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Background: The Multiple Sclerosis Quality of Life-54 (MSQOL-54) is one of the most commonly-used MS-specific health-related quality of life (HRQOL) measures. It is a multidimensional, MS-specific HRQOL inventory, which includes the generic SF-36 core items, supplemented with 18 MS-targeted items. Availability of an adaptive short version providing immediate item scoring may improve instrument usability and validity. However, multidimensional computerized adaptive testing (MCAT) has not been previously applied to MSQOL-54 items. We thus aimed to apply MCAT to the MSQOL-54 and assess its performance., Methods: Responses from a large international sample of 3669 MS patients were assessed. We calibrated 52 (of the 54) items using bifactor graded response model (10 group factors and one general HRQOL factor). Then, eight simulations were run with different termination criteria: standard errors (SE) for the general factor and group factors set to different values, and change in factor estimates from one item to the next set at < 0.01 for both the general and the group factors. Performance of the MCAT was assessed by the number of administered items, root mean square difference (RMSD), and correlation., Results: Eight items were removed due to local dependency. The simulation with SE set to 0.32 (general factor), and no SE thresholds (group factors) provided satisfactory performance: the median number of administered items was 24, RMSD was 0.32, and correlation was 0.94., Conclusions: Compared to the full-length MSQOL-54, the simulated MCAT required fewer items without losing precision for the general HRQOL factor. Further work is needed to add/integrate/revise MSQOL-54 items in order to make the calibration and MCAT performance efficient also on group factors, so that the MCAT version may be used in clinical practice and research., (© 2023. The Author(s).)

Introduction: REsilience and Activities for every DaY (READY) is an Acceptance and Commitment Therapy-based group resilience-training program that has preliminary empirical support in promoting quality of life and other psychosocial outcomes in people with multiple sclerosis (PwMS). Consistent with the Medical Research Council framework for developing and evaluating complex interventions, we conducted a pilot randomized controlled trial (RCT), followed by a phase III RCT. The present paper describes the phase III RCT protocol., Methods and Analysis: This is a multi-centre cluster RCT comparing READY with a group relaxation program (1:1 ratio) in 240 PwMS from eight centres in Italy (trial registration: isrctn.org Identifier: ISRCTN67194859). Both interventions are composed of 7 weekly sessions plus a booster session five weeks later. Resilience (primary outcome), mood, health-related quality of life, well-being and psychological flexibility will be assessed at baseline, after the booster session, and at three and six month follow-ups. If face-to-face group meetings are interrupted because of COVID-19 related-issues, participants will be invited to complete their intervention via teleconferencing. Relevant COVID-19 information will be collected and the COVID-19 Peritraumatic Distress scale will be administered (ancillary study) at baseline and 3-month follow-up. Analysis will be by intention-to-treat to show superiority of READY over relaxation. Longitudinal changes will be compared between the two arms using repeated-measures, hierarchical generalized linear mixed models., Conclusion: It is expected that his study will contribute to the body of evidence on the efficacy and effectiveness of READY by comparing it with an active group intervention in frontline MS rehabilitation and clinical settings. Results will be disseminated in peer-reviewed journals and at other relevant conferences., Competing Interests: AMG reports a grant from Rehabilitation in Multiple Sclersis (RIMS) during the conduct of the study. AS reports grants from Fondazione Italiana Sclerosi Multipla (FISM), during the conduct of the study; personal fees from Biogen Idec, Merck Serono, Novartis, Almirall, and Excemed. As a staff member of the University of Queensland and co-Author of the READY program, KP receives royalties from UniQuest for commercial (not research) licensing arrangements entered into by third parties who want to deliver the program. PC has received honoraria for speaking or consultation fees from Novartis and Biogen, has received funding for travel to attend scientific events or speaker honoraria from Merck Serono, Biogen Idec, Teva and Roche. He has also received institutional research support from Merk-Serono, Novartis and Roche. He is also principal investigator in clinical trials for Biogen, Merck Serono, and Roche. This does not alter our adherence to PLOS ONE policies on sharing data. All the other authors reports no competing interests.

Background: MSQOL-54 is a multidimensional, widely-used, health-related quality of life (HRQOL) instrument specific for multiple sclerosis (MS). Findings from the validation study suggested that the two MSQOL-54 composite scores are correlated. Given this correlation, it could be assumed that a unique total score of HRQOL may be calculated, with the advantage to provide key stakeholders with a single overall HRQOL score. We aimed to assess how well the bifactor model could account for the MSQOL-54 structure, in order to verify whether a total HRQOL score can be calculated., Methods: A large international database (3669 MS patients) was used. By means of confirmatory factor analysis, we estimated a bifactor model in which every item loads onto both a general factor and a group factor. Fit of the bifactor model was compared to that of single and two second-order factor models by means of Akaike information and Bayesian information criteria reduction. Reliability of the total and subscale scores was evaluated with Mc Donald's coefficients (omega, and omega hierarchical)., Results: The bifactor model outperformed the two second-order factor models in all the statistics. All items loaded satisfactorily (≥ 0.40) on the general HRQOL factor, except the sexual function items. Omega coefficients for total score were very satisfactory (0.98 and 0.87). Omega hierarchical for subscales ranged between 0.22 to 0.57, except for the sexual function (0.70)., Conclusions: The bifactor model is particularly useful when it is intended to acknowledge multidimensionality and at the same time take account of a single general construct, as the HRQOL related to MS. The total raw score can be used as an estimate of the general HRQOL latent score., (© 2021. The Author(s).)

Psychological distress imposed by the SARS-CoV-2 outbreak particularly affects patients with pre-existing medical conditions, and the progression of their diseases. Patients who fail to keep scheduled medical appointments experience a negative impact on care. The aim of this study is to investigate the psychosocial factors contributing to the cancellation of medical appointments during the pandemic by patients with pre-existing health conditions. Data were collected in eleven Italian hospitals during the last week of lockdown, and one month later. In order to assess the emotional impact of the SARS-CoV-2 outbreak and the subject's degree of psychological flexibility, we developed an ad hoc questionnaire (ImpACT), referring to the Acceptance and Commitment Therapy (ACT) model. The Impact of Event Scale-Revised (IES-R), the Depression, Anxiety and Stress Scale (DASS) and the Cognitive Fusion Questionnaire (CFQ) were also used. Pervasive dysfunctional use of experiential avoidance behaviours (used with the function to avoid thought, emotions, sensations), feelings of loneliness and high post-traumatic stress scores were found to correlate with the fear of COVID-19, increasing the likelihood of cancelling medical appointments. Responding promptly to the information and psychological needs of patients who cancel medical appointments can have positive effects in terms of psychological and physical health.

Background: ManTra is a mixed-methods, co-production research project for developing an intervention (resource) for people with newly diagnosed secondary progressive multiple sclerosis (pwSPMS) in Italy and Germany. In previous project actions, six resources were outlined, meeting the needs prioritized by pwSPMS. Aims: This study aims to achieve multiple-stakeholder consensus on the most suitable resource and to refine the consensus resource. Methods: Two nominal group technique (NGT) meetings were held, one in Milan and one in Hamburg. Participants were pwSPMS (five in Italy/six in Germany), pwSPMS significant others (SOs, four/five), healthcare professionals (HPs, seven/four), and health service researchers/patient and citizen organizations representatives (HPCORs, five/five). Two of the four resources discussed in each meeting were the same in Italy and Germany: "Promoting the engagement of pwSPMS: a program for the patients and the HPs" and "Enriched physiotherapy program for pwSPMS." The other two were "A personalized care plan for pwSPMS" and "Roadmap for social and economic benefits" in Italy and "Metacognitive and everyday life training for pwSPMS" and "Psychological support for pwSPMS" in Germany. Each meeting consisted of two plenary sessions and a parallel group session (four stakeholder groups: pwSPMS, SOs, HPs, and HPCORs) in between. Meetings' narratives were analyzed thematically. Results: The two meetings were rich in participation and discussion. In Italy, the consensus resource was "A personalized care plan for pwSPMS." Refinements included enrichment with pwSPMS engagement, inclusion of additional HPs, improved definition of the MS nurse's role within the interdisciplinary panel, and community care integration. In Germany, the consensus resource was "Psychological support for pwSPMS." Refinements included reshaping this resource into a more comprehensive and adaptive rehabilitation intervention and training the psychologist in recognizing client's rehabilitative needs and enhancing his/her autonomy. Conclusions: The NGT eased multiple-stakeholder deliberation and resource fine-tuning in both countries., (Copyright © 2020 Giovannetti, Barabasch, Giordano, Quintas, Barello, Graffigna, Alfieri, Schiffmann, Muche-Borowski, Borreani, Heesen, Solari and the ManTra project.)

Introduction: An Australian case series study demonstrated the effectiveness of the REsilience and Activities for every DaY for people with multiple sclerosis (READY for MS), a resilience group training program based on Acceptance and Commitment Therapy, in improving quality of life in people with MS. This study aimed to evaluate the feasibility and acceptability of the Italian READY for MS program, and to preliminary assess its efficacy when compared to an active control intervention (group relaxation)., Methods: Single-blind phase II randomized controlled trial (RCT) and nested qualitative study (ISRCTN registration number: 38971970). Health-related quality of life (primary study outcome), mood, resilience, psychological flexibility and its protective factors were measured at baseline, after seven, 12 and 24 weeks. READY participants completed the purpose-built satisfaction questionnaire after 12 weeks. After trial completion, the control group also received READY. One-to-one participant interviews were conducted within three months of finishing the READY groups., Results: Four intervention groups were conducted with 39 participants (20 READY, 19 relaxation). Two patients (READY) withdrew before beginning the intervention due to unexpected work commitments. Feasibility and acceptability of READY were good, with high participant engagement and satisfaction. No statistical effects of READY were detected vs relaxation. Thirty participants were interviewed (18 READY; 12 relaxation + READY). Content data analysis revealed seven overarching themes: "Attitudes towards participation"; "Perceptions of program composition"; "Program impacts on life domains"; "Program active elements"; "Program improvement trajectories"; "Program differences and similarities"; "Suggested READY improvements"., Conclusion: READY was well accepted by MS patients with varied socio-demographic and clinical characteristics. Qualitative (but not quantitative) data provided evidence in favour of READY. Our findings will inform methodological and intervention refinements for the multi-centre RCT that will follow., Competing Interests: AS reports grants from Fondazione Italiana Sclerosi Multipla (FISM), during the conduct of the study; personal fees from Biogen Idec, Merck Serono, Novartis, Almirall, and Excemed. As a staff member of the University of Queensland and co-Author of the READY program, KP receives royalties from UniQuest for commercial (not research) licensing arrangements entered into by third parties who want to deliver the program. PC acted as an Advisory Board member of Biogen and Novartis; received funding for traveling from Biogen, Merck, Teva, Novartis; received honoraria for speaking or writing from Biogen and Novartis. He received support for research project by Novartis and Merk and is involved as principal investigator or co-investigator in clinical trials for Teva, Novartis, Biogen and Merk. RQ received funding for conducting research and clinical projects from AIM Education (Novartis) and Fondazione Serono (Merck) in 2019. This does not alter our adherence to PLOS ONE policies on sharing data.

Background: Conversion to secondary progressive multiple sclerosis (SPMS) is associated with a relatively poor prognosis, and SPMS is responsible for the majority of the social and economic costs associated with MS. Managing the Transition to SPMS (ManTra) is a mixed methods project conducted in Italy and Germany aimed to set up a user-led resource to empower and improve the quality of life of newly diagnosed SPMS patients., Aims: To assess the experiences and the needs of Italian people who recently converted to SPMS, patient significant others (SOs), neurologists and other health professionals (HPs)., Methods: We conducted 15 personal semistructured interviews (PSIs) with SPMS patients who transitioned up to five years, and three focus group meetings (FGMs), one of SPMS SOs, one of neurologists, and one of other HPs. Participants were purposely selected from the three geographic areas of Italy, and varied in terms of gender, education and (for patients) disease severity. PSIs and FGMs were audiorecorded, transcribed and analyzed by two researchers using the framework analysis., Results: One hundred sub-categories were identified, grouped into 13 categories and four themes: 'awareness of the transition', 'communication of the transition', 'dealing with symptoms worsening', and 'needs'. The major unmet needs were collected in four dimensions 'organization and management; 'empowerment training'; 'information'; and 'policies'., Conclusions: Two are the main findings: first, the widespread lack of awareness around the transition; second, the need to improve the quality of the care pathway in the Italian context. It was particularly stressed the need for a holistic and multidisciplinary approach (with patients and SOs as members of the team), the development of an ad hoc plan of follow up visits with easy access to MS specialists' consultation/treatment; specialized training for each stakeholders; more information on SPMS, daily management and changes at policy level., Competing Interests: AS is affilliated with Biogen Idec, Merck Serono, Novartis, Almirall, and Excemed. CH is affiliated with Biogen, Genzyme, Sanofi Aventis, Merck, Novartis, and Roche. There are no patents, products in development or marketed products to declare. This does not alter our adherence to PLOS ONE policies on sharing data and materials.

Background: Few studies have investigated the experiences of patients around the conversion to secondary progressive multiple sclerosis (SPMS). ManTra is a mixed-method, co-production research project conducted in Italy and Germany to develop an intervention for newly-diagnosed SPMS patients. In previous project actions, we identified the needs and experiences of patients converting to SPMS via literature review and qualitative research which involved key stakeholders. Aims: The online patient survey aimed to assess, on a larger and independent sample of recently-diagnosed SPMS patients: (a) the characteristics associated to patient awareness of SPMS conversion; (b) the experience of conversion; (c) importance and prioritization of the needs previously identified. Methods: Participants were consenting adults with SPMS since ≤5 years. The survey consisted of three sections: on general and clinical characteristics; on experience of SPMS diagnosis disclosure (aware participants only); and on importance and prioritization of 33 pre-specified needs. Results: Of 215 participants, those aware of their SPMS diagnosis were 57% in Italy vs. 77% in Germany ( p = 0.004). In both countries, over 80% of aware participants received a SPMS diagnosis from the neurologist; satisfaction with SPMS disclosure was moderate to high. Nevertheless, 28-35% obtained second opinions, and 48-56% reported they did not receive any information on SPMS. Participants actively seeking further information were 63% in Germany vs. 31% in Italy ( p < 0.001). Variables independently associated to patient awareness were geographic area (odds ratio, OR 0.32, 95% CI 0.13-0.78 for Central Italy; OR 0.21, 95% CI 0.08-0.58 for Southern Italy [vs. Germany]) and activity limitations (OR 7.80, 95% CI 1.47-41.37 for dependent vs. autonomous patients). All pre-specified needs were scored a lot or extremely important, and two prioritized needs were shared by Italian and German patients: "physiotherapy" and "active patient care involvement." The other two differed across countries: "an individualized health care plan" and "information on social rights and policies" in Italy, and "psychological support" and "cognitive rehabilitation" in Germany. Conclusions: Around 40% of SPMS patients were not aware of their disease form indicating a need to improve patient-physician communication. Physiotherapy and active patient care involvement were prioritized in both countries.

Background: We performed a qualitative study to investigate the experiences of participants in a multicentre randomized controlled trial on a home-based palliative approach (HPA) for adults with severe multiple sclerosis (MS) and their caregivers. Our aim was to explore the strengths and challenges of the intervention, and circumstances that may have influenced its efficacy., Methods: Participants to the qualitative study were the patients, their caregivers, patient referring physicians, and the teams who delivered the HPA intervention. We performed semi-structured one-on-one interviews with 12 patients and 15 informal caregivers chosen using a maximum variation strategy, two focus group meetings with patient referring physicians (4 participants each), and one with the HPA teams (9 participants)., Results: From data analysis (framework method) 38 sub-categories emerged, which were grouped into 10 categories and 3 themes: 'expectations,' 'met and unmet needs', and 'barriers'. Intervention benefits were improved control of symptoms and reduced sense of isolation of the patient-caregiver dyads. Limitations were: factors related to experimental design (difficulty of dyads in identifying examiner and team roles, additional burden for caregivers); team issues (insufficient team building /supervision, competing priorities); limitations of the intervention itself (insufficient length, lack of rehabilitation input); and external factors (resource limitations, under-responsive services/professionals). The referring physician focus groups provided little experiential data., Conclusions: The HPA reduced patient symptoms and sense of isolation in patients and caregivers. The indirect role of the HPA teams, and insufficient length of the intervention were key limitations. The experimental design imposed additional burdens on the dyads. Key barriers were the paucity of available services, the demanding administrative procedures, and lack of networking facilities. These findings suggest that two major requirements are necessary for home palliative care to be effective in this patient population: HPA teams well-connected with MS rehabilitation services, and care delivered over the long-term, with variable intensity., Trial Registration: Current Controlled Trials ISRCTN73082124 (Registered 19/06/2014)., Competing Interests: FP has received speaking honoraria from, and has been a board member of: Alimirall, Bayer, Biogen, Celgene, Merck, Novartis, Roche, Sanofi Genzyme, and Teva. VTC has been a board member of Merck, Novartis, Sanofi Genzyme. She has received travel expenses from Almirall, Biogen, Merck; speaking/writing honoraria from Almirall, Merck, and Teva; and support from Almirall for a research project. AS has been a board member of Biogen, Merck, and Novartis; she has received speaker honoraria from Almirall, Excemed, Merck, Sanofi Genzyme, and Teva. The authors confirm that this declaration does not alter the authors’ adherence to all PLOS ONE policies on sharing data and materials.

Introduction: 15 years after clinical onset, about 50% of patients with relapsing-remitting multiple sclerosis convert to secondary progressive multiple sclerosis (SPMS). Notwithstanding the importance of this transition, knowledge of the experiences and needs of patients and carers is fragmentary, and targeted interventions are not available. Managing the Transition to SPMS (ManTra) is a mixed methodology project to develop and test a user-led resource for newly diagnosed patients with SPMS. Here, we describe the developmental phase, consisting of a literature review and a new research study involving key stakeholders, in which we construct the resource., Methods and Analysis: Round 1: The literature review and study will be conducted in parallel. The latter will identify patient needs using a qualitative approach consisting of: personal semistructured interviews with >15 recently diagnosed patients with SPMS; three focus group meetings (one with significant others of patients, one with neurologists and one with other health professionals caring for patients with SPMS). An online survey (>200 recently diagnosed Italian patients with SPMS) will follow to verify needs in a larger independent sample. An expert panel will outline a set of candidate resources/interventions that aim to satisfy the needs thus identified. Round 2: Consensus on the final resource will be obtained in a 1-day meeting of recently diagnosed patients with SPMS, significant others, health professionals and other stakeholders, using the nominal group technique. The expert panel will refine the resource, identify the outcome measures to assess its efficacy and ascertain the most suitable comparator (ManTra Phase 2, not part of this protocol)., Ethics and Dissemination: The study protocol was approved by the ethics committees of each of the involved centres: Foundation IRCCS Neurological Institute C Besta, Milan ; G D'Annunzio University of Chieti-Pescara and the Aldo Moro University of Bari. The results will be published in peer-reviewed journals, presented at conferences and a lay summary sent to participants., Competing Interests: Competing interests: AS has been a board member of Biogen Idec, Merck Serono and Novartis; she has received speaker honoraria from Almirall, Excemed, Merck Serono, Sanofi Genzyme and Teva. CT has received speaker honoraria from Biogen, Merck Serono, Novartis, Genzyme, Teva, Almirall, Schering and Excemed., (© Article author(s) (or their employer(s) unless otherwise stated in the text of the article) 2017. All rights reserved. No commercial use is permitted unless otherwise expressly granted.)

Objective: Our purpose was to provide a comprehensive understanding of how women informal caregivers of patients in vegetative state (VS) or minimally conscious state (MCS) describe, represent, and experience changes that occurred in their life after the acute event of their family member., Methods: A qualitative study was conducted and fifteen women informal caregivers, mothers, or spouses of patients in VS or MCS were interviewed., Results: Caregivers' narratives revealed (1) important personal and interpersonal changes and (2) difficulties while facing the complex situation and integrating past, present, and future, defined as a "time gap experience." This difficulty is expressed in two ways. First, the reduction of variety of roles into one, caregiver's role. Second, the relationship with the relative is characterised by fluctuation in the relational style between caregiver and relative; it shifts from an adult to adult interaction to an adult to child one. Another fluctuation can be observed in the mixed use of present and past tenses when caregivers speak about their relatives., Conclusions: Caregiving cause pervasive modifications in one's life. Targeted interventions aiming to empower the caregivers, to support them after the acute event in caregiving activities together with patient-focused interventions, and to promote their health should be implemented.

Migraine is a common disease which causes significant burden to individuals, in terms of personal suffering and activity reduction, and to societies, in terms of disease cost. The purpose of this study is to identify the most relevant psychosocial difficulties related to migraine, the variables associated with them and the most relevant determinants of their evolution over time. MEDLINE and PsychINFO were searched for studies published in English between 2000 and 2010 that examined psychosocial difficulties in persons with migraine with and without aura, from clinical trials and observational studies. Information on the description of each difficulty, its determinants of onset and change over time and associated variables were extracted and categorized at a higher level. In total, 34 difficulties have been collected from 51 papers: the most frequent were reduced vitality and fatigue, emotional problems, pain, difficulties at work, general physical and mental health, social functioning and global disability. Evidence exists that pharmacological treatments have an impact toward improvement in patients' difficulties, in particular emotional problems, physical and mental health, difficulties with employment and global disability. Migraine treatments and decreased headaches frequency are the major determinants of improvements in psychosocial difficulties, while no information is available for determinants of worsening; understanding the role of such factors is of primary public health relevance, given the high prevalence and the relevant personal and societal costs of migraine.

Caring for children in vegetative state (VS) or minimally conscious state (MCS) challenges parents and impacts on their well-being. This study aims to evaluate caregivers' health condition, coping, anxiety and depression levels, and how these issues relate to children's disability. 35 children with VS and MCS were administered the disability rating scale (DRS) and 35 caregivers completed the Coping Orientations to Problem Experiences, Short Form-12, Beck Depression Inventory, and the Spielberger State-Trait Anxiety Inventory-Y. Children were mainly males (68.6%), hosted at domicile (77.1%), and diagnosed with VS (60%), with anoxic aetiology (45.7%). Caregivers were mainly mothers (85.7%), married (82.9%), and housewives (51.4%); 60% declared financial difficulties, and 82.9% provided full-time assistance. 57.2% reported depressive symptoms, poor mental health, and high level of state and trait anxiety. "Problem-oriented" (P < 0.001) and "emotional-oriented" (P < 0.001), were more adopted than "potentially dysfunctional" ones. DRS scores (mean = 22.0; SD = 1.9) did not significantly correlate to any psychological measure. Rehabilitative programs for children with SV and SMC should also provide interventions on surrounding systems: improving the network of psychological support and social assistance may decrease the burden of caregivers and, in turn, improve caring abilities and children quality of life.

In genetically hypertensive rats of the Milan hypertensive strain (MHS) and normotensive rats (NR) developed from the same Wistar stock colony, blood pressure (BP), plasma renin activity (PRA), sodium balance, and water balance were measured from the time of weaning to the seventh week postweaning in three separate but essentially identical experiments. In a fourth experiment, total and extracellular water, total sodium, and exchangeable sodium were measured in MHS and NR at 24 and 130 days of age. Although the time course of changes varied slightly between experiments, BP of both NR and MHS rose until the second and third weeks postweaning, at which time BP in MHS was 40 to 50 mm Hg higher than in NR. PRA in MHS was one-half that of NR at weaning. Increasing BP was accompanied by falling PRA in both, and PRA was not significantly different when stable, adult blood pressure was reached. Urinary volume in MHS was 50% to 100% greater (P less than 0.001) than in NR at weaning and for a few days after. Sodium was retained to a greater extent by MHS during the period when the blood pressure difference develops, from weaning to the fourth week postweaning. This sodium retention (MHS = 97.0 plus or minus 10.3, NR = 65.2 plus or minus 6.8 SE mu-Eq Na retained/g body weight gain; P less than 0.005) is the result of significantly lower urinary excretion of dietary sodium by MHS. A causative role for the kidney is suggested in the established of high blood pressure in MHS.