Travis R Larsh, Donald L Gilbert, Sudhakar Vadivelu, Devin K Binder, Ernest V Pedapati, Steve W Wu
{"title":"Post Deep Brain Stimulation Time Course of Aperiodic Activity in Childhood and Young Adult Dystonia.","authors":"Travis R Larsh, Donald L Gilbert, Sudhakar Vadivelu, Devin K Binder, Ernest V Pedapati, Steve W Wu","doi":"10.1002/mdc3.14159","DOIUrl":"10.1002/mdc3.14159","url":null,"abstract":"","PeriodicalId":19029,"journal":{"name":"Movement Disorders Clinical Practice","volume":" ","pages":"1305-1307"},"PeriodicalIF":2.6,"publicationDate":"2024-10-01","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"https://www.ncbi.nlm.nih.gov/pmc/articles/PMC11489614/pdf/","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"141580340","PeriodicalName":null,"FirstCategoryId":null,"ListUrlMain":null,"RegionNum":4,"RegionCategory":"医学","ArticlePicture":[],"TitleCN":null,"AbstractTextCN":null,"PMCID":"OA","EPubDate":null,"PubModel":null,"JCR":null,"JCRName":null,"Score":null,"Total":0}
{"title":"Symptom Network Analysis in a Large Sample of Children and Adults with a Chronic Tic Disorder.","authors":"Caroline Garcia Forlim, Valerie Brandt, Ewgeni Jakubovski, Christos Ganos, Simone Kühn, Kirsten Müller-Vahl","doi":"10.1002/mdc3.14167","DOIUrl":"10.1002/mdc3.14167","url":null,"abstract":"<p><strong>Background: </strong>Chronic tic disorders (CTD) are multifaceted disorders characterized by multiple motor and/or vocal tics. They are often associated with complex tics including echophenomena, paliphenomena, and coprophenomena as well as psychiatric comorbidities such as attention deficit/hyperactivity disorder (ADHD) and obsessive-compulsive disorder (OCD).</p><p><strong>Objectives: </strong>Our goal was to uncover the inter-relational structure of CTD and comorbid symptoms in children and adults and to understand changes in symptom structure across development.</p><p><strong>Methods: </strong>We used network and graph analyses to uncover the structure of association of symptoms in childhood/adolescence (n = 529) and adulthood (n = 503) and how this structure might change from childhood to adulthood, pinpointing core symptoms as a main target for interventions.</p><p><strong>Results: </strong>The analysis yielded core symptom networks in young and adult patients with CTD including complex tics and tic-related phenomena as well as touching people and objects. Core symptoms in childhood also included ADHD symptoms, whereas core symptoms in adults included symptoms of OCD instead. Interestingly, self-injurious behavior did not play a core role in the young CTD network, but became one of the central symptoms in adults with CDT. In addition, we found strong connections between complex motor and vocal tics as well as echolalia and echopraxia.</p><p><strong>Conclusions: </strong>Next to other complex tics, echophenomena, paliphenomena, and coprophenomena can be regarded core symptoms of CTD. ADHD symptoms are closely related to CTD in childhood, whereas symptoms of OCD and self-injurious behavior are closely associated with CTD in adults. Our results suggest that a differentiation between motor and vocal tics is somewhat arbitrary.</p>","PeriodicalId":19029,"journal":{"name":"Movement Disorders Clinical Practice","volume":" ","pages":"1232-1240"},"PeriodicalIF":2.6,"publicationDate":"2024-10-01","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"https://www.ncbi.nlm.nih.gov/pmc/articles/PMC11489602/pdf/","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"141759927","PeriodicalName":null,"FirstCategoryId":null,"ListUrlMain":null,"RegionNum":4,"RegionCategory":"医学","ArticlePicture":[],"TitleCN":null,"AbstractTextCN":null,"PMCID":"OA","EPubDate":null,"PubModel":null,"JCR":null,"JCRName":null,"Score":null,"Total":0}
Franziska Hopfner, Anja Tietz, Yuri D'Elia, Cristian Pattaro, Jos S Becktepe, Martin Gögele, Laura Barin, Peter P Pramstaller, Gregor Kuhlenbäumer, Roberto Melotti
{"title":"Archimedes Spiral Ratings: Determinants and Population-Based Limits of Normal.","authors":"Franziska Hopfner, Anja Tietz, Yuri D'Elia, Cristian Pattaro, Jos S Becktepe, Martin Gögele, Laura Barin, Peter P Pramstaller, Gregor Kuhlenbäumer, Roberto Melotti","doi":"10.1002/mdc3.14201","DOIUrl":"10.1002/mdc3.14201","url":null,"abstract":"<p><strong>Background: </strong>Tremor is commonly found among healthy humans or prevalently a symptom of neurological dysfunctions. However, the distinction between physiological and pathological tremor is dependent on the examiner's competence. Archimedes Spiral Rating (ASR) is a valid and reproducible semi-quantitative method to assess the severity of action tremor.</p><p><strong>Objectives: </strong>(1) To assess the range and percentiles of ASR in a large sample seemingly free of tremor-related conditions or symptoms from the population-based CHRIS-study. (2) To analyze the influence of sex, age, and the drawing hand on ASR. (3) To define ASR limits of normal. (4) To supply exemplary Archimedes spiral drawings by each rating to favor consistent and proficient clinical evaluation.</p><p><strong>Methods: </strong>Accurately investigated participants were randomly sampled over 14 sex-age strata. 2686 paired spirals drawn with both hands by 1343 participants were expertly assessed on a tremor rating scale from 0 to 9.</p><p><strong>Results: </strong>ASR had a quadratic increase with age in both sexes, while it was relatively lower in the dominant compared to the non-dominant hand and in women compared to men. ASRs above sex-age specific 97.5th percentiles of 4 and 5, below and above 60 years of age, respectively, were conceivably of non-physiological nature.</p><p><strong>Conclusions: </strong>In a large population-based sample we show a steeper increase of action tremor by age as age progresses. Relatively higher ratings among the elderly, males and the non-dominant hands, appear compatible with ASR limits of \"normal\" across sex-age groups. The current operational evidence may support practitioners differentiating physiological and pathological hand tremor.</p>","PeriodicalId":19029,"journal":{"name":"Movement Disorders Clinical Practice","volume":" ","pages":"1257-1265"},"PeriodicalIF":2.6,"publicationDate":"2024-10-01","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"https://www.ncbi.nlm.nih.gov/pmc/articles/PMC11489605/pdf/","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"142133264","PeriodicalName":null,"FirstCategoryId":null,"ListUrlMain":null,"RegionNum":4,"RegionCategory":"医学","ArticlePicture":[],"TitleCN":null,"AbstractTextCN":null,"PMCID":"OA","EPubDate":null,"PubModel":null,"JCR":null,"JCRName":null,"Score":null,"Total":0}
Amir H Talebi, Sirwan K L Darweesh, Bas R Bloem, Ioan G Bucur, Tom Heskes
{"title":"Effect of Early Levodopa Treatment on Mortality in People with Parkinson's Disease.","authors":"Amir H Talebi, Sirwan K L Darweesh, Bas R Bloem, Ioan G Bucur, Tom Heskes","doi":"10.1002/mdc3.14174","DOIUrl":"10.1002/mdc3.14174","url":null,"abstract":"<p><strong>Background: </strong>The ideal timing for initiating levodopa in newly diagnosed people with Parkinson's disease (PD) is uncertain due to limited data on the long-term effects of levodopa.</p><p><strong>Objective: </strong>The aim was to investigate whether early levodopa initiation postpones mortality (primary outcome), the requirement of device-aided therapies, and the incidence of PD-related complications, such as fall-induced injuries.</p><p><strong>Methods: </strong>Using nationwide claims data from Dutch hospitals (2012-2020), we grouped newly diagnosed PD individuals as \"early initiators\" (initiating levodopa within 2 years of diagnosis) or \"nonearly initiators.\" We used the national death registry to assess mortality and health-care claims to assess PD-related complications and device-aided therapies. We used marginal structural models to compare mortality and device-aided therapy rates between groups, and a Poisson regression model to compare PD-related complication rates.</p><p><strong>Results: </strong>Among 29,943 newly diagnosed PD individuals (mean age at diagnosis: 71.6, 38.5% female), there were 24,847 early and 5096 nonearly levodopa initiators. Over a median 4.25 years, 8109 (27.1%) died. The causal risk ratio for mortality was 1.04 (95% confidence interval [CI] 0.92-1.19) for early versus nonearly initiators. The risk ratio of receiving any device-aided therapy was 3.19 (95% CI 2.56-5.80). No association was observed with incidence of PD-related complications (incidence rate ratio: 1.00, 95% CI 0.96-1.05).</p><p><strong>Conclusions: </strong>Early levodopa initiation in PD does neither postpone nor accelerate mortality or PD-related complications, nor does it precipitate earlier occurrence of PD-related complications or mortality. However, we cannot exclude that the results were influenced by residual confounding due to unmeasured risk factors of mortality.</p>","PeriodicalId":19029,"journal":{"name":"Movement Disorders Clinical Practice","volume":" ","pages":"1249-1256"},"PeriodicalIF":2.6,"publicationDate":"2024-10-01","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"https://www.ncbi.nlm.nih.gov/pmc/articles/PMC11489610/pdf/","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"141875403","PeriodicalName":null,"FirstCategoryId":null,"ListUrlMain":null,"RegionNum":4,"RegionCategory":"医学","ArticlePicture":[],"TitleCN":null,"AbstractTextCN":null,"PMCID":"OA","EPubDate":null,"PubModel":null,"JCR":null,"JCRName":null,"Score":null,"Total":0}
Claudio Togni, Sandra Carpinelli, Philipp O Valko, Christopher Bockisch, Daniel Waldvogel, Esther Werth, Konrad P Weber, Yulia Valko
{"title":"Rapid Eye Movements during REM Sleep Differentiate PSP from Parkinson's Disease.","authors":"Claudio Togni, Sandra Carpinelli, Philipp O Valko, Christopher Bockisch, Daniel Waldvogel, Esther Werth, Konrad P Weber, Yulia Valko","doi":"10.1002/mdc3.14187","DOIUrl":"10.1002/mdc3.14187","url":null,"abstract":"<p><strong>Background: </strong>Little is known about the characteristics and occurrence frequencies of rapid eye movements (REMs) during REM sleep in movement disorders.</p><p><strong>Objectives: </strong>The aim of this study was to detect and characterize REMs during polysomnographically defined REM sleep as recorded by electro-oculography (EOG) in 12 patients with progressive supranuclear palsy (PSP), 13 patients with Parkinson's disease (PD) and 12 healthy controls.</p><p><strong>Methods: </strong>Using a modified EOG montage, we developed an algorithm that automatically detects and characterizes REMs during REM sleep based on their presumptive saccadic kinematics.</p><p><strong>Results: </strong>Compared to PD and healthy controls, REM densities and REM peak velocities were significantly reduced in PSP. These effects were most pronounced in vertical REMs.</p><p><strong>Conclusion: </strong>Ocular motor dysfunction, one of the cardinal features of PSP, seems to be equally at play during REM sleep and wakefulness. For future studies, we provide a novel tool for the unbiased analysis of REMs during REM sleep in movement disorders.</p>","PeriodicalId":19029,"journal":{"name":"Movement Disorders Clinical Practice","volume":" ","pages":"1281-1285"},"PeriodicalIF":2.6,"publicationDate":"2024-10-01","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"https://www.ncbi.nlm.nih.gov/pmc/articles/PMC11489613/pdf/","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"141902425","PeriodicalName":null,"FirstCategoryId":null,"ListUrlMain":null,"RegionNum":4,"RegionCategory":"医学","ArticlePicture":[],"TitleCN":null,"AbstractTextCN":null,"PMCID":"OA","EPubDate":null,"PubModel":null,"JCR":null,"JCRName":null,"Score":null,"Total":0}
Christopher Kobylecki, Yee Yen Goh, Rahema Mohammad, Alanna Beat, Emilia Michou, Samantha Pavey, Huw Morris, Henry Houlden, Viorica Chelban
{"title":"Clinical Practices and Opinions toward Gastrostomy Use in Patients with Atypical Parkinsonian Syndromes: A National Survey in the UK.","authors":"Christopher Kobylecki, Yee Yen Goh, Rahema Mohammad, Alanna Beat, Emilia Michou, Samantha Pavey, Huw Morris, Henry Houlden, Viorica Chelban","doi":"10.1002/mdc3.14196","DOIUrl":"10.1002/mdc3.14196","url":null,"abstract":"<p><strong>Background: </strong>Severe dysphagia poses a significant challenge for clinicians regarding feeding tube choices, practices, and timing due to a lack of evidence-based guidance.</p><p><strong>Objectives: </strong>To assess national clinical practices and opinions on gastrostomy use in patients with atypical parkinsonian syndromes (APS) across the UK.</p><p><strong>Methods: </strong>Online survey was administered to clinicians and allied health professionals regarding availability of services, current use, perceived advantages, and problems associated with gastrostomy insertion.</p><p><strong>Results: </strong>We received responses from 47 respondents across 12 UK centers, including 44 clinicians specialized in APS. Consensus was observed regarding primary indications for gastrostomy insertion and circumstances justifying avoidance of the procedure. Limitations in recommending gastrostomy due to insufficient evidence on safety and outcomes, survival and quality of life were identified. Widespread agreement on delays in gastrostomy discussions was highlighted as a challenge in optimizing patient care, together with variability in current practices and concerns over the lack of a standardized gastrostomy pathway, emphasizing the need for further research to address existing evidence gaps.</p><p><strong>Conclusion: </strong>This multi-center survey highlights agreement among clinicians on key aspects of indication, challenges, and limitations such as delayed decision-making and the absence of standardized pathways regarding the timing, method, and overall approach to gastrostomy insertion in APS. This study identified next steps to facilitate a more structured approach to future research toward a consensus on best practices for gastrostomy in APS. Addressing these challenges is crucial for enhancing patient outcomes and overall care quality in APS.</p>","PeriodicalId":19029,"journal":{"name":"Movement Disorders Clinical Practice","volume":" ","pages":"1266-1273"},"PeriodicalIF":2.6,"publicationDate":"2024-10-01","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"https://www.ncbi.nlm.nih.gov/pmc/articles/PMC11489604/pdf/","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"142073333","PeriodicalName":null,"FirstCategoryId":null,"ListUrlMain":null,"RegionNum":4,"RegionCategory":"医学","ArticlePicture":[],"TitleCN":null,"AbstractTextCN":null,"PMCID":"OA","EPubDate":null,"PubModel":null,"JCR":null,"JCRName":null,"Score":null,"Total":0}
Bart R Maas, Daniël H B Speelberg, Gert-Jan de Vries, Giulio Valenti, Andreas Ejupi, Bastiaan R Bloem, Sirwan K L Darweesh, Nienke M de Vries
{"title":"Patient Experience and Feasibility of a Remote Monitoring System in Parkinson's Disease.","authors":"Bart R Maas, Daniël H B Speelberg, Gert-Jan de Vries, Giulio Valenti, Andreas Ejupi, Bastiaan R Bloem, Sirwan K L Darweesh, Nienke M de Vries","doi":"10.1002/mdc3.14169","DOIUrl":"10.1002/mdc3.14169","url":null,"abstract":"<p><strong>Background: </strong>Remote monitoring systems have the potential to measure symptoms and treatment effects in people with Parkinson's disease (PwP) in the home environment. However, information about user experience and long-term compliance of such systems in a large group of PwP with relatively severe PD symptoms is lacking.</p><p><strong>Objective: </strong>The aim was to gain insight into user experience and long-term compliance of a smartwatch (to be worn 24/7) and an online dashboard to report falls and receive feedback of data.</p><p><strong>Methods: </strong>We analyzed the data of the \"Bringing Parkinson Care Back Home\" study, a 1-year observational cohort study in 200 PwP with a fall history. User experience, compliance, and reasons for noncompliance were described. Multiple Cox regression models were used to identify determinants of 1-year compliance.</p><p><strong>Results: </strong>We included 200 PwP (mean age: 69 years, 37% women), of whom 116 (58%) completed the 1-year study. The main reasons for dropping out of the study were technical problems (61 of 118 reasons). Median wear time of the smartwatch was 17.5 h/day. The online dashboard was used by 77% of participants to report falls. Smartphone possession, shorter disease duration, more severe motor symptoms, and less-severe freezing and balance problems, but not age and gender, were associated with a higher likelihood of 1-year compliance.</p><p><strong>Conclusions: </strong>The 1-year compliance with this specific smartwatch was moderate, and the user experience was generally good, except battery life and data transfer. Future studies can build on these findings by incorporating a smartwatch that is less prone to technical issues.</p>","PeriodicalId":19029,"journal":{"name":"Movement Disorders Clinical Practice","volume":" ","pages":"1223-1231"},"PeriodicalIF":2.6,"publicationDate":"2024-10-01","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"https://www.ncbi.nlm.nih.gov/pmc/articles/PMC11489606/pdf/","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"141759926","PeriodicalName":null,"FirstCategoryId":null,"ListUrlMain":null,"RegionNum":4,"RegionCategory":"医学","ArticlePicture":[],"TitleCN":null,"AbstractTextCN":null,"PMCID":"OA","EPubDate":null,"PubModel":null,"JCR":null,"JCRName":null,"Score":null,"Total":0}
Luis Guilherme Ramanzini, Julia M Frare, Luís F M Camargo, Juliana O F Silveira, Joseph Jankovic
{"title":"Does Delaying Levodopa Prevent Motor Complications in Parkinson's Disease? A Meta-Analysis.","authors":"Luis Guilherme Ramanzini, Julia M Frare, Luís F M Camargo, Juliana O F Silveira, Joseph Jankovic","doi":"10.1002/mdc3.14198","DOIUrl":"10.1002/mdc3.14198","url":null,"abstract":"<p><strong>Background: </strong>There has been a long debate whether delaying treatment with levodopa prevents motor complications in Parkinson's disease (PD).</p><p><strong>Objectives: </strong>We performed a meta-analysis on randomized clinical trials (RCTs) that compared early- versus delayed-start treatment with levodopa in PD.</p><p><strong>Methods: </strong>A systematic review was conducted in PubMed, EMBASE, and Web of Science databases from inception to July 1, 2023. Only RCTs that compared early and delayed levodopa treatment in PD were included. Non-randomized comparisons from follow-up studies were included as well. Our primary outcomes were occurrence of overall motor complications, motor fluctuations, and dyskinesias.</p><p><strong>Results: </strong>Seven studies with a total of 1149 patients (636 in the early-start group and 513 in the delayed-start) were included in our analysis. There was no difference between groups regarding motor complications (OR 1.39; 95% CI: 0.68-1.72; P = 0.37) or dyskinesias (OR 1.52; 95% CI: 0.90-2.57; P = 0.11). Motor fluctuations occurred less frequently in the early-start group (OR 0.70; 95% CI: 0.52-0.95; P = 0.02). Nonetheless, on subgroup analysis of dopamine agonists, rate of dyskinesias was smaller in the delayed-start group (OR 1.82; 95% CI: 1.08-3.07; P = 0.03).</p><p><strong>Conclusions: </strong>Delaying treatment with levodopa does not seem to prevent levodopa-related motor complications in PD. Adjunct treatment with dopamine agonists may reduce the need for higher doses of levodopa and thus reduce the risk for dyskinesias but this practice is often associated with a higher frequency of adverse effects related to dopamine agonists.</p>","PeriodicalId":19029,"journal":{"name":"Movement Disorders Clinical Practice","volume":" ","pages":"1195-1202"},"PeriodicalIF":2.6,"publicationDate":"2024-10-01","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"https://www.ncbi.nlm.nih.gov/pmc/articles/PMC11489609/pdf/","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"142073334","PeriodicalName":null,"FirstCategoryId":null,"ListUrlMain":null,"RegionNum":4,"RegionCategory":"医学","ArticlePicture":[],"TitleCN":null,"AbstractTextCN":null,"PMCID":"OA","EPubDate":null,"PubModel":null,"JCR":null,"JCRName":null,"Score":null,"Total":0}
Franclo Henning, Kireshnee Naidu, Christopher J Record, Natalia Dominik, Jana Vandrovcova, Frans Lubbe, Marli Dercksen, Lindsay A Wilson, Francois Van Der Westhuizen, Mary M Reilly, Henry Houlden, Michael G Hanna, Jonathan Carr
{"title":"Extended Phenotype of PEX11B Pathogenic Variants: Ataxia, Tremor, and Dystonia Due to a Novel C.2T > G Variant.","authors":"Franclo Henning, Kireshnee Naidu, Christopher J Record, Natalia Dominik, Jana Vandrovcova, Frans Lubbe, Marli Dercksen, Lindsay A Wilson, Francois Van Der Westhuizen, Mary M Reilly, Henry Houlden, Michael G Hanna, Jonathan Carr","doi":"10.1002/mdc3.14178","DOIUrl":"10.1002/mdc3.14178","url":null,"abstract":"","PeriodicalId":19029,"journal":{"name":"Movement Disorders Clinical Practice","volume":" ","pages":"1298-1300"},"PeriodicalIF":2.6,"publicationDate":"2024-10-01","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"https://www.ncbi.nlm.nih.gov/pmc/articles/PMC11489608/pdf/","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"141875404","PeriodicalName":null,"FirstCategoryId":null,"ListUrlMain":null,"RegionNum":4,"RegionCategory":"医学","ArticlePicture":[],"TitleCN":null,"AbstractTextCN":null,"PMCID":"OA","EPubDate":null,"PubModel":null,"JCR":null,"JCRName":null,"Score":null,"Total":0}