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Chronic levamisole-induced leukoencephalopathy: Uncommon presentation of two cases with adult-onset progressive symptoms
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
{"title":"Chronic levamisole-induced leukoencephalopathy: Uncommon presentation of two cases with adult-onset progressive symptoms","authors":"João Victor Cabral Correia Férrer,&nbsp;Lara Santos Machado,&nbsp;Luiz José Malta Gaia Ferreira Júnior,&nbsp;Luccas Almeida Andrade,&nbsp;Marianna Pinheiro Moraes de Moraes,&nbsp;José Luiz Pedroso,&nbsp;Orlando Graziani Povoas Barsottini","doi":"10.1016/j.nerep.2025.100247","DOIUrl":"10.1016/j.nerep.2025.100247","url":null,"abstract":"<div><div>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.</div></div>","PeriodicalId":100950,"journal":{"name":"Neuroimmunology Reports","volume":"7 ","pages":"Article 100247"},"PeriodicalIF":0.0,"publicationDate":"2025-01-01","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"143171842","PeriodicalName":null,"FirstCategoryId":null,"ListUrlMain":null,"RegionNum":0,"RegionCategory":"","ArticlePicture":[],"TitleCN":null,"AbstractTextCN":null,"PMCID":"OA","EPubDate":null,"PubModel":null,"JCR":null,"JCRName":null,"Score":null,"Total":0}
引用次数: 0
Atypical initial presentation of MOGAD in a patient with probable tuberculous meningoencephalitis: Case report
Neuroimmunology Reports Pub Date : 2025-01-01 DOI: 10.1016/j.nerep.2024.100237
Diego Canales, Stefany Espinoza, Maria Burgos, Pablo Orosco
{"title":"Atypical initial presentation of MOGAD in a patient with probable tuberculous meningoencephalitis: Case report","authors":"Diego Canales,&nbsp;Stefany Espinoza,&nbsp;Maria Burgos,&nbsp;Pablo Orosco","doi":"10.1016/j.nerep.2024.100237","DOIUrl":"10.1016/j.nerep.2024.100237","url":null,"abstract":"<div><div>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.</div><div>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.</div><div>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.</div></div><div><h3>Conclusion</h3><div>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.</div></div>","PeriodicalId":100950,"journal":{"name":"Neuroimmunology Reports","volume":"7 ","pages":"Article 100237"},"PeriodicalIF":0.0,"publicationDate":"2025-01-01","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"143171843","PeriodicalName":null,"FirstCategoryId":null,"ListUrlMain":null,"RegionNum":0,"RegionCategory":"","ArticlePicture":[],"TitleCN":null,"AbstractTextCN":null,"PMCID":"OA","EPubDate":null,"PubModel":null,"JCR":null,"JCRName":null,"Score":null,"Total":0}
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]
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
{"title":"Erratum to “Atypical presentation of rare entity autoimmune autonomic ganglionopathy: Another potential cause of hypertensive crisis” [Neuroimmunology Reports Volume 2, 2022, 100062]","authors":"Baljinder Singh,&nbsp;Janani Ravichandran,&nbsp;Mohammad Rauf Chaudry,&nbsp;Simrandeep Kaur Brar,&nbsp;Isabel Narvaez,&nbsp;Salvador Cruz-Flores","doi":"10.1016/j.nerep.2024.100244","DOIUrl":"10.1016/j.nerep.2024.100244","url":null,"abstract":"","PeriodicalId":100950,"journal":{"name":"Neuroimmunology Reports","volume":"7 ","pages":"Article 100244"},"PeriodicalIF":0.0,"publicationDate":"2025-01-01","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"143171972","PeriodicalName":null,"FirstCategoryId":null,"ListUrlMain":null,"RegionNum":0,"RegionCategory":"","ArticlePicture":[],"TitleCN":null,"AbstractTextCN":null,"PMCID":"OA","EPubDate":null,"PubModel":null,"JCR":null,"JCRName":null,"Score":null,"Total":0}
引用次数: 0
Exposure to satralizumab in AQP4-positive NMOSD during pregnancy: A case report and review of the literature
Neuroimmunology Reports Pub Date : 2025-01-01 DOI: 10.1016/j.nerep.2025.100248
Ricardo Alonso
{"title":"Exposure to satralizumab in AQP4-positive NMOSD during pregnancy: A case report and review of the literature","authors":"Ricardo Alonso","doi":"10.1016/j.nerep.2025.100248","DOIUrl":"10.1016/j.nerep.2025.100248","url":null,"abstract":"<div><div>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.</div></div>","PeriodicalId":100950,"journal":{"name":"Neuroimmunology Reports","volume":"7 ","pages":"Article 100248"},"PeriodicalIF":0.0,"publicationDate":"2025-01-01","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"143171844","PeriodicalName":null,"FirstCategoryId":null,"ListUrlMain":null,"RegionNum":0,"RegionCategory":"","ArticlePicture":[],"TitleCN":null,"AbstractTextCN":null,"PMCID":"OA","EPubDate":null,"PubModel":null,"JCR":null,"JCRName":null,"Score":null,"Total":0}
引用次数: 0
Tumefactive and longitudinally extensive myelitis: A rare presentation of multiple sclerosis?
Neuroimmunology Reports Pub Date : 2025-01-01 DOI: 10.1016/j.nerep.2025.100257
Adam L. Morehead , Eoin P. Flanagan , Andrew J. Solomon
{"title":"Tumefactive and longitudinally extensive myelitis: A rare presentation of multiple sclerosis?","authors":"Adam L. Morehead ,&nbsp;Eoin P. Flanagan ,&nbsp;Andrew J. Solomon","doi":"10.1016/j.nerep.2025.100257","DOIUrl":"10.1016/j.nerep.2025.100257","url":null,"abstract":"<div><div>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.</div></div>","PeriodicalId":100950,"journal":{"name":"Neuroimmunology Reports","volume":"7 ","pages":"Article 100257"},"PeriodicalIF":0.0,"publicationDate":"2025-01-01","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"143838539","PeriodicalName":null,"FirstCategoryId":null,"ListUrlMain":null,"RegionNum":0,"RegionCategory":"","ArticlePicture":[],"TitleCN":null,"AbstractTextCN":null,"PMCID":"","EPubDate":null,"PubModel":null,"JCR":null,"JCRName":null,"Score":null,"Total":0}
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]
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
{"title":"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]","authors":"Osman Ozel ,&nbsp;Svetlana P. Eckert ,&nbsp;Dejan Jakimovski ,&nbsp;Nicholas Silvestri ,&nbsp;David Hojnacki ,&nbsp;Bianca Weinstock-Guttman","doi":"10.1016/j.nerep.2024.100243","DOIUrl":"10.1016/j.nerep.2024.100243","url":null,"abstract":"","PeriodicalId":100950,"journal":{"name":"Neuroimmunology Reports","volume":"7 ","pages":"Article 100243"},"PeriodicalIF":0.0,"publicationDate":"2025-01-01","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"143104687","PeriodicalName":null,"FirstCategoryId":null,"ListUrlMain":null,"RegionNum":0,"RegionCategory":"","ArticlePicture":[],"TitleCN":null,"AbstractTextCN":null,"PMCID":"OA","EPubDate":null,"PubModel":null,"JCR":null,"JCRName":null,"Score":null,"Total":0}
引用次数: 0
Oral cladribine for management of multiple sclerosis–Clinical experience from Hong Kong
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
{"title":"Oral cladribine for management of multiple sclerosis–Clinical experience from Hong Kong","authors":"Chun-Ho Choi ,&nbsp;Jason Ka-Yeung Fong ,&nbsp;Siu-Hung Li ,&nbsp;Kary Ka-Wai Chan ,&nbsp;Alexander Yuk-Lun Lau","doi":"10.1016/j.nerep.2024.100238","DOIUrl":"10.1016/j.nerep.2024.100238","url":null,"abstract":"<div><h3>Background</h3><div>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.</div></div><div><h3>Methods</h3><div>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.</div></div><div><h3>Results</h3><div>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.</div></div><div><h3>Conclusions</h3><div>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.</div></div>","PeriodicalId":100950,"journal":{"name":"Neuroimmunology Reports","volume":"7 ","pages":"Article 100238"},"PeriodicalIF":0.0,"publicationDate":"2025-01-01","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"143171846","PeriodicalName":null,"FirstCategoryId":null,"ListUrlMain":null,"RegionNum":0,"RegionCategory":"","ArticlePicture":[],"TitleCN":null,"AbstractTextCN":null,"PMCID":"OA","EPubDate":null,"PubModel":null,"JCR":null,"JCRName":null,"Score":null,"Total":0}
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
Neuroimmunology Reports Pub Date : 2025-01-01 DOI: 10.1016/j.nerep.2025.100254
Dong Hyun Kim , Christoph Friedli
{"title":"Contactin-associated protein-like 2 (CASPR2) antibody generation in acute herpes simplex encephalitis: Case report and review of the literature","authors":"Dong Hyun Kim ,&nbsp;Christoph Friedli","doi":"10.1016/j.nerep.2025.100254","DOIUrl":"10.1016/j.nerep.2025.100254","url":null,"abstract":"<div><h3>Introduction</h3><div>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.</div></div><div><h3>Methods</h3><div>This is a case presentation and literature review of HSE with CASPR2 antibody positivity.</div></div><div><h3>Results</h3><div>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.</div></div><div><h3>Conclusion</h3><div>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.</div></div>","PeriodicalId":100950,"journal":{"name":"Neuroimmunology Reports","volume":"7 ","pages":"Article 100254"},"PeriodicalIF":0.0,"publicationDate":"2025-01-01","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"143680775","PeriodicalName":null,"FirstCategoryId":null,"ListUrlMain":null,"RegionNum":0,"RegionCategory":"","ArticlePicture":[],"TitleCN":null,"AbstractTextCN":null,"PMCID":"OA","EPubDate":null,"PubModel":null,"JCR":null,"JCRName":null,"Score":null,"Total":0}
引用次数: 0
High-activity multiple sclerosis in feminizing hormone therapy for gender reassignment
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
{"title":"High-activity multiple sclerosis in feminizing hormone therapy for gender reassignment","authors":"Irene Expósito Ruiz,&nbsp;Cintia Tuñas Gesto,&nbsp;Alfredo Puy Núñez,&nbsp;Eva Costa Arpín","doi":"10.1016/j.nerep.2024.100234","DOIUrl":"10.1016/j.nerep.2024.100234","url":null,"abstract":"","PeriodicalId":100950,"journal":{"name":"Neuroimmunology Reports","volume":"7 ","pages":"Article 100234"},"PeriodicalIF":0.0,"publicationDate":"2025-01-01","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"143104686","PeriodicalName":null,"FirstCategoryId":null,"ListUrlMain":null,"RegionNum":0,"RegionCategory":"","ArticlePicture":[],"TitleCN":null,"AbstractTextCN":null,"PMCID":"OA","EPubDate":null,"PubModel":null,"JCR":null,"JCRName":null,"Score":null,"Total":0}
引用次数: 0
Sudden onset orthostatic dysarthria as a presenting symptom of bulbar onset myasthenia gravis: a video case report
Neuroimmunology Reports Pub Date : 2025-01-01 DOI: 10.1016/j.nerep.2024.100235
Dr. Jānis Dāvis Osipovs , Dr. Eva Šankova , Prof. Guntis Karelis , Dr. Ziedīte Želve , Dr. Elīna Polunosika , Mg. sc. sal. Līga Savicka
{"title":"Sudden onset orthostatic dysarthria as a presenting symptom of bulbar onset myasthenia gravis: a video case report","authors":"Dr. Jānis Dāvis Osipovs ,&nbsp;Dr. Eva Šankova ,&nbsp;Prof. Guntis Karelis ,&nbsp;Dr. Ziedīte Želve ,&nbsp;Dr. Elīna Polunosika ,&nbsp;Mg. sc. sal. Līga Savicka","doi":"10.1016/j.nerep.2024.100235","DOIUrl":"10.1016/j.nerep.2024.100235","url":null,"abstract":"<div><div>Myasthenia gravis (MG) is a rare autoimmune neuromuscular disorder that can present with various early clinical symptoms and signs. The diagnosis of MG with bulbar symptom onset can be clinically challenging for clinicians with limited experience with neuromuscular disorders.</div><div>This case report presents an elderly patient with an abrupt onset of orthostatic dysarthria, in whom a high level of diagnostic suspicion leads to a short period until confirmation of diagnosis. This clinical case shows symptomatic treatment initiation on the 4th day after the onset of symptoms in a patient with bulbar onset MG. Serologic confirmation of MG was received on the 11th hospitalisation day. This case report contains a short video of the evaluation of orthostatic dysarthria.</div><div>Orthostatic dysarthria is a rare presentation symptom for bulbar onset MG. Information on the evaluation of orthostatic dysarthria is sparse. Considering that bulbar onset MG can be a life-threatening condition, we would like to share our positive experience of rapid diagnosis and successful treatment.</div></div>","PeriodicalId":100950,"journal":{"name":"Neuroimmunology Reports","volume":"7 ","pages":"Article 100235"},"PeriodicalIF":0.0,"publicationDate":"2025-01-01","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"143104688","PeriodicalName":null,"FirstCategoryId":null,"ListUrlMain":null,"RegionNum":0,"RegionCategory":"","ArticlePicture":[],"TitleCN":null,"AbstractTextCN":null,"PMCID":"OA","EPubDate":null,"PubModel":null,"JCR":null,"JCRName":null,"Score":null,"Total":0}
引用次数: 0
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