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Julia Sabin,1 Elisa Salas,2 Jesús Martín-Martínez,3 Antonio Candeliere-Merlicco,4 Francisco Javier Barrero Hernández,5 Ana María Alonso Torres,6 José Luis Sánchez-Menoyo,7 Laura Borrega,8 María Rodríguez-Rodríguez,9 Montserrat Gómez-Gutiérrez,10 Sara Eichau,11 Miguel Ángel Hernández-Pérez,12 Carmen Calles,13 Eva Fernandez-Diaz,14 Olga Carmona,15 Aida Orviz,16 Ana López-Real,17 Pablo López-Muñoz,18 Amelia Mendoza Rodríguez,19 Eduardo Aguera-Morales,20 Jorge Maurino2 1Department of Neurology, Hospital Universitario Puerta de Hierro, Madrid, Spain; 2Medical Department, Roche Farma, Madrid, Spain; 3Department of Neurology, Hospital Universitario Miguel Servet, Zaragoza, Spain; 4Department of Neurology, Hospital Rafael Méndez, Lorca, Spain; 5Department of Neurology, Hospital Clínico Universitario San Cecilio, Granada, Spain; 6Department of Neurology, Hospital Regional Universitario de Málaga, Málaga, Spain; 7Department of Neurology, Hospital de Galdakao-Usansolo, Galdakao, Spain; 8Department of Neurology, Hospital Universitario Fundación Alcorcón, Alcorcón, Spain; 9Department of Neurology, Hospital Universitario Lucus Augusti, Lugo, Spain; 10Department of Neurology, Hospital San Pedro de Alcántara, Cáceres, Spain; 11Department of Neurology, Hospital Universitario Virgen Macarena, Sevilla, Spain; 12Department of Neurology, Hospital Universitario Nuestra Señora de Candelaria, Tenerife, Spain; 13Department of Neurology, Hospital Universitario Son Espases, Palma de Mallorca, Spain; 14Department of Neurology, Hospital Universitario de Albacete, Albacete, Spain; 15Department of Neurology, Fundació Salut Empordà, Figueres, Spain; 16Department of Neurology, Hospital Universitario Fundación Jiménez Díaz, Madrid, Spain; 17Department of Neurology, Complejo Hospitalario Universitario A Coruña, A Coruña, Spain; 18Department of Neurology, Hospital Arnau de Vilanova, Llíria, Spain; 19Department of Neurology, Complejo Asistencial de Segovia, Segovia, Spain; 20Department of Neurology, Hospital Universitario Reina Sofía, Córdoba, SpainCorrespondence: Jorge Maurino, Ribera del Loira 50, Madrid, 28042, Spain, Tel + 34 913 24 81 00, Email jorge.maurino@roche.comPurpose: Shared decision-making is critical in multiple sclerosis (MS) due to the uncertainty of the disease trajectory over time and the large number of treatment options with differing efficacy, safety and administration characteristics. The aim of this study was to assess patients’ decisional conflict regarding the choice of a disease-modifying therapy and its associated factors in patients with mid-stage relapsing-remitting multiple sclerosis (RRMS).Methods: A multicenter, non-interventional study was conducted. Adult patients with a diagnosis of RRMS (2017 revised McDonald criteria) and disease duration of 3 to 8 years were included. The level of uncertainty experienced by a patient when faced with making a treatment choice was assessed using the 4-item Decisional Conflict Scale. A battery of patient-reported and clinician-rated measures was administered to obtain information on symptom severity, illness perception, illness-related uncertainty, regret, MS knowledge, risk taking behavior, preferred role in the decision-making process, cognition, and self-management. Patients were recruited during routine follow-up visits and completed all questionnaires online using electronic tablets at the hospital. A multivariate logistic regression analysis was conducted.Results: A total of 201 patients were studied. Mean age (Standard deviation) was 38.7 (8.4) years and 74.1% were female. Median disease duration (Interquartile range) was 6.0 (4.0– 7.0) years. Median EDSS score was 1.0 (0– 2.0). Sixty-seven (33.3%) patients reported a decisional conflict. These patients had lower MS knowledge and more illness uncertainty, anxiety, depressive symptoms, fatigue, subjective symptom severity, a threatening illness perception, and poorer quality of life than their counterparts. Lack of decisional conflict was associated with MS knowledge (Odds ratio [OR]=1.195, 95% CI 1.045, 1.383, p=0.013), self-management (OR=1.049, 95% CI 1.013, 1.093, p=0.018), and regret after a healthcare decision (OR=0.860, 95% CI 0.756, 0.973, p=0.018) in the multivariate analysis.Conclusion: Decisional conflict regarding the selection of a disease-modifying therapy was a common phenomenon in patients with mid-stage RRMS. Identifying factors associated with decisional conflict may be useful to implement preventive strategies that help patients better understand their condition and strengthen their self-management resources.Keywords: multiple sclerosis, disease-modifying therapies, decision-making, decisional conflict, disease-related knowledge, self-management

Susana Sainz de la Maza,1 Jorge Maurino,2 Tamara Castillo-Triviño,3 Mónica Borges,4 Berta Sebastián Torres,5 Javier Sotoca,6 Ana María Alonso Torres,7 Ana B Caminero,8 Laura Borrega,9 José L Sánchez-Menoyo,10 Francisco J Barrero-Hernández,11 Carmen Calles,12 Luis Brieva,13 María Rosario Blasco,14 Julio Dotor García-Soto,4 Ana Rodríguez-Regal,15 Laura Navarro-Cantó,16 Eduardo Agüera-Morales,17 Moisés Garcés,18 Olga Carmona,19 Laura Gabaldón-Torres,20 Lucía Forero,21 Mariona Hervás,22 Rocío Gómez-Ballesteros2 1Department of Neurology, Hospital Universitario Ramón y Cajal, Madrid, Spain; 2Medical Department, Roche Farma, Madrid, Spain; 3Department of Neurology, Hospital Universitario Donostia, San Sebastián, Spain; 4Department of Neurology, Hospital Universitario Virgen Macarena, Sevilla, Spain; 5Department of Neurology, Hospital Universitario Miguel Servet, Zaragoza, Spain; 6Department of Neurology, Hospital Universitari Vall d´Hebrón, Barcelona, Spain; 7Department of Neurology, Hospital Regional Universitario de Málaga, Málaga, Spain; 8Department of Neurology, Complejo Asistencial de Ávila, Ávila, Spain; 9Department of Neurology, Hospital Universitario Fundación Alcorcón, Alcorcón, Spain; 10Department of Neurology, Hospital de Galdakao-Usansolo, Galdakao, Spain; 11Department of Neurology, Hospital Universitario Clínico San Cecilio, Granada, Spain; 12Department of Neurology, Hospital Universitari Son Espases, Palma de Mallorca, Spain; 13Department of Neurology, Hospital Universitari Arnau de Vilanova, Lleida, Spain; 14Department of Neurology, Hospital Universitario Puerta de Hierro, Madrid, Spain; 15Department of Neurology, Complexo Hospitalario Universitario de Pontevedra, Pontevedra, Spain; 16Department of Neurology, Hospital General Universitario de Elche, Elche, Spain; 17Department of Neurology, Hospital Universitario Reina Sofía, Córdoba, Spain; 18Department of Neurology, Hospital Clínico Universitario Lozano Blesa, Zaragoza, Spain; 19Department of Neurology, Fundació Salut Empordà, Figueres, Spain; 20Department of Neurology, Hospital Francesc de Borja, Gandía, Spain; 21Department of Neurology, Hospital Universitario Puerta del Mar, Cádiz, Spain; 22Department of Neurology, Consorci Corporació Sanitària Parc Taulí, Sabadell, SpainCorrespondence: Jorge Maurino, Medical Department, Roche Farma, Ribera del Loira, 50, Madrid, 28042, Spain, Tel +34 913 24 81 00, Email jorge.maurino@roche.comBackground: Hopelessness is a risk factor for depression and suicide. There is little information on this phenomenon among patients with relapsing-remitting multiple sclerosis (RRMS), one of the most common causes of disability and loss of autonomy in young adults. The aim of this study was to assess state hopelessness and its associated factors in early-stage RRMS.Methods: A multicenter, non-interventional study was conducted. Adult patients with a diagnosis of RRMS, a disease duration ≤ 3 years, and an Expanded Disability Status Scale (EDSS) score of 0– 5.5 were included. The State-Trait Hopelessness Scale (STHS) was used to measure patients´ hopelessness. A battery of patient-reported and clinician-rated measurements was used to assess clinical status. A multivariate logistic regression analysis was conducted to determine the association between patients’ characteristics and state hopelessness.Results: A total of 189 patients were included. Mean age (standard deviation-SD) was 36.1 (9.4) years and 71.4% were female. Median disease duration (interquartile range-IQR) was 1.4 (0.7, 2.1) years. Symptom severity and disability were low with a median EDSS (IQR) score of 1.0 (0, 2.0). A proportion of 65.6% (n=124) of patients reported moderate-to-severe hopelessness. Hopelessness was associated with older age (p=0.035), depressive symptoms (p=< 0.001), a threatening illness perception (p=0.001), and psychological and cognitive barriers to workplace performance (p=0.029) in the multivariate analysis after adjustment for confounders.Conclusion: Hopelessness was a common phenomenon in early-stage RRMS, even in a population with low physical disability. Identifying factors associated with hopelessness may be critical for implementing preventive strategies helping patients to adapt to the new situation and cope with the disease in the long term.Keywords: relapsing-remitting multiple sclerosis, hopelessness, depressive symptoms, workplace difficulties, suicide

Jose M García-Domínguez,1 María L Martínez-Ginés,1 Olga Carmona,2 Ana B Caminero,3 Daniel Prefasi,4 Jorge Maurino,4 Javier Ballesteros5 On behalf of the W-IMPACT Clinical Investigators 1Department of Neurology, Hospital Universitario Gregorio Marañón, Madrid, Spain; 2Department of Neurology, Hospital de Figueres, Figueres, Spain; 3Department of Neurology, Hospital Nuestra Señora de Sonsoles, Complejo Asistencial de Ávila, Ávila, Spain; 4Medical Department, Roche Farma, Madrid, Spain; 5Department of Neurosciences and CIBERSAM, Universidad del País Vasco, Leioa, Spain Background: Understanding caregiver strain may be crucial to determine which interventions are most needed to mitigate the negative impact of caring for people with multiple sclerosis (MS). The Caregiver Strain Index (CSI) is a brief self-assessment tool for measuring the caregivers’ perceived level of burden. Limited information is available on the psychometric performance of the CSI in MS.Objective: The objective of this study was to assess the factor structure and construct validity of the CSI in MS.Methods: A multicenter, cross-sectional study in adults with relapsing-remitting and primary-progressive MS (McDonald 2010 criteria) was conducted. A non-parametric item response theory (IRT) procedure, Mokken analysis, was conducted to assess the dimensional structure of the CSI. A parametric IRT model for dichotomous responses, Rasch model, was conducted to assess item characteristics. Discriminative validity was assessed comparing the distribution of its overall score between people with mild and moderate-severe disability according to the Expanded Disability Status Scale.Results: A total of 72 MS caregivers were studied. The prevalence of a high level of strain was 23.6% (n=17). Internal reliability was high (Cronbach’s alpha =0.91). According to Mokken analysis, CSI represented a unidimensional construct of caregiver burden although two of the total 13 items (#1 and #13) could not be assigned to any factor by an automatic item selection procedure. Without these items, the scalability moved from a weak (Hi =0.37) to a medium scale (Hi =0.44). However, the item characteristic curve of the Rasch model showed a range of appropriate difficulty and the item and person parameters showed good fit (Andersen likelihood ratio test =18.40, df =11; P-value =0.07; all item values for the infit). The CSI score showed a good discriminative validity between the levels of disability of the care recipient.Conclusion: The CSI questionnaire shows appropriate psychometric characteristics being a useful instrument to assess different aspects of burden in MS caregivers in clinical practice. Keywords: caregivers, multiple sclerosis, psychometrics, caregiver burden, strain

Abstract Background Multiple sclerosis (MS) is an incurable chronic disease that predominantly affects young adults. It has a high socio-economic impact which increases as disability progresses. An assessment of the real costs of MS may contribute to our knowledge of the disease and to treat it more efficiently. Our objective is to assess the direct and indirect costs of MS from a societal perspective, in patients monitored in our MS Unit (Baix Llobregat, Catalonia) and grouped according to their disability (EDSS). Methods We analysed data from 200 MS patients, who answered a questionnaire on resource consumption, employment and economical status. Mean age was 41.6 years, mean EDSS 2.7, 65.5% of patients were female, 79.5% had a relapsing-remitting course, and 67.5% of them were receiving immunomodulatory treatment (IT). Patients were grouped into five EDSS stages. Data from the questionnaires, hospital charts, Catalan Health Service tariffs, and figures from Catalan Institute of Statistics were used to calculate the direct and indirect costs. The cost-of-illness method, and the human capital approach for indirect costs, were applied. Sensitivity analyses were performed to strengthen results. Results The mean total annual cost of MS per patient results 24272 euros. This cost varied according to EDSS: 14327 euros (EDSS = 0), 18837 euros (EDSS = 1–3), 27870 euros (EDSS = 3.5–5.5), 41198 euros (EDSS = 6–7) and 52841 euros (EDSS>7.5). When the mean total annual costs was adjusted by the mean % of patients on IT in our Unit (31%) the result was 19589 euros. The key-drivers for direct costs were IT in low EDSS stages, and caregiver costs in high stages. Indirect costs were assessed in terms of the loss of productivity when patients stop working. Direct costs accounted for around 60% of total costs in all EDSS groups. IT accounts from 78% to 11% of direct costs, and decreased as disability progressed. Conclusion The total mean social costs of MS in a cohort from Baix Llobregat (Catalonia) were estimated at 24272 euros per patient/year, and ranged between 14327 euros (EDSS = 0) and 52841 euros (EDSS = 7.5–9.5). Total costs, and particularly informal and direct costs, increased as the disability progressed. IT should be able to delay the progression of disability to be efficient and not only effective.