Annals of Neurology最新文献

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Track Sign Distal to Internal Carotid Artery Occlusion 颈内动脉闭塞远端轨道征。
IF 8.1 1区 医学
Annals of Neurology Pub Date : 2025-03-11 DOI: 10.1002/ana.27234
Ying Yu MD, Jiabao Yang MD, Ning Ma MD
{"title":"Track Sign Distal to Internal Carotid Artery Occlusion","authors":"Ying Yu MD, Jiabao Yang MD, Ning Ma MD","doi":"10.1002/ana.27234","DOIUrl":"10.1002/ana.27234","url":null,"abstract":"","PeriodicalId":127,"journal":{"name":"Annals of Neurology","volume":"98 1","pages":"183-184"},"PeriodicalIF":8.1,"publicationDate":"2025-03-11","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"143595938","PeriodicalName":null,"FirstCategoryId":null,"ListUrlMain":null,"RegionNum":1,"RegionCategory":"医学","ArticlePicture":[],"TitleCN":null,"AbstractTextCN":null,"PMCID":"","EPubDate":null,"PubModel":null,"JCR":null,"JCRName":null,"Score":null,"Total":0}
引用次数: 0
Network Localization of Pediatric Lesion-Induced Dystonia 小儿病变性肌张力障碍的网络定位。
IF 8.1 1区 医学
Annals of Neurology Pub Date : 2025-03-10 DOI: 10.1002/ana.27224
Rose Gelineau-Morel MD, Nomazulu Dlamini MD, PhD, Joel Bruss BA, Alexander L. Cohen MD, PhD, Amanda Robertson MSc, Dimitrios Alexopoulos MS,, Christopher D. Smyser MD, Aaron D. Boes MD, PhD
{"title":"Network Localization of Pediatric Lesion-Induced Dystonia","authors":"Rose Gelineau-Morel MD,&nbsp;Nomazulu Dlamini MD, PhD,&nbsp;Joel Bruss BA,&nbsp;Alexander L. Cohen MD, PhD,&nbsp;Amanda Robertson MSc,&nbsp;Dimitrios Alexopoulos MS,,&nbsp;Christopher D. Smyser MD,&nbsp;Aaron D. Boes MD, PhD","doi":"10.1002/ana.27224","DOIUrl":"10.1002/ana.27224","url":null,"abstract":"<div>\u0000 \u0000 <section>\u0000 \u0000 <h3> Objective</h3>\u0000 \u0000 <p>Dystonia is a movement disorder defined by involuntary muscle contractions leading to abnormal postures or twisting and repetitive movements. Classically dystonia has been thought of as a disorder of the basal ganglia, but newer results in idiopathic dystonia and lesion-induced dystonia in adults point to broader motor network dysfunction spanning the basal ganglia, cerebellum, premotor cortex, sensorimotor, and frontoparietal regions. It is unclear whether a similar network is shared between different etiologies of pediatric lesion-induced dystonia.</p>\u0000 </section>\u0000 \u0000 <section>\u0000 \u0000 <h3> Methods</h3>\u0000 \u0000 <p>Three cohorts of pediatric patients with lesion-induced dystonia were identified. The lesion etiologies included hypoxia, kernicterus, and stroke versus comparison subjects with acquired lesions not associated with dystonia. Multivariate lesion-symptom mapping and lesion network mapping were used to evaluate the anatomy and networks associated with dystonia.</p>\u0000 </section>\u0000 \u0000 <section>\u0000 \u0000 <h3> Results</h3>\u0000 \u0000 <p>Multivariate lesion-symptom mapping showed that lesions of the putamen and globus pallidus were associated with dystonia (<i>r</i> = 0.41, <i>p</i> &lt; 0.001). Lesion network mapping using normative connectome data from healthy children demonstrated that these regional findings occurred within a common brain-wide network that involves the basal ganglia, anterior and medial cerebellum, and cortical regions that overlap the cingulo-opercular action-mode and somato-cognitive-action networks.</p>\u0000 </section>\u0000 \u0000 <section>\u0000 \u0000 <h3> Interpretation</h3>\u0000 \u0000 <p>We interpret these findings as novel evidence for a unified dystonia brain network that involves the somato-cognitive-action network, which is implicated in the coordination of movement. Elucidation of this network gives insight into the functional origins of dystonia and provides novel targets to investigate for therapeutic intervention. ANN NEUROL 2025;98:152–162</p>\u0000 </section>\u0000 </div>","PeriodicalId":127,"journal":{"name":"Annals of Neurology","volume":"98 1","pages":"152-162"},"PeriodicalIF":8.1,"publicationDate":"2025-03-10","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"143584003","PeriodicalName":null,"FirstCategoryId":null,"ListUrlMain":null,"RegionNum":1,"RegionCategory":"医学","ArticlePicture":[],"TitleCN":null,"AbstractTextCN":null,"PMCID":"","EPubDate":null,"PubModel":null,"JCR":null,"JCRName":null,"Score":null,"Total":0}
引用次数: 0
Annals of Neurology: Volume 97, Number 4, April 2025 神经病学年鉴:第97卷,第4号,2025年4月
IF 8.1 1区 医学
Annals of Neurology Pub Date : 2025-03-08 DOI: 10.1002/ana.26979
{"title":"Annals of Neurology: Volume 97, Number 4, April 2025","authors":"","doi":"10.1002/ana.26979","DOIUrl":"https://doi.org/10.1002/ana.26979","url":null,"abstract":"","PeriodicalId":127,"journal":{"name":"Annals of Neurology","volume":"97 4","pages":"C1"},"PeriodicalIF":8.1,"publicationDate":"2025-03-08","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"https://onlinelibrary.wiley.com/doi/epdf/10.1002/ana.26979","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"143571219","PeriodicalName":null,"FirstCategoryId":null,"ListUrlMain":null,"RegionNum":1,"RegionCategory":"医学","ArticlePicture":[],"TitleCN":null,"AbstractTextCN":null,"PMCID":"OA","EPubDate":null,"PubModel":null,"JCR":null,"JCRName":null,"Score":null,"Total":0}
引用次数: 0
Blood DDIT4 and TRIM13 Transcript Levels Mark the Early Stages of Machado–Joseph Disease 血液DDIT4和TRIM13转录物水平标志着马查多-约瑟夫病的早期阶段。
IF 8.1 1区 医学
Annals of Neurology Pub Date : 2025-03-05 DOI: 10.1002/ana.27222
Ana F. Ferreira PhD, Mafalda Raposo PhD, Emily D. Shaw MSc, Louisa Liu, João Vasconcelos MD, Teresa Kay MD, Conceição Bettencourt PhD, Maria Luiza Saraiva-Pereira MD, PhD, Laura Bannach Jardim MD, PhD, Maria do Carmo Costa PhD, Manuela Lima PhD
{"title":"Blood DDIT4 and TRIM13 Transcript Levels Mark the Early Stages of Machado–Joseph Disease","authors":"Ana F. Ferreira PhD,&nbsp;Mafalda Raposo PhD,&nbsp;Emily D. Shaw MSc,&nbsp;Louisa Liu,&nbsp;João Vasconcelos MD,&nbsp;Teresa Kay MD,&nbsp;Conceição Bettencourt PhD,&nbsp;Maria Luiza Saraiva-Pereira MD, PhD,&nbsp;Laura Bannach Jardim MD, PhD,&nbsp;Maria do Carmo Costa PhD,&nbsp;Manuela Lima PhD","doi":"10.1002/ana.27222","DOIUrl":"10.1002/ana.27222","url":null,"abstract":"<div>\u0000 \u0000 <section>\u0000 \u0000 <h3> Objective</h3>\u0000 \u0000 <p>An abundance of select transcripts and proteins has been found to be dysregulated in blood samples of Machado–Joseph disease (MJD) carriers. Here, we aimed to: (1) identify blood transcriptional changes as potential biomarkers of MJD; (2) correlate levels of differentially expressed blood transcripts with MJD carriers features; and (3) evaluate whether the identified differential abundance of blood transcripts in MJD patients is preserved in MJD brains.</p>\u0000 </section>\u0000 \u0000 <section>\u0000 \u0000 <h3> Methods</h3>\u0000 \u0000 <p>We used unbiased RNA microarray and quantitative polymerase chain reaction to assess transcript levels in blood and brain samples, and western blot analysis to evaluate the abundance of specific proteins in brain samples.</p>\u0000 </section>\u0000 \u0000 <section>\u0000 \u0000 <h3> Results</h3>\u0000 \u0000 <p>We observed consistent dysregulation of <i>DDIT4</i>, <i>TRIM13</i>, and <i>P2RY13</i> transcriptional levels in the blood of MJD patients from preclinical to symptomatic stages in Azorean and Brazilian cohorts. Combined blood <i>DDIT4</i> and <i>TRIM13</i> transcriptional levels show a very high accuracy to discriminate MJD carriers from matched controls (AUC ≥0.90). Levels of <i>P2RY13</i> transcripts correlate with age at onset, and an abundance of <i>DDIT4</i> and <i>TRIM13</i> transcripts correlate with the expanded CAG repeat size in combined Azorean and Brazilian patients; and levels of <i>TRIM13</i> transcripts correlate with age at onset of early-stage Azorean patients. Moreover, the abundance of TRIM13 protein is increased in the cerebral cortex of MJD patients.</p>\u0000 </section>\u0000 \u0000 <section>\u0000 \u0000 <h3> Interpretation</h3>\u0000 \u0000 <p>Overall, blood <i>DDIT4</i> and <i>TRIM13</i> transcript levels are potential biomarkers of MJD. Cellular processes involving <i>DDIT4</i>, <i>TRIM13</i>, and <i>P2RY13</i> appear to be commonly dysregulated in the blood and brain of MJD patients, indicating the involvement of these genes in MJD pathogenesis. ANN NEUROL 2025;98:107–119</p>\u0000 </section>\u0000 </div>","PeriodicalId":127,"journal":{"name":"Annals of Neurology","volume":"98 1","pages":"107-119"},"PeriodicalIF":8.1,"publicationDate":"2025-03-05","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"https://onlinelibrary.wiley.com/doi/epdf/10.1002/ana.27222","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"143555371","PeriodicalName":null,"FirstCategoryId":null,"ListUrlMain":null,"RegionNum":1,"RegionCategory":"医学","ArticlePicture":[],"TitleCN":null,"AbstractTextCN":null,"PMCID":"OA","EPubDate":null,"PubModel":null,"JCR":null,"JCRName":null,"Score":null,"Total":0}
引用次数: 0
Purine Metabolism and Dystonia: Perspectives of a Long-Promised Relationship 嘌呤代谢和肌张力障碍:一个长期承诺的关系的观点。
IF 8.1 1区 医学
Annals of Neurology Pub Date : 2025-03-03 DOI: 10.1002/ana.27227
Ugo Sorrentino MD, Audrey G. O'Neill BSc, Justin M. Kollman PhD, Hyder A. Jinnah MD, PhD, Michael Zech MD
{"title":"Purine Metabolism and Dystonia: Perspectives of a Long-Promised Relationship","authors":"Ugo Sorrentino MD,&nbsp;Audrey G. O'Neill BSc,&nbsp;Justin M. Kollman PhD,&nbsp;Hyder A. Jinnah MD, PhD,&nbsp;Michael Zech MD","doi":"10.1002/ana.27227","DOIUrl":"10.1002/ana.27227","url":null,"abstract":"<p>Dystonia research focuses on the identification of converging biological pathways, allowing to define molecular drivers that serve as treatment targets. We summarize evidence supporting the concept that aberrations in purine metabolism intersect with dystonia pathogenesis. The recent discovery of <i>IMPDH2</i>-related dystonia introduced a gain-of-function paradigm in purinergic system defects, offering new perspectives to understand purine-pool imbalances in brain diseases. We discuss commonalities between known dystonia-linked mechanisms and mechanisms emerging from studies of purine metabolism disorders including Lesch–Nyhan disease. Together, we hypothesize that a greater appreciation of the relevance of purine perturbances in dystonia can offer fresh avenues for therapeutic intervention. ANN NEUROL 2025;97:809–825</p>","PeriodicalId":127,"journal":{"name":"Annals of Neurology","volume":"97 5","pages":"809-825"},"PeriodicalIF":8.1,"publicationDate":"2025-03-03","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"https://onlinelibrary.wiley.com/doi/epdf/10.1002/ana.27227","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"143539683","PeriodicalName":null,"FirstCategoryId":null,"ListUrlMain":null,"RegionNum":1,"RegionCategory":"医学","ArticlePicture":[],"TitleCN":null,"AbstractTextCN":null,"PMCID":"OA","EPubDate":null,"PubModel":null,"JCR":null,"JCRName":null,"Score":null,"Total":0}
引用次数: 0
The Starry Cortex of Aβ-Related Angiitis a β相关性脉管炎的星形皮质。
IF 8.1 1区 医学
Annals of Neurology Pub Date : 2025-03-03 DOI: 10.1002/ana.27228
Shiyuan Fang MD, Nan Jiang MD, Lixin Zhou MD, Jun Ni MD
{"title":"The Starry Cortex of Aβ-Related Angiitis","authors":"Shiyuan Fang MD,&nbsp;Nan Jiang MD,&nbsp;Lixin Zhou MD,&nbsp;Jun Ni MD","doi":"10.1002/ana.27228","DOIUrl":"10.1002/ana.27228","url":null,"abstract":"","PeriodicalId":127,"journal":{"name":"Annals of Neurology","volume":"97 6","pages":"1219-1221"},"PeriodicalIF":8.1,"publicationDate":"2025-03-03","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"143539695","PeriodicalName":null,"FirstCategoryId":null,"ListUrlMain":null,"RegionNum":1,"RegionCategory":"医学","ArticlePicture":[],"TitleCN":null,"AbstractTextCN":null,"PMCID":"","EPubDate":null,"PubModel":null,"JCR":null,"JCRName":null,"Score":null,"Total":0}
引用次数: 0
Plasma Glucosylceramide Levels Are Regulated by ATP10D and Are Not Involved in Parkinson's Disease Pathogenesis 血浆葡萄糖甘油三酯水平受 ATP10D 调节,与帕金森病发病机制无关
IF 8.1 1区 医学
Annals of Neurology Pub Date : 2025-03-01 DOI: 10.1002/ana.27219
Emma N. Somerville BSc, Alva James PhD, Christian Beetz PhD, Robert Schwieger PhD, Gal Barrel PhD, Krishna K. Kandaswamy PhD, Marius I. Iurascu PhD, Peter Bauer MD, Michael Ta MS, Hirotaka Iwaki MD, PhD, Konstantin Senkevich MD, PhD, Eric Yu PhD, Roy N. Alcalay MD, MS, Ziv Gan-Or MD, PhD
{"title":"Plasma Glucosylceramide Levels Are Regulated by ATP10D and Are Not Involved in Parkinson's Disease Pathogenesis","authors":"Emma N. Somerville BSc,&nbsp;Alva James PhD,&nbsp;Christian Beetz PhD,&nbsp;Robert Schwieger PhD,&nbsp;Gal Barrel PhD,&nbsp;Krishna K. Kandaswamy PhD,&nbsp;Marius I. Iurascu PhD,&nbsp;Peter Bauer MD,&nbsp;Michael Ta MS,&nbsp;Hirotaka Iwaki MD, PhD,&nbsp;Konstantin Senkevich MD, PhD,&nbsp;Eric Yu PhD,&nbsp;Roy N. Alcalay MD, MS,&nbsp;Ziv Gan-Or MD, PhD","doi":"10.1002/ana.27219","DOIUrl":"10.1002/ana.27219","url":null,"abstract":"<p><i>GBA1</i> variants and decreased glucocerebrosidase activity are implicated in Parkinson's disease (PD). We investigated the hypothesis that increased levels of glucosylceramide (GlcCer), a main substrate of glucocerebrosidase, are involved in PD pathogenesis. Using multiple genetic methods, we show that ATPase phospholipid transporting 10D (<i>ATP10D</i>), not <i>GBA1</i>, is the main regulator of plasma GlcCer levels, yet it is not involved in PD pathogenesis. Plasma GlcCer levels were associated with PD, but not in a causative manner, and are not predictive of disease status. These results argue against targeting GlcCer in <i>GBA1</i>-PD, and underscore the need to explore alternative mechanisms and biomarkers for PD. ANN NEUROL 2025;97:873–878</p>","PeriodicalId":127,"journal":{"name":"Annals of Neurology","volume":"97 5","pages":"873-878"},"PeriodicalIF":8.1,"publicationDate":"2025-03-01","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"https://onlinelibrary.wiley.com/doi/epdf/10.1002/ana.27219","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"143530828","PeriodicalName":null,"FirstCategoryId":null,"ListUrlMain":null,"RegionNum":1,"RegionCategory":"医学","ArticlePicture":[],"TitleCN":null,"AbstractTextCN":null,"PMCID":"OA","EPubDate":null,"PubModel":null,"JCR":null,"JCRName":null,"Score":null,"Total":0}
引用次数: 0
Longitudinal Proteomic Profiling of Cognition across an Aerobic Exercise Intervention 有氧运动干预中认知的纵向蛋白质组学分析。
IF 8.1 1区 医学
Annals of Neurology Pub Date : 2025-02-27 DOI: 10.1002/ana.27210
Alison M. H. Donald, Luiz G. N. de Almeida, Mohamed Ziad Dabaja, Isabella Orchard, Kaia Ybema, Veronica Tsegai, Victoria Armstrong, Sophie Smith, Daniel Young, Richard Stewart Longman, Amanda V. Tyndall, Jean M. Rawling, Michael D. Hill, Willis H. Tsai, Ejaife Agbani, Marc J. Poulin, Antoine Dufour
{"title":"Longitudinal Proteomic Profiling of Cognition across an Aerobic Exercise Intervention","authors":"Alison M. H. Donald,&nbsp;Luiz G. N. de Almeida,&nbsp;Mohamed Ziad Dabaja,&nbsp;Isabella Orchard,&nbsp;Kaia Ybema,&nbsp;Veronica Tsegai,&nbsp;Victoria Armstrong,&nbsp;Sophie Smith,&nbsp;Daniel Young,&nbsp;Richard Stewart Longman,&nbsp;Amanda V. Tyndall,&nbsp;Jean M. Rawling,&nbsp;Michael D. Hill,&nbsp;Willis H. Tsai,&nbsp;Ejaife Agbani,&nbsp;Marc J. Poulin,&nbsp;Antoine Dufour","doi":"10.1002/ana.27210","DOIUrl":"10.1002/ana.27210","url":null,"abstract":"<p>The physiological basis of cognitive decline remains largely uncharacterized. We identified a protein panel signature, in living humans, that correlates to improvement in neurocognition over a period of 5 years. Our signature is composed of complement proteins, coagulation cascade, and extracellular matrix regulators. In our cohort, SERPINF1 is associated with greater maximal oxygen uptake after an aerobic exercise intervention. Sleep quality is also a key factor in relation to inter-alpha-trypsin inhibitor heavy chain H2, which was associated with greater sleep efficiency. Additionally, we validate that the coagulation profile of decliners’ plasma contains procoagulant agonists, leading to greater platelet activation. ANN NEUROL 2025;97:1007–1018</p>","PeriodicalId":127,"journal":{"name":"Annals of Neurology","volume":"97 5","pages":"1007-1018"},"PeriodicalIF":8.1,"publicationDate":"2025-02-27","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"https://onlinelibrary.wiley.com/doi/epdf/10.1002/ana.27210","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"143514152","PeriodicalName":null,"FirstCategoryId":null,"ListUrlMain":null,"RegionNum":1,"RegionCategory":"医学","ArticlePicture":[],"TitleCN":null,"AbstractTextCN":null,"PMCID":"OA","EPubDate":null,"PubModel":null,"JCR":null,"JCRName":null,"Score":null,"Total":0}
引用次数: 0
Clinical, Prognostic, and Longitudinal Functional and Neuropsychological Features of West Nile Virus Neuroinvasive Disease in the United States: A Systematic Review and Meta-Analysis 美国西尼罗河病毒神经侵袭性疾病的临床、预后、纵向功能和神经心理学特征:系统回顾与元分析》。
IF 8.1 1区 医学
Annals of Neurology Pub Date : 2025-02-26 DOI: 10.1002/ana.27220
Jackson A. Roberts MD, Carla Y. Kim PhD, Soonmyung A. Hwang BS, Amir Hassan MD, Ethan Covington BS, Kimia Heydari BS, Mac Lyerly BS, James J. Sejvar MD, Rodrigo Hasbun MD, Manya Prasad MD, Kiran T. Thakur MD
{"title":"Clinical, Prognostic, and Longitudinal Functional and Neuropsychological Features of West Nile Virus Neuroinvasive Disease in the United States: A Systematic Review and Meta-Analysis","authors":"Jackson A. Roberts MD,&nbsp;Carla Y. Kim PhD,&nbsp;Soonmyung A. Hwang BS,&nbsp;Amir Hassan MD,&nbsp;Ethan Covington BS,&nbsp;Kimia Heydari BS,&nbsp;Mac Lyerly BS,&nbsp;James J. Sejvar MD,&nbsp;Rodrigo Hasbun MD,&nbsp;Manya Prasad MD,&nbsp;Kiran T. Thakur MD","doi":"10.1002/ana.27220","DOIUrl":"10.1002/ana.27220","url":null,"abstract":"<div>\u0000 \u0000 <section>\u0000 \u0000 <h3> Objective</h3>\u0000 \u0000 <p>West Nile virus (WNV) is the most common cause of arboviral disease in the United States. Approximately 1% of infections involve the nervous system, most commonly resulting in West Nile encephalitis (WNE), West Nile meningitis (WNM), or acute flaccid paralysis (AFP).</p>\u0000 </section>\u0000 \u0000 <section>\u0000 \u0000 <h3> Methods</h3>\u0000 \u0000 <p>In this systematic review, we characterized comprehensively the diagnostic and clinical features of WNV neuroinvasive disease (WNND) in the United States, as well as the evidence regarding prognostic factors and long-term outcomes of WNND.</p>\u0000 </section>\u0000 \u0000 <section>\u0000 \u0000 <h3> Results</h3>\u0000 \u0000 <p>We identified 47 relevant studies reporting data on acute or longitudinal features of WNND. Across studies, the most common presenting symptoms were fever (88%), nausea/vomiting (58%), and fatigue (50%) coupled neurologically with headache (50%), altered mental status (39%), and focal weakness (32%). Pooled mortality was 9.2%, and 42.1% of reported cases required intensive care unit (ICU) admission. In meta-analyses, chronic kidney disease (odds ratio [OR] = 5.99, 95% confidence interval [CI] = 2.71–13.23), diabetes mellitus (OR = 2.43, 95% CI = 1.54–3.84), and hypertension (OR = 4.01, 95% CI = 2.39–6.72) were associated with an increased risk of mortality. Multidomain neurocognitive impairment was reported in several studies at post-hospitalization follow-up, although with marked heterogeneity between study methodology. Subjective neurocognitive impairment, most notably fatigue (37–75%), memory concerns (11–57%), concentration deficits (17–48%), and depression (17–38%), were also common at post-hospitalization follow-up.</p>\u0000 </section>\u0000 \u0000 <section>\u0000 \u0000 <h3> Interpretation</h3>\u0000 \u0000 <p>These findings underscore the significant mortality and morbidity of WNND in the acute and long-term setting. Our findings may additionally provide utility for risk stratification of hospitalized patients with WNND and suggest the need for further evaluation of novel therapeutics to prevent substantial disease-associated acute and long-term disability. ANN NEUROL 2025;98:93–106</p>\u0000 </section>\u0000 </div>","PeriodicalId":127,"journal":{"name":"Annals of Neurology","volume":"98 1","pages":"93-106"},"PeriodicalIF":8.1,"publicationDate":"2025-02-26","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"143497763","PeriodicalName":null,"FirstCategoryId":null,"ListUrlMain":null,"RegionNum":1,"RegionCategory":"医学","ArticlePicture":[],"TitleCN":null,"AbstractTextCN":null,"PMCID":"","EPubDate":null,"PubModel":null,"JCR":null,"JCRName":null,"Score":null,"Total":0}
引用次数: 0
Efficacy and Safety of Ofatumumab Treatment for Anti-NMDA Receptor Autoimmune Encephalitis (OFF-AE): A Prospective, Multicenter Cohort Study Ofatumumab治疗抗nmda受体自身免疫性脑炎(OFF-AE)的疗效和安全性:一项前瞻性、多中心队列研究
IF 8.1 1区 医学
Annals of Neurology Pub Date : 2025-02-24 DOI: 10.1002/ana.27218
Kundian Guo MD, Fuhua Peng MD, Jia Liu MD, Youming Long MD, Shougang Guo MD, Honghao Wang MD, Gang Yu MD, Yanlin Zhang MD, Xiong Han MD, Ewen Tu MD, Yake Zheng MD, Jialu Huang MD, Yanxia Zhou MD, Dongmei An MD, Guanyan Lin MD, Baojie Wang MD, Yuanyuan Wang MD, Ping Yang MD, Yu Jiang MD, Beijia Cui MD, Zhenyu Yang MD, Maiqi Du MD, Meiling Jiang MD, Limin Qin MD, Xueying Kong MD, Xue Gong MD, Xu Liu MD, Linjun Cai MD, Jinmei Li MD, PhD, Dong Zhou MD, PhD, Zhen Hong MD, PhD
{"title":"Efficacy and Safety of Ofatumumab Treatment for Anti-NMDA Receptor Autoimmune Encephalitis (OFF-AE): A Prospective, Multicenter Cohort Study","authors":"Kundian Guo MD,&nbsp;Fuhua Peng MD,&nbsp;Jia Liu MD,&nbsp;Youming Long MD,&nbsp;Shougang Guo MD,&nbsp;Honghao Wang MD,&nbsp;Gang Yu MD,&nbsp;Yanlin Zhang MD,&nbsp;Xiong Han MD,&nbsp;Ewen Tu MD,&nbsp;Yake Zheng MD,&nbsp;Jialu Huang MD,&nbsp;Yanxia Zhou MD,&nbsp;Dongmei An MD,&nbsp;Guanyan Lin MD,&nbsp;Baojie Wang MD,&nbsp;Yuanyuan Wang MD,&nbsp;Ping Yang MD,&nbsp;Yu Jiang MD,&nbsp;Beijia Cui MD,&nbsp;Zhenyu Yang MD,&nbsp;Maiqi Du MD,&nbsp;Meiling Jiang MD,&nbsp;Limin Qin MD,&nbsp;Xueying Kong MD,&nbsp;Xue Gong MD,&nbsp;Xu Liu MD,&nbsp;Linjun Cai MD,&nbsp;Jinmei Li MD, PhD,&nbsp;Dong Zhou MD, PhD,&nbsp;Zhen Hong MD, PhD","doi":"10.1002/ana.27218","DOIUrl":"10.1002/ana.27218","url":null,"abstract":"<div>\u0000 \u0000 <section>\u0000 \u0000 <h3> Objective</h3>\u0000 \u0000 <p>Ofatumumab presents a potentially promising alternative to current second-line immunotherapy for refractory anti-N-methyl-D-aspartate receptor autoimmune encephalitis (NMDAR-AE). We aimed to evaluate the efficacy and safety of ofatumumab as a novel second-line immunotherapy for NMDAR-AE.</p>\u0000 </section>\u0000 \u0000 <section>\u0000 \u0000 <h3> Methods</h3>\u0000 \u0000 <p>This prospective, multicenter, nested cohort study compared patients with NMDAR-AE from the CHina Autoimmune encephalitiS outcomE study registry (CHASE) recruited between October 2011 and February 2024, treated with and without ofatumumab. The primary outcome was the proportion reaching a favorable functional outcome (modified Rankin Scale [mRS] score ≤2) at the last follow-up. Secondary outcomes included mRS scores and Clinical Assessment Scale in Autoimmune Encephalitis (CASE) scores over the first 24-month follow-up and the proportion with further mRS score improvement after ofatumumab initiation. A propensity score matching was performed to balance major confounders.</p>\u0000 </section>\u0000 \u0000 <section>\u0000 \u0000 <h3> Results</h3>\u0000 \u0000 <p>A total of 715 patients with AE were screened. Fifty-eight propensity score-matched patients with NMDAR-AE each in the ofatumumab group and non-ofatumumab group were analyzed. Fifty-four patients (93.1%) in the ofatumumab group achieved further mRS score improvement with a median time of 14 days from ofatumumab initiation, and 53 (91.4%) reached a favorable functional outcome at the last follow-up. For those who failed first-line immunotherapy, the ofatumumab group demonstrated a faster mRS score and CASE score improvement and more frequently reached a favorable functional outcome at the last follow-up compared with the non-ofatumumab group (87.9% vs. 64.7%, odds ratio [OR] 3.95; 95% confidence interval [CI] 1.12–13.94; <i>p</i> = 0.026). No serious adverse events associated with ofatumumab treatment were reported.</p>\u0000 </section>\u0000 \u0000 <section>\u0000 \u0000 <h3> Interpretation</h3>\u0000 \u0000 <p>Ofatumumab showed substantial efficacy and safety, particularly in patients who failed first-line immunotherapy, warranting its consideration in NMDAR-AE management. ANN NEUROL 2025;98:80–92</p>\u0000 </section>\u0000 </div>","PeriodicalId":127,"journal":{"name":"Annals of Neurology","volume":"98 1","pages":"80-92"},"PeriodicalIF":8.1,"publicationDate":"2025-02-24","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"143481799","PeriodicalName":null,"FirstCategoryId":null,"ListUrlMain":null,"RegionNum":1,"RegionCategory":"医学","ArticlePicture":[],"TitleCN":null,"AbstractTextCN":null,"PMCID":"","EPubDate":null,"PubModel":null,"JCR":null,"JCRName":null,"Score":null,"Total":0}
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