Pub Date : 2022-10-01DOI: 10.1177/20552173221144226
Javier Ballesteros, Guillermo Bueno-Gil, Alfredo Rodríguez-Antigüedad, Ángel P Sempere, Beatriz Del Río, Mar Baz, Nicolás Medrano, Gustavo Saposnik, Jorge Maurino
Experiences of regret associated with caring for patients with multiple sclerosis (MS) can affect medical decisions. A non-interventional study was conducted to assess the dimensionality and item characteristics of a battery including the Regret Intensity Scale (RIS-10) and 15 items evaluating common situations experienced by nurses in MS care. A total of 97 nurses were included. The RIS-10 showed good internal reliability and a unidimensional structure according to Mokken analysis. All-item homogeneity coefficients exceeded 0.30, whereas scalability for the overall RIS-10 was 0.66, indicating a strong scale. This battery showed adequate psychometric properties to evaluate regret among MS nurses.
{"title":"Assessing care-related regret among nurses specialized in multiple sclerosis: A psychometric analysis of a new assessment battery.","authors":"Javier Ballesteros, Guillermo Bueno-Gil, Alfredo Rodríguez-Antigüedad, Ángel P Sempere, Beatriz Del Río, Mar Baz, Nicolás Medrano, Gustavo Saposnik, Jorge Maurino","doi":"10.1177/20552173221144226","DOIUrl":"https://doi.org/10.1177/20552173221144226","url":null,"abstract":"<p><p>Experiences of regret associated with caring for patients with multiple sclerosis (MS) can affect medical decisions. A non-interventional study was conducted to assess the dimensionality and item characteristics of a battery including the Regret Intensity Scale (RIS-10) and 15 items evaluating common situations experienced by nurses in MS care. A total of 97 nurses were included. The RIS-10 showed good internal reliability and a unidimensional structure according to Mokken analysis. All-item homogeneity coefficients exceeded 0.30, whereas scalability for the overall RIS-10 was 0.66, indicating a strong scale. This battery showed adequate psychometric properties to evaluate regret among MS nurses.</p>","PeriodicalId":18961,"journal":{"name":"Multiple Sclerosis Journal - Experimental, Translational and Clinical","volume":"8 4","pages":"20552173221144226"},"PeriodicalIF":2.8,"publicationDate":"2022-10-01","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"https://ftp.ncbi.nlm.nih.gov/pub/pmc/oa_pdf/42/09/10.1177_20552173221144226.PMC9742695.pdf","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"10362511","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}
Pub Date : 2022-10-01DOI: 10.1177/20552173221128170
Negar Molazadeh, Gauruv Bose, Itay Lotan, Michael Levy
Background: Myelin oligodendrocyte glycoprotein antibody-associated disease (MOGAD) has various similarities with AQP4-IgG-seropositive Neuromyelitis Optica Spectrum Disorder (AQP4-IgG + NMOSD) in terms of clinical presentations, magnetic resonance imaging (MRI) findings, and response to treatment. But unlike AQP4-IgG + NMOSD, which is known to coexist with various autoimmune diseases and cancers, an association of MOGAD with these conditions is less clear.
Methods: We conducted a systematic search in PubMed, Scopus, Web of Science, and Embase based on the preferred reporting items for systematic reviews and meta-analysis (PRISMA). Duplicates were removed using Mendeley 1.19.8 (USA production) and the citations were uploaded into Covidence systematic review platform for screening.
Results: The most common autoimmune disease overlapping with MOGAD was anti-N-Methyl-D-Aspartate receptor encephalitis (anti-NMDAR-EN), followed by autoimmune thyroid disorders, and the most common autoantibody was antinuclear antibody (ANA), followed by AQP4-IgG (double-positive MOG-IgG and AQP4-IgG). A few sporadic cases of cancers and MOG-IgG-associated paraneoplastic encephalomyelitis were found.
Conclusion: Unlike AQP4-IgG + NMOSD, MOGAD lacks clustering of autoimmune diseases and autoantibodies associated with systemic and organ-specific autoimmunity. Other than anti-NMDAR-EN and perhaps AQP4-IgG + NMOSD, the evidence thus far does not support the need for routine screening of overlapping autoimmunity and neoplasms in patients with MOGAD.
{"title":"Autoimmune diseases and cancers overlapping with myelin oligodendrocyte glycoprotein antibody-associated disease (MOGAD): A systematic review.","authors":"Negar Molazadeh, Gauruv Bose, Itay Lotan, Michael Levy","doi":"10.1177/20552173221128170","DOIUrl":"https://doi.org/10.1177/20552173221128170","url":null,"abstract":"<p><strong>Background: </strong>Myelin oligodendrocyte glycoprotein antibody-associated disease (MOGAD) has various similarities with AQP4-IgG-seropositive Neuromyelitis Optica Spectrum Disorder (AQP4-IgG + NMOSD) in terms of clinical presentations, magnetic resonance imaging (MRI) findings, and response to treatment. But unlike AQP4-IgG + NMOSD, which is known to coexist with various autoimmune diseases and cancers, an association of MOGAD with these conditions is less clear.</p><p><strong>Methods: </strong>We conducted a systematic search in PubMed, Scopus, Web of Science, and Embase based on the preferred reporting items for systematic reviews and meta-analysis (PRISMA). Duplicates were removed using Mendeley 1.19.8 (USA production) and the citations were uploaded into Covidence systematic review platform for screening.</p><p><strong>Results: </strong>The most common autoimmune disease overlapping with MOGAD was anti-N-Methyl-D-Aspartate receptor encephalitis (anti-NMDAR-EN), followed by autoimmune thyroid disorders, and the most common autoantibody was antinuclear antibody (ANA), followed by AQP4-IgG (double-positive MOG-IgG and AQP4-IgG). A few sporadic cases of cancers and MOG-IgG-associated paraneoplastic encephalomyelitis were found.</p><p><strong>Conclusion: </strong>Unlike AQP4-IgG + NMOSD, MOGAD lacks clustering of autoimmune diseases and autoantibodies associated with systemic and organ-specific autoimmunity. Other than anti-NMDAR-EN and perhaps AQP4-IgG + NMOSD, the evidence thus far does not support the need for routine screening of overlapping autoimmunity and neoplasms in patients with MOGAD.</p>","PeriodicalId":18961,"journal":{"name":"Multiple Sclerosis Journal - Experimental, Translational and Clinical","volume":"8 4","pages":"20552173221128170"},"PeriodicalIF":2.8,"publicationDate":"2022-10-01","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"https://ftp.ncbi.nlm.nih.gov/pub/pmc/oa_pdf/b7/19/10.1177_20552173221128170.PMC9597055.pdf","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"10277551","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}
Pub Date : 2022-10-01DOI: 10.1177/20552173221132469
Jennifer Lyons, Richard Hughes, Kerry McCarthy, Nicholas Everage, Shivani Kapadia, Catherine Miller, Priya Singhal, Karen Smirnakis
Background and objectives: Dimethyl fumarate (DMF), an oral disease-modifying therapy with an established benefit and well-described safety profile, is among the most commonly used therapies for relapsing forms of multiple sclerosis. As of 31 December 2021, >560,000 patients have been treated with DMF, representing >1,190,000 person-years of exposure. Of these, 6413 patients (14,292 person-years) were from clinical trials.
Methods and results: Progressive multifocal leukoencephalopathy (PML) has occurred in the setting of lymphopenia (<0.91 × 109/L) in patients treated with DMF. We present detailed clinical characteristics and outcomes of the 12 confirmed PML cases occurring in MS patients on DMF as of 21 July 2021. The PML incidence in DMF-treated patients is 1.07 per 100,000 person-years of DMF exposure. Lymphopenia is the common risk for PML in DMF treatment.
Discussion: DMF-related PML is rare but has occurred in the setting of lymphopenia, supporting the current recommendations for absolute lymphocyte count monitoring in all patients, regardless of age and time on therapy.
{"title":"Progressive multifocal leukoencephalopathy outcomes in patients with multiple sclerosis treated with dimethyl fumarate.","authors":"Jennifer Lyons, Richard Hughes, Kerry McCarthy, Nicholas Everage, Shivani Kapadia, Catherine Miller, Priya Singhal, Karen Smirnakis","doi":"10.1177/20552173221132469","DOIUrl":"https://doi.org/10.1177/20552173221132469","url":null,"abstract":"<p><strong>Background and objectives: </strong>Dimethyl fumarate (DMF), an oral disease-modifying therapy with an established benefit and well-described safety profile, is among the most commonly used therapies for relapsing forms of multiple sclerosis. As of 31 December 2021, >560,000 patients have been treated with DMF, representing >1,190,000 person-years of exposure. Of these, 6413 patients (14,292 person-years) were from clinical trials.</p><p><strong>Methods and results: </strong>Progressive multifocal leukoencephalopathy (PML) has occurred in the setting of lymphopenia (<0.91 × 10<sup>9</sup>/L) in patients treated with DMF. We present detailed clinical characteristics and outcomes of the 12 confirmed PML cases occurring in MS patients on DMF as of 21 July 2021. The PML incidence in DMF-treated patients is 1.07 per 100,000 person-years of DMF exposure. Lymphopenia is the common risk for PML in DMF treatment.</p><p><strong>Discussion: </strong>DMF-related PML is rare but has occurred in the setting of lymphopenia, supporting the current recommendations for absolute lymphocyte count monitoring in all patients, regardless of age and time on therapy.</p>","PeriodicalId":18961,"journal":{"name":"Multiple Sclerosis Journal - Experimental, Translational and Clinical","volume":"8 4","pages":"20552173221132469"},"PeriodicalIF":2.8,"publicationDate":"2022-10-01","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"https://ftp.ncbi.nlm.nih.gov/pub/pmc/oa_pdf/34/c5/10.1177_20552173221132469.PMC9661630.pdf","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"10829756","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}
Pub Date : 2022-09-07eCollection Date: 2022-07-01DOI: 10.1177/20552173221117784
Alessandra Spanu, Hélène E Aschmann, Jürg Kesselring, Milo A Puhan
Background: Fingolimod is a disease-modifying drug approved for multiple sclerosis but its benefit-harm balance has never been assessed compared to other active treatments.
Objectives: Our aim was to compare the benefits and harms of fingolimod with interferon beta-1a using individual patient data from TRial Assessing injectable interferon versus FTY720 Oral in RRMS trial.
Methods: We modelled the health status of patients over time including Expanded Disability Status Scale measurements, relapses and any adverse events. We assessed the mean health status between arms and the proportion of patients whose health deteriorated or improved relatively to baseline, using a prespecified minimal important difference of 4.6. We performed sensitivity analyses to test our assumptions.
Results: Main and sensitivity analyses favoured fingolimod 0.5 mg over interferon beta-1a. The average health status difference was 1.01 (95% CI 0.93-1.08). Patients on fingolimod 0.5 mg were 0.47 (95% CI: 0.35-0.63, p < 0.001) times less likely to experience a relevant decline in health status compared to interferon beta-1a patients, with a number needed to treat of 7.10 [5.18, 11.23].
Conclusions: Fingolimod's net benefit over interferon beta-1a did not reach the clinical relevance over 1 year, but the decreased risk for health status deterioration may be more pronounced more long term and patients may prefer less treatment burden associated with fingolimod.
{"title":"Fingolimod versus interferon beta 1-a: Benefit-harm assessment approach based on TRANSFORMS individual patient data.","authors":"Alessandra Spanu, Hélène E Aschmann, Jürg Kesselring, Milo A Puhan","doi":"10.1177/20552173221117784","DOIUrl":"https://doi.org/10.1177/20552173221117784","url":null,"abstract":"<p><strong>Background: </strong>Fingolimod is a disease-modifying drug approved for multiple sclerosis but its benefit-harm balance has never been assessed compared to other active treatments.</p><p><strong>Objectives: </strong>Our aim was to compare the benefits and harms of fingolimod with interferon beta-1a using individual patient data from TRial Assessing injectable interferon versus FTY720 Oral in RRMS trial.</p><p><strong>Methods: </strong>We modelled the health status of patients over time including Expanded Disability Status Scale measurements, relapses and any adverse events. We assessed the mean health status between arms and the proportion of patients whose health deteriorated or improved relatively to baseline, using a prespecified minimal important difference of 4.6. We performed sensitivity analyses to test our assumptions.</p><p><strong>Results: </strong>Main and sensitivity analyses favoured fingolimod 0.5 mg over interferon beta-1a. The average health status difference was 1.01 (95% CI 0.93-1.08). Patients on fingolimod 0.5 mg were 0.47 (95% CI: 0.35-0.63, <i>p</i> < 0.001) times less likely to experience a relevant decline in health status compared to interferon beta-1a patients, with a number needed to treat of 7.10 [5.18, 11.23].</p><p><strong>Conclusions: </strong>Fingolimod's net benefit over interferon beta-1a did not reach the clinical relevance over 1 year, but the decreased risk for health status deterioration may be more pronounced more long term and patients may prefer less treatment burden associated with fingolimod.</p>","PeriodicalId":18961,"journal":{"name":"Multiple Sclerosis Journal - Experimental, Translational and Clinical","volume":"8 3","pages":"20552173221117784"},"PeriodicalIF":2.8,"publicationDate":"2022-09-07","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"https://www.ncbi.nlm.nih.gov/pmc/articles/PMC9459487/pdf/","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"33461572","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}
Pub Date : 2022-09-04eCollection Date: 2022-07-01DOI: 10.1177/20552173221116282
Eea van Egmond, K van der Hiele, Dam van Gorp, P J Jongen, Jjl van der Klink, M F Reneman, Eac Beenakker, Jjj van Eijk, Stfm Frequin, K de Gans, B M van Geel, Ohh Gerlach, Gjd Hengstman, J P Mostert, Wim Verhagen, Ham Middelkoop, L H Visser
Background: Symptoms of anxiety and depression affect the daily life of people with multiple sclerosis (MS). This study examined work difficulties and their relationship with anxiety, depression and coping style in people with MS.
Methods: 219 employed people with MS (median age = 43 years, 79% female) completed questionnaires on anxiety, depression, coping style, demographics and work difficulties, and underwent a neurological examination. Two regression analyses were performed with work difficulties as the dependent variable and either anxiety or depression as continuous independent variables. Coping style, age, gender, educational level, MS-related disability and disease duration were added as additional predictors, as well as interaction terms between coping style and either symptoms of depression or anxiety.
Results: A significant model was found (F(10,205) = 13.14, p < 0.001, R2 = 0.39) in which anxiety, emotion- and avoidance-oriented coping and MS-related disability were positively related to work difficulties. The analysis of depression resulted in a significant model (F(10,205) = 14.98, p < 0.001, R2 = 0.42) in which depression, emotion- and avoidance-oriented coping and MS-related disability were positively related to work difficulties. None of the interaction effects were significant.
Conclusions: Work difficulties were positively related to anxiety, depression, emotion- and avoidance-oriented coping and MS-related disability in workers with MS.
背景:焦虑和抑郁症状影响多发性硬化症(MS)患者的日常生活。方法:219名在职多发性硬化症患者(中位年龄43岁，女性79%)完成了焦虑、抑郁、应对方式、人口统计学和工作困难问卷调查，并进行了神经学检查。以工作困难为因变量，焦虑或抑郁为连续自变量，进行两次回归分析。应对方式、年龄、性别、教育水平、ms相关残疾和疾病持续时间被添加为额外的预测因素，以及应对方式与抑郁或焦虑症状之间的相互作用项。结果:存在显著模型(F (10,205) = 13.14, p r2 = 0.39)，焦虑、情绪和回避型应对和ms相关残疾与工作困难呈正相关。对抑郁的分析得出显著模型(F (10,205) = 14.98, p r2 = 0.42)，抑郁、情绪和回避型应对和ms相关残疾与工作困难呈正相关。交互作用均不显著。结论:工作困难与多发性硬化症患者的焦虑、抑郁、情绪和回避型应对以及多发性硬化症相关残疾呈正相关。
{"title":"Work difficulties in people with multiple sclerosis: The role of anxiety, depression and coping.","authors":"Eea van Egmond, K van der Hiele, Dam van Gorp, P J Jongen, Jjl van der Klink, M F Reneman, Eac Beenakker, Jjj van Eijk, Stfm Frequin, K de Gans, B M van Geel, Ohh Gerlach, Gjd Hengstman, J P Mostert, Wim Verhagen, Ham Middelkoop, L H Visser","doi":"10.1177/20552173221116282","DOIUrl":"https://doi.org/10.1177/20552173221116282","url":null,"abstract":"<p><strong>Background: </strong>Symptoms of anxiety and depression affect the daily life of people with multiple sclerosis (MS). This study examined work difficulties and their relationship with anxiety, depression and coping style in people with MS.</p><p><strong>Methods: </strong>219 employed people with MS (median age = 43 years, 79% female) completed questionnaires on anxiety, depression, coping style, demographics and work difficulties, and underwent a neurological examination. Two regression analyses were performed with work difficulties as the dependent variable and either anxiety or depression as continuous independent variables. Coping style, age, gender, educational level, MS-related disability and disease duration were added as additional predictors, as well as interaction terms between coping style and either symptoms of depression or anxiety.</p><p><strong>Results: </strong>A significant model was found (<i>F</i> <sub>(10,205)</sub> = 13.14, <i>p</i> < 0.001, <i>R</i> <sup>2</sup> = 0.39) in which anxiety, emotion- and avoidance-oriented coping and MS-related disability were positively related to work difficulties. The analysis of depression resulted in a significant model (<i>F</i> <sub>(10,205)</sub> = 14.98, <i>p</i> < 0.001, <i>R</i> <sup>2</sup> = 0.42) in which depression, emotion- and avoidance-oriented coping and MS-related disability were positively related to work difficulties. None of the interaction effects were significant.</p><p><strong>Conclusions: </strong>Work difficulties were positively related to anxiety, depression, emotion- and avoidance-oriented coping and MS-related disability in workers with MS.</p>","PeriodicalId":18961,"journal":{"name":"Multiple Sclerosis Journal - Experimental, Translational and Clinical","volume":"8 3","pages":"20552173221116282"},"PeriodicalIF":2.8,"publicationDate":"2022-09-04","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"https://www.ncbi.nlm.nih.gov/pmc/articles/PMC9445483/pdf/","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"33454050","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}
Pub Date : 2022-04-01DOI: 10.1177/20552173221091475
Benjamin M. Greenberg, J. D. Bowen, E. Alvarez, Moses Rodriguez, A. Caggiano, A. Warrington, Ping Zhao, A. Eisen
Background Recombinant human immunoglobulin M22 (rHIgM22) has promoted remyelination in animal models and was well tolerated in people with clinically stable multiple sclerosis. Objective Safety/tolerability of a single rHIgM22 dose was investigated following an acute relapse and to determine whether this enhanced CNS/CSF concentrations. Methods Adults (N = 27) with acute relapse were assigned to rHIgM22 (0.5 or 2.0 mg/kg) or placebo. Study included screening/steroid administration periods and 10 study visits over 6 months. rHIgM22 CSF concentrations were assessed on days 2 and 29. Pharmacokinetic and safety samples were taken for up to 60 days. Assessments included adverse events and other clinical measures. Brain magnetic resonance imaging was performed with/without gadolinium. Results rHIgM22 CSF levels were consistent with dose-dependent concentration on both days 2 and 29. Infusion was generally well tolerated during an acute relapse. Immunogenicity was mild. Most adverse events did not appear to be dose dependent, were mild/moderate, and were events often associated with multiple sclerosis. Conclusion Although limited by high variability and small sample size, the data suggest enhanced CNS uptake associated with a drop in CSF levels. This study demonstrated safety of an antibody directed to myelin and oligodendrocytes in the course of active demyelinating disease. Further research into rHIgM22 is warranted. ClinicalTrials.gov: NCT02398461 https://clinicaltrials.gov/ct2/show/NCT02398461
{"title":"A double-blind, placebo-controlled, single-ascending-dose intravenous infusion study of rHIgM22 in subjects with multiple sclerosis immediately following a relapse","authors":"Benjamin M. Greenberg, J. D. Bowen, E. Alvarez, Moses Rodriguez, A. Caggiano, A. Warrington, Ping Zhao, A. Eisen","doi":"10.1177/20552173221091475","DOIUrl":"https://doi.org/10.1177/20552173221091475","url":null,"abstract":"Background Recombinant human immunoglobulin M22 (rHIgM22) has promoted remyelination in animal models and was well tolerated in people with clinically stable multiple sclerosis. Objective Safety/tolerability of a single rHIgM22 dose was investigated following an acute relapse and to determine whether this enhanced CNS/CSF concentrations. Methods Adults (N = 27) with acute relapse were assigned to rHIgM22 (0.5 or 2.0 mg/kg) or placebo. Study included screening/steroid administration periods and 10 study visits over 6 months. rHIgM22 CSF concentrations were assessed on days 2 and 29. Pharmacokinetic and safety samples were taken for up to 60 days. Assessments included adverse events and other clinical measures. Brain magnetic resonance imaging was performed with/without gadolinium. Results rHIgM22 CSF levels were consistent with dose-dependent concentration on both days 2 and 29. Infusion was generally well tolerated during an acute relapse. Immunogenicity was mild. Most adverse events did not appear to be dose dependent, were mild/moderate, and were events often associated with multiple sclerosis. Conclusion Although limited by high variability and small sample size, the data suggest enhanced CNS uptake associated with a drop in CSF levels. This study demonstrated safety of an antibody directed to myelin and oligodendrocytes in the course of active demyelinating disease. Further research into rHIgM22 is warranted. ClinicalTrials.gov: NCT02398461 https://clinicaltrials.gov/ct2/show/NCT02398461","PeriodicalId":18961,"journal":{"name":"Multiple Sclerosis Journal - Experimental, Translational and Clinical","volume":" ","pages":""},"PeriodicalIF":2.8,"publicationDate":"2022-04-01","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"46747270","PeriodicalName":null,"FirstCategoryId":null,"ListUrlMain":null,"RegionNum":0,"RegionCategory":"","ArticlePicture":[],"TitleCN":null,"AbstractTextCN":null,"PMCID":"","EPubDate":null,"PubModel":null,"JCR":null,"JCRName":null,"Score":null,"Total":0}
Pub Date : 2022-04-01DOI: 10.1177/20552173221103436
Marc Hilty, Pietro Oldrati, Liliana Barrios, T. Müller, Claudia Blumer, M. Foege, Phrt consortium, Christian Holz, A. Lutterotti
Background Dysfunction of the autonomic nervous system is common in multiple sclerosis patients, and probably present years before diagnosis, but its role in the disease is poorly understood. Objectives To study the autonomic nervous system in patients with multiple sclerosis using cardiac autonomic regulation measured with a wearable. Methods In a two-week study, we present a method to standardize the measurement of heart rate variability using a wearable sensor that allows the investigation of circadian trends. Using this method, we investigate the relationship of cardiac autonomic dysfunction with clinical hallmarks and subjective burden of fatigue and autonomic symptoms. Results In 55 patients with multiple sclerosis and 24 healthy age- and gender-matched controls, we assessed the cumulative circadian heart-rate variability trend of two weeks. The trend analysis revealed an effect of inflammation (P = 0.0490, SMD = -0.5466) and progressive neurodegeneration (P = 0.0016, SMD = 1.1491) on cardiac autonomic function. No association with subjective symptoms could be found. Conclusions Trend-based heart rate variability measured with a wearable provides the opportunity for unobtrusive long-term assessment of autonomic functions in patients with multiple sclerosis. It revealed a general dysregulation in patients with multiple sclerosis.
{"title":"Continuous monitoring with wearables in multiple sclerosis reveals an association of cardiac autonomic dysfunction with disease severity","authors":"Marc Hilty, Pietro Oldrati, Liliana Barrios, T. Müller, Claudia Blumer, M. Foege, Phrt consortium, Christian Holz, A. Lutterotti","doi":"10.1177/20552173221103436","DOIUrl":"https://doi.org/10.1177/20552173221103436","url":null,"abstract":"Background Dysfunction of the autonomic nervous system is common in multiple sclerosis patients, and probably present years before diagnosis, but its role in the disease is poorly understood. Objectives To study the autonomic nervous system in patients with multiple sclerosis using cardiac autonomic regulation measured with a wearable. Methods In a two-week study, we present a method to standardize the measurement of heart rate variability using a wearable sensor that allows the investigation of circadian trends. Using this method, we investigate the relationship of cardiac autonomic dysfunction with clinical hallmarks and subjective burden of fatigue and autonomic symptoms. Results In 55 patients with multiple sclerosis and 24 healthy age- and gender-matched controls, we assessed the cumulative circadian heart-rate variability trend of two weeks. The trend analysis revealed an effect of inflammation (P = 0.0490, SMD = -0.5466) and progressive neurodegeneration (P = 0.0016, SMD = 1.1491) on cardiac autonomic function. No association with subjective symptoms could be found. Conclusions Trend-based heart rate variability measured with a wearable provides the opportunity for unobtrusive long-term assessment of autonomic functions in patients with multiple sclerosis. It revealed a general dysregulation in patients with multiple sclerosis.","PeriodicalId":18961,"journal":{"name":"Multiple Sclerosis Journal - Experimental, Translational and Clinical","volume":" ","pages":""},"PeriodicalIF":2.8,"publicationDate":"2022-04-01","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"47771762","PeriodicalName":null,"FirstCategoryId":null,"ListUrlMain":null,"RegionNum":0,"RegionCategory":"","ArticlePicture":[],"TitleCN":null,"AbstractTextCN":null,"PMCID":"","EPubDate":null,"PubModel":null,"JCR":null,"JCRName":null,"Score":null,"Total":0}
Pub Date : 2022-04-01DOI: 10.1177/20552173211058862
M. Huang, L. Doyle, A. Burnham, D. Fry, K. Shea
Background Inspiratory muscle training (IMT) using a threshold device improves inspiratory muscle strength. What factors influence the IMT outcome has not been examined. Objective To identify predictors of the positive outcome following IMT in persons with advanced multiple sclerosis (PwAMS). Methods Inclusion criteria were non-ambulatory PwAMS, Expanded Disability Status Scale (EDSS) ≥6.5, age >18 years, no acute medical conditions, current non-smokers, and ability to consent. Participants (n = 38) performed daily inspiratory exercises using a resistive threshold device for 10 weeks. Baseline measurements included age, sex, body mass index, year post multiple sclerosis diagnosis, comorbidities, EDSS, Modified Fatigue Impact Scale-5, and oral Symbol Digit Modality Test. The percentage of completed prescribed exercise trials (Trials%) during the 10-week intervention was calculated. Age- and sex-adjusted predicted values of maximum inspiratory pressure (MIP%pred) and maximum expiratory pressure (MEP%pred) were obtained before and after the 10-week intervention. Backward multivariable regression analyses for the primary outcome (MIP%pred) were conducted. Results After controlling for the initial MIP%pred, perceived fatigue at the baseline and Trial% were significant and independent predictors of MIP%pred after IMT. Conclusion Less fatigue at the baseline and higher adherence to the prescribed exercise repetitions were positive predictors of the positive outcome following IMT in PwAMS.
{"title":"Predictors of positive outcomes following resistive inspiratory muscle training in non-ambulatory persons with advanced multiple sclerosis","authors":"M. Huang, L. Doyle, A. Burnham, D. Fry, K. Shea","doi":"10.1177/20552173211058862","DOIUrl":"https://doi.org/10.1177/20552173211058862","url":null,"abstract":"Background Inspiratory muscle training (IMT) using a threshold device improves inspiratory muscle strength. What factors influence the IMT outcome has not been examined. Objective To identify predictors of the positive outcome following IMT in persons with advanced multiple sclerosis (PwAMS). Methods Inclusion criteria were non-ambulatory PwAMS, Expanded Disability Status Scale (EDSS) ≥6.5, age >18 years, no acute medical conditions, current non-smokers, and ability to consent. Participants (n = 38) performed daily inspiratory exercises using a resistive threshold device for 10 weeks. Baseline measurements included age, sex, body mass index, year post multiple sclerosis diagnosis, comorbidities, EDSS, Modified Fatigue Impact Scale-5, and oral Symbol Digit Modality Test. The percentage of completed prescribed exercise trials (Trials%) during the 10-week intervention was calculated. Age- and sex-adjusted predicted values of maximum inspiratory pressure (MIP%pred) and maximum expiratory pressure (MEP%pred) were obtained before and after the 10-week intervention. Backward multivariable regression analyses for the primary outcome (MIP%pred) were conducted. Results After controlling for the initial MIP%pred, perceived fatigue at the baseline and Trial% were significant and independent predictors of MIP%pred after IMT. Conclusion Less fatigue at the baseline and higher adherence to the prescribed exercise repetitions were positive predictors of the positive outcome following IMT in PwAMS.","PeriodicalId":18961,"journal":{"name":"Multiple Sclerosis Journal - Experimental, Translational and Clinical","volume":" ","pages":""},"PeriodicalIF":2.8,"publicationDate":"2022-04-01","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"47320141","PeriodicalName":null,"FirstCategoryId":null,"ListUrlMain":null,"RegionNum":0,"RegionCategory":"","ArticlePicture":[],"TitleCN":null,"AbstractTextCN":null,"PMCID":"","EPubDate":null,"PubModel":null,"JCR":null,"JCRName":null,"Score":null,"Total":0}
Pub Date : 2022-04-01DOI: 10.1177/20552173221086662
K. Al, Laura J. Craven, Shaeley Gibbons, S. Parvathy, Ana Christina Wing, Chantelle Graf, Kate A. Parham, S. Kerfoot, Hannah Wilcox, J. Burton, M. Kremenchutzky, S. Morrow, C. Casserly, J. Meddings, Manas Sharma, M. Silverman
Background Patients with MS have an altered gut microbiota compared to healthy individuals, as well as elevated small intestinal permeability, which may be contributing to the development and progression of the disease. Objective We sought to investigate if fecal microbiota transplantation was safe and tolerable in MS patients and if it could improve abnormal intestinal permeability. Methods Nine patients with MS were recruited and provided monthly FMTs for up to six months. The primary outcome investigated was change in peripheral blood cytokine concentrations. The secondary outcomes were gut microbiota composition, intestinal permeability, and safety (assessed with EDSS and MRI). Results The study was terminated early and was subsequently underpowered to assess whether peripheral blood cytokines were altered following FMTs. FMTs were safe in this group of patients. Two of five patients had elevated small intestinal permeability at baseline that improved to normal values following FMTs. Significant, donor-specific, beneficial alterations to the MS patient gut microbiota were observed following FMT. Conclusion FMT was safe and tolerable in this cohort of RRMS patients, may improve elevated small intestinal permeability, and has the potential to enrich for an MS-protective microbiota. Further studies with longer follow-up and larger sample sizes are required to determine if FMT is a suitable therapy for MS.
{"title":"Fecal microbiota transplantation is safe and tolerable in patients with multiple sclerosis: A pilot randomized controlled trial","authors":"K. Al, Laura J. Craven, Shaeley Gibbons, S. Parvathy, Ana Christina Wing, Chantelle Graf, Kate A. Parham, S. Kerfoot, Hannah Wilcox, J. Burton, M. Kremenchutzky, S. Morrow, C. Casserly, J. Meddings, Manas Sharma, M. Silverman","doi":"10.1177/20552173221086662","DOIUrl":"https://doi.org/10.1177/20552173221086662","url":null,"abstract":"Background Patients with MS have an altered gut microbiota compared to healthy individuals, as well as elevated small intestinal permeability, which may be contributing to the development and progression of the disease. Objective We sought to investigate if fecal microbiota transplantation was safe and tolerable in MS patients and if it could improve abnormal intestinal permeability. Methods Nine patients with MS were recruited and provided monthly FMTs for up to six months. The primary outcome investigated was change in peripheral blood cytokine concentrations. The secondary outcomes were gut microbiota composition, intestinal permeability, and safety (assessed with EDSS and MRI). Results The study was terminated early and was subsequently underpowered to assess whether peripheral blood cytokines were altered following FMTs. FMTs were safe in this group of patients. Two of five patients had elevated small intestinal permeability at baseline that improved to normal values following FMTs. Significant, donor-specific, beneficial alterations to the MS patient gut microbiota were observed following FMT. Conclusion FMT was safe and tolerable in this cohort of RRMS patients, may improve elevated small intestinal permeability, and has the potential to enrich for an MS-protective microbiota. Further studies with longer follow-up and larger sample sizes are required to determine if FMT is a suitable therapy for MS.","PeriodicalId":18961,"journal":{"name":"Multiple Sclerosis Journal - Experimental, Translational and Clinical","volume":" ","pages":""},"PeriodicalIF":2.8,"publicationDate":"2022-04-01","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"45607029","PeriodicalName":null,"FirstCategoryId":null,"ListUrlMain":null,"RegionNum":0,"RegionCategory":"","ArticlePicture":[],"TitleCN":null,"AbstractTextCN":null,"PMCID":"","EPubDate":null,"PubModel":null,"JCR":null,"JCRName":null,"Score":null,"Total":0}
Pub Date : 2022-04-01DOI: 10.1177/20552173221104009
T. Titcomb, Wei Bao, Yang Du, Buyun Liu, L. Snetselaar, T. Wahls
Background Multiple sclerosis (MS) has been associated with increased mortality ratios, but few studies have investigated the independent association of MS with mortality. Objective To examine the prospective association of MS with risk of mortality in a nationally representative sample of U.S. adults. Methods This prospective study included 23,053 adults aged 45–79 years who participated in the National Health Interview Survey in 2002 and 2008. Physician-diagnosed MS was reported by participants during household interviews. These participants were linked to death records from survey date through December 31, 2015. Results Among the 23,053 participants included in this study, 120 reported a physician’s diagnosis of MS, with a higher prevalence in females (0.85%) than in males (0.31%). During on average 9.4 years (maximum 13.8 years) of observation, 4208 deaths occurred. After adjustment for age, sex, race/ethnicity, socioeconomic factors, lifestyle factors, and BMI, participants with MS had an 80% higher risk of mortality (HR 1.80; 95% CI, 1.11–2.92), compared with those without MS. The association remained significant (HR 1.75; 95% CI, 1.07–2.87) after further adjustment for baseline diabetes, cardiovascular disease, chronic lung disease, and cancer. Conclusion In this nationally representative sample of U.S. adults, MS was associated with an increased risk of mortality.
{"title":"Association of multiple sclerosis with risk of mortality among a nationally representative sample of adults in the United States","authors":"T. Titcomb, Wei Bao, Yang Du, Buyun Liu, L. Snetselaar, T. Wahls","doi":"10.1177/20552173221104009","DOIUrl":"https://doi.org/10.1177/20552173221104009","url":null,"abstract":"Background Multiple sclerosis (MS) has been associated with increased mortality ratios, but few studies have investigated the independent association of MS with mortality. Objective To examine the prospective association of MS with risk of mortality in a nationally representative sample of U.S. adults. Methods This prospective study included 23,053 adults aged 45–79 years who participated in the National Health Interview Survey in 2002 and 2008. Physician-diagnosed MS was reported by participants during household interviews. These participants were linked to death records from survey date through December 31, 2015. Results Among the 23,053 participants included in this study, 120 reported a physician’s diagnosis of MS, with a higher prevalence in females (0.85%) than in males (0.31%). During on average 9.4 years (maximum 13.8 years) of observation, 4208 deaths occurred. After adjustment for age, sex, race/ethnicity, socioeconomic factors, lifestyle factors, and BMI, participants with MS had an 80% higher risk of mortality (HR 1.80; 95% CI, 1.11–2.92), compared with those without MS. The association remained significant (HR 1.75; 95% CI, 1.07–2.87) after further adjustment for baseline diabetes, cardiovascular disease, chronic lung disease, and cancer. Conclusion In this nationally representative sample of U.S. adults, MS was associated with an increased risk of mortality.","PeriodicalId":18961,"journal":{"name":"Multiple Sclerosis Journal - Experimental, Translational and Clinical","volume":" ","pages":""},"PeriodicalIF":2.8,"publicationDate":"2022-04-01","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"43615168","PeriodicalName":null,"FirstCategoryId":null,"ListUrlMain":null,"RegionNum":0,"RegionCategory":"","ArticlePicture":[],"TitleCN":null,"AbstractTextCN":null,"PMCID":"","EPubDate":null,"PubModel":null,"JCR":null,"JCRName":null,"Score":null,"Total":0}
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