{"title":"Ocrelizumab-associated cryptogenic organizing pneumonia in multiple sclerosis: Two case reports and comprehensive literature review.","authors":"Alessandro Gans, Elena Pinuccia Verrengia, Elisabetta Ricchiuti, Serena Leva, Simona Brajkovic, Daniele Colombo, Antonino Mazzone, Alessandro Prelle","doi":"10.1177/13524585241295677","DOIUrl":"https://doi.org/10.1177/13524585241295677","url":null,"abstract":"<p><strong>Background: </strong>Cryptogenic organizing pneumonia (COP) is an interstitial lung disease, with causes including anti-CD20 antibodies. Ocrelizumab is a humanized monoclonal antibody against CD20 approved for use in relapsing-remitting or primary progressive multiple sclerosis (MS), with no conclusive data regarding pulmonary toxicity.</p><p><strong>Cases: </strong>We describe two cases of COP associated with ocrelizumab use in multiple sclerosis patients.</p><p><strong>Conclusions: </strong>We suggest considering COP whenever respiratory symptoms arise in MS patients receiving ocrelizumab therapy. COP diagnosis requires clinical and radiological exclusion of alternative diagnoses such as opportunistic infections, autoimmunity, drugs, and neoplasms. Appropriate steroid therapy has an excellent clinical response rate.</p>","PeriodicalId":18874,"journal":{"name":"Multiple Sclerosis Journal","volume":" ","pages":"13524585241295677"},"PeriodicalIF":4.8,"publicationDate":"2024-11-21","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"142681901","PeriodicalName":null,"FirstCategoryId":null,"ListUrlMain":null,"RegionNum":2,"RegionCategory":"医学","ArticlePicture":[],"TitleCN":null,"AbstractTextCN":null,"PMCID":"","EPubDate":null,"PubModel":null,"JCR":null,"JCRName":null,"Score":null,"Total":0}
Sonia Darvishi, Ewan Donnachie, Christiane Gasperi, Alexander Hapfelmeier, Bernhard Hemmer
{"title":"Vaccination as a risk factor for pediatric multiple sclerosis: Insights from a retrospective case-control study.","authors":"Sonia Darvishi, Ewan Donnachie, Christiane Gasperi, Alexander Hapfelmeier, Bernhard Hemmer","doi":"10.1177/13524585241297003","DOIUrl":"https://doi.org/10.1177/13524585241297003","url":null,"abstract":"<p><p>This study evaluated the association between pediatric multiple sclerosis and vaccinations within 5 years before diagnosis using German ambulatory claims data. Children with multiple sclerosis (<i>n</i> = 346) aged 9-17 were analyzed with logistic and Poisson regression. Control groups included children with Crohn's disease, psoriasis, and no autoimmune diseases. The results indicated a negative association between vaccinations and pediatric multiple sclerosis, with no significant risk identified. This negative relationship was consistent in sensitivity and clinically isolated syndrome analyses. Overall, the study's findings do not support the hypothesis that vaccination is a risk factor for pediatric multiple sclerosis.</p>","PeriodicalId":18874,"journal":{"name":"Multiple Sclerosis Journal","volume":" ","pages":"13524585241297003"},"PeriodicalIF":4.8,"publicationDate":"2024-11-14","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"142624291","PeriodicalName":null,"FirstCategoryId":null,"ListUrlMain":null,"RegionNum":2,"RegionCategory":"医学","ArticlePicture":[],"TitleCN":null,"AbstractTextCN":null,"PMCID":"","EPubDate":null,"PubModel":null,"JCR":null,"JCRName":null,"Score":null,"Total":0}
René Carvajal, Breogán Rodríguez-Acevedo, Lorena García-Vasco, Ana Zabalza, Helena Ariño, Luca Bollo, Noemí Cabello-Clotet, Joaquín Castilló, Álvaro Cobo-Calvo, Manuel Comabella, Anna Falcó-Roget, Ingrid Galán, Alexis García-Sarreón, Irene Gómez-Estévez, Galo Granados, Delon La Puma, Gloria Mato Chain, Luciana Midaglia, Asunción Nieto-García, Susana Otero-Romero, Agustín Pappolla, Marta Rodriguez, Irene Sansano, Jordi Río, Paula Tagliani, Carmen Tur, Ángela Vidal-Jordana, Andreu Vilaseca, Ana Villar, Jaume Sastre-Garriga, Celia Oreja-Guevara, Mar Tintoré, Xavier Montalban, Georgina Arrambide
{"title":"Secondary organising pneumonia associated to COVID-19 infection in patients with central nervous system inflammatory demyelinating diseases treated with anti-CD20 therapies.","authors":"René Carvajal, Breogán Rodríguez-Acevedo, Lorena García-Vasco, Ana Zabalza, Helena Ariño, Luca Bollo, Noemí Cabello-Clotet, Joaquín Castilló, Álvaro Cobo-Calvo, Manuel Comabella, Anna Falcó-Roget, Ingrid Galán, Alexis García-Sarreón, Irene Gómez-Estévez, Galo Granados, Delon La Puma, Gloria Mato Chain, Luciana Midaglia, Asunción Nieto-García, Susana Otero-Romero, Agustín Pappolla, Marta Rodriguez, Irene Sansano, Jordi Río, Paula Tagliani, Carmen Tur, Ángela Vidal-Jordana, Andreu Vilaseca, Ana Villar, Jaume Sastre-Garriga, Celia Oreja-Guevara, Mar Tintoré, Xavier Montalban, Georgina Arrambide","doi":"10.1177/13524585241297038","DOIUrl":"https://doi.org/10.1177/13524585241297038","url":null,"abstract":"<p><strong>Background: </strong>Organizing pneumonia (OP), an interstitial lung disease, has been observed in patients with inflammatory demyelinating diseases (IDDs) treated with anti-CD20, particularly after COVID-19, but data are limited.</p><p><strong>Aim: </strong>To provide a detailed characterization of COVID-19-associated OP in IDD patients treated with anti-CD20.</p><p><strong>Methods: </strong>Bi-centric retrospective cohort study including patients with multiple sclerosis (MS), aquaporin-4-positive neuromyelitis optica spectrum disorder (AQP4 + NMOSD), and myelin oligodendrocyte glycoprotein antibody disease (MOGAD) who received anti-CD20 and were diagnosed with COVID-19-associated OP between March 2020 and October 2023.</p><p><strong>Results: </strong>Nineteen patients were included (mean age 46.8 years; 52.6% female; 63% rituximab, 37% ocrelizumab). Sixteen had MS, two MOGAD, and one AQP4 + NMOSD. Intermittent fever was the predominant symptom. Hospitalization occurred in all but one patient, without fatalities. Chest CT consistently showed OP patterns. Thirteen patients had positive severe acute respiratory syndrome coronavirus 2 (SARS-CoV-2) PCR in bronchoalveolar lavage. Treatments included corticosteroids, antivirals, monoclonal antibodies, and convalescent plasma. Fourteen patients postponed infusions; nine resumed post-recovery (median 11.9 months), two switched due to hypogammaglobulinemia, and three stopped. After a mean follow-up of 1.5 years, lung abnormalities and clinical manifestations resolved in 18 patients; however, 13 experienced long-COVID.</p><p><strong>Conclusions: </strong>In anti-CD20-treated patients with recurrent fever and distinctive CT features, COVID-19-associated OP should be considered.</p>","PeriodicalId":18874,"journal":{"name":"Multiple Sclerosis Journal","volume":" ","pages":"13524585241297038"},"PeriodicalIF":4.8,"publicationDate":"2024-11-09","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"142624290","PeriodicalName":null,"FirstCategoryId":null,"ListUrlMain":null,"RegionNum":2,"RegionCategory":"医学","ArticlePicture":[],"TitleCN":null,"AbstractTextCN":null,"PMCID":"","EPubDate":null,"PubModel":null,"JCR":null,"JCRName":null,"Score":null,"Total":0}
{"title":"Emerging MRI biomarkers for the diagnosis of multiple sclerosis.","authors":"Pietro Maggi, Martina Absinta","doi":"10.1177/13524585241293579","DOIUrl":"https://doi.org/10.1177/13524585241293579","url":null,"abstract":"<p><p>The need to improve diagnostic precision in multiple sclerosis (MS) is widely recognized. In recent years, several novel magnetic resonance imaging (MRI) biomarkers have been proposed to enhance diagnostic specificity and reduce misdiagnosis. Some of these imaging biomarkers are deemed highly specific for MS and are likely ready to enter the MS diagnostic work-up, while others are still in their exploratory phase. In addition, new synthetic MRI contrasts and artificial intelligence-based diagnostic algorithms are being tested to reduce the time burden related to imaging data acquisition and analysis. In this review, we summarize the most recent advancement in the field, focusing on the adoption of these novel MRI biomarkers-whether used alone or in combination-for the differential diagnosis of MS.</p>","PeriodicalId":18874,"journal":{"name":"Multiple Sclerosis Journal","volume":" ","pages":"13524585241293579"},"PeriodicalIF":4.8,"publicationDate":"2024-11-07","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"142604740","PeriodicalName":null,"FirstCategoryId":null,"ListUrlMain":null,"RegionNum":2,"RegionCategory":"医学","ArticlePicture":[],"TitleCN":null,"AbstractTextCN":null,"PMCID":"","EPubDate":null,"PubModel":null,"JCR":null,"JCRName":null,"Score":null,"Total":0}
Anne Kever, Lauren B Heuer, Leila Simani, Victoria M Leavitt
{"title":"Development and initial validation of the Cognitive Change Scale (CCS).","authors":"Anne Kever, Lauren B Heuer, Leila Simani, Victoria M Leavitt","doi":"10.1177/13524585241290102","DOIUrl":"https://doi.org/10.1177/13524585241290102","url":null,"abstract":"<p><strong>Background: </strong>Cognitive impairment is common in neurologic diseases. Precise measurement of cognitive change over time is necessary for isolating disease-related patterns from normal age-related decline. Existing measures of subjective cognition, however, focus on present status. There is, to our knowledge, no currently available self-report measure of cognitive change. We therefore developed the Cognitive Change Scale (CCS), which assesses perceived cognitive change in neurologic populations.</p><p><strong>Methods: </strong>A systematic mixed-methods process was followed for the scale design and validation. Associations of CCS responses to demographics, mood, and fatigue were examined in 131 persons with multiple sclerosis. A total of 46 participants also completed a cognitive test battery. Correlations of test scores with CCS responses were calculated.</p><p><strong>Results: </strong>The 17-item CCS showed good reliability and validity. Results of exploratory and confirmatory factor analyses supported a four-factor structure, with items reflecting change in (1) general cognition, (2) language and executive function, (3) external feedback, and (4) use of coping strategies. Positive relationships of CCS scores with fatigue, depression, and anxiety were observed. Correlations of CCS scores with cognitive test performance did not reach significance.</p><p><strong>Conclusion: </strong>The CCS may be a useful cognitive outcome tool for treatment trials in neurologic populations.</p>","PeriodicalId":18874,"journal":{"name":"Multiple Sclerosis Journal","volume":" ","pages":"13524585241290102"},"PeriodicalIF":4.8,"publicationDate":"2024-11-07","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"142604736","PeriodicalName":null,"FirstCategoryId":null,"ListUrlMain":null,"RegionNum":2,"RegionCategory":"医学","ArticlePicture":[],"TitleCN":null,"AbstractTextCN":null,"PMCID":"","EPubDate":null,"PubModel":null,"JCR":null,"JCRName":null,"Score":null,"Total":0}
{"title":"Choroid plexus as a mediator of CNS inflammation in multiple sclerosis.","authors":"Alexandra Hochstetler, Maria K Lehtinen","doi":"10.1177/13524585241292974","DOIUrl":"https://doi.org/10.1177/13524585241292974","url":null,"abstract":"<p><p>The pathophysiology of multiple sclerosis (MS) remains poorly understood despite decades of tremendous research efforts. Advances in neuroradiography coupled with availability of unbiased approaches including spatial transcriptomics, proteomics, metabolomics, and lipidomics that are compatible with brain and fluid specimens from patients raise hope that discovery of novel disease drivers is on the horizon. Once thought to be little more than salty bathwater, our modern understanding of cerebrospinal fluid (CSF) suggests the CSF as a compelling, critical regulator of brain function in health and disease. Recent studies in the field have reinvigorated interest in CSF as a medium to better understand MS and to deliver disease-modifying therapies. In turn, the choroid plexus, an epithelial tissue located within each brain ventricle that regulates CSF and forms a key blood-CSF barrier, is uniquely positioned to orchestrate neuroinflammation associated with MS. In this perspective review, we will discuss what is known about the choroid plexus as a conductor of immune responses and how it may propagate neuroinflammation associated with MS via the CSF.</p>","PeriodicalId":18874,"journal":{"name":"Multiple Sclerosis Journal","volume":" ","pages":"13524585241292974"},"PeriodicalIF":4.8,"publicationDate":"2024-11-06","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"142583577","PeriodicalName":null,"FirstCategoryId":null,"ListUrlMain":null,"RegionNum":2,"RegionCategory":"医学","ArticlePicture":[],"TitleCN":null,"AbstractTextCN":null,"PMCID":"","EPubDate":null,"PubModel":null,"JCR":null,"JCRName":null,"Score":null,"Total":0}
Yuan Jiang, Carolyn E Cesta, Qianwen Liu, Elaine Kingwell, Pernilla Stridh, Klementy Shchetynsky, Tomas Olsson, Ingrid Kockum, Elisabet Stener-Victorin, Xia Jiang, Ali Manouchehrinia
{"title":"Exploring the relationship between polycystic ovarian syndrome, testosterone, and multiple sclerosis in women: A nationwide cohort study and genome-wide cross-trait analysis.","authors":"Yuan Jiang, Carolyn E Cesta, Qianwen Liu, Elaine Kingwell, Pernilla Stridh, Klementy Shchetynsky, Tomas Olsson, Ingrid Kockum, Elisabet Stener-Victorin, Xia Jiang, Ali Manouchehrinia","doi":"10.1177/13524585241292802","DOIUrl":"10.1177/13524585241292802","url":null,"abstract":"<p><strong>Background: </strong>Women have a higher risk of developing multiple sclerosis (MS), potentially due to hormonal factors. Elevated testosterone levels, common in polycystic ovary syndrome (PCOS), might influence MS risk.</p><p><strong>Objective: </strong>To investigate the relationship between PCOS, as a proxy for elevated testosterone levels, and MS risk through phenotypic and genomic analysis.</p><p><strong>Methods: </strong>Cox regression models analysed the association between PCOS and MS risk. The genome-wide cross-trait analysis examined the genetic architecture.</p><p><strong>Results: </strong>In a Swedish cohort of 1,374,529 women, 77 (0.3%) with PCOS and 3,654 (0.3%) without PCOS were diagnosed with MS. After adjusting for birth year and obesity, no association was found between PCOS and MS (<i>HR</i> = 0.91, 95% CI = 0.72-1.15), which was confirmed by Mendelian randomization analysis, where genetically predicted PCOS propensity, sex hormone-binding globulin (SHBG), or testosterone levels did not causally affect MS risk (all <i>p</i>-values > 0.05). By exploring horizontal pleiotropy, we identified shared genetic regions and 19 independent pleiotropic SNPs for SHBG with MS and 11 for testosterone with MS.</p><p><strong>Conclusion: </strong>We did not find evidence for a causal role of PCOS, as a proxy of elevated testosterone, in reducing the risk of MS in women. The shared genetic loci between testosterone, SHBG, and MS provide biological insights.</p>","PeriodicalId":18874,"journal":{"name":"Multiple Sclerosis Journal","volume":" ","pages":"13524585241292802"},"PeriodicalIF":4.8,"publicationDate":"2024-11-06","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"142583580","PeriodicalName":null,"FirstCategoryId":null,"ListUrlMain":null,"RegionNum":2,"RegionCategory":"医学","ArticlePicture":[],"TitleCN":null,"AbstractTextCN":null,"PMCID":"","EPubDate":null,"PubModel":null,"JCR":null,"JCRName":null,"Score":null,"Total":0}
{"title":"Impact of symptomatic multiple sclerosis therapy on pregnancy outcome.","authors":"Laura Witt, Sabrina Haben, Karen Dost-Kovalsky, Natalia Friedmann, Nadine Bast, Theresa Oganowski, Ralf Gold, Sandra Thiel, Kerstin Hellwig","doi":"10.1177/13524585241293363","DOIUrl":"10.1177/13524585241293363","url":null,"abstract":"<p><strong>Background: </strong>Information on symptomatic therapy (ST) use in women of childbearing age with multiple sclerosis is sparse, and data on the impact of ST pregnancy exposure on pregnancy outcome are lacking.</p><p><strong>Objective: </strong>To investigate (1) ST use patterns pre-conception, during pregnancy and postpartum and (2) pregnancy outcomes.</p><p><strong>Methods: </strong>Pregnancy data from the German Multiple Sclerosis and Pregnancy Registry were analyzed for the ST use from pre-conception to postpartum. Pregnancy outcomes were compared between ST-exposed (<i>n</i> = 282) and matched (disease modifying therapy and age) ST-naive (n = 536) pregnancies.</p><p><strong>Results: </strong>Of 2,449 pregnancies, 1,053 (43.0%) received ST anytime between pre-conception and postpartum, 282 (11.5%) at pre-conception and during pregnancy. The most commonly used drug classes were antidepressants (24.8%), analgetics (31.0%), and anticonvulsives (8.7%). Exposure to ST during pregnancy did not result in an increased incidence of adverse pregnancy outcomes, major congenital abnormalities, or pregnancy complications.</p><p><strong>Conclusion: </strong>Nearly 50% of women used ST between pre-conception and postpartum, but only 12% pre-conception and during pregnancy. ST use during pregnancy did not adversely affect pregnancy outcomes in our cohort. More data are needed to analyze the effect of ST on pregnancy and fetal outcomes stratified by drug to improve recommendations for ST use in family planning.</p>","PeriodicalId":18874,"journal":{"name":"Multiple Sclerosis Journal","volume":" ","pages":"13524585241293363"},"PeriodicalIF":4.8,"publicationDate":"2024-11-06","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"142583599","PeriodicalName":null,"FirstCategoryId":null,"ListUrlMain":null,"RegionNum":2,"RegionCategory":"医学","ArticlePicture":[],"TitleCN":null,"AbstractTextCN":null,"PMCID":"","EPubDate":null,"PubModel":null,"JCR":null,"JCRName":null,"Score":null,"Total":0}
E Matthew Hoffman, Lucille Brown, Evan Jolliffe, Elia Sechi, William S Harmsen, Nathan D Schilaty, Brian G Weinshenker
{"title":"McArdle sign and neck flexion-induced change in central motor conduction in multiple sclerosis.","authors":"E Matthew Hoffman, Lucille Brown, Evan Jolliffe, Elia Sechi, William S Harmsen, Nathan D Schilaty, Brian G Weinshenker","doi":"10.1177/13524585241295868","DOIUrl":"https://doi.org/10.1177/13524585241295868","url":null,"abstract":"<p><strong>Background: </strong>Rapidly reversible weakness with neck flexion (McArdle sign) is common in patients with multiple sclerosis (MS). The pathophysiology is unknown.</p><p><strong>Objective: </strong>To evaluate changes in central motor conduction time (CMCT) in patients with and without McArdle sign.</p><p><strong>Methods: </strong>We measured McArdle sign with a torque cell and CMCT with neck flexed and extended in patients with MS, other causes of myelopathy, and healthy controls.</p><p><strong>Results: </strong>CMCT was prolonged with neck flexion disproportionately in those with MS-associated myelopathy (MSAM) with prominent McArdle sign compared to MS patients with lesser degrees of McArdle sign, and to controls.</p><p><strong>Conclusion: </strong>McArdle sign may result from stretch-induced slowing of conduction due to demyelination.</p>","PeriodicalId":18874,"journal":{"name":"Multiple Sclerosis Journal","volume":" ","pages":"13524585241295868"},"PeriodicalIF":4.8,"publicationDate":"2024-11-06","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"142583613","PeriodicalName":null,"FirstCategoryId":null,"ListUrlMain":null,"RegionNum":2,"RegionCategory":"医学","ArticlePicture":[],"TitleCN":null,"AbstractTextCN":null,"PMCID":"","EPubDate":null,"PubModel":null,"JCR":null,"JCRName":null,"Score":null,"Total":0}