Elina Myller, Rolle Halonen, Daniel T Corp, Juho Joutsa
{"title":"Lesion-Induced Blepharospasm: Epidemiology and Clinical Characteristics.","authors":"Elina Myller, Rolle Halonen, Daniel T Corp, Juho Joutsa","doi":"10.5334/tohm.1025","DOIUrl":"https://doi.org/10.5334/tohm.1025","url":null,"abstract":"<p><strong>Background: </strong>Lesion-induced blepharospasm is considered rare. However, this information is based on a small number of heterogenous retrospective cohorts without routine neuroimaging.</p><p><strong>Objectives: </strong>To study the epidemiology and clinical characteristics of lesion-induced blepharospasm.</p><p><strong>Methods: </strong>Patients with blepharospasm with uncertain etiology prior to brain imaging were systematically searched from the electronic medical records of Turku University Hospital (1996-2022). Clinical information and imaging data were extracted from the patients' records and re-evaluated by the investigators. Etiology of blepharospasm was evaluated by an in-depth review of the clinical information in the context of available literature on lesion-induced dystonias. The prevalence and annual incidence of blepharospasm were calculated based on the annual population count in the area.</p><p><strong>Results: </strong>The search identified 57 patients, of whom four (7.0%) were considered to have lesion-induced blepharospasm, corresponding to a population-based prevalence of 2.5 per million and annual incidence of 0.3 per million. All patients with lesion-induced blepharospasm had atypical features, which were all significantly more common than in patients with idiopathic blepharospasm (<i>P</i> < 0.05).</p><p><strong>Conclusions: </strong>Lesion-induced blepharospasm is more common than thought previously. However, all these patients showed atypical features, suggesting that brain imaging in blepharospasm can be limited to patients with atypical features only.</p><p><strong>Highlights: </strong>This study investigated the epidemiology of lesion-induced blepharospasm by systematically re-evaluating all patients with blepharospasm with uncertain etiology prior to structural brain imaging from a university hospital (1996-2022).Our results show that lesion-induced blepharospasm is more common that thought previously (7.0% of included patients, prevalence 2.5 per million, annual incidence 0.3 per million).All patients with lesion-induced blepharospasm showed clinical features that were considered atypical for idiopathic blepharospasm, indicating that routine brain imaging is not needed in patients with typical symptoms.</p>","PeriodicalId":23317,"journal":{"name":"Tremor and Other Hyperkinetic Movements","volume":"15 ","pages":"25"},"PeriodicalIF":2.5,"publicationDate":"2025-06-09","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"https://www.ncbi.nlm.nih.gov/pmc/articles/PMC12164745/pdf/","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"144302935","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":"Severe Whip-Like Cervical Tics as an Indication For Thalamic Deep Brain Stimulation: Report of Two Cases.","authors":"Masamune Tsuji, Kei Yamashiro, Takashi Morishita, Atsushi Hirota, Hitoshi Iida, Yasuhiko Baba, Hiroshi Abe","doi":"10.5334/tohm.1010","DOIUrl":"10.5334/tohm.1010","url":null,"abstract":"<p><strong>Background: </strong>Cervical spinal cord injury caused by cervical tics associated with Tourette syndrome (TS) is a recognized complication; however, the role of deep brain stimulation (DBS) in mitigating the risk of such injuries remains unclear.</p><p><strong>Case report: </strong>We report two cases of TS with severe cervical tics, both of which responded favorably to DBS. In one case, DBS prevented the progression of cervical spinal cord injury, whereas in the other case, it prevented its onset.</p><p><strong>Discussion: </strong>Poor control of severe cervical tics is a significant risk factor for cervical spinal cord injury, and early consideration of DBS is recommended.</p><p><strong>Highlights: </strong>This case report presents two cases in which deep brain stimulation (DBS) was effective for patients with Tourette syndrome exhibiting severe cervical tics. Through this report, we demonstrate the potential effectiveness of DBS as a treatment to reduce the risk of cervical spinal cord injury caused by severe cervical tics.</p>","PeriodicalId":23317,"journal":{"name":"Tremor and Other Hyperkinetic Movements","volume":"15 ","pages":"24"},"PeriodicalIF":2.5,"publicationDate":"2025-05-16","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"https://www.ncbi.nlm.nih.gov/pmc/articles/PMC12083071/pdf/","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"144094993","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":"\"Hyperkinetic\" and \"Hypokinetic\": Is There a Need for a Third Category (i.e., \"Mixed\")?","authors":"Sara M Schaefer, Elan D Louis","doi":"10.5334/tohm.997","DOIUrl":"10.5334/tohm.997","url":null,"abstract":"<p><p>Terminology in the field of movement disorders has evolved multiple times over the years. Traditionally, classification schema have utilized a two-category approach, with a hyper- and hypokinetic branchpoint as the first step towards describing movement disorders and elucidating phenomenological diagnoses. However, this terminology falls short, as it does not adequately capture the complexity of several abnormalities of movement- including ataxia and rigidity- at the electrophysiologic and phenotypic levels. Rather, these movement disorders are characterized by mixed hyper- and hypokinetic phenomena. We propose a third category, \"mixed\", which would optimally classify the full range of movements.</p>","PeriodicalId":23317,"journal":{"name":"Tremor and Other Hyperkinetic Movements","volume":"15 ","pages":"23"},"PeriodicalIF":2.5,"publicationDate":"2025-05-13","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"https://www.ncbi.nlm.nih.gov/pmc/articles/PMC12082446/pdf/","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"144094992","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":"Dyskinesia-Hyperpyrexia Syndrome in Parkinson's Disease May Benefit from GPi Deep Brain Stimulation: A Case Report.","authors":"Shi-Shi Huang, Lu-Lu Ma, Han-Song Sheng, Rong-Pei Liu, Shi-Guo Zhu, Shan-Shan Xu, Mei-Ling Sheng, Zhu-Ling Chen, Nu Zhang, Xiong Zhang, Jian-Yong Wang","doi":"10.5334/tohm.912","DOIUrl":"10.5334/tohm.912","url":null,"abstract":"<p><strong>Background: </strong>Dyskinesia-hyperpyrexia syndrome (DHS) is a rare but life-threatening complication in Parkinson's disease (PD). Its prevention and treatment are clinical challenges.</p><p><strong>Case report: </strong>We herein present a case of a female PD patient who experienced recurrent episodes of DHS and levodopa-induced dyskinesia. The complications were alleviated after bilateral globus pallidus internus (GPi) deep brain stimulation (DBS).</p><p><strong>Discussion: </strong>Our report provides evidence that GPi DBS may reduce the susceptibility to DHS. More evidences are warranted to validate this hypothesis.</p>","PeriodicalId":23317,"journal":{"name":"Tremor and Other Hyperkinetic Movements","volume":"15 ","pages":"20"},"PeriodicalIF":2.5,"publicationDate":"2025-05-07","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"https://www.ncbi.nlm.nih.gov/pmc/articles/PMC12063572/pdf/","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"144028689","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}
Shohei Okusa, Toshiki Tezuka, Yoshihiro Nihei, Jin Nakahara, Morinobu Seki
{"title":"Clinical Features of Essential Tremor and its Impact on Quality of Life in Japan.","authors":"Shohei Okusa, Toshiki Tezuka, Yoshihiro Nihei, Jin Nakahara, Morinobu Seki","doi":"10.5334/tohm.1006","DOIUrl":"https://doi.org/10.5334/tohm.1006","url":null,"abstract":"<p><strong>Background: </strong>Essential tremor (ET) is primarily characterized by action tremor, but is also associated with various non-motor symptoms (NMS). However, the diagnostic relevance of NMS in ET remains unclear. This study aimed to compare NMS and motor symptoms of ET with those of Parkinson's disease-tremor dominant type (PD-TDT) and healthy controls (HCs) and to identify the presence and diagnostic relevance of NMS.</p><p><strong>Methods: </strong>Twenty-three ET patients, 23 PD-TDT patients, and 22 HCs were enrolled. Diagnoses of ET and PD were confirmed using Movement Disorder Society (MDS) criteria and Dopamine transporter single-photon emission computed tomography. Motor symptoms, NMS and quality of life (QOL) were evaluated using validated scales, including the Clinical Rating Scale for Tremor (CRST), MDS-Unified Parkinson's Disease Rating Scale (UPDRS), Non-Motor Symptoms Scale for Parkinson's Disease (NMSS), Odor Stick Identification Test for Japanese (OSIT-J), and Quality of Life in Essential Tremor Questionnaire (QUEST).</p><p><strong>Results: </strong>ET patients had significantly higher NMSS total scores and MDS-UPDRS part IB scores than HCs, with more severe sleep disturbances, fatigue, and urinary problems. CRST scores were significantly correlated with QUEST scores. Logistic regression identified CRST Part B and OSIT-J as key factors distinguishing ET from PD-TDT, with 87% sensitivity and 90% specificity.</p><p><strong>Discussion: </strong>ET patients showed more severe NMS than HCs. Differentiating ET from PD-TDT requires motor and sensory assessments, highlighting the diagnostic relevance of NMS. Comprehensive evaluation is essential for accurate diagnosis and management of ET.</p>","PeriodicalId":23317,"journal":{"name":"Tremor and Other Hyperkinetic Movements","volume":"15 ","pages":"21"},"PeriodicalIF":2.5,"publicationDate":"2025-05-07","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"https://www.ncbi.nlm.nih.gov/pmc/articles/PMC12063571/pdf/","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"144016952","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}
Wietske A Babeliowsky, Bart Swinnen, Jeroen Hoogland, Rob M A de Bie, Anne-Fleur van Rootselaar
{"title":"Effect of Orthostatic Tremor on Quality of Life - a Cohort Study.","authors":"Wietske A Babeliowsky, Bart Swinnen, Jeroen Hoogland, Rob M A de Bie, Anne-Fleur van Rootselaar","doi":"10.5334/tohm.1008","DOIUrl":"https://doi.org/10.5334/tohm.1008","url":null,"abstract":"<p><strong>Background: </strong>Orthostatic tremor (OT) is characterized by a lower extremity tremor causing unsteadiness while standing, urging patients to lean, walk, or sit to ease symptoms. This severely disrupts daily life and reduces quality of life (QoL), though the extent of QoL impairment remains largely unknown. The objective of this cohort study was to evaluate the effect of OT on daily functioning and well-being over time.</p><p><strong>Methods: </strong>In this prospective single-center and community based cohort study, OT patients annually completed self-report scales from 2018 to 2024, including the Dutch OT-questionnaire, Hospital Anxiety and Depression Scale (HADS), (instrumental) Activities of Daily Living, and the Short Form-36 (SF-36) to evaluate QoL.</p><p><strong>Results: </strong>Fifty-three OT patients participated in the study. OT patients showed reduced well-being compared to the general population based on outcomes from the SF-36 and HADS, with 30 out of 53 patients experiencing potential or suspected depression and/or anxiety. Despite viewing OT as progressive, most patients saw no significant changes in overall well-being or daily functioning, although a significant difference was found between baseline and last follow-up for physical role limitation.</p><p><strong>Discussion: </strong>Although OT patients report reduced well-being, both daily functioning and overall well-being remained stable over time, despite progressive symptoms. This is likely due to patients increased ability to adapt to OT symptoms. Additionally, a substantial portion had potential or suspected depression or anxiety.</p><p><strong>Highlights: </strong>Orthostatic tremor (OT) patients report reduced quality of life, with worsened walking ability and increased weather sensitivity over time. Despite this, daily functioning and overall well-being remained stable throughout the study. A significant portion of patients also showed potential or suspected depression and/or anxiety.</p>","PeriodicalId":23317,"journal":{"name":"Tremor and Other Hyperkinetic Movements","volume":"15 ","pages":"22"},"PeriodicalIF":2.5,"publicationDate":"2025-05-07","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"https://www.ncbi.nlm.nih.gov/pmc/articles/PMC12063594/pdf/","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"143987888","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":"Current Management of Neurological Wilson's Disease.","authors":"V H Ganaraja, Vikram V Holla, Pramod Kumar Pal","doi":"10.5334/tohm.938","DOIUrl":"https://doi.org/10.5334/tohm.938","url":null,"abstract":"<p><p>Wilson's disease (WD) is a disorder of copper metabolism due to variants in the <i>ATP7B</i> gene. This autosomal recessively inherited disorder is characterized by the accumulation of copper in various body parts, mainly the liver, brain, and kidneys. Initially, WD was described to involve the hepatic and neurological systems. Subsequently, diverse presentations have been reported with skeletal and hematological manifestations and various constellations of symptoms. Neurological manifestations of WD are varied, ranging from asymptomatic neurological state to refractory dystonia. Earlier, the diagnosis was based only on measuring serum ceruloplasmin levels, urinary copper levels, and imaging. Advanced genetic testing has provided an additional mode of diagnosis in the patient, screening of the family members and, a way to better understand the genotype-phenotype associations of the disease if there are any. In the last few decades, the treatment of WD has evolved from symptomatic treatment and chelation therapy to many new advanced measures for both copper chelation and symptomatic relief. With a better understanding of the genetic aspects of WD in recent years, there has been more focus on gene therapy, novel therapies targeting ATP7B genes, and therapies targeting mutant proteins to prevent copper accumulation. This article highlights the advances in diagnostic methods and treatment modalities in WD.</p>","PeriodicalId":23317,"journal":{"name":"Tremor and Other Hyperkinetic Movements","volume":"15 ","pages":"17"},"PeriodicalIF":2.5,"publicationDate":"2025-05-05","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"https://www.ncbi.nlm.nih.gov/pmc/articles/PMC12063596/pdf/","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"144051632","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}
Emily Houston, Amanda G Kennedy, Terry Rabinowitz, Gail L Rose, James Boyd
{"title":"Feasibility, Effectiveness, and Acceptability of a Telemedicine Neurological Consultation for Drug-Induced Movement Disorders; A Randomized Pilot Study.","authors":"Emily Houston, Amanda G Kennedy, Terry Rabinowitz, Gail L Rose, James Boyd","doi":"10.5334/tohm.1007","DOIUrl":"10.5334/tohm.1007","url":null,"abstract":"<p><strong>Background: </strong>Individuals exposed to neuroleptics are at risk of developing a drug-induced movement disorder (DIMD). Early identification and appropriate management are necessary to minimize the risk of DIMDs worsening and becoming irreversible. Movement disorder neurologists can assist other clinicians in assessing the cause of the DIMD and make care recommendations. The aim of this study was to evaluate if telemedicine can be used to provide a neurological consultation service to patients with DIMDs.</p><p><strong>Methods: </strong>Patients referred by mental health clinicians (MHCs) in a rural state were randomized to have a neurological consult in-person or through telemedicine. Participants completed two visits with a neurologist and completed surveys about their experience and well-being. MHCs provided feedback on the service through a survey and qualitative interviews.</p><p><strong>Results: </strong>In the IP group, 79% or participants completed Visit 1 and 71% attended Visit 2, as compared to 86% of participants completing Visit 1 in the TM group and 57% were present for Visit 2. Satisfaction scores were slightly higher for the IP group at both visits. MHCs were satisfied with the consult, reporting that it was helpful and improved care for the patients.</p><p><strong>Discussion: </strong>Providing a consult service through telemedicine is feasible, effective, and acceptable, and can be improved further following feedback provided by the MHCs. Overall, participants and MHCs were pleased to have the opportunity to meet and collaborate with a neurologist.</p><p><strong>Highlights: </strong>The results from our study address gaps in knowledge related to providing specialist care to people with drug-induced movement disorders in a rural setting. Telemedicine consultation is feasible, with low rates of technological issues, and participants with drug-induced movement disorders were satisfied with telemedicine as a mode of care delivery.</p>","PeriodicalId":23317,"journal":{"name":"Tremor and Other Hyperkinetic Movements","volume":"15 ","pages":"19"},"PeriodicalIF":2.5,"publicationDate":"2025-05-05","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"https://www.ncbi.nlm.nih.gov/pmc/articles/PMC12063570/pdf/","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"144015015","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}
Betsy Thomas, Gabriele Bellini, Wen-Yu Lee, Yidan Shi, Alon Mogilner, Michael H Pourfar
{"title":"High Intensity Focused Ultrasound - Longitudinal Data on Efficacy and Safety.","authors":"Betsy Thomas, Gabriele Bellini, Wen-Yu Lee, Yidan Shi, Alon Mogilner, Michael H Pourfar","doi":"10.5334/tohm.987","DOIUrl":"https://doi.org/10.5334/tohm.987","url":null,"abstract":"<p><strong>Background: </strong>High intensity focused ultrasound (HiFU) is a relatively new incisionless intervention used for treatment of essential tremor and Parkinson's disease tremor. Understanding the indications, benefits, risks and limitations of HiFU, as well as how it compares to deep brain stimulation (DBS), is important in guiding appropriate recommendations for prospective patients.</p><p><strong>Methods: </strong>Current literature on efficacy and safety of HiFU in essential tremor and Parkinson's disease was reviewed. We additionally reviewed data on the patients who presented to our center for HiFU consultation, including outcomes of patients with low skull density ratios, and distances traveled for the procedure.</p><p><strong>Results/discussion: </strong>HiFU is an effective and generally well-tolerated treatment for tremor. Adverse events, especially gait instability, are typically temporary but should be discussed with patients. The risk of tremor recurrence in certain patients with Parkinson's disease is also of note. Identifying appropriate candidates for either intervention remains crucial and involves considering each patient's circumstances and preferences, potential adverse effects, and practical aspects like access to follow-up and expectations. Data on bilateral HiFU lesioning, use of HiFU in patients with low skull density ratios, and emerging targets like the pallidothalamic tract are discussed as well.</p>","PeriodicalId":23317,"journal":{"name":"Tremor and Other Hyperkinetic Movements","volume":"15 ","pages":"18"},"PeriodicalIF":2.5,"publicationDate":"2025-05-05","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"https://www.ncbi.nlm.nih.gov/pmc/articles/PMC12063574/pdf/","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"144020379","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}
Harleen Kaur, Tim J Goble, Albert Fenoy, Ritesh A Ramdhani
{"title":"Deep Brain Stimulation for Post-Hypoxic Myoclonus: A Case Correlating Local Field Potentials to Clinical Outcome.","authors":"Harleen Kaur, Tim J Goble, Albert Fenoy, Ritesh A Ramdhani","doi":"10.5334/tohm.999","DOIUrl":"https://doi.org/10.5334/tohm.999","url":null,"abstract":"<p><strong>Background: </strong>Post-hypoxic myoclonus (PHM) is characterized by generalized myoclonus after hypoxic brain injury. PHM is often functionally impairing and refractory to medical therapies. There are a handful of reports utilizing deep brain stimulation (DBS) to treat medically refractory PHM.</p><p><strong>Case report: </strong>A 56-year-old woman developed PHM following an anoxic brain injury. Utilizing a stimulating and sensing DBS system, we show clinical improvement in myoclonus at 6 months and correlate it to local field potential (LFP) activity.</p><p><strong>Discussion: </strong>We present the first case to utilize DBS sensing to correlate LFP activity to myoclonus improvement. Our case contributes to the growing evidence of DBS for PHM.</p>","PeriodicalId":23317,"journal":{"name":"Tremor and Other Hyperkinetic Movements","volume":"15 ","pages":"16"},"PeriodicalIF":2.5,"publicationDate":"2025-04-24","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"https://www.ncbi.nlm.nih.gov/pmc/articles/PMC12023146/pdf/","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"144064759","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}