Neuroimmunology Reports最新文献

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Neuroimmunology Reports

Pub Date : 2025-01-01

引用次数: 0

Neuroimmunology Reports

Pub Date : 2025-01-01

引用次数: 0

Oral cladribine for management of multiple sclerosis–Clinical experience from Hong Kong 口服克拉德滨治疗多发性硬化症——香港临床经验

Neuroimmunology Reports

Pub Date : 2025-01-01 DOI: 10.1016/j.nerep.2024.100238

Chun-Ho Choi , Jason Ka-Yeung Fong , Siu-Hung Li , Kary Ka-Wai Chan , Alexander Yuk-Lun Lau

Background

Oral cladribine, a selective immune reconstitution therapy, offers a short-course option for disease-modifying therapy (DMT) in multiple sclerosis (MS), a chronic neurodegenerative disease characterized by inflammation and demyelination affecting 2.8 million people worldwide. Despite its proven safety and efficacy in phase 2 and 3 trials, data for oral cladribine treatment in Asian populations remain limited. This case series aims to contribute real-world experience across the clinical spectrum of disease by presenting Asian MS patients treated in Hong Kong.

Methods

This retrospective case series comprises 9 patients diagnosed with relapsing-remitting MS and received oral cladribine treatment in Hong Kong between 2019 and 2021. These patients, treated in both public and private hospitals, were followed up to assess the outcomes of oral cladribine therapy. Relevant data were extracted from patient's medical records.

Results

Outcomes were generally positive, with 6 out of 9 patients showing stable or improved Expanded Disability Status Scale (EDSS) scores 2–4 years after treatment initiation and 7 remaining relapse-free within 2–5 years of treatment initiation. Only 2 patients experienced mild relapses. Follow-up MRI scans revealed no new lesion development in any patient. One case of transient lymphopenia was noted, which did not require discontinuation of treatment.

Conclusions

Our clinical experience with these patients aligned with the safety and efficacy outcomes reported in clinical trials. These observations support the efficacy of a short-course regimen in reducing overall treatment burden for patients and potentially improving adherence to therapy. To substantiate and build upon these findings and explore long-term outcomes in larger cohorts, further research and continued monitoring of patients is necessary.

背景：口服克拉德滨是一种选择性免疫重建疗法，为多发性硬化症（MS）的疾病改善治疗（DMT）提供了一种短期选择。多发性硬化症（MS）是一种慢性神经退行性疾病，以炎症和脱髓鞘为特征，影响全球280万人。尽管在2期和3期试验中证明了其安全性和有效性，但亚洲人群口服氯德里滨治疗的数据仍然有限。本病例系列旨在通过介绍在香港接受治疗的亚洲多发性硬化症患者，为疾病的临床谱提供真实的经验。方法：本回顾性病例系列包括2019年至2021年在香港诊断为复发-缓解型多发性硬化症并接受口服克拉德里滨治疗的9例患者。这些患者在公立和私立医院接受治疗，随访以评估口服克拉德里滨治疗的效果。从患者病历中提取相关数据。结果总体上是积极的，9例患者中有6例在治疗开始2-4年后显示稳定或改善的扩展残疾状态量表（EDSS）评分，7例在治疗开始2-5年内无复发。仅有2例患者出现轻度复发。后续MRI扫描未发现任何患者出现新的病变。1例短暂性淋巴细胞减少被注意到，这并不需要停止治疗。结论我们对这些患者的临床经验与临床试验报告的安全性和有效性结果一致。这些观察结果支持短期治疗方案在减轻患者总体治疗负担和提高治疗依从性方面的有效性。为了证实和建立这些发现，并在更大的队列中探索长期结果，有必要进一步研究和持续监测患者。

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引用次数: 0

Erratum to “Persistent spinal cord enhancement in longitudinal extensive transverse myelitis associated with α-1-antitrypisn deficiency: A case report” [Neuroimmunology Reports Volume 2, 2022, 100090] 对“α-1-抗胰蛋白酶缺乏相关的纵向广泛横贯脊髓炎的持续脊髓强化：一例报告”的勘误[神经免疫学报告第2卷，2022,100090]

Neuroimmunology Reports

Pub Date : 2025-01-01 DOI: 10.1016/j.nerep.2024.100243

Osman Ozel , Svetlana P. Eckert , Dejan Jakimovski , Nicholas Silvestri , David Hojnacki , Bianca Weinstock-Guttman

引用次数: 0

Contactin-associated protein-like 2 (CASPR2) antibody generation in acute herpes simplex encephalitis: Case report and review of the literature 急性单纯疱疹脑炎中接触相关蛋白样2 （CASPR2）抗体的产生：病例报告及文献复习

Neuroimmunology Reports

Pub Date : 2025-01-01 DOI: 10.1016/j.nerep.2025.100254

Dong Hyun Kim , Christoph Friedli

Introduction

Herpes simplex encephalitis (HSE) is the most common cause of sporadic viral encephalitis, and can lead to auto-antibody generation. HSE has mainly been reported in association with N-methyl-D-aspartate receptor (NMDAR) antibodies, but there have also been cases associated with contactin-associated protein-like 2 (CASPR2). This article aims to highlight the relevance of CASPR2 antibody generation in HSE.

Methods

This is a case presentation and literature review of HSE with CASPR2 antibody positivity.

Results

A 62-year-old male initially presented with fever, headache and confusion in September 2023. He then developed status epilepticus requiring intubation. MRI of the brain showed extensive bilateral mesiotemporal lobe abnormalities with an haemorrhagic component. Cerebrospinal fluid (CSF) analysis was positive for herpes simplex virus 1 DNA, and CSF and serum were also positive for CASPR2 antibodies. He was treated with intravenous aciclovir for 3 weeks and short course of high dose intravenous dexamethasone. The patient continued to deteriorate despite treatment and died from the complications of the disease. In this case, it was difficult to ascertain if the patient had symptoms of CASPR2 associated disease or if it was an epiphenomenon triggered by HSE.

Conclusion

A number of case reports have described HSE with CASPR2 positivity. Whereas our patient succumbed to the disease, other cases showed clinical response with aciclovir, corticosteroids and intravenous immunoglobulin. More research on this topic is warranted as there are no specific treatment guidelines due to the lack of robust clinical trial data.

简介：单纯疱疹病毒性脑炎（HSE）是散发性病毒性脑炎最常见的病因，可导致自身抗体的产生。HSE主要与n-甲基- d -天冬氨酸受体（NMDAR）抗体相关，但也有与接触蛋白相关蛋白样2 （CASPR2）相关的病例。本文旨在强调CASPR2抗体生成在HSE中的相关性。方法对CASPR2抗体阳性的HSE进行病例报告和文献复习。结果患者男性62岁，于2023年9月首次出现发热、头痛、意识不清等症状。随后他出现了需要插管的癫痫持续状态。脑部MRI显示广泛的双侧中颞叶异常伴出血成分。脑脊液（CSF）分析单纯疱疹病毒1型DNA阳性，CSF和血清CASPR2抗体也阳性。阿昔洛韦静脉注射3周，短疗程大剂量地塞米松静脉注射。尽管接受了治疗，病人的病情仍在恶化，最终死于该病的并发症。在这种情况下，很难确定患者是否有CASPR2相关疾病的症状，或者是否是HSE引发的附带现象。结论CASPR2阳性的HSE病例报告较多。虽然我们的病人死于这种疾病，但其他病例在阿昔洛韦、皮质类固醇和静脉注射免疫球蛋白后表现出临床反应。由于缺乏可靠的临床试验数据，没有具体的治疗指南，因此需要对这一主题进行更多的研究。

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引用次数: 0

Persistent severe immunoglobulin deficiency after treatment with rituximab in a patient with MOGAD: A case report on how rituximab may, or may not, unmask primary immunodeficiencies 1例MOGAD患者在接受利妥昔单抗治疗后出现持续严重的免疫球蛋白缺乏：一份关于利妥昔单抗如何揭示原发性免疫缺陷的病例报告

Neuroimmunology Reports

Pub Date : 2025-01-01 DOI: 10.1016/j.nerep.2025.100258

A. Ali , L. Bernasconi , N. Cantoni , A.C. Lecourt , A.K. Pröbstel , A. Conen , O. Findling

We present the clinical scenario of a young female patient, who presented with a progressive sensory motor tetraparesis and fatigue. Upon investigation anti-MOG-associated disease (MOGAD) was diagnosed. After high dose intravenous steroid therapy, an immunosuppressive treatment with rituximab, in six-monthly cycles, was initiated. With this, the neurological condition stabilized without any further relapses. But the patient started to suffer from recurrent respiratory tract infections, even after the treatment with rituximab was paused for two years. Investigations revealed an underlying strikingly severe IgA deficiency alongside an IgG Deficiency.

我们提出了一个年轻的女性患者的临床方案，谁提出了进行性感觉运动四肢瘫痪和疲劳。经调查诊断为抗mog相关疾病（MOGAD）。在高剂量静脉注射类固醇治疗后，开始了以6个月为周期的利妥昔单抗免疫抑制治疗。有了这个，神经系统的状况稳定下来，没有任何进一步的复发。但患者开始出现复发性呼吸道感染，甚至在停止使用利妥昔单抗治疗两年之后。调查显示，潜在的严重IgA缺乏症和IgG缺乏症。

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Neuroimmunology Reports

Pub Date : 2025-01-01

引用次数: 0

Neuroimmunology Reports

Pub Date : 2025-01-01

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Neuroimmunology Reports

Pub Date : 2025-01-01

引用次数: 0

High-activity multiple sclerosis in feminizing hormone therapy for gender reassignment 高活性多发性硬化症女性化激素治疗的性别重新分配

Neuroimmunology Reports

Pub Date : 2025-01-01 DOI: 10.1016/j.nerep.2024.100234

Irene Expósito Ruiz, Cintia Tuñas Gesto, Alfredo Puy Núñez, Eva Costa Arpín

引用次数: 0

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