A case of ocular flutter associated with sole anti-sulphatide antibody positivity: successful treatment with intravenous immunoglobulin in a pediatric patient
{"title":"A case of ocular flutter associated with sole anti-sulphatide antibody positivity: successful treatment with intravenous immunoglobulin in a pediatric patient","authors":"Merve Akcay, Safiye Günes Sager, Aysin Tuba Kaplan, Omar Alomari, Yasemin Akin","doi":"10.1007/s13760-024-02645-3","DOIUrl":null,"url":null,"abstract":"<div><h3>Background</h3><p>Ocular flutter is a neurological disorder characterized by irregular, rapid horizontal eye movements and is often associated with autoimmune diseases, infections, drug intoxication, or paraneoplastic syndromes. The brain regions involved in ocular flutter have not been definitively determined. Sulfatide is an acidic glycolipid crucial for maintaining myelin sheath integrity and neuronal transmission. Antibodies against sulfatide can disrupt neuronal signals, and their formation is linked to autoimmune conditions such as Guillain–Barré syndrome and GALOP syndrome. To our knowledge, no pediatric cases of ocular flutter associated with sulfatide antibody-positive neuropathy have been reported.</p><h3>Case description</h3><p>A 15-year-old male with no medical history presented with oscillopsia and blurred vision. His prenatal, natal, and developmental history were unremarkable. Neurological examination revealed rapid, low-amplitude horizontal saccadic oscillations (ocular flutter) with no other neurological abnormalities. Extensive testing, including MRI of the brain and spine; blood tests; lumbar puncture; and screenings for viral, bacterial, and autoimmune conditions, returned normal or negative results. A high titer of anti-sulfatide IgM antibodies was detected. The patient was treated with intravenous immunoglobulin (IVIG), which led to complete resolution of ocular flutter. At the 3-month follow-up, his neurological examination was normal, and he remained asymptomatic with monthly IVIG infusions.</p><h3>Conclusion</h3><p>This is the first reported case of ocular flutter associated solely with anti-sulfatide antibody positivity. This finding underscores the importance of considering sulfatide antibody testing in atypical or treatment-resistant cases of ocular flutter. The resolution of symptoms following IVIG treatment suggests its potential effectiveness in managing sulfatide antibody-positive conditions. Further research is needed to explore the role of sulfatide antibodies in ocular flutter and the benefits of targeted immunotherapy.</p></div>","PeriodicalId":7042,"journal":{"name":"Acta neurologica Belgica","volume":"124 6","pages":"2063 - 2065"},"PeriodicalIF":2.0000,"publicationDate":"2024-09-28","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"","citationCount":"0","resultStr":null,"platform":"Semanticscholar","paperid":null,"PeriodicalName":"Acta neurologica Belgica","FirstCategoryId":"3","ListUrlMain":"https://link.springer.com/article/10.1007/s13760-024-02645-3","RegionNum":4,"RegionCategory":"医学","ArticlePicture":[],"TitleCN":null,"AbstractTextCN":null,"PMCID":null,"EPubDate":"","PubModel":"","JCR":"Q3","JCRName":"CLINICAL NEUROLOGY","Score":null,"Total":0}
引用次数: 0
Abstract
Background
Ocular flutter is a neurological disorder characterized by irregular, rapid horizontal eye movements and is often associated with autoimmune diseases, infections, drug intoxication, or paraneoplastic syndromes. The brain regions involved in ocular flutter have not been definitively determined. Sulfatide is an acidic glycolipid crucial for maintaining myelin sheath integrity and neuronal transmission. Antibodies against sulfatide can disrupt neuronal signals, and their formation is linked to autoimmune conditions such as Guillain–Barré syndrome and GALOP syndrome. To our knowledge, no pediatric cases of ocular flutter associated with sulfatide antibody-positive neuropathy have been reported.
Case description
A 15-year-old male with no medical history presented with oscillopsia and blurred vision. His prenatal, natal, and developmental history were unremarkable. Neurological examination revealed rapid, low-amplitude horizontal saccadic oscillations (ocular flutter) with no other neurological abnormalities. Extensive testing, including MRI of the brain and spine; blood tests; lumbar puncture; and screenings for viral, bacterial, and autoimmune conditions, returned normal or negative results. A high titer of anti-sulfatide IgM antibodies was detected. The patient was treated with intravenous immunoglobulin (IVIG), which led to complete resolution of ocular flutter. At the 3-month follow-up, his neurological examination was normal, and he remained asymptomatic with monthly IVIG infusions.
Conclusion
This is the first reported case of ocular flutter associated solely with anti-sulfatide antibody positivity. This finding underscores the importance of considering sulfatide antibody testing in atypical or treatment-resistant cases of ocular flutter. The resolution of symptoms following IVIG treatment suggests its potential effectiveness in managing sulfatide antibody-positive conditions. Further research is needed to explore the role of sulfatide antibodies in ocular flutter and the benefits of targeted immunotherapy.
期刊介绍：
Peer-reviewed and published quarterly, Acta Neurologica Belgicapresents original articles in the clinical and basic neurosciences, and also reports the proceedings and the abstracts of the scientific meetings of the different partner societies. The contents include commentaries, editorials, review articles, case reports, neuro-images of interest, book reviews and letters to the editor.
Acta Neurologica Belgica is the official journal of the following national societies:
Belgian Neurological Society
Belgian Society for Neuroscience
Belgian Society of Clinical Neurophysiology
Belgian Pediatric Neurology Society
Belgian Study Group of Multiple Sclerosis
Belgian Stroke Council
Belgian Headache Society
Belgian Study Group of Neuropathology
求助内容：
应助结果提醒方式：
京公网安备 11010802042870号
