Case Reports in Neurology最新文献

{"title":"Superior Ophthalmic Vein Thrombosis Presenting as Isolated Headache: A Case Report.","authors":"Vincenzo Laterza, Alfredo De Liso, Francesco Macchione, Alessandra Bavaro, Marco Rossi, Giuseppe Rinaldi","doi":"10.1159/crn/abyag020","DOIUrl":"https://doi.org/10.1159/crn/abyag020","url":null,"abstract":"<p><strong>Introduction: </strong>Superior ophthalmic vein thrombosis (SOVT) is a rare condition that can present with varied symptoms, including headache and orbital pain. Its diagnosis requires advanced imaging techniques, including CT angiography and MRI. This case report describes a patient presenting with headache and orbital pain attributed to superior ophthalmic vein dilation and thrombosis.</p><p><strong>Case presentation: </strong>An 81-year-old female patient presented with headache, orbital pain, periorbital edema, and mild palpebral ptosis. Initial investigations included a brain CT scan and CT angiography, which revealed tortuous and dilated superior ophthalmic veins with massive thrombosis of the left superior ophthalmic vein. Brain MRI and MR angiography were also performed. Other examinations included ophthalmological evaluation, fluorescein angiography, and blood tests. The patient's symptoms, including headache and orbital pain, were attributed to the confirmed dilation and massive thrombosis of the left superior ophthalmic vein. CT angiography initially suggested a carotid-cavernous fistula, which was subsequently excluded by brain MRI and MR angiography. Fluorescein angiography revealed severe myopic chorioretinopathy with peripapillary atrophy and disc hyperfluorescence consistent with venous congestion in the left eye. Thrombophilia screening identified elevated anticardiolipin IgM antibodies, raising suspicion for antiphospholipid syndrome (APS). Anticoagulant therapy was initiated with enoxaparin and subsequently transitioned to warfarin, in accordance with current guidelines that favor vitamin K antagonists over direct oral anticoagulants for APS-associated thrombosis.</p><p><strong>Conclusion: </strong>This case highlights the importance of considering SOVT in the differential diagnosis of headache accompanied by orbital symptoms.</p>","PeriodicalId":9639,"journal":{"name":"Case Reports in Neurology","volume":"18 1","pages":"654-660"},"PeriodicalIF":0.4,"publicationDate":"2026-10-10","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"https://www.ncbi.nlm.nih.gov/pmc/articles/PMC13688602/pdf/","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"149263192","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}

{"title":"Methyldigoxin-Induced Visual Hallucinations in an Elderly Patient with Advanced Fazekas Scale 3 Leukoaraiosis: A Diagnostic Challenge and Case Report.","authors":"Mario Ćurković, Tamer Salha, Mladen Oršolić, Nika Ćurković","doi":"10.1159/crn/abyag018","DOIUrl":"https://doi.org/10.1159/crn/abyag018","url":null,"abstract":"<p><strong>Introduction: </strong>Digoxin and its derivatives remain valuable adjunctive therapies for rate control in atrial fibrillation, yet their narrow therapeutic index poses a significant risk for neurotoxicity, particularly in the geriatric population.</p><p><strong>Case presentation: </strong>We report the case of a 79-year-old female with heart failure and chronic kidney disease who developed vivid, complex visual hallucinations 2 weeks after initiating methyldigoxin therapy. The patient's medical history and subsequent magnetic resonance imaging revealed advanced subcortical vascular encephalopathy, characterized by confluent perivascular gliosis (Fazekas scale 3). This structural background initially raised suspicion of a transient ischemic attack or progressing vascular dementia. However, the patient maintained a high cognitive baseline (MMSE 28/30), and the hallucinations - which included perceiving her deceased husband and grandson - resolved completely within 4 days of methyldigoxin discontinuation. This rapid recovery, combined with the absence of new focal neurological deficits, suggests digitalis induced neurotoxicity rather than a primary cerebrovascular event.</p><p><strong>Conclusion: </strong>This case underscores the \"clinical-radiological discrepancy\" often encountered in elderly patients, where advanced neuroimaging findings may act as a confounding factor. It highlights the necessity of maintaining a high index of suspicion for drug-induced causes of acute neurological symptoms in patients with a \"vulnerable brain\" due to severe leukoaraiosis. Careful monitoring of digitalis compounds in patients with impaired renal function is mandatory to prevent misdiagnosis and unnecessary interventions.</p>","PeriodicalId":9639,"journal":{"name":"Case Reports in Neurology","volume":"18 1","pages":"648-653"},"PeriodicalIF":0.4,"publicationDate":"2026-10-09","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"https://www.ncbi.nlm.nih.gov/pmc/articles/PMC13688626/pdf/","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"149263242","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}

{"title":"Rebound-Like Inflammatory Activity Presenting as a Tumefactive Demyelinating Lesion after Teriflunomide Discontinuation: A Case Report.","authors":"Alpkağan Kale, Nermin Tepe","doi":"10.1159/crn/abyag014","DOIUrl":"https://doi.org/10.1159/crn/abyag014","url":null,"abstract":"<p><strong>Introduction: </strong>Rebound after teriflunomide discontinuation is uncommon and difficult to distinguish from ordinary disease reactivation without a documented pretreatment baseline. Tumefactive demyelination is a rarely reported post-cessation phenotype.</p><p><strong>Case presentation: </strong>This report presents a 39-year-old woman with relapsing-remitting multiple sclerosis had no relapses, Expanded Disability Status Scale (EDSS) worsening, or new MRI lesions during approximately 5 years of teriflunomide treatment. She stopped treatment without medical supervision or an accelerated elimination procedure for pregnancy planning. Four months later, persistent headache prompted MRI, which showed a 2.6 × 2.6-cm right frontal periventricular tumefactive demyelinating lesion with edema, peripheral diffusion restriction, and mild ring enhancement, plus a new left cerebellar plaque. EDSS remained 0. Absolute lymphocyte count rose from 600 cells/μL at the last on-treatment visit to 1,700 cells/μL at presentation. Serum MOG-IgG and AQP4-IgG and CSF cytology were negative; a type 2 oligoclonal-band pattern was present, and targeted infectious screening showed no evidence of active infection. Teriflunomide concentration was <0.02 mg/L twice, 14 days apart. Intravenous methylprednisolone (1 g/day for 5 days) resolved the headache. Cladribine was then started, and 1-month MRI showed marked regression. EDSS remained 0 and no new or enhancing lesion appeared through 12 months.</p><p><strong>Conclusion: </strong>The tumor-like lesion exceeded the patient's pretreatment radiological phenotype, but the absence of objective deficit, EDSS worsening, and a high lesion burden makes definitive rebound uncertain. Rebound-like post-cessation inflammatory activity is the most defensible description. Supervised treatment transition and clinical and MRI monitoring are important when teriflunomide is stopped.</p>","PeriodicalId":9639,"journal":{"name":"Case Reports in Neurology","volume":"18 1","pages":"640-647"},"PeriodicalIF":0.4,"publicationDate":"2026-10-05","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"https://www.ncbi.nlm.nih.gov/pmc/articles/PMC13688590/pdf/","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"149263175","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}

{"title":"An Unusual Mechanism of Stroke in Neurobrucellosis: Middle Cerebral Artery Infarction Secondary to Direct Abscess Compression - A Case Report and Literature Review.","authors":"Ruba Adel Aweer, Majd A AbuAlrob, Khaled Zammar, Ahmad Elsotouhy","doi":"10.1159/crn/abyag015","DOIUrl":"https://doi.org/10.1159/crn/abyag015","url":null,"abstract":"<p><strong>Introduction: </strong>Brucellosis is a zoonotic infection that remains endemic across the Middle East, Central Asia, the Mediterranean basin, and parts of Africa and Latin America. Neurobrucellosis (NB) develops in fewer than 5% of cases and constitutes the most severe systemic complication. Cerebrovascular involvement is uncommon and most often attributed to small-vessel vasculitis, immune-mediated endarteritis, or septic embolization. Ischemic stroke caused by direct mechanical compression of an intracranial artery by a Brucella abscess has not been previously reported.</p><p><strong>Case presentation: </strong>A 47-year-old previously healthy male with occupational livestock exposure presented with sudden onset of left-sided weakness, facial droop, and global aphasia. Initial imaging demonstrated an acute right middle cerebral artery (MCA) territory infarct, and standard ischemic stroke management was initiated. Over the next 24 h, intermittent low-grade fever, persistent confusion, and focal motor seizures emerged despite levetiracetam. Subsequent magnetic resonance imaging showed a 2.5 cm rim-enhancing lesion in the right temporal lobe with central diffusion restriction, consistent with a pyogenic abscess. Magnetic resonance angiography demonstrated focal occlusion of the anterior segment of the right MCA, immediately adjacent to the abscess. Cerebrospinal fluid showed lymphocytic pleocytosis (36 cells/mm³, 85% lymphocytes) and elevated protein (112 mg/dL). Serum Brucella IgG returned positive on day 20, confirming neurobrucellosis. Triple antimicrobial therapy with intravenous ceftriaxone, oral doxycycline, and oral rifampicin produced gradual neurological improvement, and the patient was discharged on long-term oral therapy.</p><p><strong>Conclusion: </strong>This case illustrates a novel cerebrovascular mechanism in NB, in which mass effect from a peri-vascular abscess produced ischemic stroke. Coexisting perilesional inflammation likely contributed through a vasculitic component, and the two mechanisms are not mutually exclusive. The absence of systemic brucellosis features delayed recognition and underscores the protean clinical presentation of NB. Brucellosis warrants consideration in the differential diagnosis of acute ischemic stroke when imaging reveals concurrent intracranial infectious lesions, particularly in endemic regions. Early antimicrobial therapy is critical to limit progression and improve neurological outcomes.</p>","PeriodicalId":9639,"journal":{"name":"Case Reports in Neurology","volume":"18 1","pages":"630-639"},"PeriodicalIF":0.4,"publicationDate":"2026-10-05","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"https://www.ncbi.nlm.nih.gov/pmc/articles/PMC13688592/pdf/","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"149263162","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}

{"title":"Cortical Spreading Depression Mimicking Ischemic Stroke: A Case Report of Complex Hemiplegic Migraine with Reversible Cortical Magnetic Resonance Imaging Changes.","authors":"Ali Hammed, Salah Hammed, Thomas Stein, Ulrich Niemöller, Marco Meyer, Christian Tanislav","doi":"10.1159/000551416","DOIUrl":"https://doi.org/10.1159/000551416","url":null,"abstract":"<p><strong>Introduction: </strong>Hemiplegic migraine (HM) is a rare subtype of migraine with aura characterized by transient motor weakness and focal neurological deficits that can closely resemble acute ischemic stroke. Cortical spreading depression, the underlying electrophysiological mechanism, produces transient neuronal and vascular changes that may lead to stroke-like imaging findings, creating diagnostic challenges in emergency settings.</p><p><strong>Case presentation: </strong>We describe a 66-year-old man with a long-standing history of migraine with aura who presented with acute left-sided hemiparesis, dysarthria, and visual disturbances. Initial computed tomography revealed subtle sulcal effacement without evidence of hemorrhage. Brain MRI demonstrated right hemispheric diffusion-weighted hyperintensities without corresponding apparent diffusion coefficient (ADC) reduction, suggesting transient cytotoxic edema rather than infarction. Electroencephalography showed right hemispheric slowing without epileptiform discharges, and cerebrospinal fluid analysis was unremarkable. The patient experienced gradual clinical improvement over 7 days, with mild residual fine motor deficits at discharge. Preventive therapy with flunarizine was initiated.</p><p><strong>Conclusion: </strong>This case underscores the importance of recognizing HM as a stroke mimic. Integration of the clinical context with MRI findings - particularly the absence of ADC restriction - can help differentiate HM from acute ischemic stroke and guide appropriate clinical management. Early and accurate diagnosis, together with individualized prophylactic treatment, may help reduce recurrence and long-term morbidity.</p>","PeriodicalId":9639,"journal":{"name":"Case Reports in Neurology","volume":"18 1","pages":"558-566"},"PeriodicalIF":0.4,"publicationDate":"2026-09-21","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"https://www.ncbi.nlm.nih.gov/pmc/articles/PMC13630380/pdf/","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"149104572","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}

{"title":"Hemorrhagic Transformation of HSV-1 Encephalitis Presenting as Ischemic Stroke and Discussion of Uncertainties in Clinical Management: A Case Report.","authors":"Jonathan I Eini, Janak Patel, Nerses Sanossian, Roy A Poblete","doi":"10.1159/crn/abyag013","DOIUrl":"https://doi.org/10.1159/crn/abyag013","url":null,"abstract":"<p><strong>Introduction: </strong>Herpes simplex virus (HSV) encephalitis is the most common cause of sporadic encephalitis in the USA. Both ischemic and hemorrhagic brain injury are commonly recognized complications of central nervous system (CNS) HSV infections, with radiographic findings such as diffusion restriction that can mimic acute ischemic stroke (AIS). The risk of hemorrhage complicates the use of antithrombotic therapy in patients with HSV encephalitis and concurrent ischemic injury or venous thromboembolic disease. Still, no clear guidelines exist on the use of antithrombotic drugs in patients with HSV encephalitis.</p><p><strong>Case presentation: </strong>In this case report, we describe a patient with history of stroke who was initially diagnosed with AIS and treated with antiplatelet therapy and venous thromboembolism chemoprophylaxis, later found to have superimposed HSV-1 encephalitis and subsequently developed hemorrhagic transformation. The clinical course, including evolving neurologic deficits, seizure activity, and imaging progression, raised uncertainty as to whether the lesions represented true ischemic infarction, HSV-related cytotoxic injury, or a combination of both. Antithrombotic therapy was continued due to the history of stroke, vascular risk factors, and a concern for concurrent ischemic injury, highlighting competing risks.</p><p><strong>Conclusion: </strong>Hemorrhagic transformation occurring in HSV encephalitis during antithrombotic therapy for suspected AIS is a rarely reported finding in the literature. This case underscores the limitations of applying standard stroke paradigms in CNS infection, where hemorrhagic transformation may reflect disease evolution and causality related to antithrombotic therapy is uncertain.</p>","PeriodicalId":9639,"journal":{"name":"Case Reports in Neurology","volume":"18 1","pages":"612-620"},"PeriodicalIF":0.4,"publicationDate":"2026-09-05","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"https://www.ncbi.nlm.nih.gov/pmc/articles/PMC13688603/pdf/","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"149263170","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}

{"title":"Transcutaneous Auricular Vagus Nerve Stimulation in a Pacemaker-Dependent Child with Syndromic Drug-Resistant Epilepsy: A Case Report with Prospective 9-Month Follow-Up.","authors":"Redab Al-Ghawanmeh, Hamzeh Al-Momani, Rawan Abed Alrahman, Yahya Badaineh, Dima Alarameen","doi":"10.1159/crn/abyag011","DOIUrl":"https://doi.org/10.1159/crn/abyag011","url":null,"abstract":"<p><strong>Introduction: </strong>Transcutaneous auricular vagus nerve stimulation (taVNS) is an emerging noninvasive neuromodulation therapy for drug-resistant epilepsy (DRE). However, evidence regarding its use in patients with cardiac implantable electronic devices, particularly pacemaker-dependent children, remains extremely limited.</p><p><strong>Case presentation: </strong>We report a 6-year-old boy with genetically confirmed TAF6-related Alazami-Yuan syndrome, severe developmental impairment, complete atrioventricular block requiring permanent epicardial VVIR pacing, and syndromic DRE refractory to multiple antiseizure medications (ASMs). Auricular taVNS was initiated at the cymba conchae under multidisciplinary supervision using stimulation parameters of 25 Hz, pulse width 250 μs, intensity titrated to tolerance up to 1.7 mA, for 4 h/day. Treatment initiation was performed in the pediatric intensive care unit with continuous electrocardiographic monitoring for 5 days. The patient underwent prospective follow-up over 9 months using caregiver-maintained seizure diaries, serial electroencephalography (EEG), serial electrocardiography, repeated pacemaker interrogation, adverse event surveillance, and Pediatric Quality of Life Family Impact Module (PedsQL-FIM) assessments. No arrhythmias, pacing abnormalities, device malfunction, or clinically significant pacemaker parameter changes were observed during follow-up. A mild transient local skin irritation resolved with conservative care. Seizure burden decreased from 2 to 4 prolonged seizures daily at baseline to approximately two brief generalized tonic seizures weekly, with sustained improvement through month 9. Reduction of ASM burden began only after clinical improvement had already been observed. Follow-up EEGs remained markedly abnormal with only limited interval electrographic change. PedsQL-FIM improved progressively from 10.4 at baseline to 54.9 at month 3, 80.6 at month 7, and 84.0 at month 9.</p><p><strong>Conclusion: </strong>Auricular taVNS was successfully administered in a pacemaker-dependent child under structured multidisciplinary monitoring without identifiable cardiac or pacemaker-related adverse events during 9 months of follow-up. Although efficacy and safety conclusions cannot be inferred from a single case, this report supports the clinical feasibility of combining noninvasive vagal neuromodulation with permanent cardiac pacing in carefully selected pediatric patients.</p>","PeriodicalId":9639,"journal":{"name":"Case Reports in Neurology","volume":"18 1","pages":"580-588"},"PeriodicalIF":0.4,"publicationDate":"2026-08-31","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"https://www.ncbi.nlm.nih.gov/pmc/articles/PMC13688625/pdf/","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"149263186","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}

{"title":"From Mitochondrial Myopathy to <i>SLC18A3</i>-Related Congenital Presynaptic Myasthenic Syndrome: A Case Report.","authors":"Anke Geertje Boxum, Dennis Klaas Bos, Gea Drost, Jan-Willem Elting, Tom Jozef de Koning, Sonja Leonie van Ockenburg, Lisette Henriëtta Koens","doi":"10.1159/crn/abyag009","DOIUrl":"https://doi.org/10.1159/crn/abyag009","url":null,"abstract":"<p><strong>Introduction: </strong>Recognizing congenital myasthenic syndromes is essential, as appropriate medication can improve muscle weakness.</p><p><strong>Case presentation: </strong>We report on a 25-year-old male who was diagnosed with a mitochondrial myopathy based on muscle biopsy in childhood, although genetic testing of the mitochondrial DNA did not show abnormalities. New analysis, including a gene panel, revealed two novel variants in the SLC18A3 gene, and characteristic electromyography findings changed his diagnosis to a congenital myasthenic syndrome. Treatment was started, which significantly improved his quality of life.</p><p><strong>Conclusion: </strong>Our report underscores the importance of re-evaluating diagnoses in patients without genetic confirmation, so that treatable conditions are not overlooked.</p>","PeriodicalId":9639,"journal":{"name":"Case Reports in Neurology","volume":"18 1","pages":"589-593"},"PeriodicalIF":0.4,"publicationDate":"2026-08-28","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"https://www.ncbi.nlm.nih.gov/pmc/articles/PMC13688596/pdf/","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"149263181","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}

{"title":"Reversible Cerebral Vasoconstriction Syndrome Secondary to Dermatologic Cream: A Case Report.","authors":"Mark Edward Wilhelm, Andrew Joseph Sessions, Gitanjali Das, Benson H Sederholm, Jennifer Juhl Majersik","doi":"10.1159/crn/abyag007","DOIUrl":"https://doi.org/10.1159/crn/abyag007","url":null,"abstract":"&lt;p&gt;&lt;strong&gt;Introduction: &lt;/strong&gt;Imiquimod is an FDA-approved prescription topical medication used in the treatment of skin lesions and neoplasms, such as actinic keratosis, superficial basal cell carcinoma, and external genital and perianal warts. The etiology of rare CNS effects, including headache and stroke, is not completely understood. Headache is a commonly reported adverse reaction, while stroke has been identified through postmarketing pharmacovigilance reports without a definitively established causal relationship.&lt;/p&gt;&lt;p&gt;&lt;strong&gt;Case presentation: &lt;/strong&gt;A 60-year-old female with mild depression and basal cell carcinoma, and an absence of traditional vascular risk factors, presented with a 2-week history of new recurrent thunderclap headaches. CT angiogram showed 40-50% narrowing of the proximal right A1. Conventional angiography demonstrated multiple regions of vasoconstriction which improved with intra-arterial verapamil, supporting the diagnosis of reversible cerebral vasoconstrictive syndrome (RCVS) diagnosis. Oral verapamil was up-titrated to 40 mg every 3 h for vasospasm and headache control. At 4-week follow-up, there was only residual A1 stenosis. Pre-RCVS, the patient had been diagnosed with basal cell carcinoma and was prescribed imiquimod 5% cream, with instructions to apply one 0.25-g prefilled packet topically once daily, which she used for 8.5 weeks prior to symptom onset. The patient had been on a stable low dose (2.5 mg daily) of escitalopram, a selective serotonin reuptake inhibitor and recognized potential trigger of RCVS, for several years without prior neurological symptoms. Discontinuation after the first headache did not prevent subsequent episodes, making it less likely to be the sole precipitant, though a contributory role cannot be excluded. In the absence of other vasoactive substances or identifiable etiologies for vasculitis, we suspect that topical imiquimod was responsible for RCVS in this case.&lt;/p&gt;&lt;p&gt;&lt;strong&gt;Conclusion: &lt;/strong&gt;Imiquimod is an immune modulating agent that acts as a toll-like receptor (TLR) 7 agonist, causing cytokine induction, inflammatory infiltration, and tumor cell apoptosis. Though headache and stroke are reported as rare side effects, stroke subtype and etiology are uncharacterized, with no RCVS reported to our knowledge. A moyamoya animal model, a pathophysiologically distinct entity from RCVS, demonstrated that using imiquimod to induce TLR-7 activation resulted in a phenotypic change in vascular smooth muscle cells. In vivo, imiquimod also led to diminished internal carotid artery blood flow, while inhibiting TLR-7 resulted in decreased stenosis. These findings suggest that imiquimod-mediated TLR-7 agonism can directly alter cerebrovascular smooth muscle behavior - a mechanism that is biologically plausible as a contributor to the acute vasospasm observed in RCVS. Topical imiquimod is thought to trigger systemic reactions secondary to increased production and circulation of cy","PeriodicalId":9639,"journal":{"name":"Case Reports in Neurology","volume":"18 1","pages":"594-599"},"PeriodicalIF":0.4,"publicationDate":"2026-08-26","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"https://www.ncbi.nlm.nih.gov/pmc/articles/PMC13688591/pdf/","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"149263224","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}

{"title":"Bilateral Foot Drop as the Presenting Manifestation of Systemic Lupus Erythematosus-Associated Mononeuritis Multiplex: A Case Report.","authors":"Fadel Alqatati, Mohammad Ghabashneh, Muntasir Alanasweh, Mohamad Wajeh Dulli, Mustafa Abufouda, Hasan Hani Awad, Muhammad Naeem","doi":"10.1159/crn/abyag002","DOIUrl":"https://doi.org/10.1159/crn/abyag002","url":null,"abstract":"<p><strong>Introduction: </strong>Peripheral nervous system involvement in systemic lupus erythematosus (SLE) is uncommon, and mononeuritis multiplex as the presenting neurological manifestation is particularly rare. Early recognition may be difficult when the initial clinical picture resembles focal compressive neuropathy.</p><p><strong>Case presentation: </strong>A 31-year-old previously healthy woman developed acute right foot numbness and foot drop after right knee pain and earlier nonspecific arthralgia. Initial assessments favored peroneal neurapraxia. Over the following 3 weeks, her deficits progressed to persistent right foot drop followed by new left lower limb numbness and weakness, resulting in asymmetric bilateral foot drop. Electrophysiological studies demonstrated an asymmetric, non-length-dependent motor and sensory axonal neuropathy consistent with mononeuritis multiplex. Magnetic resonance imaging of the lumbosacral spine excluded a compressive lumbosacral explanation and focused ultrasound showed no compressive lesion involving the right common peroneal nerve. Serological evaluation revealed high-titer antinuclear antibody, positive anti-double-stranded DNA, anti-Ro, anti-ribosomal P antibodies, elevated inflammatory markers, and a positive lupus anticoagulant screen, with negative infectious screening and no evidence of active lupus nephritis. She was treated with intravenous methylprednisolone, intravenous immunoglobulin, hydroxychloroquine, and rituximab, followed by oral corticosteroid tapering and rehabilitation. Her pain improved early, distal motor function partially recovered, and she later demonstrated meaningful functional improvement with orthotic and rehabilitation support.</p><p><strong>Conclusion: </strong>This case suggests that SLE may initially present predominantly as mononeuritis multiplex, with evolving bilateral foot drop as the dominant neurological and function-threatening manifestation, through a suspected vasculitic mechanism, even when more typical renal, serosal, or major mucocutaneous features are absent at neurological presentation. In patients with progressive or sequential foot drop, especially when the presentation becomes bilateral or multifocal, prompt electrodiagnostic testing and evaluation for systemic autoimmune disease are essential to avoid diagnostic delay and allow timely immunotherapy.</p>","PeriodicalId":9639,"journal":{"name":"Case Reports in Neurology","volume":"18 1","pages":"567-579"},"PeriodicalIF":0.4,"publicationDate":"2026-08-11","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"https://www.ncbi.nlm.nih.gov/pmc/articles/PMC13688621/pdf/","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"149263216","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}