3","JCRName":"IMMUNOLOGY","Score":null,"Total":0}
Occult cartilaginous choristoma in a child with myelin oligodendrocyte glycoprotein antibody-associated encephalomyelitis
Myelin oligodendrocyte glycoprotein antibody-associated disease is a central inflammatory demyelinating disorder that cause diverse neurological symptoms. Paraneoplastic neurologic syndromes occur in association with malignant neoplasms and manifest with a wide range of neuropsychiatric symptoms. Paraneoplastic neurological syndrome is rare in myelin oligodendrocyte glycoprotein antibody-associated disease and, when reported, is typically seen in adults. Herein, we report a 3-year-old girl who presented with lower limb paralysis, urinary retention, and seropositivity for myelin oligodendrocyte glycoprotein antibody. Her symptoms did not respond to various immunotherapies. She was incidentally found to have a cartilaginous choristoma in the neck, after surgical resection of which, she showed symptomatic recovery without recurrence. Immunohistochemical analysis of the resected tumor revealed expression of myelin oligodendrocyte glycoprotein within the cartilage tissue. Based on this case experience, children presenting with myelin oligodendrocyte glycoprotein antibody-associated disease who fail to respond to immunotherapies may need prompt screening for occult tumors.
期刊介绍:
The Journal of Neuroimmunology affords a forum for the publication of works applying immunologic methodology to the furtherance of the neurological sciences. Studies on all branches of the neurosciences, particularly fundamental and applied neurobiology, neurology, neuropathology, neurochemistry, neurovirology, neuroendocrinology, neuromuscular research, neuropharmacology and psychology, which involve either immunologic methodology (e.g. immunocytochemistry) or fundamental immunology (e.g. antibody and lymphocyte assays), are considered for publication.