Tremor and Other Hyperkinetic Movements最新文献

{"title":"Continued Dialogue about the Clinical Heterogeneity and Degenerative Pathology That Characterizes the Essential Tremors.","authors":"Elan D Louis","doi":"10.5334/tohm.1199","DOIUrl":"10.5334/tohm.1199","url":null,"abstract":"","PeriodicalId":23317,"journal":{"name":"Tremor and Other Hyperkinetic Movements","volume":"16 ","pages":"20"},"PeriodicalIF":1.9,"publicationDate":"2026-03-30","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"https://www.ncbi.nlm.nih.gov/pmc/articles/PMC13045786/pdf/","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"147623749","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":"Paroxysmal Hemidystonia as the Presenting Manifestation of Previously Undiagnosed Diabetes Mellitus.","authors":"Subhankar Chatterjee, Samya Sengupta, Ritwik Ghosh, Julián Benito-León, Souvik Dubey","doi":"10.5334/tohm.1185","DOIUrl":"10.5334/tohm.1185","url":null,"abstract":"<p><strong>Background: </strong>Hyperglycemia-related movement disorders classically present as hemichorea-hemiballismus, whereas dystonia is far less common and may occasionally precede the recognition of diabetes mellitus.</p><p><strong>Case report: </strong>A 72-year-old man presented with a 3-month history of recurrent, stereotyped dystonic spells involving the right side of the face, arm, and leg. These episodes occurred approximately twice per minute and lasted about 5 seconds each. Initial laboratory testing revealed a plasma glucose level of 494 mg/dL and an HbA1c of 16.1%, consistent with previously unrecognized diabetes mellitus. Electrolyte levels, electroencephalography, cerebrospinal fluid analysis, and evaluation for autoimmune encephalopathy were unremarkable. Brain MRI showed only a chronic lacunar infarct in the left basal ganglia, with no evidence of an acute striatal lesion. The abnormal movements resolved completely within 3 days after correction of hyperglycemia. Follow-up 18F-FDG PET/CT performed after clinical recovery demonstrated subtle relative hypometabolism in the left cerebellum, with preserved uptake in the basal ganglia and cerebral cortex.</p><p><strong>Discussion: </strong>This case broadens the phenomenologic spectrum of hyperglycemia-related movement disorders and highlights paroxysmal hemidystonia as a potential presenting manifestation of previously undiagnosed diabetes mellitus. Bedside glucose testing should therefore be routinely performed in the evaluation of new-onset paroxysmal hyperkinetic movements. In addition, isolated extra-striatal metabolic abnormalities detected after symptom resolution should be interpreted with caution.</p>","PeriodicalId":23317,"journal":{"name":"Tremor and Other Hyperkinetic Movements","volume":"16 ","pages":"19"},"PeriodicalIF":1.9,"publicationDate":"2026-03-24","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"https://www.ncbi.nlm.nih.gov/pmc/articles/PMC13025155/pdf/","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"147575479","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":"Spectrum of Movement Disorders in Hematological Malignancies: A Comprehensive Systematic Review of Clinical Phenotypes, Mechanisms, and Outcomes.","authors":"Ravindra Kumar Garg, Amita Jain, Ritu Karoli, Shweta Pandey, Vimal Paliwal, Vinay Suresh, Sanjay Singhal","doi":"10.5334/tohm.1147","DOIUrl":"10.5334/tohm.1147","url":null,"abstract":"<p><strong>Background: </strong>Movement disorders associated with hematologic malignancies remain incompletely defined. This review synthesized published articles to clarify clinical patterns, mechanisms, and outcomes across hematologic malignancies.</p><p><strong>Methods: </strong>A PRISMA-compliant systematic search of PubMed, Embase, Scopus, and Google Scholar was conducted to identify reports describing movement disorders associated with hematologic malignancies. Eligible reports were screened according to predefined criteria, and data were extracted on demographics, hematologic diagnosis, central nervous system (CNS) involvement, movement-disorder phenotype, investigative findings, treatments administered, and neurological outcomes.</p><p><strong>Results: </strong>A total of 252 cases were included: 152 lymphoid, 78 myeloid, and 22 plasma-cell neoplasms. Lymphoid malignancies most commonly presented with cerebellar ataxia (55.3 percent), followed by parkinsonism (18.4 percent) and chorea (11.8 percent), with CNS involvement frequent, including infiltration (33.6 percent) and immune-mediated injury (30.3 percent). Myeloid disorders showed a predominantly hyperkinetic profile, especially generalized chorea (41 percent) and hemichorea or hemiballismus (17.9 percent), while structural CNS disease was rare (2.6 percent) and most cases reflected systemic or treatment-related mechanisms. Plasma-cell neoplasms were characterized mainly by parkinsonism (45.5 percent) and cerebellar syndromes (27.3 percent). Neurological improvement occurred in most patients, particularly after treatment of underlying hematologic disorder. In a cohort of 232 CNS lymphoma patients, prodromal neuropsychiatric features occurred in 9 percent, parkinsonism in 25 percent of those, and 80 percent improved following lymphoma-directed therapy.</p><p><strong>Conclusions: </strong>Movement disorders in hematologic cancers show distinct, disease-specific profiles, with cerebellar, hyperkinetic, and parkinsonian patterns corresponding to lymphoid, myeloid, and plasma-cell neoplasms. CNS involvement mechanisms differ, providing essential diagnostic and therapeutic guidance.</p>","PeriodicalId":23317,"journal":{"name":"Tremor and Other Hyperkinetic Movements","volume":"16 ","pages":"18"},"PeriodicalIF":1.9,"publicationDate":"2026-03-19","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"https://www.ncbi.nlm.nih.gov/pmc/articles/PMC13004057/pdf/","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"147499811","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":"Multidimensional Phenotyping of Orthostatic Tremor and Orthostatic Myoclonus: Baseline Findings from a Longitudinal Clinical Study.","authors":"Giridhar S Immanni, Anish Mehta, Prabhudev M Hiremath, Thyagarajan Shivashanmugam, Chandrasekhar Enuguri, Pradeep Rangaiah, Mahendra Javali, Purushottam Acharya","doi":"10.5334/tohm.1121","DOIUrl":"10.5334/tohm.1121","url":null,"abstract":"<p><strong>Background: </strong>Orthostatic tremor (OT) and orthostatic myoclonus (OM) are rare weight-bearing hyperkinetic disorders defined electrophysiologically but often overlap clinically. Prior studies were limited to small series with little assessment of comorbidities, functional outcomes, or treatments. The Longitudinal Orthostatic Tremor Study (LOTS) was initiated to address these gaps through multidimensional phenotyping.</p><p><strong>Methods: </strong>Baseline data from 58 consecutively identified patients with OT or OM at a tertiary neurology center in India were analyzed. Clinical evaluation, surface electromyography (sEMG), neuroimaging, and comorbidity screening were performed. Severity was assessed with the OT-10 scale, functional status with the Orthostatic Tremor Impact Profile (OTIP), and quality of life with the 36-item Short Form Health Survey (SF-36). Variables were organized into five analytical domains-demographics, phenomenology, comorbidities, functional outcomes, and treatments-and compared across OT vs OM, primary vs secondary OT, and high- vs low-frequency OT.</p><p><strong>Results: </strong>Fifty-two patients had OT (89.7%) and six had OM (10.3%). Demographics were broadly similar, though age was associated with high-frequency OT (P = .012). Phenomenological features overlapped, with twitching linked to secondary OT (P = .022). Dementia, polyneuropathy, and diabetes were more common in OM and secondary OT (P < .05). Functional outcomes were comparable, except for higher pain scores in secondary OT (P = .026). Clonazepam was most prescribed; other agents showed inconsistent associations.</p><p><strong>Discussion: </strong>Baseline LOTS findings show broad overlap across diagnostic domains, indicating that sEMG-based labels alone do not capture heterogeneity. Multidimensional approaches, including Disease burden index and latent class analysis, may refine classification and guide individualized management.</p><p><strong>Highlights: </strong>This study presents the first structured multidimensional phenotyping of orthostatic tremor and orthostatic myoclonus, integrating clinical, electrophysiological, comorbidity, functional, and treatment domains. Findings demonstrate diagnostic overlap and support multidimensional approaches for refining classification and guiding individualized management across orthostatic hyperkinetic syndromes.</p>","PeriodicalId":23317,"journal":{"name":"Tremor and Other Hyperkinetic Movements","volume":"16 ","pages":"17"},"PeriodicalIF":1.9,"publicationDate":"2026-03-19","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"https://www.ncbi.nlm.nih.gov/pmc/articles/PMC13004056/pdf/","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"147499837","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":"Resilience Mitigates the Link between Adverse Childhood Experiences and Musician's Dystonia: A Neuroendocrine and Psychological Perspective.","authors":"Julian Burek, Stine Alpheis, Christopher Sinke, Tillmann H C Krüger, Michael Großbach, Daniel S Scholz, Florian Worschech, André Lee, Eckart Altenmüller","doi":"10.5334/tohm.1161","DOIUrl":"10.5334/tohm.1161","url":null,"abstract":"<p><strong>Background: </strong>Musician's dystonia (MD) is a task-specific movement disorder affecting up to 1% of all professional musicians. Although adverse childhood experiences (ACEs) have been proposed as risk factors, the etiology of MD is not fully understood, and protective factors remain largely unexplored.</p><p><strong>Objective: </strong>This study investigated a possible protective role of psychological resilience on the association between MD and adverse childhood experiences and examined the influence of dystonia, resilience and ACEs on the stress reactivity of musicians.</p><p><strong>Methods: </strong>Forty participants with MD were compared to 39 matched healthy musicians. While undergoing the \"Montreal Imaging Stress Task\", cortisol responses of the participants were measured. Furthermore, participants completed two psychological assessments, the Connor-Davidson Resilience Scale and the Childhood Trauma Questionnaire. Mediation and moderation analyses evaluated the mitigating role of resilience on the relationship between ACEs and MD. Bayesian multilevel models were used to analyze links between cortisol responses, ACEs and MD.</p><p><strong>Results: </strong>Healthy musicians showed higher resilience than MD patients, especially when looking at the dimensions \"adaptability/flexibility\" (<i>W</i> = 517, <i>p</i> = 0.004) and \"regulation of emotion and cognition\" (<i>W</i> = 554, <i>p</i> = 0.011). Resilience moderated the association between ACEs and dystonia (-0.29 [-0.47, -0.10]). MD patients and healthy participants did not differ in their cortisol output.</p><p><strong>Discussion: </strong>While links between acute stress reactivity, ACEs and MD were more equivocal, resilience seems to decrease the negative effects of ACEs on MD development.The results of this study emphasize the need to implement resilience enhancing interventions at an early state of musical education.</p><p><strong>Highlights: </strong>Musicians suffering from musician's dystonia (MD) and healthy musicians were compared concerning resilience, adverse childhood experiences (ACEs) and their stress reactivity.Healthy musicians showed tendencies of higher resilience than MD patients.Resilience appears to decrease the negative effects of ACEs on the development of musician's dystonia.MD patients and healthy musicians did not display differences in their cortisol output when presented with an acute stress task.</p>","PeriodicalId":23317,"journal":{"name":"Tremor and Other Hyperkinetic Movements","volume":"16 ","pages":"16"},"PeriodicalIF":1.9,"publicationDate":"2026-03-13","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"https://www.ncbi.nlm.nih.gov/pmc/articles/PMC12985811/pdf/","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"147463718","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":"Clinical Characteristics of Primary Orthostatic Tremor - a Comprehensive Clinical Assessment of Patients in Sweden.","authors":"Karolina Af Edholm, Mathias Sundgren, Erik Fransén, Henrik Sjöström, Anders Svenningsson","doi":"10.5334/tohm.1143","DOIUrl":"10.5334/tohm.1143","url":null,"abstract":"<p><strong>Background: </strong>Primary orthostatic tremor (POT) is a rare neurological disease presenting as a bilaterally coherent tremor of 13-18 Hz and a subjective sensation of unsteadiness while standing. Patients are severely affected by the inability to stand and often eventually referred to walking aids and dependence on others.</p><p><strong>Objectives: </strong>This study aimed to investigate the clinical characteristics of POT in a Swedish patient population by interviews and questionnaires.</p><p><strong>Methods: </strong>Patients with POT were recruited nationwide in Sweden. All participants underwent neurological examination, structured interview and evaluation according to nine standardized rating scales and questionnaires, including the novel orthostatic tremor scales OTIP and OT-10.</p><p><strong>Results: </strong>Fifty-two participants with EMG-verified POT were included in the final analysis. Disease duration was not significantly correlated to disease severity, while OTIP and OT-10 were highly correlated with severity of POT. Postural or action tremor in the arms were present in 58%. Mild signs of parkinsonism were common, and the combination of mild rigidity and bradykinesia was present in 25%. Symptoms of depression and anxiety were present in 25%. Although quality of life was often severely affected, 65% performed activities of daily life independently.</p><p><strong>Discussion: </strong>Patients with POT may be severely affected by their disease, independently of disease duration. Multiple associated symptoms like tremor in the upper extremities and mild Parkinsonian features need to be recognized by healthcare professionals.</p><p><strong>Highlights: </strong>52 patients with POT were clinically examined with rating scales and questionnaires. Additional symptoms like other tremors, parkinsonian signs, and depressive symptoms were common. Disease severity did not correlate to additional symptoms or disease duration. Questionnaires like OTIP and OT-10 can be recommended for assessment of disease severity.</p>","PeriodicalId":23317,"journal":{"name":"Tremor and Other Hyperkinetic Movements","volume":"16 ","pages":"14"},"PeriodicalIF":1.9,"publicationDate":"2026-03-05","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"https://www.ncbi.nlm.nih.gov/pmc/articles/PMC12962244/pdf/","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"147378706","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":"Clinical, Radiological, and Genetic Profile of Patients with <i>FA2H</i>-Associated Neurodegeneration: Eight Cases from India and a Review of the Literature.","authors":"Vikram V Holla, Riyanka Kumari, Neeharika Sriram, Nitish Kamble, Jitender Saini, Ravi Yadav, Babylakshmi Muthusamy, Pramod Kumar Pal","doi":"10.5334/tohm.1162","DOIUrl":"10.5334/tohm.1162","url":null,"abstract":"<p><strong>Background: </strong>Autosomal recessive spastic paraplegia 35 (SPG35), also known as Fatty acid hydroxylase-associated neurodegeneration (FAHN), is a rare recessive neurodegenerative disorder with or without ataxia, dystonia, and other neurological findings. It is caused by genetic variants in <i>FA2H</i>, which encodes fatty acid 2-hydroxylase.</p><p><strong>Objective: </strong>To report the clinical, electrophysiological, radiological, and genetic profile of patients diagnosed with FAHN.</p><p><strong>Methods: </strong>We performed a retrospective chart review of genetically proven cases of FAHN from our database.</p><p><strong>Results: </strong>We identified eight patients (6 females) with genetically proven FAHN. All patients presented with first-decade onset pyramidal syndrome with or without ataxia and with radiological findings of callosal atrophy, peri-ventricular white matter hyperintensity, and cerebellar atrophy. Iron accumulation was observed in four of them. Whole exome sequencing revealed seven unique variants including three missense variants (c.83G>C;p.Arg28Pro, c.130C>A;p.Pro44Thr, and c.703C>T;p.Arg235Cys), a stop-gain variant (c.379C>T;p.Arg127Ter), a frameshift deletion variant (c.536delT;p.Leu179Argfs*62), a in-frame deletion variant (c.200_202del;p.His67del) and a in-frame duplication variant (c.86_97dup;p.Arg29_Arg32dup). The variants p.Pro44Thr, p.Arg28Pro, p.Arg29_Arg32dup, and p.His67del are located in the iron-binding region, and the p.Arg235Cys in the hydroxylase domain. The other two variants, p.Arg127Ter and p.Leu179Argfs*62, predictively cause protein truncation, leading to loss of the transmembrane domain and the fatty acid hydroxylase domain, which in turn may result in disruption of fatty acid alpha-hydroxylase activity of FA2H.</p><p><strong>Conclusion: </strong>Our study identifies novel variants associated with FA2H in FAHN patients, highlighting their possible roles in iron binding and in the loss of the transmembrane and catalytic domains.</p>","PeriodicalId":23317,"journal":{"name":"Tremor and Other Hyperkinetic Movements","volume":"16 ","pages":"15"},"PeriodicalIF":1.9,"publicationDate":"2026-03-05","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"https://www.ncbi.nlm.nih.gov/pmc/articles/PMC12962245/pdf/","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"147378703","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":"Generalized Dystonia in a Patient With Wilson Disease 5 Years After Liver Transplant: A Case Report.","authors":"Elise Edwards, Benjamin Coleman, Matthew Feldman, Jude Hassan Charles, Danielle S Shpiner","doi":"10.5334/tohm.1120","DOIUrl":"10.5334/tohm.1120","url":null,"abstract":"<p><strong>Background: </strong>Liver transplant (LT) is considered curative for Wilson disease (WD) with hepatic failure refractory to medical therapy, particularly when neurologic symptoms are absent.</p><p><strong>Case report: </strong>A 29-year-old man with WD developed progressive generalized dystonia five years after LT. He presented with acute-on-chronic neck pain, dysphagia, and dystonic posturing of the neck, trunk, and upper and lower extremities. MRI brain and copper studies were normal. Genetic testing confirmed two heterozygous pathogenic <i>ATP7B</i> variants. Symptoms improved with botulinum toxin injections.</p><p><strong>Discussion: </strong>Post-LT neurologic complications may arise from copper dysregulation, immunosuppressant neurotoxicity, or unrelated primary dystonia. Early recognition enables effective symptomatic management.</p>","PeriodicalId":23317,"journal":{"name":"Tremor and Other Hyperkinetic Movements","volume":"16 ","pages":"13"},"PeriodicalIF":1.9,"publicationDate":"2026-02-13","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"https://www.ncbi.nlm.nih.gov/pmc/articles/PMC12904119/pdf/","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"146202941","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":"A Case of Ganglionopathy Presenting with Hyperkinetic Movements.","authors":"Sanuri Gunawardena, Umar Shuaib, Junaid Siddiqui","doi":"10.5334/tohm.1110","DOIUrl":"10.5334/tohm.1110","url":null,"abstract":"<p><strong>Background: </strong>Sensory ganglionopathy is a rare, often underrecognized neurological manifestation of Sjogren's disease, presenting with non-length-dependent sensory symptoms that may mimic other neuropathies.</p><p><strong>Case report: </strong>We report a case of Sjogren's-related sensory ganglionopathy presenting as severe sensory ataxia and pseudoathetosis. A 73-year-old man developed involuntary movements, sensory loss, and gait ataxia. Exam showed absent proprioception, areflexia, and worsening ataxia with eye closure. MRI revealed degenerative spine disease; EMG showed demyelinating and axonal polyneuropathy. CSF protein was elevated and Sjogren's antibodies were positive.</p><p><strong>Discussion: </strong>Progressive sensory ataxia despite immunotherapy confirmed Sjogren's-associated sensory ganglionopathy emphasizing the importance of early recognition.</p>","PeriodicalId":23317,"journal":{"name":"Tremor and Other Hyperkinetic Movements","volume":"16 ","pages":"12"},"PeriodicalIF":1.9,"publicationDate":"2026-02-11","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"https://www.ncbi.nlm.nih.gov/pmc/articles/PMC12904112/pdf/","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"146202828","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":"Pseudoathetotic Pseudodystonia as a Manifestation of Isolated Medullary Demyelination in Neuromyelitis Optica Spectrum Disorder.","authors":"Prachi Mohapatra, Lekshmi Sambhu Hema, Aditya Mahadevan, Divyani Garg, Ayush Agarwal, Awadh Kishor Pandit, Ajay Garg, Achal Kumar Srivastava, Divya M Radhakrishnan","doi":"10.5334/tohm.1153","DOIUrl":"10.5334/tohm.1153","url":null,"abstract":"<p><strong>Background: </strong>Pseudoathetosis and pseudodystonia are rare sensory-driven hyperkinetic movement disorders that may mimic primary dystonia. Although these manifestations have been reported in neuromyelitis optica spectrum disorder (NMOSD), they are typically associated with cervical spinal cord lesions, and occurrence due to isolated medullary demyelination is extremely uncommon.</p><p><strong>Case report: </strong>A 32-year-old woman with aquaporin-4-positive NMOSD developed pseudoathetotic movements with dystonic-appearing posturing of both hands following an isolated demyelinating lesion of the medulla with normal spinal cord imaging. The abnormal movements persisted after corticosteroids but resolved completely following plasmapheresis and rituximab.</p><p><strong>Discussion: </strong>This case expands the known anatomic spectrum of NMOSD-associated movement disorders by demonstrating isolated medullary demyelination as a rare substrate for pseudoathetotic pseudodystonia and emphasizes the importance of recognizing sensory-driven hyperkinetic movements to ensure timely immunotherapy.</p>","PeriodicalId":23317,"journal":{"name":"Tremor and Other Hyperkinetic Movements","volume":"16 ","pages":"11"},"PeriodicalIF":1.9,"publicationDate":"2026-02-10","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"https://www.ncbi.nlm.nih.gov/pmc/articles/PMC12904110/pdf/","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"146202133","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}