Tremor and Other Hyperkinetic Movements最新文献

{"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":"https://doi.org/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":2.1,"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":2.1,"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":2.1,"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":2.1,"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":2.1,"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":2.1,"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":2.1,"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}

{"title":"Copy Number Variant Duplications Associated with Essential Tremor.","authors":"Miranda Medeiros, Calwing Liao, Allison A Dilliott, Jay P Ross, Veikko Vuokila, Farah Aboasali, Charles-Etienne Castonguay, Dan Spiegelman, Franziska Hopfner, Carles Vilariño-Güell, Elena García-Martín, Hortensia Alonso-Navarro, José A G Agúndez, Félix Javier Jiménez-Jiménez, Pau Pastor, Alex Rajput, Ali Rajput, Günther Deuschl, Gregor Kuhlenbäumer, Simon L Girard, Sali M K Farhan, Patrick A Dion, Guy A Rouleau","doi":"10.5334/tohm.1132","DOIUrl":"10.5334/tohm.1132","url":null,"abstract":"<p><strong>Background: </strong>Essential tremor (ET) is a complex neurological disorder with a strong genetic basis, yet there remains a disparity between its estimated heritability and currently known genetic risk. This missing heritability has led to a lack of appropriate treatments and has exacerbated the misdiagnosis of patients.</p><p><strong>Methods: </strong>To address the missing heritability of ET, we called copy number variants (CNVs) in a large cohort of ET patients (n = 1,853) and unaffected controls (n = 10,336). CNVs were called from single nucleotide polymorphism (SNP) microarray data using PennCNV and QuantiSNP and only rare CNVs (frequency < 1%) intersecting protein coding regions of the genome were analyzed. To investigate whether CNV occurrence was associated with ET, global burden, pathogenicity burden, gene set enrichment, and gene burden tests were conducted.</p><p><strong>Results: </strong>Global duplication burden by CNV number, CNV length, and number of genes affected by CNVs were all significantly elevated in ET patients compared to controls. Across gene-sets, duplications affecting Mendeliome genes, genes highly expressed in the brain, and genes expressed in the cerebellum were significantly enriched in patients compared to controls. Gene-based burden testing indicated that duplications involving <i>ZNF813</i> were significantly less frequent in ET patients than in controls. No associations with deletion events were observed.</p><p><strong>Discussion: </strong>Our results point to rare copy number duplications affecting protein coding regions of the genome as likely contributors to ET genetic risk. However, specific susceptibility genes could not be reliably identified, highlighting the need for larger studies of diverse variant types to clarify the genetic architecture of ET.</p>","PeriodicalId":23317,"journal":{"name":"Tremor and Other Hyperkinetic Movements","volume":"16 ","pages":"10"},"PeriodicalIF":2.1,"publicationDate":"2026-02-10","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"https://www.ncbi.nlm.nih.gov/pmc/articles/PMC12904121/pdf/","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"146202955","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 Profiles of Eight Patients with Combined Dystonic Manifestation of Type-III GM1 Gangliosidosis: A Video Case Series from India.","authors":"Subhajit Roy, Cheshta Arora, Vikram V Holla, Shweta Prasad, Prashant Phulpagar, Nitish Kamble, Babylakshmi Muthusamy, Jitender Saini, Ravi Yadav, Pramod Kumar Pal","doi":"10.5334/tohm.1152","DOIUrl":"10.5334/tohm.1152","url":null,"abstract":"<p><strong>Background: </strong>Type-III (adult/chronic) GM1 gangliosidosis is an uncommon, late-onset lysosomal disorder that frequently presents as a complex movement disorder.</p><p><strong>Methods: </strong>In this retrospective case series, clinical details, neuroimaging, electrophysiology, and genetics were extracted from standardized records and videos.</p><p><strong>Results: </strong>Eight patients were identified with median age at symptom onset of 6 years (range 3-18), and age at presentation of 23 years (12-27). All exhibited generalized dystonia with early, conspicuous oro-mandibular-cranio-cervical involvement; dysarthria was universal, parkinsonism occurred in two, and corticospinal signs in six. Ocular motor abnormalities were frequent; kyphoscoliosis was common. Where performed, nerve conduction studies, electroencephalography, evoked potentials, and abdominal ultrasound were unremarkable. MRI consistently demonstrated bilateral posterior putaminal T2/FLAIR change and the pathognomonic pallidal SWI \"wishbone\" pattern. All patients harboured biallelic <i>GLB1</i> variants: seven were compound heterozygous and one was homozygous. The recurrent variant c.1325G>A;p.Arg442Gln was present in seven patients. One novel variant (c.1022G>T;p.Gly341Val) was identified. Symptomatic therapies yielded variable, generally modest benefits over available follow-up.</p><p><strong>Discussion: </strong>A prominent oromandibular-cranio-cervical dystonia, posterior putaminal atrophy, and hyperintensity, with a SWI \"wishbone\" sign, strongly point to Type-III GM1 gangliosidosis. Recognizing this clinico-radiologic-genetic constellation can streamline targeted testing and counselling.</p>","PeriodicalId":23317,"journal":{"name":"Tremor and Other Hyperkinetic Movements","volume":"16 ","pages":"9"},"PeriodicalIF":2.1,"publicationDate":"2026-02-09","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"https://www.ncbi.nlm.nih.gov/pmc/articles/PMC12904113/pdf/","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"146202960","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":"Rescue Thalamotomy for Habituation to Deep Brain Stimulation in Essential Tremor: Case Report.","authors":"Roman Kiselev, Vladislav Babchenko","doi":"10.5334/tohm.1050","DOIUrl":"10.5334/tohm.1050","url":null,"abstract":"<p><strong>Background: </strong>Habituation to deep brain stimulation (DBS) remains a therapeutic challenge in essential tremor (ET), with 4-42% of patients experiencing progressive loss of benefit.</p><p><strong>Case report: </strong>A 68-year-old man with ET presented with debilitating bilateral tremor. The patient underwent bilateral DBS to posterior subthalamic area (PSA). Despite initial tremor relief, symptoms gradually recurred three months later. After 12 months of unsuccessful programming, the patient underwent left-sided radiofrequency Vim thalamotomy. Due to severe tremor rebound phenomenon, tremor control was achieved in 1.5 months.</p><p><strong>Discussion: </strong>To our knowledge, this study represents the first report of successful thalamotomy performed for PSA-DBS habituation, accompanied by delayed postoperative tremor improvement. The clinical trajectory and putative mechanisms underlying both habituation and lesion-induced tremor suppression are discussed based on imaging analysis.</p>","PeriodicalId":23317,"journal":{"name":"Tremor and Other Hyperkinetic Movements","volume":"16 ","pages":"8"},"PeriodicalIF":2.1,"publicationDate":"2026-02-09","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"https://www.ncbi.nlm.nih.gov/pmc/articles/PMC12904111/pdf/","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"146202135","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}