{"title":"Unusual Manifestation of Bilateral Meralgia Paresthetica Following Laparoscopic Appendectomy: A Case Report.","authors":"Sunny Chou, Poyin Huang","doi":"10.4103/ANT.ANT_113_0012","DOIUrl":"https://doi.org/10.4103/ANT.ANT_113_0012","url":null,"abstract":"<p><p>Meralgia paresthetica (MP), a mononeuropathy of the lateral femoral cutaneous nerve (LFCN), presents with dysesthesias in the anterolateral thigh. While often idiopathic, iatrogenic causes are increasingly recognized. We report a rare case of bilateral MP following laparoscopic appendectomy, highlighting its severe impact on quality of life and the need for surgical awareness. A 47-year-old male presented with persistent bilateral thigh numbness, severe pain, and allodynia after laparoscopic appendectomy. He reported significant functional limitations and sleep disturbances. Bilateral MP was diagnosed based on clinical findings and magnetic resonance imaging evidence of inflammation near the LFCN course. This case underscored the atypical presentation of bilateral MP following laparoscopic appendectomy and expands the literature on iatrogenic causes. Clinicians should include MP in the differential diagnosis of postoperative lower limb paresthesia. Surgical awareness of the LFCN vulnerability, coupled with precise trocar placement, could minimize the risk of this potentially debilitating complication.</p>","PeriodicalId":93852,"journal":{"name":"Acta neurologica Taiwanica","volume":"34 3","pages":"170-172"},"PeriodicalIF":0.0,"publicationDate":"2025-07-01","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"145187822","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}
{"title":"Seronegative but Cerebrospinal Fluid-positive NMDA Receptor Encephalitis in an Alzheimer's Dementia Patient with Good Treatment Outcome: A Case Report.","authors":"Yu-Chung Lee, Yi-Cheng Tai","doi":"10.4103/ANT.ANT_113_0041","DOIUrl":"https://doi.org/10.4103/ANT.ANT_113_0041","url":null,"abstract":"<p><p>We present a rare case of elderly-onset anti-N-methyl-D-aspartate (NMDA) receptor encephalitis combined with underlying Alzheimer dementia, characterized by seronegativity but cerebrospinal fluid (CSF) antibody positivity, which showed a significant improvement following intravenous immunoglobulin (IVIG) treatment. The case highlights serial electroencephalogram (EEG) changes. We focus the discussion on factors contributing to favorable recovery. A 78-year-old woman with Alzheimer's dementia and hypertension experienced rapid onset of further cognitive decline over 1 week, presenting with disorientation, irrelevant speech, and disorganized behaviors. Myoclonus of both upper limbs and focal seizures 10 days later which lead her into intensive care unit (ICU) admission. Test results revealed positive anti-NMDA antibodies in CSF but negative in serum. Serial electroencephalograms (EEG) exhibited extreme delta brush activity transitioning to excess beta activity. Initial treatment with a 5-day course of steroid pulse therapy showed limited efficacy. Subsequently, 5 days of IVIG therapy provided marked improvement in consciousness and cognitive function and also normalized the EEG. This patient presented with severe neurological dysfunction, the need for ICU management, and presence of extreme delta brush EEG pattern. Generally, the prognosis was unfavorable. However, advanced age onset and anti-NMDA antibody positivity in CSF with seronegativity in serum may imply better prognosis. Thus, testing anti-NMDA receptor antibody in both serum and CSF is mandatory and under this complicated situation, timely immunotherapy may be the influencing factors of good prognosis.</p>","PeriodicalId":93852,"journal":{"name":"Acta neurologica Taiwanica","volume":"34 3","pages":"161-164"},"PeriodicalIF":0.0,"publicationDate":"2025-07-01","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"145187905","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}
Arthur de Oliveira Veras, Ícaro Araújo de Sousa, Manoella Guerra de Albuquerque Bueno, Ellen Riedi Oliveira, Frederico Fernandes Alessio-Alves, Octávio Marques Pontes-Neto
{"title":"Stroke after Erysipelothrix Rhusiopathiae Endocarditis: A Case Report.","authors":"Arthur de Oliveira Veras, Ícaro Araújo de Sousa, Manoella Guerra de Albuquerque Bueno, Ellen Riedi Oliveira, Frederico Fernandes Alessio-Alves, Octávio Marques Pontes-Neto","doi":"10.4103/ANT.ANT_113_0021","DOIUrl":"10.4103/ANT.ANT_113_0021","url":null,"abstract":"<p><p>Erysipelothrix rhusiopathiae is a rare cause of infective endocarditis (IE). This study aims to report the fifth known case of intracranial manifestation resulting from E. rhusiopathiae endocarditis. Moreover, we aim to address the main epidemiological and clinical aspects associated with this issue. A 62-year-old retired bird breeder presented with left-sided appendicular ataxia and hemiparesis. Diagnostic tests revealed endocarditis caused by E. rhusiopathiae . He received a 6-week antibiotic course, including ampicillin, which resulted in neurological and cardiac improvement. Follow-ups demonstrated no recurrence, and he was discharged from both neurology and cardiology 1 year later. The low virulence of the E. rhusiopathiae pathogen explains the minimal frequency of intracranial manifestations associated with the infection. However, patient's overall condition is a decisive factor in this context. This is the first reported case concerning presumed transmission by birds. Physical manifestations are more commonly found in IE involving the mitral valve compared to the aortic and tricuspid valves. Even with appropriate treatment, mortality from endocarditis associated with E. rhusiopathiae can exceed 38%. Furthermore, IE by this microorganism often causes extensive valve damage. The positive outcome of this case emphasizes the significance of prompt recognition and appropriate management. This report highlights the need for clinical awareness regarding E. rhusiopathiae as an etiological agent for IE and its potential complications, including stroke.</p>","PeriodicalId":93852,"journal":{"name":"Acta neurologica Taiwanica","volume":" ","pages":"165-169"},"PeriodicalIF":0.0,"publicationDate":"2025-07-01","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"144577181","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}
{"title":"Intrafamilial Phenotypic Variation in Taiwanese Patients with Hereditary Spastic Paraplegia and Charcot-Marie-Tooth Disease Due to KIF5A Mutations: A Cross-Sectional Observational Study.","authors":"Po-Yu Lin, Cheng-Tsung Hsiao, Han-Wei Huang, Yi-Jen Wu, Ssu-Ju Fu, Yi-Chung Lee","doi":"10.4103/ant.ANT-D-25-00042","DOIUrl":"https://doi.org/10.4103/ant.ANT-D-25-00042","url":null,"abstract":"<p><strong>Background: </strong>Hereditary spastic paraplegia (HSP) type 10 (SPG10) is an autosomal-dominantly inherited disease caused by pathogenic variants in KIF5A, presenting as either pure or complex HSP.</p><p><strong>Objectives: </strong>This study aims to investigate the clinical and genetic features of KIF5A variants in a Taiwanese cohort diagnosed with HSP.</p><p><strong>Materials and methods: </strong>We analyzed KIF5A coding regions in 219 unrelated Taiwanese patients clinically diagnosed with HSP using a targeted resequencing panel. Clinical, electrophysiological, and neuroimaging features of patients with SPG10 were characterized.</p><p><strong>Results: </strong>Only one (0.5%) patient carried a heterozygous KIF5A variant, c.838C>T (p.Arg280Cys). This patient had a complex HSP phenotype with sensorimotor polyneuropathy, neuropathic pain, appendicular ataxia, and late disease onset at 39 years. Three family members also carried the variant, with one presented with HSP and two with axonal polyneuropathy, diagnosed as axonal Charcot-Marie-Tooth disease (CMT2).</p><p><strong>Conclusions: </strong>SPG10 is a rare HSP subtype in the Taiwanese population. This is the first report of SPG10 in Taiwan, highlighting the coexistence of SPG10 and CMT2 within a single family and the significant intra-familial phenotypic variation.</p>","PeriodicalId":93852,"journal":{"name":"Acta neurologica Taiwanica","volume":" ","pages":""},"PeriodicalIF":0.0,"publicationDate":"2025-06-11","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"144303868","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}
{"title":"Ischemic Stroke Due to Septic Emboli in a 78-Year-Old Man With Portal Vein Thrombosis.","authors":"Cheng-Pang Tsai, Pei-Hao Chen, Chih-Po Chang","doi":"10.4103/ANT.ANT_112_0103","DOIUrl":"10.4103/ANT.ANT_112_0103","url":null,"abstract":"","PeriodicalId":93852,"journal":{"name":"Acta neurologica Taiwanica","volume":" ","pages":"118-119"},"PeriodicalIF":0.0,"publicationDate":"2025-04-01","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"144112210","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}
{"title":"Recent Advancement of Neurofibromatosis Type 1: A Narrative Review.","authors":"Po-Yuan Huang, Ming-Jen Lee","doi":"10.4103/ant.ANT-D-24-00042","DOIUrl":"10.4103/ant.ANT-D-24-00042","url":null,"abstract":"<p><strong>Abstract: </strong>Neurofibromatosis type 1 (NF1) is a complex autosomal dominant genetic disorder caused by mutations in the NF1 gene on chromosome 17, which encodes the tumor-suppressor protein, neurofibromin. Neurofibromin plays a critical role in regulating the RAS signaling pathway, and its loss leads to uncontrolled cell proliferation and tumor development. Clinically, NF1 presents with variable manifestations, including café-au-lait spots, neurofibromas, optic gliomas, skeletal abnormalities, and cognitive deficits. The updated diagnostic criteria now incorporate genetic testing and choroidal anomalies, facilitating earlier and more accurate diagnoses. Recent molecular insights have revealed that neurofibromin interacts with multiple cellular pathways, contributing to diverse clinical presentations such as plexiform neurofibromas, which may transform into malignant peripheral nerve sheath tumors, and developmental issues such as scoliosis and learning disabilities. Current therapeutic approaches focus on targeting specific signaling molecules involved in tumorigenesis. This review aims to summarize the recent findings on the clinical features, molecular mechanisms, and potential treatments of NF1, offering a comprehensive understanding of the disorder's complexities and highlighting future research directions.</p>","PeriodicalId":93852,"journal":{"name":"Acta neurologica Taiwanica","volume":" ","pages":"64-75"},"PeriodicalIF":0.0,"publicationDate":"2025-04-01","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"144129737","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}
{"title":"Acute Cauda Equina Syndrome Following Spinal Anesthesia During Cesarean Section: A Case Report.","authors":"Shin-Rung Tsai, Wei-Hao Lin","doi":"10.4103/ANT.ANT_113_0032","DOIUrl":"https://doi.org/10.4103/ANT.ANT_113_0032","url":null,"abstract":"<p><strong>Abstract: </strong>This case report aims to discuss the prompt recognition and the possible mechanism of acute cauda equina syndrome (CES) following intrathecal bupivacaine administration for cesarean section. A 30-year-old primigravida developed sudden-onset bilateral lower-extremity weakness, numbness, and paresthesia, along with urinary and bowel incontinence, after an uncomplicated cesarean delivery under spinal anesthesia. Magnetic resonance imaging revealed engorgement and gadolinium enhancement of the cauda equina nerve roots, consistent with a diagnosis of CES. The patient was immediately started on high-dose intravenous steroids and intrathecal triamcinolone, followed by intensive inpatient rehabilitation. However, despite maximal medical treatment, the patient exhibited only minimal improvement, with persistent neurological deficits and dysfunction. This case highlights the importance of maintaining a high index of suspicion for CES following intrathecal bupivacaine administration and the need for prompt recognition and multidisciplinary management to optimize maternal outcomes. Understanding the potential mechanisms underlying bupivacaine-induced neurotoxicity, including direct nerve toxicity, vascular changes, and synergistic effects, may guide the development of targeted therapeutic strategies to mitigate this devastating complication. Early spinal imaging, high-dose steroid therapy, and a collaborative approach involving obstetrics, anesthesiology, neurology, and rehabilitation services are crucial for the successful management of acute CES.</p>","PeriodicalId":93852,"journal":{"name":"Acta neurologica Taiwanica","volume":"34 2","pages":"98-101"},"PeriodicalIF":0.0,"publicationDate":"2025-04-01","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"144236209","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}
Marsal Risfandi, Celia Celia, Robert Shen, Andre Marolop Pangihutan Siahaan, Adeodata Lily Wibisono
{"title":"Intracranial Atypical Cartilaginous Tumor (Low-grade Chondrosarcoma) Involving Temporal Bone, Clivus, Sphenoclival Synchondrosis, and Cavernous Sinus: A Rare Case Report.","authors":"Marsal Risfandi, Celia Celia, Robert Shen, Andre Marolop Pangihutan Siahaan, Adeodata Lily Wibisono","doi":"10.4103/ANT.ANT_113_0024","DOIUrl":"10.4103/ANT.ANT_113_0024","url":null,"abstract":"<p><strong>Abstract: </strong>Intracranial atypical cartilaginous tumors (ACTs) or low-grade chondrosarcoma are rare among all skull base and intracranial tumors. Late diagnosis leads to higher morbidity and worsened neurological deficits caused by neurovascular compression. Intracranial ACTs have a higher mortality rate than other ACTs due to their location, difficulty of surgical management, local invasiveness, and recurrence rate. This report aims to highlight the challenges of diagnosing and managing intracranial ACTs, which can present with symptoms mimicking other cranial base tumors. It also aims to raise awareness about the possibility of misdiagnosis, which can lead to futile pharmacological treatment. To the best of the authors' knowledge, no published cases of intracranial ACTs from Indonesia have been reported. A 43-year-old female with complaints of chronic left facial numbness and paralysis, diplopia, tinnitus, and posture and exertion-related headache with worsening symptoms for the last 2 months, especially after waking up in the morning, with a disturbance of the left cranial nerves V-2, V-3, VI, VII, and VIII. The patient had been receiving symptomatic treatment repeatedly with several differential diagnoses for one-prior-year due to an underdiagnosis. Magnetic resonance imaging showed a mass with bone destruction located on the left temporal bone involving sphenoclival synchondrosis, clivus, and cavernous sinus with a ring-and-arch-forming calcification with the lytic pattern. After total gross resection, the diagnosis of ACTs was confirmed by a histopathological specimen that shows an abundant lobulated mass composed of uniform hyaline cartilage matrix, increased cellularity, and myxoid changes. Continuous follow-up is necessary because ACTs have a mild recurrence rate despite a high postsurgical survival rate.</p>","PeriodicalId":93852,"journal":{"name":"Acta neurologica Taiwanica","volume":" ","pages":"93-97"},"PeriodicalIF":0.0,"publicationDate":"2025-04-01","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"144129731","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}