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Interferon-beta-1a induced retinopathy in a 14-year-old female patient with multiple sclerosis 一名 14 岁女性多发性硬化症患者因干扰素-β-1a 引起的视网膜病变
Pub Date : 2024-01-01 DOI: 10.1016/j.nerep.2024.100202
C. Tsimakidi, M. Gontika, D. Gkougka, K Rizonaki, S. Fanouraki, C. Kotsalis

Interferon (INF) b-1a is a widely used medication, administered as first-line treatment in adult and pediatric patients with multiple sclerosis (MS). Common adverse reactions include flu-like symptoms, skin changes, allergic reactions and depression.

Interferon-induced retinopathy commonly arises in HCV patients treated with INFa, but has rarely been associated with IFNb administration in adults.

We present a 14-year-old female, newly diagnosed with MS, that presented with interferon-induced retinopathy, five months after IFNb-1a initiation. The medication was discontinued with complete recovery.

To our knowledge, this is the first reported case of IFNb-induced retinopathy in a pediatric patient.

干扰素(INF)b-1a 是一种广泛使用的药物,是成人和儿童多发性硬化症(MS)患者的一线治疗药物。常见的不良反应包括流感样症状、皮肤变化、过敏反应和抑郁。干扰素诱发的视网膜病变通常发生在接受 INFa 治疗的 HCV 患者身上,但在成人患者中却很少与 IFNb 相关。我们报告了一名 14 岁的女性患者,她刚被诊断为多发性硬化症,在开始接受 IFNb-1a 治疗五个月后出现了干扰素诱发的视网膜病变。据我们所知,这是首例报道的儿童患者因 IFNb 引起视网膜病变的病例。
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引用次数: 0
Multiple sclerosis and optic neuritis triggered by COVID-19 mRNA 由 COVID-19 mRNA 引发的多发性硬化症和视神经炎
Pub Date : 2024-01-01 DOI: 10.1016/j.nerep.2023.100198
Giovanna Thais Torques Moretti , Giorgio Fabiani , Georgette Mouchaileh Evangelista Ferreira , Jessica Pizatto de Araujo , Eduardo Hummelgen

Background

In December 2019, the World Health Organization declared COVID-19 a pandemic; an unprecedented health crisis has rocked the world. Enormous efforts by governments, the pharmaceutical industry, and health professionals, including a new mRNA vaccine technology against SARS-CoV-2, have been massively employed to control the COVID-19 pandemic. The vaccine's primary mechanism is based on S glycoprotein, the leading viral surface antigen, which induces protective neutralizing antibodies.

Case presentation

This project aims to present two cases of demyelinating syndromes in previously asymptomatic patients. The triggering factor in both cases was vaccination against COVID-19 with mRNA vaccines.

Case Report

The first case is a young female, 25 years old, previously asymptomatic, one week after the ChAdOx1 Vaccine developed right hemiparesis. Brain MRI and MRI of the spinal cord demonstrated multiple hyperintense lesions, acute and chronic. The second case is an 8-year-old Caucasian male; 12 days after the double dose of the Pfizer-BioNTech vaccine, the patient complained of bilateral visual blurring. The patient was pulsed with methylprednisolone one g/day/5 days with complete recovery.

Conclusion

Rarely can vaccines trigger multiple sclerosis or optic neuritis. This report demonstrated two demyelinating syndromes triggered by the m-RNA COVID-19 vaccine.

背景2019年12月,世界卫生组织宣布COVID-19为大流行病;一场前所未有的健康危机震撼了世界。为控制 COVID-19 大流行,各国政府、制药业和卫生专业人员付出了巨大努力,其中包括针对 SARS-CoV-2 的新型 mRNA 疫苗技术。该疫苗的主要机制是基于 S 糖蛋白(主要的病毒表面抗原),它能诱导保护性中和抗体。第一个病例是一名年轻女性,25 岁,之前无症状,接种 ChAdOx1 疫苗一周后出现右侧偏瘫。脑部核磁共振成像和脊髓核磁共振成像显示多处急性和慢性高强度病变。第二个病例是一名 8 岁的白种男性;在接种两剂辉瑞生物技术疫苗 12 天后,患者主诉双侧视力模糊。结论疫苗很少会引发多发性硬化或视神经炎。本报告展示了 m-RNA COVID-19 疫苗引发的两种脱髓鞘综合征。
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引用次数: 0
Transitory recurrent diplopia and dysarthria with MRI mimicking central pontine myelinolysis: a peculiar NMOSD onset 短暂性复发性复视和构音障碍,核磁共振成像模拟中枢神经髓鞘溶解:一种特殊的 NMOSD 发病情况
Pub Date : 2024-01-01 DOI: 10.1016/j.nerep.2024.100207
C. Chiavazza , E. Genovese , M. Narracci , S. Gasverde , C. Baima , D.M. Papurello
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引用次数: 0
Allopurinol treatment in pediatric multiple sclerosis 别嘌醇治疗小儿多发性硬化症
Pub Date : 2024-01-01 DOI: 10.1016/j.nerep.2024.100209
Yousef A Assaleh , Farah Thabet , Kalthoum Graies-Tlili , Brahim Tabarki

We report a 12-year-old male patient with relapsing-remitting multiple sclerosis who showed a strong response to allopurinol with no safety concern. Researchers should further evaluate this drug option in multiple sclerosis.

我们报告了一名 12 岁的男性复发性多发性硬化症患者,他对别嘌呤醇的反应强烈,而且没有安全问题。研究人员应进一步评估这种治疗多发性硬化症的药物。
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引用次数: 0
Chronic inflammatory demyelinating polyneuropathy or subacute Guillain-Barré syndrome? Not always an easy differential diagnosis 慢性炎症性脱髓鞘多发性神经病还是亚急性格林-巴利综合征?鉴别诊断并非易事
Pub Date : 2024-01-01 DOI: 10.1016/j.nerep.2023.100196
Elisabetta Cecconi , Sara Torricelli , Gabriele Rosario Rodolico , Martina Sperti , Maddalena Spalletti , Roberto Fratangelo , Valentina Bessi , Leonello Guidi , Sabrina Matà

Guillain-Barré syndrome (GBS) is an immuno-mediated disorder of the peripheral nervous system with an acute onset of 2–4 weeks, and a monophasic course. In the subacute variant the symtpoms nadir is reached in 4–8 weeks. However, in sporadic cases the onset and the evolution of the disease may be different than expected, leading to significant diagnostic difficulties.

Case report: A 78-yrs old man presented at our Hospital with a 4-month history of progressive, diffuse motor and sensory deficit. He had previously undergone two electrophysiological examinations with uncertain findings. At hospitalization the nerve conduction study (NCS) and the cerebrospinal fluid examination were consistent with inflammatory demyelinating polyradiculoneuropathy. While a 5-day high-dose of intravenous methylprednisolone proved to be ineffective, a single intravenous immunoglobulin cycle risulted in a significant clinical improvement without relapse after an 18-month follow-up. Based on clinical and neurophysiological findings, a diagnosis of atypical subacute GBS was finally made.

Conclusion: Although generally accepted that GBS has an acute onset within a few weeks, with a maximum of 8 weeks in the subacute variant, this case report shows that it may initially present with a very slow clinical progression and inconsistent NCS findings. However, its recognition and differentiation from the chronic inflammatory nerve disorders, which have a chronic, long-term evolution, is mandatory to provide the correct therapy protocol.

吉兰-巴雷综合征(GBS)是一种免疫介导的周围神经系统疾病,急性起病期为 2-4 周,病程呈单相。在亚急性变异型中,症状在 4-8 周内达到最低点。然而,在散发性病例中,起病和疾病的演变可能与预期的不同,从而导致诊断上的重大困难:病例报告:一名 78 岁的男性患者因进行性、弥漫性运动和感觉障碍 4 个月来我院就诊。此前,他曾接受过两次电生理检查,但结果均不确定。住院时,神经传导检查(NCS)和脑脊液检查结果与炎症性脱髓鞘多发性神经病一致。虽然为期 5 天的大剂量甲基强的松龙静脉注射被证明无效,但经过 18 个月的随访,单次免疫球蛋白静脉注射周期使患者的临床症状明显改善,且没有复发。根据临床和神经电生理检查结果,最终诊断为非典型亚急性 GBS:尽管一般认为 GBS 在数周内急性起病,亚急性变异型最长不超过 8 周,但本病例报告显示,GBS 最初的临床表现可能非常缓慢,NCS 结果也不一致。然而,要提供正确的治疗方案,就必须对其进行识别,并将其与慢性炎症性神经疾病区分开来,因为后者具有慢性、长期的演变过程。
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引用次数: 0
Erratum to “Atypical Myelin Oligodendrocyte Glycoprotein Antibody Disease Presenting with Isolated Elevated Intracranial Pressure” [Neuroimmunology Reports Volume 1, December 2021, 100028] 非典型髓鞘少突胶质细胞糖蛋白抗体病伴有孤立性颅内压升高》勘误 [《神经免疫学报告》第 1 卷,2021 年 12 月,100028]
Pub Date : 2024-01-01 DOI: 10.1016/j.nerep.2024.100203
Alexandria Valdrighi , Jeffrey Russ , Emmanuelle Waubant , Nailyn Rasool , Carla Francisco
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引用次数: 0
Rapidly progressive cerebral dysfunction in a 5-year-old child: A reminder of subacute sclerosing panencephalitis (SSPE) and the importance of measles vaccination coverage 一名 5 岁儿童出现快速进展性脑功能障碍:提醒人们注意亚急性硬化性泛脑炎(SSPE)和麻疹疫苗接种的重要性
Pub Date : 2023-12-12 DOI: 10.1016/j.nerep.2023.100195
Mustafa Bakir , Nadir Khan , Jonathan Gehlbach

We present a case of a preschool boy admitted to the pediatric intensive care unit with generalized weakness and encephalopathy that progressed to coma and spastic paralysis over the next few weeks. Extensive evaluation of a wide range of possible diagnoses, including infectious, post-infectious, autoimmune, and paraneoplastic disorders, proved unrevealing. Owing to concerns regarding autoimmune encephalitis, the patient received plasmapheresis, intravenous immunoglobulin, high-dose glucocorticoids, anakinra, rituximab, and empirical botulinum antitoxin. Despite these treatments, the patient's neurological condition continued to deteriorate, requiring endotracheal intubation. The patient developed repeated tremors and dystonic events, which progressed to hypertonia with gaze deviation. His-EEG in the third week of admission showed Radermecker complexes most consistent with SSPE. Because of his history of travel to Afghanistan at 8 months of age, before he was vaccinated for measles, and after a measles-like illness upon return, we checked the measles IgG titer in the CSF from the initial lumbar puncture and found it to be elevated. Global coverage with the first dose of the measles vaccine has dropped to 81 % by 2021, the lowest rate since 2008. This decline raises the concern of a possible increase in the incidence of measles and its fatal complications such as SSPE.

我们介绍了一例因全身乏力和脑病而被送入儿科重症监护室的学龄前男孩的病例,该病在接下来的几周内发展为昏迷和痉挛性瘫痪。对各种可能的诊断进行了广泛的评估,包括感染、感染后、自身免疫和副肿瘤性疾病,但都没有结果。出于对自身免疫性脑炎的担忧,患者接受了浆细胞吸出术、静脉注射免疫球蛋白、大剂量糖皮质激素、阿那金拉、利妥昔单抗和经验性肉毒杆菌抗毒素治疗。尽管接受了这些治疗,但患者的神经状况仍在继续恶化,需要进行气管插管。患者反复出现震颤和肌张力障碍,并发展为肌张力亢进,伴有凝视偏移。入院第三周,他的电子脑电图显示出与 SSPE 最为一致的 Radermecker 复极。由于他在 8 个月大时曾去阿富汗旅行,当时他还没有接种麻疹疫苗,回国后又出现了类似麻疹的疾病,因此我们检查了首次腰椎穿刺采集的脑脊液中的麻疹 IgG 滴度,发现滴度升高。到 2021 年,全球麻疹疫苗首剂接种率将降至 81%,这是自 2008 年以来的最低值。这一下降引起了人们对麻疹及其致命并发症(如 SSPE)发病率可能上升的担忧。
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引用次数: 0
Regaining walking and restoring corticospinal tract excitability after neuromyelitis optica spectrum disorder: A case report 视神经脊髓炎后恢复行走和皮质脊髓束兴奋性1例
Pub Date : 2023-11-20 DOI: 10.1016/j.nerep.2023.100193
Hui-Ting Shih , Hui-Ting Goh , Seema Sikka , Rita Hamilton , Chad Swank

Neuromyelitis optica spectrum disorder (NMOSD) is a rare autoimmune disease causing axonal damage with corresponding functional deficits. In this case report, we prospectively tracked walking recovery and corticospinal excitability of a female diagnosed with NMOSD through six months after her inpatient rehabilitation (IPR) stay. She recovered independent walking function in home and community settings. Neurophysiological measures acquired using transcranial magnetic stimulation showed two temporal evolution patterns. There was a remarkably reduced intra-cortical inhibition and increased intra-cortical facilitation at the early recovery phase whereas increased corticospinal pathway excitability was noted at 6 months after IPR discharge.

视神经脊髓炎谱系障碍(NMOSD)是一种罕见的自身免疫性疾病,引起轴突损伤并伴有相应的功能缺陷。在本病例报告中,我们前瞻性地跟踪了一名确诊为NMOSD的女性患者在住院康复(IPR)后6个月的行走恢复和皮质脊髓兴奋性。她在家庭和社区环境中恢复了独立行走功能。经颅磁刺激获得的神经生理测量显示两种时间演化模式。在恢复早期,皮质内抑制明显减少,皮质内促进明显增加,而在IPR出院后6个月,皮质脊髓通路兴奋性明显增加。
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引用次数: 0
Decades-delayed onset of subcutaneous granulomata and calcifications from interferon-beta-1a in a patient with multiple sclerosis 多发性硬化症患者皮下肉芽肿和钙化的干扰素- β -1a延迟发作数十年
Pub Date : 2023-11-17 DOI: 10.1016/j.nerep.2023.100190
Mara Bahri , Kristi Epstein , Yinan Zhang

Multiple sclerosis (MS) is a chronic autoimmune inflammatory disease affecting the central nervous system. Injectable disease-modifying therapies such as interferon-beta have had longstanding and widespread use in MS treatment. We report the case of a 54-year-old woman with relapsing-remitting MS, who developed multiple bilateral subcutaneous granulomata and calcifications on both hips 11 years after a 9-year course of treatment with interferon-beta-1a (IFN-β-1a). We highlight the potential for delayed severe skin responses with subcutaneous IFN-β-1a injections, outline preventative measures, and discuss treatment options for this treatment complication.

多发性硬化症(MS)是一种影响中枢神经系统的慢性自身免疫性炎症疾病。可注射的疾病修饰疗法,如干扰素- β,在多发性硬化症治疗中已有长期和广泛的应用。我们报告一例54岁女性复发缓解型多发性硬化症患者,在接受干扰素-β-1a (IFN-β-1a)治疗9年后,双侧髋关节出现多发皮下肉芽肿和钙化。我们强调皮下注射IFN-β-1a可能会延迟严重的皮肤反应,概述预防措施,并讨论这种治疗并发症的治疗方案。
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引用次数: 0
A case of refractory tumefactive multiple sclerosis in an African American woman 非裔美国妇女难治性肿瘤性多发性硬化症1例
Pub Date : 2023-11-15 DOI: 10.1016/j.nerep.2023.100192
Conor Kelly , Jennings Gyedu , Rock Heyman , Cigdem Isitan Alkawadri

Background

Tumefactive multiple sclerosis (TMS) is a rare subtype of multiple sclerosis (MS) that poses a diagnostic and therapeutic challenge, with relatively little available published data. Though studies have demonstrated similar disease course in TMS with non-tumefactive disease, refractory cases requiring early escalation of therapy are noted and risk factors are unclear. Furthermore, no studies have presented data specifically on adult African American patients with TMS.

Case report

We present a case of TMS refractory to steroids and plasma exchange in an African-American woman. Disease progression was halted after treatment with cyclophosphamide. The patient was later transitioned to rituximab maintenance therapy.

Conclusions

This case contributes to the limited data on disease course and treatment response of this rare disease process in a population that is under-represented in the literature. We note the importance of further studies to identify risk factors for refractory disease requiring early initiation of high-efficacy disease modifying treatment (DMT).

背景:肿瘤性多发性硬化症(TMS)是多发性硬化症(MS)的一种罕见亚型,其诊断和治疗具有挑战性,可获得的已发表数据相对较少。虽然研究表明经颅磁刺激与非肿瘤性疾病的病程相似,但需要早期升级治疗的难治性病例被注意到,危险因素尚不清楚。此外,还没有研究提供针对成年非裔美国人经颅磁刺激患者的具体数据。病例报告:我们报告一例非裔美国妇女经颅磁刺激对类固醇和血浆置换难治的病例。环磷酰胺治疗后疾病进展停止。患者随后转入利妥昔单抗维持治疗。结论:该病例有助于在文献中代表性不足的人群中对这种罕见疾病的病程和治疗反应提供有限的数据。我们注意到进一步研究确定需要早期开始高效疾病修饰治疗(DMT)的难治性疾病的危险因素的重要性。
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引用次数: 0
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Neuroimmunology Reports
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