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Navigating the uncharted-Progressive multifocal leukoencephalopathy in a child with NMOSD treated with rituximab. 利妥昔单抗治疗NMOSD患儿的进展性多灶性脑白质病
IF 5 2区 医学 Q1 CLINICAL NEUROLOGY Pub Date : 2025-11-01 Epub Date: 2025-04-18 DOI: 10.1177/13524585251331875
Kumaran Deiva
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引用次数: 0
Baló's concentric sclerosis successfully treated with alemtuzumab: Long-term follow-up. Baló的同心硬化成功治疗阿仑单抗:长期随访。
IF 5 2区 医学 Q1 CLINICAL NEUROLOGY Pub Date : 2025-11-01 Epub Date: 2025-04-12 DOI: 10.1177/13524585251331536
Sandra Elias, Todd A Hardy, Aijaz Khan, Jessica Redgrave, Nigel Hoggard, Stuart Coley, Richard Dyde, Ronan O'Malley, Zeid Yasiry, David Paling

Baló's concentric sclerosis (BCS) is regarded as a rare variant of multiple sclerosis (MS), characterised by multi-layered ring-like lesions in cerebral white matter. Despite pathological overlap with MS, the effect of treatment with MS disease-modifying therapies remains unclear. The only extant case report of alemtuzumab in BCS described a lack of clinical response in a patient who had previously not responded to corticosteroids, plasmapheresis and cyclophosphamide. The authors speculated that alemtuzumab may have been effective if started earlier in the disease process. We present the outcomes of a patient with BCS who responded clinically and radiologically to alemtuzumab over a 6-year follow-up.

Baló的同心圆硬化(BCS)被认为是多发性硬化症(MS)的一种罕见变体,其特征是大脑白质中的多层环状病变。尽管病理上与多发性硬化症有重叠,但多发性硬化症的治疗效果尚不清楚。阿仑单抗在BCS中唯一存在的病例报告描述了先前对皮质类固醇、血浆置换和环磷酰胺无反应的患者缺乏临床反应。作者推测,如果在疾病过程的早期开始使用阿仑单抗,可能是有效的。我们报告了一位对阿仑单抗有临床和放射学反应的BCS患者的结果,随访6年。
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引用次数: 0
Treating Balo's concentric sclerosis in the monoclonal antibody era. 单克隆抗体时代治疗Balo同心圆硬化。
IF 5 2区 医学 Q1 CLINICAL NEUROLOGY Pub Date : 2025-11-01 Epub Date: 2025-04-12 DOI: 10.1177/13524585251331551
Jun-Ichi Kira
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引用次数: 0
A first report of progressive multifocal leukoencephalopathy in childhood-onset NMOSD. 儿童期发病NMOSD中进行性多灶性脑白质病的首次报道。
IF 5 2区 医学 Q1 CLINICAL NEUROLOGY Pub Date : 2025-11-01 Epub Date: 2025-04-18 DOI: 10.1177/13524585251331855
Pakeeran Siriratnam, Simon Gosling, Maneesh Bhojak, Michael Griffiths, Rachel Kneen, Evangeline Wassmer, Saif Huda

Background: Progressive multifocal leukoencephalopathy (PML) has not been reported in pediatric neuromyelitis optica spectrum disorder (NMOSD) and rarely described in children.

Objectives: To report a case of PML in childhood-onset NMOSD.

Methods: A single retrospective case report.

Results: Nine years after diagnosis of aquaporin-4 antibody positive NMOSD, a 17-year-old boy treated with rituximab presented with painless right visual loss over 6 weeks coinciding with CD19 repopulation. Acute relapse treatment was given but he continued to deteriorate, and JC virus was detected in cerebrospinal fluid confirming PML.

Conclusion: PML can occur in childhood-onset NMOSD and protracted clinical presentations with unusual radiological features should prompt JCV testing. Balancing effective immunosuppression while mitigating the risks of associated complications in long-term relapsing conditions remains a challenge.

背景:进行性多灶性脑白质病(PML)在小儿视神经脊髓炎谱系障碍(NMOSD)中尚未报道,在儿童中也很少报道。目的:报告1例儿童期发病NMOSD的PML。方法:单例回顾性病例报告。结果:在诊断为水通道蛋白-4抗体阳性NMOSD的9年后,一名17岁男孩接受利妥昔单抗治疗后,在6周内出现无痛性右侧视力丧失,并伴有CD19重新聚集。急性复发治疗后病情持续恶化,脑脊液中检出JC病毒,证实PML。结论:PML可发生在儿童期发病的NMOSD中,具有不寻常放射学特征的长期临床表现应提示进行JCV检测。平衡有效的免疫抑制,同时减轻长期复发条件下相关并发症的风险仍然是一个挑战。
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引用次数: 0
Commentary on: Acute retinal necrosis associated with dimethyl fumarate. 评论:富马酸二甲酯引起的急性视网膜坏死。
IF 5 2区 医学 Q1 CLINICAL NEUROLOGY Pub Date : 2025-10-01 Epub Date: 2025-05-04 DOI: 10.1177/13524585251326487
Angela Vidal-Jordana
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引用次数: 0
Acute retinal necrosis associated with dimethyl fumarate. 富马酸二甲酯引起的急性视网膜坏死。
IF 5 2区 医学 Q1 CLINICAL NEUROLOGY Pub Date : 2025-10-01 Epub Date: 2025-05-04 DOI: 10.1177/13524585251326475
Christopher Paisey, Karen Curtin, Simon J Epps, Claire M Rice

Background: The full range of side effects of disease-modifying treatment for multiple sclerosis (MS) remains to be determined. To date, four serious varicella zoster virus (VZV) infections have been reported in association with dimethyl fumarate (DMF).

Report: A 44-year-old man on DMF for relapsing remitting MS presented with monocular pain and visual impairment. Ophthalmological examination revealed panuveitis with arteritis, retinitis and vitritis. Vitreous PCR was positive for VZV confirming the clinical impression of acute retinal necrosis (ARN).

Discussion: ARN due to VZV may mimic optic neuritis in people on disease-modifying therapy for MS.

背景:多发性硬化症(MS)疾病改善治疗的全部副作用仍有待确定。迄今为止,已报告了4例与富马酸二甲酯(DMF)有关的严重水痘带状疱疹病毒(VZV)感染。报告:一名44岁男性因复发缓解型MS而接受DMF治疗,表现为单眼疼痛和视力障碍。眼科检查发现全葡萄膜炎合并动脉炎、视网膜炎和玻璃体炎。玻璃体PCR检测VZV阳性,证实急性视网膜坏死(ARN)的临床表现。讨论:VZV引起的ARN可能在接受疾病改善治疗的MS患者中模拟视神经炎。
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引用次数: 0
Opportunistic enteroviral infections in MS patients treated with anti-CD20 therapies. 接受抗cd20治疗的MS患者的机会性肠病毒感染。
IF 5 2区 医学 Q1 CLINICAL NEUROLOGY Pub Date : 2025-09-01 Epub Date: 2025-01-20 DOI: 10.1177/13524585241310395
James A Varley, Nicholas Ws Davies
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引用次数: 0
A case of neuromyelitis optica spectrum disorder complicated with aquaporin-4-antibody-associated organizing pneumonia. 视神经脊髓炎伴水通道蛋白-4抗体相关性组织性肺炎1例。
IF 5 2区 医学 Q1 CLINICAL NEUROLOGY Pub Date : 2025-09-01 Epub Date: 2025-01-06 DOI: 10.1177/13524585241310397
Tomoya Shibahara, Kei Yamanaka, Mikiaki Matsuoka, Masaki Tachibana, Junya Kuroda, Hiroshi Nakane

An 80-year-old man with aquaporin-4-antibody-positive neuromyelitis optica spectrum disorder presented with a 2-week history of cough and hiccups, followed by progressive bilateral lower limb weakness, a bandlike burning sensation in the upper body, and urinary retention. Magnetic resonance imaging showed area postrema and thoracic central medullary lesions. Thorax computed tomography showed bilateral upper lung lobe consolidations. Lung biopsy confirmed organizing pneumonia. Immunohistochemistry of these lesions demonstrated aquaporin-4 loss, immunoglobulin G-binding, and complement deposits, akin to central nervous system lesions found in neuromyelitis optica spectrum disorder. Thus, aquaporin-4-antibodies may be involved in extra-central nervous system manifestations of neuromyelitis optica spectrum disorder.

80岁男性,患有水通道蛋白-4抗体阳性的视神经脊髓炎频谱障碍,有2周的咳嗽和打嗝史,随后进行性双侧下肢无力,上半身烧灼感,尿潴留。磁共振成像显示后脑区及胸椎中央髓质病变。胸部电脑断层显示双侧上肺叶实变。肺活检证实为组织性肺炎。这些病变的免疫组织化学显示水通道蛋白-4缺失、免疫球蛋白g结合和补体沉积,类似于光性神经脊髓炎谱系障碍中发现的中枢神经系统病变。因此,水通道蛋白-4抗体可能参与视神经脊髓炎谱系障碍的中枢外神经系统表现。
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引用次数: 0
Severe coxsackie virus B5 encephalitis mimics autoimmune limbic encephalitis in a young woman under long-term B-cell depletion with ocrelizumab: A case report. 严重柯萨奇病毒B5脑炎模拟自身免疫性边缘脑炎在长期使用奥克雷单抗b细胞消耗的年轻女性:一个病例报告。
IF 5 2区 医学 Q1 CLINICAL NEUROLOGY Pub Date : 2025-09-01 Epub Date: 2025-02-27 DOI: 10.1177/13524585251322327
Jakob Stögbauer, Victoria Schegerer, Fabian K Berger, Walter Schulz-Schaeffer, Klaus Fassbender, Jasmin Naumann, Sigrun Smola, Janina Eisenbeis, Moritz Bewarder, Florian Rosar, Michael Kettner, Thomas Gilcher, Sabine Diedrich, Mathias Fousse

Background: B-cell-depleting therapies are increasingly being used to treat autoimmune diseases. Although thousands of patients are and have been treated with these agents, the data are not yet strong enough to identify rare side effects with certainty.

Methods: We report the case of a patient undergoing ocrelizumab therapy for relapsing multiple sclerosis who developed a severe limbic syndrome.

Results: Autoimmune pathogenesis was initially suspected, with worsening on immunosuppressive therapy. Later, after diagnosis of an enterovirus infection, treatment with ribavirin and favipiravir in combination with intravenous immunoglobulins was initiated. After 4 weeks of therapy, the patient's clinical condition had stabilized with residual cognitive deficits.

Conclusion: Diagnosis and treatment of enterovirus infections remain challenging, especially in patients receiving immunosuppressive therapy.

背景:b细胞消耗疗法越来越多地被用于治疗自身免疫性疾病。尽管成千上万的患者正在或已经接受了这些药物的治疗,但这些数据还不足以确定罕见的副作用。方法:我们报告了一例接受ocrelizumab治疗复发性多发性硬化症的患者,他发展为严重的边缘综合征。结果:最初怀疑自身免疫发病,免疫抑制治疗后病情加重。后来,在诊断出肠道病毒感染后,开始使用利巴韦林和法匹拉韦联合静脉注射免疫球蛋白进行治疗。经过4周的治疗,患者的临床状况趋于稳定,但仍存在认知缺陷。结论:肠病毒感染的诊断和治疗仍然具有挑战性,特别是在接受免疫抑制治疗的患者中。
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引用次数: 0
Neurofilament light biomarkers in MS: Effects of extended natalizumab dosing. 多发性硬化症的神经丝光生物标志物:延长纳他珠单抗用药时间的影响。
IF 5 2区 医学 Q1 CLINICAL NEUROLOGY Pub Date : 2025-09-01 Epub Date: 2025-02-11 DOI: 10.1177/13524585251315342
Gloria Dalla Costa, Aiden Haghikia, Ralf Gold, Roberto Furlan, Giancarlo Comi
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引用次数: 0
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Multiple Sclerosis Journal
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