Case Reports in Neurology最新文献

Therapeutic Role of Amantadine in Prolonged Postictal Encephalopathy: A Case Report in an Elderly Patient. 金刚烷胺在延长后脑病中的治疗作用：1例老年患者报告。

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Case Reports in Neurology Pub Date : 2025-08-20 eCollection Date: 2025-01-01 DOI: 10.1159/000548013

Abdullah M Shadid, Rahaf S Alabbad, Sohaila Alshimemeri

{"title":"Therapeutic Role of Amantadine in Prolonged Postictal Encephalopathy: A Case Report in an Elderly Patient.","authors":"Abdullah M Shadid, Rahaf S Alabbad, Sohaila Alshimemeri","doi":"10.1159/000548013","DOIUrl":"https://doi.org/10.1159/000548013","url":null,"abstract":"Introduction: Prolonged postictal encephalopathy in elderly patients can be difficult to manage, especially when standard treatments fail. Amantadine, a dopaminergic agent with NMDA antagonist properties, is approved for use in Parkinson's disease and has demonstrated efficacy in enhancing recovery in TBI. However, its role in nontraumatic etiologies such as postictal states is not well established.Case presentation: We describe a 95-year-old man with multiple comorbidities who remained somnolent and minimally responsive for 14 days after convulsive status epilepticus, despite seizure control and metabolic correction. EEGs showed no epileptiform activity, and imaging revealed no acute pathology. A trial of amantadine 100 mg daily was initiated for suspected postictal encephalopathy. Within 48 h, the patient showed significant improvement in alertness, orientation, and interaction, returning near baseline by day 10.Conclusion: This case suggests a potential role for amantadine in treating prolonged postictal encephalopathy in elderly patients. Further research is needed to assess its efficacy in nontraumatic disorders of consciousness.","PeriodicalId":9639,"journal":{"name":"Case Reports in Neurology","volume":"17 1","pages":"113-118"},"PeriodicalIF":0.6,"publicationDate":"2025-08-20","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"https://www.ncbi.nlm.nih.gov/pmc/articles/PMC12503772/pdf/","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"145250015","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 Lipoprotein (a) Levels Causing Recurrent Deep Vein Thrombosis and Cerebral Venous Sinus Thrombosis in Different Pregnancies: A Case Report. 高脂蛋白(a)水平引起不同妊娠期深静脉血栓和脑静脉窦血栓复发1例报告。

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Case Reports in Neurology Pub Date : 2025-07-14 eCollection Date: 2025-01-01 DOI: 10.1159/000546461

Akihiro Okada, Takeshi Kawauchi, Yukinori Terada, Kenji Hashimoto

{"title":"High Lipoprotein (a) Levels Causing Recurrent Deep Vein Thrombosis and Cerebral Venous Sinus Thrombosis in Different Pregnancies: A Case Report.","authors":"Akihiro Okada, Takeshi Kawauchi, Yukinori Terada, Kenji Hashimoto","doi":"10.1159/000546461","DOIUrl":"10.1159/000546461","url":null,"abstract":"Introduction: High levels of lipoprotein (a) [Lp (a)] have gained attention as a risk factor for venous thrombosis. Venous thrombosis during pregnancy in women without thrombophilic predisposition is attributed to pregnancy itself. Herein, we report a case of high Lp (a) levels manifesting as recurrent deep vein thrombosis (DVT) and cerebral venous sinus thrombosis (CVST) in different pregnancies.Case presentation: A 29-year-old Nepalese woman developed DVT during her first pregnancy. Examination revealed no thrombophilic predisposition. The thrombus resolved with oral anticoagulant medication, which was discontinued after 3 months. During the second pregnancy, prophylactic subcutaneous heparin injections were initiated to prevent venous embolism. Following several days of non-administration of heparin, she experienced left occipital pain, and magnetic resonance venogram showed left CVST. Oral anticoagulants were initiated, and her headache resolved within a few days. Additional blood tests showed abnormally high levels of Lp (a) at 113 mg/dL. Six months later, the CVST was partially recanalized.Conclusion: If venous thrombosis develops during pregnancy in patients without thrombophilic predisposition, Lp (a) levels should be monitored, and long-term anticoagulant medication may be desirable in those with high Lp (a) levels to prevent recurrence of venous thrombosis.","PeriodicalId":9639,"journal":{"name":"Case Reports in Neurology","volume":"17 1","pages":"107-112"},"PeriodicalIF":0.6,"publicationDate":"2025-07-14","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"https://www.ncbi.nlm.nih.gov/pmc/articles/PMC12342702/pdf/","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"144834085","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

Bilateral Meckel's Cave Cephalocele Presenting as Cranial Nerve VI Palsy: A Case Report. 双侧Meckel洞穴型头膨出表现为脑神经麻痹1例。

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Case Reports in Neurology Pub Date : 2025-06-25 eCollection Date: 2025-01-01 DOI: 10.1159/000546760

Cesar Prugue, Delaney C Abood, Paul LaPenna

{"title":"Bilateral Meckel's Cave Cephalocele Presenting as Cranial Nerve VI Palsy: A Case Report.","authors":"Cesar Prugue, Delaney C Abood, Paul LaPenna","doi":"10.1159/000546760","DOIUrl":"10.1159/000546760","url":null,"abstract":"Introduction: Meckel's cave cephalocele, also known as petrous apex cephalocele (PAC), is a rare cystic lesion caused by herniation of the cerebrospinal fluid into the Meckel's cave. Bilateral PACs are exceptionally rare, with only 21 reported cases. PACs may present with headache, diplopia, tinnitus, or cranial nerve palsy.Case presentation: A 74-year-old male presented with acute onset headache, vision changes, and gait instability. His medical history included hypertension and melanoma. Examination revealed a left cranial nerve VI palsy. Initial imaging with head CT and CTA showed no acute intracranial abnormalities but revealed moderate chronic sinusitis. Magnetic resonance imaging demonstrated large bilateral Meckel's cave cephaloceles, more prominent on the left, with mass effect on the inferior margins of the cavernous sinus. Steroid treatment was initiated for suspected influenza-associated cranial neuropathy. Follow-up did not reveal an immediate improvement. However, after several weeks of steroid treatment, his symptoms improved significantly. The patient deferred surgical intervention.Conclusion: This case highlights a rare presentation of bilateral PACs with cranial nerve VI palsy. Although surgical intervention may be definitive, conservative treatment can provide relief of symptoms in select cases. More research is needed to guide optimal management strategies.","PeriodicalId":9639,"journal":{"name":"Case Reports in Neurology","volume":"17 1","pages":"102-106"},"PeriodicalIF":0.6,"publicationDate":"2025-06-25","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"https://www.ncbi.nlm.nih.gov/pmc/articles/PMC12316450/pdf/","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"144774738","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

Neurological Immune-Related Adverse Events in Patients Treated with Anti-Programmed Death 1 Agent, Pembrolizumab: A Case Series. 抗程序性死亡1药物（Pembrolizumab）治疗患者的神经免疫相关不良事件：一个病例系列

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Case Reports in Neurology Pub Date : 2025-06-19 eCollection Date: 2025-01-01 DOI: 10.1159/000547004

Sepehr Khosravi, Tara Khoeini, Fateme Rahattalab, Bahram Haghi Ashtiani

{"title":"Neurological Immune-Related Adverse Events in Patients Treated with Anti-Programmed Death 1 Agent, Pembrolizumab: A Case Series.","authors":"Sepehr Khosravi, Tara Khoeini, Fateme Rahattalab, Bahram Haghi Ashtiani","doi":"10.1159/000547004","DOIUrl":"10.1159/000547004","url":null,"abstract":"Background: This case series highlights the various clinical manifestations of pembrolizumab-induced neurological adverse events, offering insights into the spectrum of immune-related neurological adverse events associated with immune checkpoint inhibitor (ICI) therapy.Case presentation: We present 5 patients who received pembrolizumab as a part of their treatment. They exhibited a variety of clinical manifestations, which included central nervous system involvement and necrotizing myopathy, each responding differently to therapeutic interventions. Among the participants, there were two cases of myopathy, one case of demyelinating polyneuropathy, one individual whose myasthenia gravis had worsened, and 1 patient diagnosed with encephalitis. The patients experienced symptoms with varying degrees of severity, leading to different responses in their clinical treatment, and 1 patient ultimately passed away due to complications.Conclusion: Neurological immune-related adverse effects (n-irAEs) are rare, and their identification poses a challenge due to their complexity and the presence of underlying autoimmune and paraneoplastic disorders. The expected prognosis for n-irAEs is unclear, presenting a range of complete recovery rates and differing mortality outcomes. The increased frequency of ICI usage increases the probability of an immune response, necessitating healthcare professionals to identify new symptoms in immunotherapy patients. Side effects differ from conventional chemotherapy and necessitate different therapeutic approaches.","PeriodicalId":9639,"journal":{"name":"Case Reports in Neurology","volume":"17 1","pages":"94-101"},"PeriodicalIF":0.6,"publicationDate":"2025-06-19","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"https://www.ncbi.nlm.nih.gov/pmc/articles/PMC12274057/pdf/","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"144673999","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

Challenges in the Diagnosis and Care of Tuberous Sclerosis Complex in Resource-Limited Settings: A Case Report from Tanzania. 在资源有限的情况下结节性硬化症的诊断和护理的挑战：来自坦桑尼亚的病例报告。

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Case Reports in Neurology Pub Date : 2025-06-10 eCollection Date: 2025-01-01 DOI: 10.1159/000546098

Basil Tumaini, John Calori, Ernest Ibenzi

{"title":"Challenges in the Diagnosis and Care of Tuberous Sclerosis Complex in Resource-Limited Settings: A Case Report from Tanzania.","authors":"Basil Tumaini, John Calori, Ernest Ibenzi","doi":"10.1159/000546098","DOIUrl":"10.1159/000546098","url":null,"abstract":"Introduction: Tuberous sclerosis complex (TSC) is a rare, multisystem genetic disorder caused by mutations in the TSC1 or TSC2 genes, resulting in dysregulation of the mTOR pathway. Clinical manifestations include epilepsy, cutaneous lesions, and hamartomas in various organs, presenting diagnostic challenges, especially in resource-limited settings. In low- and middle-income countries (LMICs), barriers such as lack of access to genetic testing, advanced imaging, and targeted therapies contribute to underdiagnosis and delayed care.Case presentation: We present a 22-year-old male from rural Tanzania with a 20-year history of intractable, treatment-refractory epilepsy. He also exhibited cutaneous findings, including facial angiofibromas, ash-leaf macules, and a shagreen patch. Brain imaging revealed subependymal nodules, and renal imaging identified angiomyolipomas. Despite the absence of genetic testing, a clinical diagnosis of TSC was made. The patient's condition was further complicated by aspiration pneumonia. Management included anticonvulsant therapy, antibiotics, and supportive care. Multisystem evaluation revealed cystic lung disease and renal involvement, underscoring the need for comprehensive follow-up. This case highlights the diagnostic reliance on clinical expertise and basic imaging in resource-limited settings.Conclusion: This report emphasizes the importance of clinical recognition and multidisciplinary management of TSC in resource-constrained settings. Strengthening healthcare infrastructure, raising awareness, and fostering collaborations to enhance access to genetic testing and mTOR-targeted therapies are critical to improving outcomes for TSC patients in LMICs. The lessons from this case underscore the global relevance of addressing healthcare disparities in rare genetic disorders.","PeriodicalId":9639,"journal":{"name":"Case Reports in Neurology","volume":"17 1","pages":"79-87"},"PeriodicalIF":0.6,"publicationDate":"2025-06-10","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"https://www.ncbi.nlm.nih.gov/pmc/articles/PMC12252377/pdf/","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"144625447","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

Subclavian Artery-Internal Carotid Artery Bypass Using a Radial Artery Graft for Common Carotid Artery Occlusion with Immediate Improvement of Cognitive Function: A Case Report. 锁骨下动脉-颈内动脉搭桥桡动脉移植治疗颈总动脉闭塞，认知功能即刻改善1例报告。

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Case Reports in Neurology Pub Date : 2025-06-05 eCollection Date: 2025-01-01 DOI: 10.1159/000546580

Kazuya Morita, Tomoya Kamide, Riho Nakajima, Seiya Kudou, Taishi Tsutsui, Kouichi Misaki, Mitsutoshi Nakada

{"title":"Subclavian Artery-Internal Carotid Artery Bypass Using a Radial Artery Graft for Common Carotid Artery Occlusion with Immediate Improvement of Cognitive Function: A Case Report.","authors":"Kazuya Morita, Tomoya Kamide, Riho Nakajima, Seiya Kudou, Taishi Tsutsui, Kouichi Misaki, Mitsutoshi Nakada","doi":"10.1159/000546580","DOIUrl":"10.1159/000546580","url":null,"abstract":"Introduction: Although rare, common carotid artery occlusion (CCAO) causes ischemic neurological dysfunction, which can be treated by revascularization. Although several bypass approaches for CCAO have been suggested, no consensus on the surgical revascularization approach and its functional outcome have been reached. Herein, we present a case of Rile's type 1A CCAO in which a subclavian artery (SclA)-internal carotid artery (ICA) bypass using a radial artery graft (RAG) resulted in immediate recovery of cognitive function and successfully prevented ischemic stroke.Case description: A 58-year-old man presented with recurrent episodes of right-sided weakness. Brain magnetic resonance imaging revealed multiple cerebral infarcts. Digital subtraction angiography confirmed left CCAO and no anterograde blood flow in the left ICA and ipsilateral external carotid artery. A bypass was performed from the left SclA to the left cervical ICA using a left RAG, and supraclavicular anastomosis between the SclA and ICA was performed without graft-vessel kinking. Postoperatively, no neurological deficits were observed, and his cognitive function was successfully improved.Conclusion: SclA-ICA bypass using an RAG can be a reasonable treatment option for CCAO. CCAO revascularization can prevent ischemic stroke and improve cognitive function.","PeriodicalId":9639,"journal":{"name":"Case Reports in Neurology","volume":"17 1","pages":"88-93"},"PeriodicalIF":0.6,"publicationDate":"2025-06-05","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"https://www.ncbi.nlm.nih.gov/pmc/articles/PMC12263144/pdf/","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"144641902","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

Lyme Neuroborreliosis Presenting with Isolated Intracranial Hypertension: A Case Report. 以孤立性颅内高压为表现的莱姆病神经螺旋体病1例。

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Case Reports in Neurology Pub Date : 2025-05-26 eCollection Date: 2025-01-01 DOI: 10.1159/000546097

Dax Bourcier, Joyce Beshara, Griffin Pauli, Tyler Henry, Michael Peckford, Richard Huntsman, Bashar M Bata, Tobias R Kollmann

{"title":"Lyme Neuroborreliosis Presenting with Isolated Intracranial Hypertension: A Case Report.","authors":"Dax Bourcier, Joyce Beshara, Griffin Pauli, Tyler Henry, Michael Peckford, Richard Huntsman, Bashar M Bata, Tobias R Kollmann","doi":"10.1159/000546097","DOIUrl":"10.1159/000546097","url":null,"abstract":"Background: Lyme borreliosis, a tick-borne illness caused by Borrelia burgdorferi, is increasingly prevalent in Nova Scotia, Canada, which has the highest incidence in North America. While most cases present as early localized disease, approximately 20% develop early disseminated disease, which can include neurological symptoms, an entity called Lyme neuroborreliosis (LNB). This case report describes an unusual LNB presentation with isolated intracranial hypertension (IH).Case presentation: A 6-year-old female presented to our pediatric hospital with binocular horizontal diplopia, headache, fever, malaise, and suspected papilledema 47 days after an embedded tick bite. A diagnosis of Lyme disease had been made 10 days prior in the community based on positive serologies and erythema migrans, but she developed a Jarisch-Herxheimer reaction within 24 h of oral doxycycline, leading to an antibiotic change to amoxicillin. During the hospital admission, an ophthalmological examination revealed papilledema and IH was evidenced by an opening pressure of 36 mm Hg and brain MRI findings. The lumbar puncture revealed pleocytosis and positive cerebrospinal fluid antibodies for Borrelia. The patient was initially treated with 2 days of ceftriaxone, followed by a 12-day outpatient course of doxycycline for LNB. High doses of acetazolamide (500 mg TID) were needed to achieve symptom control. Two months after her hospital discharge, there was resolution of papilledema and the acetazolamide was weaned.Conclusion: This case highlights the importance of considering Lyme disease in the differential diagnosis of IH, particularly in endemic regions. Early recognition, diagnostic workup, and appropriate treatment are crucial for optimal outcomes in LNB.","PeriodicalId":9639,"journal":{"name":"Case Reports in Neurology","volume":"17 1","pages":"72-78"},"PeriodicalIF":0.6,"publicationDate":"2025-05-26","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"https://www.ncbi.nlm.nih.gov/pmc/articles/PMC12187110/pdf/","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"144483264","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

Delayed Intracerebral Hemorrhage after Deep Brain Stimulation for Parkinson's Disease. 帕金森病深部脑刺激后迟发性脑出血。

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Case Reports in Neurology Pub Date : 2025-05-06 eCollection Date: 2025-01-01 DOI: 10.1159/000546056

Hana Chudy, Marina Raguž, Petar Marčinković, Valentino Rački, Eliša Papić, Mario Hero, Vladimira Vuletić, Darko Chudy

{"title":"Delayed Intracerebral Hemorrhage after Deep Brain Stimulation for Parkinson's Disease.","authors":"Hana Chudy, Marina Raguž, Petar Marčinković, Valentino Rački, Eliša Papić, Mario Hero, Vladimira Vuletić, Darko Chudy","doi":"10.1159/000546056","DOIUrl":"10.1159/000546056","url":null,"abstract":"Introduction: Deep brain stimulation (DBS) of the subthalamic nucleus (STN) is a well-established treatment for advanced Parkinson's disease (PD), offering significant symptomatic relief. Although DBS is generally considered safe, it carries risks, including the potential for delayed complications such as intracerebral hemorrhage (ICH).Case presentation: We present a rare case of a 67-year-old male with PD who developed delayed ICH after undergoing bilateral STN DBS. Initially, the patient showed no neurological deficits postoperatively, with imaging confirming correct lead placement and no signs of hemorrhage. However, on the second postoperative day, the patient developed sudden right-sided hemiparesis. A CT scan revealed ICH alongside the left lead. The hemorrhage was managed conservatively, and the patient underwent extensive physical therapy, leading to significant improvement. Over the next 2 weeks, the patient's condition improved, and follow-up CT scans showed complete resolution of the ICH. At this point, the left lead stimulation was initiated, further improving the patient's PD symptoms. This case illustrates the potential for delayed ICH following STN DBS, emphasizing the need for ongoing monitoring and individualized treatment strategies.Conclusion: This case underscores the importance of vigilant postoperative monitoring and individualized management strategies in STN DBS patients. Early detection and appropriate management of complications such as ICH are crucial for minimizing risks and ensuring optimal patient outcomes. The potential for delayed complications highlights the need for continuous follow-up, even in the absence of immediate postoperative issues.","PeriodicalId":9639,"journal":{"name":"Case Reports in Neurology","volume":"17 1","pages":"62-71"},"PeriodicalIF":0.6,"publicationDate":"2025-05-06","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"https://www.ncbi.nlm.nih.gov/pmc/articles/PMC12162119/pdf/","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"144282460","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

Trigeminal Neuralgia as a Rare Complication of Idiopathic Intracranial Hypertension. 三叉神经痛是特发性颅内高压的罕见并发症。

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Case Reports in Neurology Pub Date : 2025-04-07 eCollection Date: 2025-01-01 DOI: 10.1159/000544077

Mariana Sarov-Riviere, Claire Ancelet, Ghaidaa Nasser, Jildaz Caroff, Nozar Aghakhani, Christian Denier

{"title":"Trigeminal Neuralgia as a Rare Complication of Idiopathic Intracranial Hypertension.","authors":"Mariana Sarov-Riviere, Claire Ancelet, Ghaidaa Nasser, Jildaz Caroff, Nozar Aghakhani, Christian Denier","doi":"10.1159/000544077","DOIUrl":"https://doi.org/10.1159/000544077","url":null,"abstract":"Introduction: Idiopathic intracranial hypertension (IIH) is a rare cause of chronic headaches. Usually, patients with IIH present headaches and papilledema with no focal neurological signs. Classical MRI findings feature characteristic signs, i.e., dilated perioptic nerves sheath and empty sella turcica. Rarely, cranial nerve palsies existed, especially abducens nerve palsy.Case presentation: We reported herein another rare clinical feature of IIH: trigeminal neuralgia in association with meningocele. This 35-year-old obese woman initially presented with chronic headaches and papilledema. Cerebral MRI showed classical IIH findings. The CSF opening pressure was increased. A treatment coupling acetazolamide and iterative lumbar punctures led to the regression of papilledema, but headaches were difficult to control. Ten years later, while she was lost to follow up and treated with acetazolamide by her general practitioner, she developed extremely painful neuropathic pain in the left trigeminal nerve territory, in association with a recurrence of the chronic headaches. A new MRI showed new bilateral cavum trigeminal meningoceles, predominantly on the left side, associated with an atrophy of the cisternal segment of the left trigeminal nerve. Angio-CT showed transverse sinus stenosis, treated by stenting. After this treatment, IIH symptoms disappeared, while trigeminal neuralgia amplified: a surgical procedure led to its complete disappearance.Conclusion: To our knowledge, this is the first case reported of trigeminal neuralgia associated with meningocele formation in IIH. Our case illustrates the great efficacy of venous stenting in IIH, and one may wonder whether earlier stenting could have avoided the subsequent development of meningoceles and subsequent neuralgia.","PeriodicalId":9639,"journal":{"name":"Case Reports in Neurology","volume":"17 1","pages":"57-61"},"PeriodicalIF":0.6,"publicationDate":"2025-04-07","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"https://www.ncbi.nlm.nih.gov/pmc/articles/PMC12068838/pdf/","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"143977715","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

Seronegative Autoimmune Encephalomyelitis with Area Postrema Symptoms. 血清阴性自身免疫性脑脊髓炎伴区域后发症状。

IF 0.6

Case Reports in Neurology Pub Date : 2025-03-26 eCollection Date: 2025-01-01 DOI: 10.1159/000545402

David Andrew Prentice, Ravi Ambati, Lay K Kho, Thomas Jenkins, Paul M Parizel

{"title":"Seronegative Autoimmune Encephalomyelitis with Area Postrema Symptoms.","authors":"David Andrew Prentice, Ravi Ambati, Lay K Kho, Thomas Jenkins, Paul M Parizel","doi":"10.1159/000545402","DOIUrl":"https://doi.org/10.1159/000545402","url":null,"abstract":"Introduction: Patients presenting with encephalopathy and longitudinally extensive myelitis pose a significant diagnostic challenge. Area postrema-related symptoms, such as intractable hiccoughs, can aid in narrowing the differential diagnosis. Neuromyelitis optica spectrum disorders and glial fibrillary acidic protein (GFAP) autoimmune encephalitis are known causes; however, some cases remain seronegative, suggesting the presence of unidentified autoantibodies or immune targets.Case presentation: A previously healthy man in his 70s presented with headache, fever, and confusion, followed by a seizure and persistent hiccoughs. MRI revealed brainstem involvement and extensive transverse myelitis. Cerebrospinal fluid (CSF) analysis showed inflammatory features, but testing for AQP4, MOG, and GFAP antibodies was initially negative. He was treated with intravenous corticosteroids and plasma exchange, after which serum GFAP-IgG was weakly positive, though CSF remained negative. His condition improved with immunotherapy, but significant lower limb weakness persisted. Based on clinical and radiological findings, we hypothesize that tanycytes - specialized glial cells in the area postrema - may be an additional immune target in GFAP encephalitis.Conclusion: This case highlights a seronegative encephalomyelitis syndrome with area postrema involvement, possibly implicating glial cells beyond astrocytes. Further studies are needed to explore the role of tanycytes in autoimmune neuroinflammation.","PeriodicalId":9639,"journal":{"name":"Case Reports in Neurology","volume":"17 1","pages":"50-56"},"PeriodicalIF":0.6,"publicationDate":"2025-03-26","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"https://www.ncbi.nlm.nih.gov/pmc/articles/PMC12058113/pdf/","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"144062466","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