Neuroimmunology Reports最新文献

英文中文

Myasthenic crisis following multiple bee stings: A rare case of neuromuscular dysfunction triggered by hymenoptera envenomation 多次蜜蜂蜇伤后的肌无力危机：一个罕见的由膜翅目昆虫蜇伤引发的神经肌肉功能障碍病例

Neuroimmunology Reports

Pub Date : 2025-08-30 DOI: 10.1016/j.nerep.2025.100265

Vivek Hari , Basavaprabhu Achappa , Ayushi Gupta , Shivananda Pai D

Introduction

Hymenoptera stings which include stings from bees, wasps, hornets, and fire ants, typically cause minor local reactions. However, systemic responses, including anaphylaxis and rare delayed complications, can occur. One such rare complication is Myasthenia Gravis (MG), a neuromuscular junction disorder characterized by skeletal muscle weakness due to antibodies against acetylcholine receptors (AChR). We report a case of acute neuromuscular weakness resembling a myasthenic crisis shortly after multiple bee stings.

Case Presentation

A 64-year-old male with type 2 diabetes and a history of Lumbar IVDP, presented after being stung by approximately 200 bees. He initially complained of facial puffiness, limb swelling, and generalized itching. He was febrile, bradycardic, and had diffuse erythema and edema. Initial management included antihistamines, systemic steroids, and stinger removal. Twelve hours later, he developed nausea, vomiting, and sudden onset dysphagia, ptosis, and proximal muscle weakness. Shortly after, he became dyspneic and required ICU admission with ventilatory support. Laboratory investigations revealed elevated CPK (2468 mcg/L) and negative Anti-AChR antibodies. On clinical grounds, he was treated with pyridostigmine and emergency plasmapheresis, resulting in gradual neuromuscular improvement. Supportive care was continued, and he was subsequently weaned off ventilator and was tracheostomized.

Conclusion

This case illustrates a rare myasthenia-like crisis triggered by multiple bee stings. Although confirmatory electrophysiological and serological evidence was lacking, the clinical presentation and therapeutic response strongly suggested neuromuscular junction dysfunction. The sequential association of bee sting and the onset of myasthenia gravis is too close to suppose chance association. Lack of immunologic abnormality may suggest the same. Though few cases of wasp sting causing Myasthenia have been reported, bee sting leading to neurological symptoms are unheard of. Our case highlights the importance of anticipating as well as early recognition of such complications.

膜翅目昆虫的蜇伤包括蜜蜂、黄蜂、大黄蜂和火蚁的蜇伤，通常会引起轻微的局部反应。然而，可发生全身反应，包括过敏反应和罕见的延迟并发症。其中一种罕见的并发症是重症肌无力（MG），这是一种神经肌肉连接障碍，以抗乙酰胆碱受体（AChR）抗体引起的骨骼肌无力为特征。我们报告一例急性神经肌肉无力类似肌无力危机后不久，多次蜜蜂蜇伤。病例介绍男性，64岁，2型糖尿病，腰椎IVDP病史，被约200只蜜蜂叮咬后出现。他最初主诉面部浮肿、四肢肿胀和全身瘙痒。他发热，心动过缓，有弥漫性红斑和水肿。最初的治疗包括抗组胺药、全身类固醇和去除毒刺。12小时后，患者出现恶心、呕吐、突发性吞咽困难、上睑下垂和近端肌无力。不久之后，他出现呼吸困难，需要在呼吸支持下住进ICU。实验室检查显示CPK升高（2468 mcg/L），抗achr抗体阴性。由于临床原因，他接受了吡哆斯的明和紧急血浆置换治疗，导致神经肌肉逐渐改善。继续进行支持性治疗，随后他断开呼吸机并进行气管造口术。结论本病例是一例罕见的由多次蜜蜂蜇伤引起的肌无力危象。虽然缺乏确切的电生理和血清学证据，但临床表现和治疗反应强烈提示神经肌肉连接功能障碍。蜜蜂蜇伤与重症肌无力发病的顺序关系非常密切，不能认为是偶然的联系。缺乏免疫异常可能提示相同。虽然很少有黄蜂蜇伤导致重症肌无力的病例报道，但蜜蜂蜇伤导致神经系统症状是闻所未闻的。我们的病例强调了预测和早期识别这些并发症的重要性。

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引用次数: 0

“When HIV meets MG: Is immune reconstitution in the mix?” “当HIV与MG相遇时：是否存在免疫重建？”

Neuroimmunology Reports

Pub Date : 2025-08-18 DOI: 10.1016/j.nerep.2025.100263

Aditya Vijayakrishnan Nair, Lesley Ponraj, Ajith Sivadasan, Sanjit Aaron

Background

Autoimmune disorders and human immunodeficiency virus (HIV) positivity rarely coexist. We report a case of anti-muscle-specific kinase (MuSK) antibody-positive myasthenia gravis (MuSK+ MG) in an HIV positive patient. Antiretroviral therapy resulted in an immune reconstitution syndrome, resulting in worsening of MG symptoms. However, the role of additional immunomodulation in this setting has yet to be clarified.

Case Presentation

A 44-year-old woman with virologically suppressed HIV infection presented with fluctuating oculobulbar symptoms, limb weakness, and respiratory failure. These symptoms were noticed around 6 months following antiretroviral therapy (ART) modification. Electrophysiological studies and elevated anti-MuSK antibody levels confirmed MuSK + MG. An increase in the CD4 count from 115 to 681 cells/µL following the ART modification was consistent with immune reconstitution. Treatment with corticosteroids, Rituximab, and opportunistic infection prophylaxis resulted in a reduction in her MG symptoms with no adverse side effects.

Conclusions

This case underscores the complexities of managing autoimmune diseases in HIV, where ART-induced immune reconstitution may unmask or trigger autoimmunity. Early diagnosis and targeted therapies can improve outcomes. In this case, Rituximab initiation facilitated symptom control and steroid tapering. The need for a multidisciplinary approach to balance immunosuppression and infection risks has been emphasized.

自身免疫性疾病和人类免疫缺陷病毒（HIV）阳性很少共存。我们报告一例抗肌肉特异性激酶（MuSK）抗体阳性的重症肌无力（MuSK+ MG）在HIV阳性患者。抗逆转录病毒治疗导致免疫重建综合征，导致MG症状恶化。然而，在这种情况下，额外的免疫调节的作用尚未得到澄清。病例表现：一名44岁女性，病毒学抑制HIV感染，表现为波动性眼球症状、肢体无力和呼吸衰竭。这些症状在抗逆转录病毒治疗（ART）修改后约6个月被注意到。电生理研究和抗MuSK抗体水平升高证实了MuSK + MG。在ART修饰后，CD4细胞计数从115个增加到681个/µL，这与免疫重建是一致的。皮质类固醇、利妥昔单抗和机会性感染预防治疗使她的MG症状减轻，无不良副作用。结论：本病例强调了管理HIV自身免疫性疾病的复杂性，art诱导的免疫重建可能揭示或触发自身免疫。早期诊断和靶向治疗可以改善预后。在这种情况下，利妥昔单抗开始促进症状控制和类固醇逐渐减少。需要一个多学科的方法来平衡免疫抑制和感染风险已被强调。

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引用次数: 0

Serum and cerebrospinal fluid AQP4-MOG double-positive longitudinally extensive transverse myelitis: a rare case report and literature review 血清和脑脊液AQP4-MOG双阳性纵向广泛横贯脊髓炎1例罕见报告并文献复习

Neuroimmunology Reports

Pub Date : 2025-08-18 DOI: 10.1016/j.nerep.2025.100264

Lulu Chu , Wenbo Yang , Hai Yu , Xiang Zhang , Xiaoni Liu , Xiangjun Chen

Objective

The following case report details a patient with serum and cerebrospinal fluid AQP4-MOG double-positive longitudinally extensive transverse myelitis, whose condition was found to be progressively exacerbated and which resulted in severe disability.

Case presentation

A 51-year-old female patient initially presented with pain and discomfort in the left upper limb and posterior occipital region for a period of 10 days. Unfortunately, she then developed hypoesthesia and flaccid paralysis, in addition to urinary retention within a few days. Concurrently, she exhibited symptoms consistent with area postrema syndrome (nausea, vomiting, and hiccups) and brain-stem syndrome (hoarseness and dysphagia). Concomitantly, AQP4 and MOG antibodies were identified as positive in both the serum and cerebrospinal fluid (CSF). MRI findings were consistent with longitudinally extensive transverse myelitis (LETM), spanning cervical through thoracic spinal cord levels. Despite presenting with meningismus, the patient's gadolinium-enhanced brain MRI revealed no abnormal leptomeningeal enhancement. Following treatment with pulse intravenous methylprednisolone and plasmapheresis, a stabilisation of symptoms was observed.

Conclusion

Patients presenting with longitudinally extensive transverse myelitis may exhibit a positive double-immunofluorescence test result for aquaporin-4 (AQP4) and myelin oligodendrocyte glycoprotein (MOG) in both serum and CSF. The administration of early, more potent immunotherapy has the potential to enhance outcomes.

目的报告1例血清和脑脊液AQP4-MOG双阳性的纵向广泛横断性脊髓炎患者，病情逐渐加重，导致严重残疾。患者女，51岁，首发表现为左上肢及枕后区疼痛不适，持续10天。不幸的是，她随后出现了感觉减退和弛缓性麻痹，并在几天内出现尿潴留。同时，她表现出与区域后综合征（恶心、呕吐和打嗝）和脑干综合征（声音嘶哑和吞咽困难）一致的症状。同时，血清和脑脊液中AQP4和MOG抗体均为阳性。MRI结果与纵向广泛横贯脊髓炎（LETM）一致，跨越颈椎至胸脊髓水平。尽管表现为脑膜炎，但患者的钆增强脑MRI未显示异常的脑膜增强。经脉搏静脉注射甲基强的松龙和血浆置换治疗后，观察到症状稳定。结论纵向广泛横断性脊髓炎患者血清和脑脊液中水通道蛋白-4 （AQP4）和髓鞘少突胶质细胞糖蛋白（MOG）的双免疫荧光检测结果均为阳性。早期给予更有效的免疫治疗有可能提高预后。

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引用次数: 0

Interferon beta-induced paracentral acute middle maculopathy: A case report 干扰素诱发的中央旁急性中黄斑病变1例

Neuroimmunology Reports

Pub Date : 2025-07-14 DOI: 10.1016/j.nerep.2025.100262

Hamlet Ghukasyan , Laura Gabaldón Torres , María García-Pous

引用次数: 0

Decoding a cryptic brain 解码一个神秘的大脑

Neuroimmunology Reports

Pub Date : 2025-07-09 DOI: 10.1016/j.nerep.2025.100261

Sreelakshmi Narayanan, Mahima Sriram, Tejas Shivarthi, Sudheeran Kannoth, Siby Gopinath, Vivek Nambiar, Udit Saraf, Gopikrishnan Unnikrishnan, Anandkumar Anandakuttan

Background

Cryptococcosis is a deadly infection caused by Cryptococcus which majorly affects immunosuppressed individuals but may also be seen in immunocompetent hosts. It may present as meningitis, meningoencephalitis and CNS cryptococcomas. We report a unique presentation of CNS cryptococcosis.

Results

We report a case of a 48 year old lady with a background of diabetes mellitus and hypothyroidism, who presented with apathy and aphasia. Her routine blood investigations were normal and her MRI Brain revealed T2 fluid attenuated inversion recovery (FLAIR) hyperintensities with post contrast enhancement and diffusion restriction seen in cortical and deep white matter.

CSF showed mild pleocytosis and all infective panels and routine auto-antibody panels were negative. Considering a possible etiology of demyelination, she was pulsed with steroids during the course of disease yet worsened clinico-radiologically. Her brain biopsy was indicative of demyelination, and Cryptococcus PCR in CSF and biopsy specimen were positive. Repeat India ink and fungal culture persisted to be negative. The patient was treated with amphotericin and fluconazole.

Conclusions

The immunopathogenesis in cryptococcal infection is complex and the fungus-host interaction determines the outcome. Our case showed extensive demyelination due to the excess inflammation mounted by the host, but the process was ineffective in clearing the fungus as evidenced by PCR positivity on CSF and biopsy specimens. Worsening on steroids and positive response to anticryptococcal regime gives the evidence of a fungus mediated disease. This case shows that any demyelination should be extensively investigated for secondary causes before labelling it resistant.

隐球菌病是一种由隐球菌引起的致命感染，主要影响免疫抑制的个体，但也可以在免疫正常的宿主中看到。它可能表现为脑膜炎、脑膜脑炎和中枢神经系统隐球菌病。我们报告一个独特的CNS隐球菌病的表现。结果我们报告了一位48岁的女性，她有糖尿病和甲状腺功能减退的背景，她表现出冷漠和失语。她的血常规检查正常，她的脑部MRI显示T2液体衰减反转恢复（FLAIR）高信号，在皮质和深部白质中可见增强和扩散限制。脑脊液显示轻度多细胞增多，所有感染面板和常规自身抗体面板均为阴性。考虑到脱髓鞘可能的病因，她在疾病过程中使用类固醇，但临床放射学恶化。她的脑组织活检提示脱髓鞘，脑脊液和活检标本隐球菌PCR阳性。重复印墨和真菌培养仍然呈阴性。患者给予两性霉素和氟康唑治疗。结论隐球菌感染的免疫发病机制复杂，真菌与宿主的相互作用决定了感染的结果。我们的病例显示了由于宿主过度炎症引起的广泛脱髓鞘，但该过程对清除真菌无效，脑脊液和活检标本的PCR阳性证明了这一点。类固醇治疗的恶化和抗隐球菌治疗的积极反应提供了真菌介导疾病的证据。这个病例表明，任何脱髓鞘应广泛调查的继发性原因之前，标记其耐药。

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引用次数: 0

Chronic levamisole-induced leukoencephalopathy: Uncommon presentation of two cases with adult-onset progressive symptoms 慢性左旋咪唑诱导的脑白质病：两例成人发病进行性症状的罕见表现

Neuroimmunology Reports

Pub Date : 2025-01-01 DOI: 10.1016/j.nerep.2025.100247

João Victor Cabral Correia Férrer, Lara Santos Machado, Luiz José Malta Gaia Ferreira Júnior, Luccas Almeida Andrade, Marianna Pinheiro Moraes de Moraes, José Luiz Pedroso, Orlando Graziani Povoas Barsottini

Toxic leukoencephalopathies are abnormalities in the cerebral white matter associated with the use of legal or illegal substances. Levamisole-induced leukoencephalopathy (LILE) is a rare condition that typically presents with a subacute progression and has a clear temporal relationship to compound exposure. However, we describe two cases with a chronic progression of adult-onset progressive spastic-ataxia syndrome, likely due to recurrent exposure to levamisole. Both cases exhibited typical abnormalities on brain magnetic resonance imaging (MRI) and had normal cerebrospinal fluid (CSF) analysis. In both instances, clinical stability was closely associated with drug abstinence. These cases are distinct from the literature due to their chronic presentation, and LILE should always be considered as a differential diagnosis when toxic exposure is suspected.

中毒性脑白质病是与使用合法或非法物质有关的脑白质异常。左旋咪唑诱导的白质脑病（LILE）是一种罕见的疾病，通常表现为亚急性进展，与化合物暴露有明确的时间关系。然而，我们描述了两例慢性进展的成人发作进行性痉挛性共济失调综合征，可能是由于反复暴露于左旋咪唑。两例均表现出典型的脑磁共振成像（MRI）异常，脑脊液分析正常。在这两种情况下，临床稳定性与药物戒断密切相关。这些病例由于其慢性表现而与文献不同，当怀疑毒性暴露时，应始终将LILE视为鉴别诊断。

引用次数: 0

Atypical initial presentation of MOGAD in a patient with probable tuberculous meningoencephalitis: Case report 一例疑似结核性脑膜脑炎患者MOGAD的不典型初始表现：病例报告

Neuroimmunology Reports

Pub Date : 2025-01-01 DOI: 10.1016/j.nerep.2024.100237

Diego Canales, Stefany Espinoza, Maria Burgos, Pablo Orosco

Tuberculosis (TB) is an endemic disease in our country that effects on various organs and at the nervous system. The diagnosis requires confirmatory tests, however if the suspicion is high, empirical treatment can be initiated with subsequent follow-up of the patient.

TB has been associated with neuromyelitis optica, however it didn't associate with MOG antibody disease (MOGAD). MOGAD is a rare entity in the adult population; its main manifestation is bilateral optic neuritis or acute disseminated encephalitis. Which also requires confirmatory tests and immunosuppressive treatment.

We report the case of a patient diagnosed with TB meningoencephalitis, in the second month of empirical treatment with anti-TB drugs, who was admitted to our service due to persistence of epileptic seizures and greater deterioration in the level of alertness. She was tested for infectious, autoimmune, tumor and metabolic diseases, which came out within the normal range. Magnetic resonance imaging showed apparently sequelae diffuse bilateral leukodystrophy without contrast enhancement. The possibility of autoimmune encephalitis was evaluated. Therefore, it decided to start treatment with immunoglobulin for 6 cycles. During her evolution, clinical improvement was evident, without epileptic seizures, with better interaction with his environment and she began to walk with a walker. At the end of the anti-TB and immunoglobulin treatment, she presented an episode of dysarthria and facial asymmetry. The new image showed nodular lesions in the midbrain and cerebellum, some of which captured contrast. Therefore, the studies were expanded and an anti-MOG test was performed, which was positive. It was decided to start immunoglobulin cycles. With apparently good clinical evolution.

Conclusion

We present the case of an atypical initial presentation of MOGAD. This should be taken into account in cases where there is no adequate clinical improvement, since treatment with corticosteroids or immunoglobulins may be effective in these patients and prevent serious sequelae.

结核病（TB）是我国一种影响人体各器官和神经系统的地方病。诊断需要确认性检查，但如果怀疑程度高，可通过对患者的后续随访开始经验性治疗。结核与视神经脊髓炎相关，但与MOG抗体病（MOGAD）无关。MOGAD在成年人群中是一个罕见的实体；主要表现为双侧视神经炎或急性播散性脑炎。这也需要确认性检查和免疫抑制治疗。我们报告的情况下，诊断为结核脑膜脑炎的病人，在第二个月的经验治疗抗结核药物，谁是入院我们的服务，由于持续的癫痫发作和警觉性水平更大的恶化。她接受了传染病、自身免疫性疾病、肿瘤和代谢疾病的检查，结果都在正常范围内。磁共振成像显示明显的双侧弥漫性脑白质营养不良后遗症，无增强。评估自身免疫性脑炎的可能性。因此，决定开始使用免疫球蛋白治疗6个周期。在她的发展过程中，临床改善很明显，没有癫痫发作，与环境的互动更好，她开始用助行器走路。在抗结核和免疫球蛋白治疗结束时，她出现构音障碍和面部不对称。新图像显示了中脑和小脑的结节状病变，其中一些得到了对比。因此，我们扩大了研究范围，进行了抗mog试验，结果为阳性。决定开始免疫球蛋白循环。临床进展明显良好。结论我们提出一例非典型的MOGAD的初始表现。在没有充分临床改善的情况下，应考虑到这一点，因为皮质类固醇或免疫球蛋白治疗可能对这些患者有效，并防止严重的后遗症。

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引用次数: 0

Erratum to “Atypical presentation of rare entity autoimmune autonomic ganglionopathy: Another potential cause of hypertensive crisis” [Neuroimmunology Reports Volume 2, 2022, 100062] 对“罕见实体自身免疫性自主神经节病的非典型表现：高血压危象的另一个潜在原因”的勘误[神经免疫学报告第2卷，2022,100062]

Neuroimmunology Reports

Pub Date : 2025-01-01 DOI: 10.1016/j.nerep.2024.100244

Baljinder Singh, Janani Ravichandran, Mohammad Rauf Chaudry, Simrandeep Kaur Brar, Isabel Narvaez, Salvador Cruz-Flores

引用次数: 0

Exposure to satralizumab in AQP4-positive NMOSD during pregnancy: A case report and review of the literature 妊娠期aqp4阳性NMOSD暴露于satralizumab：一例报告和文献回顾

Neuroimmunology Reports

Pub Date : 2025-01-01 DOI: 10.1016/j.nerep.2025.100248

Ricardo Alonso

Neuromyelitis optica spectrum disorder (NMOSD) is a severe autoimmune disease of the central nervous system. It mainly affects young women and can lead to serious episodes involving the spinal cord and optic nerves. Treatments, may pose risks to the fetus, with limited safety data available. Satralizumab, is effective in reducing relapse rates but lacks sufficient safety data for use during pregnancy. The case of a 32-year-old woman with NMOSD who was treated with satralizumab during her first trimester is presented. This case provides preliminary evidence on satralizumab use in pregnancy, suggesting it might be a viable option with careful management.

视神经脊髓炎谱系障碍（NMOSD）是一种严重的中枢神经系统自身免疫性疾病。它主要影响年轻女性，并可导致严重的脊髓和视神经发作。治疗可能对胎儿构成风险，可获得的安全性数据有限。Satralizumab在降低复发率方面有效，但缺乏足够的安全性数据用于妊娠期间使用。一个32岁的妇女NMOSD谁是治疗与satralizumab在她的前三个月提出。本病例提供了妊娠期使用satralizumab的初步证据，表明在谨慎管理下，它可能是一个可行的选择。

引用次数: 0

Tumefactive and longitudinally extensive myelitis: A rare presentation of multiple sclerosis? 浸润性和纵向广泛的脊髓炎：多发性硬化症的罕见表现？

Neuroimmunology Reports

Pub Date : 2025-01-01 DOI: 10.1016/j.nerep.2025.100257

Adam L. Morehead , Eoin P. Flanagan , Andrew J. Solomon

Cerebral tumefactive demyelinating lesions are well described in multiple sclerosis (MS). Yet few cases of MS-associated tumefactive spinal cord demyelination have been described. We report a patient with a clinical course typical of relapsing-remitting MS who presented with a relapse associated with a tumefactive and longitudinally extensive cervical spinal cord lesion. Despite extensive evaluation, and with two years of longitudinal follow up, no better explanation than MS could be identified. This case highlights the diagnostic and prognostic challenges associated with presumed tumefactive spinal cord demyelination in MS, and the unmet need for further cases to guide therapeutic decision-making.

脑膨胀性脱髓鞘病变在多发性硬化症（MS）中有很好的描述。然而，很少有病例ms相关的肿瘤性脊髓脱髓鞘已被描述。我们报告了一个典型的复发-缓解型多发性硬化症的患者，他的复发与肿瘤性和纵向广泛的颈脊髓病变有关。尽管进行了广泛的评估，并进行了两年的纵向随访，但没有比MS更好的解释。该病例强调了与多发性硬化症推定的肿瘤性脊髓脱髓鞘相关的诊断和预后挑战，以及对进一步病例指导治疗决策的未满足需求。

引用次数: 0

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