TYPE Study Protocol PUBLISHED 18 May 2023 DOI 10.3389/fneur.2023.1172640 An expert patient program to OPEN ACCESS improve the empowerment and quality of life of people with EDITED BY Christoph Heesen, University Medical Center Hamburg-Eppendorf, Germany REVIEWED BY multiple sclerosis: protocol for a Andrea Giordano, IRCCS Carlo Besta Neurological Institute Foundation, Italy multicenter pre-post intervention Birgitta Langhammer, Oslo Metropolitan University, Norway Pavol Mikula, study University of Pavol Jozef Šafárik, Slovakia *CORRESPONDENCE Miguel Angel Robles-Sanchez 1,2,3*, Paloma Amil-Bujan 4, Miguel Angel Robles-Sanchez Cristina Bosch-Farré 1, Clàudia Coll-Martínez 5,6, marobles@cem-cat.org Maria Jesús Arévalo 2, Elisenda Anglada 2, Rebeca Menéndez 2, RECEIVED 23 February 2023 ACCEPTED 21 April 2023 Xavier Montalban 2, Jaume Sastre-Garriga 2, PUBLISHED 18 May 2023 Lluís Ramió-Torrentà 5,6,7 and Carme Bertran-Noguer 1 CITATION Robles-Sanchez MA, Amil-Bujan P, 1 Grup de Recerca Salut i Atenció Sanitaria, University of Girona, Girona, Spain, 2 Department of Bosch-Farré C, Coll-Martínez C, Arévalo MJ, Neurology, Centre d’Esclerosi Múltiple de Catalunya (Cemcat), Hospital Universitari Vall d’Hebron, Anglada E, Menéndez R, Montalban X, Barcelona, Spain, 3 Grup d’Investigació Multidisciplinari d’Infermeria, Vall d’Hebron Institut de Recerca Sastre-Garriga J, Ramió-Torrentà L and (VHIR), Hospital Universitari Vall d’Hebron, Barcelona, Spain, 4 Expert Patient Program Catalonia, General Bertran-Noguer C (2023) An expert patient Directorate of Health Planning and Research, Department of Health, Generalitat de Catalunya, program to improve the empowerment and Barcelona, Spain, 5 Neurodegeneration and Neuroinflammation Research Group, Girona Biomedical quality of life of people with multiple sclerosis: Research Institute (IDIBGI), Salt, Spain, 6 Girona Multiple Sclerosis and Neuroimmunology Unit. protocol for a multicenter pre-post Neurology Department, Dr. Josep Trueta University Hospital and Santa Caterina Hospital, Girona-Salt, intervention study. Spain, 7 Department of Medical Sciences, University of Girona, Girona, Spain Front. Neurol. 14:1172640. doi: 10.3389/fneur.2023.1172640 COPYRIGHT © 2023 Robles-Sanchez, Amil-Bujan, Bosch- Introduction: Multiple sclerosis (MS) causes a progressive disability, which Farré, Coll-Martínez, Arévalo, Anglada, substantially impacts the quality of life (QoL). Health interventions that meet the Menéndez, Montalban, Sastre-Garriga, Ramió- Torrentà and Bertran-Noguer. This is an open- needs and demands of people with MS are essential to minimize QoL impairment. access article distributed under the terms of Expert patient programs (EPPs) facilitate health-related empowerment through the Creative Commons Attribution License peer learning. Based on a previous focus group study, we designed an EPP for (CC BY). The use, distribution or reproduction in other forums is permitted, provided the MS coordinated by nursing professionals for implementation in the different MS original author(s) and the copyright owner(s) reference units of Catalonia (Southwestern Europe). This study aims to evaluate are credited and that the original publication in the effects on quality of life, disease-related knowledge, and self-management this journal is cited, in accordance with accepted academic practice. No use, related to the health process of the participants of the Expert Patient Program distribution or reproduction is permitted which Catalonia™ for people with multiple sclerosis (EPPC-MS). does not comply with these terms. Methods: Pre-post intervention multicenter clinical study involving 12 groups of 12 participants: six groups including relapsing and six groups including progressive MS patients, with 144 participants from 7 MS reference units from all over Catalonia, organized in six teams. The intervention will consist of nine telematic learning peer-led sessions (one weekly session). The expert patient (EP) leading the sessions will be an individual with MS with disease-related knowledge, who will be further trained by nurses to lead the sessions. Study variables will be measured before and immediately after the intervention and 6 and 12months after the end of the sessions and will include: QoL, emotional impact, activation of the person, MS-related knowledge, fatigue, habits and lifestyles, health services use, and program-related experience. Baseline characteristics considered will be sociodemographic data, date of MS diagnosis and type, family history, and treatment characteristics. Variables related to disease follow-up will be new Frontiers in Neurology 01 frontiersin.org Robles-Sanchez et al. 10.3389/fneur.2023.1172640 relapses and characteristics and changes in the ongoing treatment. The number of sessions attended will also be collected. Study variables will be analyzed using a pre-post comparison. Discussion: Peer-led learning programs led by EP help empower people with chronic conditions and offer them tools to improve their autonomy and QoL. This study’s intervention will be performed remotely, offering advantages both for people with chronic conditions and the healthcare system regarding the facilitation of family and work conciliation, saving time, simplifying attendance to meetings, lowering costs, and using fewer material resources. Trial registration: NCT04988880 on September 22, 2021. KEYWORDS clinical research protocol, multiple sclerosis, health education, health literacy, patient empowerment, peer group, quality of life Introduction In Catalonia, which has a population of over 7 million, more than 10,000 people have MS, of which almost 7,000 are women (11). A Multiple sclerosis (MS) is a chronic autoimmune degenerative previous qualitative study based on focus groups including people disorder of the central nervous system with a variable clinical course with MS allowed to identify the topics to be addressed in the sessions, characterized by a progressive disability, causing a substantial impact the ideal profile of the EP and the participants, the program’s design, on quality of life (QoL) (1). Identifying and solving the needs and and the potential benefits of a specific Multiple Sclerosis Expert demands of individuals with MS is fundamental to minimizing their Patient Program of Catalonia (EPPC-MS) (12). As a result of this QoL impairment and improving their well-being (1). However, the pivotal study, the research team defined the final design and contents needs and demands related to MS diagnosis and evolution generate a of the EPPC-MS, which will be implemented across the Catalan dynamic and complex scenario, requiring an individualized and high- territory and remains to be evaluated for its ability to improve the quality healthcare planning focused on solving these needs (2). In this empowerment and QoL of people with MS. regard, patient-centered healthcare systems are more cost-effective and improve health literacy and patient participation (3). In addition, it is necessary to simultaneously incorporate a positive view of health Objectives that promotes individuals’ empowerment to solve health challenges (2). The primary objective of this study is to deploy the EPPC-MS Expert patient programs (EPPs) facilitate patient-centered across Catalonia. Secondary objectives are to evaluate the effectiveness healthcare delivery by improving individuals’ understanding of of the EPPC-MS in reducing the emotional impact of MS and chronic diseases through knowledge transfer and exchange, thus improving QoL, fatigue management, patient activation, habits and promoting autonomy and self-care through a proactive approach, lifestyles, and MS knowledge of participants after completing the improving health indicators and reducing the use of healthcare program. Additional secondary objectives are to evaluate the resources (4). In addition, they facilitate the empowerment of people effectiveness of EPPC-MS in improving health outcomes according to with MS and improve their well-being and overall perception of health the type of MS, the use of healthcare services by participants, the (5, 6), enabling them to reorient their priorities, plan their activities, satisfaction of the EP and the participants with the EPPC-MS, and the and choose their care and health preferences (7). long-term persistence of program benefits (at 6 and 12 months). An Expert Patient Program of Catalonia™ (EPPC), consisting of a peer-led learning intervention, was included in the Health Plan of Catalonia 2011–2015 (Spain) and maintained in subsequent Methods plans. The EPPC focused on self-responsibility, self-care, and promotion of people’s autonomy (8, 9). The Expert Patient (EP), a Overall study design non-healthcare professional with the same chronic condition, leads the program sessions, developed in groups of 10–12 participants This research will be conducted using a pre-post intervention with the same long-term condition, guided by a nurse (4). EPs are multicenter clinical study design to evaluate the territorial people with significant knowledge of their disease and treatment in implementation of a specific EPPC-MS for people with MS. The study addition to self-management skills, who take responsibility for self- is planned for development during a period of 36 months (January care and disease management, know how to identify and respond 2021–December 2023). The common goal of this kind of study is to properly to symptoms, and acquire skills enabling them to manage find whether a health intervention affects the participants and their the impact of the disease, resulting in improved QoL (10). The clinical variables and health-related outcomes (13). The intervention EPPC complements other health interventions led by healthcare sessions will be conducted remotely using Microsoft Teams platform professionals and is not currently implemented for the to avoid face-to-face meetings during the current pandemic situation MS population. (SARS-CoV-2) as part of a risk mitigation strategy (14). EPPC Frontiers in Neurology 02 frontiersin.org Robles-Sanchez et al. 10.3389/fneur.2023.1172640 FIGURE 1 Location of the seven participating multiple sclerosis units. The line between the hospital in Tarragona and the hospital in Reus indicates that these 2units will participate as a joint team, resulting in total of six participating teams. managers will provide this platform linked to the Health Department with progressive MS, resulting in a total of 12 groups. The program of Catalonia, who will endorse its use. Each intervention will consist will be implemented in four consecutive phases, including three of nine weekly sessions, lasting an average of 3 months. groups of participants from 3 MS units per phase that will undergo This study protocol adheres to the Standard Protocol Items: the 9-week intervention simultaneously. After completing the recommendations for Interventional Trials (SPIRIT) guidelines1 for intervention in Phase 1, the same 3 units will start Phase 2 with the reporting study protocols. remaining three groups in any order (e.g., first the relapsing group and then the progressive one). Hence, 3 MS units will participate in the first two phases. Immediately after the first two phases are Study setting completed, the three remaining units will start the intervention in their groups in Phase 3 and, after completing the three groups, in The Catalan healthcare system provides free and universal Phase 4. Each group will be invited to attend a follow-up session 6 healthcare to its over 7 million population. The estimated 10,000 and 12 months after the end of the intervention to share any individuals who live with MS in Catalonia receive care in changes on their daily habits resulting from the learning program, multidisciplinary reference MS units. The study will be carried by and the application of disease management strategies (see six teams from 7 MS units located across Catalonia: one from Figure 2). Girona, three from Barcelona, one from Lleida, and two from the Tarragona area (Tarragona unit and Reus unit) pariticipating as a joint team (Figure 1). The participating MS units are the main Selection of participants and EPs multidisciplinary reference units of the territory for individuals diagnosed or suspected of demyelinating disease and specifically Patients with a diagnosis of MS treated in the participating MS MS. The study will start after the approval of each local Clinical units will be eligible to participate as participants or EPs in the Research Ethics Committee. deployment of the EPPC-MS. All members of the MS multidisciplinary teams will identify participant and EP candidates according to their criteria based on their MS-related knowledge and ability for self-care Participant and unit timeline and self-management, and will invite them to participate. The teams coordinate the process of selection and assessment of participants’ Each unit will identify and recruit two groups of people with inclusion criteria, including a visit with a trained neuropsychologist two forms of MS: one group of people with relapsing MS and one to administer the Brief International Cognitive Assessment for Multiple Sclerosis (BICAMS) (15) and Hospital Anxiety and Depression Scales (HADS) (16). Furthermore, the teams initiate and coordinate all the steps for the selection of EPs among pre-selected 1 http://www.spirit-statement.org/ candidates. A nurse [i.e., the study’s principal investigator (PI)] will Frontiers in Neurology 03 frontiersin.org Robles-Sanchez et al. 10.3389/fneur.2023.1172640 FIGURE 2 Flow diagram and timeline of participants. BICAMS, Brief International Cognitive Assessment for Multiple Sclerosis; HADS, Hospital Anxiety and Depression Scale; M, month; MS, multiple sclerosis. *Participant candidates with BICAMS and HADS data obtained in the routine neurorehabilitation screening will not be required to sign an informed consent for the screening procedure, only to participate in the study. coordinate and provide support for these processes to the participating appraise the patients’ characteristics and skills. Furthermore, the MS units, and will monitor the process to ensure protocol compliance. selection of patients diagnosed before 2018 ensures a long-term Eligible participants must meet the following inclusion criteria: relationship with the multidisciplinary teams. Irrespective of these (1) be ≥18 years old; (2) have reported the need for support for disease criteria, potential EP candidates could meet other characteristics, such self-management or to improve MS-related knowledge; (3) confirm as information and communication technologies skills, availability to attend at least 80% of the sessions; (4) sign an informed communication and leadership skills, and comprehensive experience consent. Patients meeting any of the following exclusion criteria will and knowledge of the health process. The information and not be eligible: (1) be unable to speak or write in Spanish or Catalan; communication technologies skills are partly appraised during (2) have aphasia or an auditory disorder preventing them from contacts with the MS units using electronic communication tools, interacting with the group; (3) have a severe cognitive impairment, such as email or other platforms, and by evaluating the use of reference defined as a score of 1.5-fold the standard deviation below the information websites. During the selection process, EPs are informed population mean in one of the three subtests of the Spanish version of that their participation will be altruistic and should expect the BICAMS (15); (4) have severe emotional impairment, defined as no compensation. a score above 11/21 on any of the two Hospital Anxiety and Depression Each of the participating teams will select three EP candidates, Scale (HADS) subscales (anxiety and depression) (16); and (5) have who will be scheduled for interviews with the PI and the institutional any psychopathological comorbidity or mental disorder diagnosis that managers of the EPPC to further assess their adequacy to fulfill the EP may restrict their interaction with the group. role. Disease-related knowledge will be assessed using questions from To be eligible as EPs, individuals must meet the same inclusion the disease knowledge, habits and lifestyle, and quality of life criteria as participants. However, they must lack the need for support questionnaires used to evaluate the program, and candidates will for disease self-management and should have MS-related knowledge. be engaged to speak about self-management, self-care, and their future Healthcare or educational professionals and individuals meeting role as EP to assess their predisposition, leadership skills, and participants’ exclusion criteria will be ineligible as EPs. Furthermore, communication abilities. This strategy for EP selection has been used EPs are selected among patients diagnosed before 2018 to ensure previously in other versions of the program (17). Moreover, the sufficient disease-related knowledge based on their own experience interviews will be conducted remotely using the same platform that and interactions with the health care system and professionals. will be used for the program sessions to confirm the ability of the EP Additionally, EPs are selected among patients showing the ability for candidates to use it. The PI and EPPC managers will select one EP per self-management and self-care and those with a positive perception of team, who will be provided with the session materials and will the disease. No objective instrument or scale will be used to assess EP be trained for their role. The training sessions will allow us to confirm inclusion criteria. However, health care professionals from the MS the adequacy of the EPs and, if necessary, the selection may units follow patients closely, getting to know them and, therefore, can be reconsidered. Frontiers in Neurology 04 frontiersin.org Robles-Sanchez et al. 10.3389/fneur.2023.1172640 Sample size Sample size is estimated based on the deployment of the EPPC-MS across Catalonia (primary outcome). Sample size was calculated by prioritizing those units willing to participate in the study and interested in implementing the program in the future to offer it to the people with MS followed up at the unit regularly. Of the 7 units willing to participate, the 2 units located in the Tarragona area (Tarragona and Reus units) will particpate as joint team, resulting in a total of six participating teams. Each MS team will include two groups of people with MS, of which one group will include people with relapsing MS and the other group people with progressive MS, resulting in a total of 12 groups, with six groups for each MS type. Based on the methodology used in the program, the recruitment target will be 12 people with MS per group (144 individuals in total). We expect this group size will compensate for possible losses during the intervention and ensure a minimum of 8–10 participants per group throughout the program (96–120 participants) (4). We estimated this sample size based on the number of people with MS in Catalonia (⁓10,000) and the number cared for at each of the MS units. Ideally, the exact calculation of the sample size required to assess the study’s secondary FIGURE 3 Expert Patient Program timeline. EP, expert patient; EPPC-MS- outcomes (EPPC-MS effects) should be based on the results of a pilot Relapsing and EPPC-RMS, expert patient group including participants study testing the program’s effects in a smaller sample of patients. with relapsing multiple sclerosis; EPPC-MS-Progressive and EPPC- However, this study has not yet been completed. If the 12 groups are PMS, expert patient group including participants with progressive multiple sclerosis; G1 to G12, expert patient groups 1 to 12; M, insufficient to reach an appropriate sample, the participating month. investigators will contact the Ethics Committees to request their approval to expand the number of groups. information sheet and be asked to sign a written informed consent Recruitment before assessing the selection criteria, such as cognitive and emotional impairment (when required). The participant timeline summarizes Participants: follow-up of people with MS involves therapeutic the recruitment process (Figure 3). support by the multidisciplinary team during clinical visits at the unit Expert Patient candidates will be selected among the MS facilities (i.e., regular follow-up visits), the day care clinic, or through population followed up at each unit and according to the eligibility non-scheduled consultations for any reason (i.e., needing additional criteria. The study PI will evaluate the knowledge of EP candidates to care due to treatment secondary effects or disease symptoms, confirm their eligibility. Additionally, people with MS who participated information, and support). After completing a routine follow-up visit as EP in the pilot study testing the program effects or other MS units by a healthcare professional, people with MS self-reporting the need may be invited to participate again as EPs in this study. for support for disease management or the need to improve MS-related knowledge will be invited to participate in the study. To ensure that the target sample size is reached, healthcare professionals, members Intervention of the MS multidisciplinary teams may actively identify candidates by asking people with MS about their need for additional support and The intervention aims to reinforce positive knowledge concisely information for disease management. People with MS initially willing using plain language that all participants can understand. Throughout to participate will be referred to the study coordinator at each nine 90-min sessions (one per week) led by an EP, participants in the participating center to receive complete information about the study. group will share their knowledge and experiences on all aspects of MS Neuropsychologists within the MS multidisciplinary teams will and its self-management. The organization of session and its contents assess the inclusion and exclusion criteria of participant candidates. are summarized in Table 1. To assess severe cognitive and emotional impairment (exclusion The study’s PI is a nurse specialized in MS and will train the EPs criteria), a trained and experienced staff in assessing cognitive and and support them throughout the intervention, together with emotional impairment will screen participant candidates using the professionals in each of the participating units. Before starting the BICAMS (15) and HADS (16) instruments. These tests are part of the intervention, the study’s PI will train the EPs in a presentation of all routine neurorehabilitation screening procedure, but may the learning materials and contents of each session. Furthermore, be administered to other participant candidates outside the routine using role-playing activities, the nurse PI will ask example questions screening that require a specific consent. Before the study starts, a related to the contents of each session to the EPs to be answered senior neuropsychologist will lead an update session for all jointly. The training and activities aim to expand the EPs’ knowledge neuropsychologists of the participant centers to ensure uniformity of of the sessions’ contents and gain confidence to lead and answer data collection (18). Eligible candidates will receive the patient the questions. Frontiers in Neurology 05 frontiersin.org Robles-Sanchez et al. 10.3389/fneur.2023.1172640 TABLE 1 Expert Patient Program Catalonia-Multiple Sclerosis (EPPC-MS): sessions and contents. Session Content description 1. Introduction Presentation of group members, program objectives, session contents, session development, and questionnaires 2. The disease What is multiple sclerosis? Concept, causes, types of MS, and diagnosis 3. Signs and symptoms Relapses (relapsing MS), progression (progressive MS), and fatigue 4. Treatment Existing treatment, future treatments, treatments during pregnancy, and alternative therapies 5. Health promotion Healthy lifestyles: nutritional facts, activity and exercise, tobacco, vitamin D and sun exposure, and obesity 6. Taboo subjects Urinary and fecal incontinence, sexuality, and sexual dysfunction 7. Emotional impact and relationships Coping with the disease, work and family relationships, and available resources for emotional support 8. Social environment and quality of life Rehabilitation and physical aspects, social resources, and benefits 9. Program finalization Wrap-up session Throughout the intervention, a professional nurse in the MS team Secondary outcomes are those related to the effectiveness of the and the EP will meet before each session to prepare the contents and program and include changes in QoL, measured using the Multiple solve any doubts from the previous sessions. In all sessions, two Sclerosis Quality of Life 54 items (MSQOL-54) questionnaire (19, nurses, on from the participating team and one member of the EPPC 20); emotional impact, measured using the HADS (16); fatigue panel of directors, will act as an observers, provide help and support management, measured using the Fatigue Severity Scale (FSS) (21); to the EP if needed, and assess each session jointly with the EP at patient activation and engagement, measured using the 13-item the end. Patient Activation Measure (PAM-13) (22); patient knowledge, Sessions are divided into two blocks: a first block with a theoretical measured using the EPPC specific questionnaire about MS-related introduction to focus on the day’s topic, conducted by the EP (30 min), knowledge; individuals’ habits and lifestyles, measured using the and a practical block (60 min). A previous pivotal study characterized EPPC specific questionnaire about habits and lifestyles; use of the topics addressed in the sessions (12). After the first block healthcare services, such as visits to the MS unit, visits to the introducing the topic, the EP will encourage interaction, facilitating general physician (GP), and visits to the accident and emergency the communication of doubts, questions, or experiences related to the (A&E); and Patient Reported Satisfaction (i.e., experience), contents, which will be solved or answered by participants or measured using the EPPC-specific questionnaire about patient- collectively through discussion within the group (practical block). reported experience measures (PREMs), designed ad hoc for this This intervention strategy with an EP leading the session and the study. Except for the latter, we will compare the scores obtained participation of all the group members aims to empower them to gain before the intervention and at post-intervention timepoints control over their health situation. The knowledge acquired will come (immediately after the intervention and at 6 and/or 12 months after from the information provided by the EP and the knowledge resulting the end of the intervention). The participants and the EPs will from the life experiences regarding MS of all the participants involved answer the patient-reported satisfaction questionnaire, and scores in the intervention. will be compared immediately after the intervention and at 6 and Participants will complete questionnaires according to the study 12 months. schedule (prior and immediately after the intervention, and at the evaluation periods 6 and 12 months after the end of the intervention) to assess the secondary outcomes. The completed questionnaires will Study variables and data collection be collected at post-intervention sessions (at 6 and 12 months), allowing participants to clarify concepts and expose doubts regarding Data will be collected according to the study timeline depicted in the contents of the previous nine sessions. Figure 4. Baseline sociodemographic characteristics of EPs and To ensure adherence, the platform will send reminders before participants, including age, gender, marital status, family support, each session and, if necessary, sessions will be rescheduled. educational level, and employment situation, will be collected during the recruitment visit. Disease variables, including, among others, date of diagnosis, type of MS, MS treatment, and disability, will be collected Outcomes from the clinical records before the intervention (Week 1) and after the intervention (Week 9 and at 6 and 12 months after the end of the The primary outcome of this study is the deployment and intervention). Variables related to the use of healthcare services implementation of the EPPC-MS in the participating MS units across include the number of visits to the MS unit (both scheduled routine Catalonia. The feasibility of the EPPC-MS implementation will follow-up visits and unscheduled visits), primary care visits, and A&E be appraised in terms of the correct inclusion of the six participating department visits during 1 year before and after the intervention, and teams and the successful execution of all the procedures. Participation will be collected prior to the intervention (Week 1) and after the in the study entails sufficient training of professionals at MS units for intervention (Month 12), respectively, by the research team using a the correct identification and recruitment of participants and EPs and questionnaire. This information is routinely registered during visits to for appropriate data collection and monitorization of study clinics and healthcare consultations. It is made available by the health procedures. At the end of the study, the participating units will be able registry of the region to the healthcare institutions after institutional to implement the EPPC-MS independently and autonomously based authorization from the Ethics Committee of each center. Before the on the methodologies of the EPPC-MS. study starts, all the healthcare professionals involved will receive an Frontiers in Neurology 06 frontiersin.org Robles-Sanchez et al. 10.3389/fneur.2023.1172640 FIGURE 4 Assessments and data collection timeline. BICAMS, Brief International Cognitive Assessment for Multiple Sclerosis; EP, expert patient; FSS, Fatigue Severity Scale; HADS, Hospital Anxiety and Depression Scale; MSQoL-54; Multiple Sclerosis Quality of Life-54 items; PAM-13, patient activation measure; PRT, participants; RT, research team. update session led by the study’s PI regarding data entry to ensure The study’s PI, a nurse, will maintain a field notebook to record uniform data collection (18). the basic information for each session, including participants, the Patient-reported outcomes (PROs) and PREMs evaluating the session’s contents, the group, and the MS reference unit. The PI will impact of the intervention (secondary outcomes) will be assessed additionally collect participants’ comments and questions related to using specific instruments (i.e., questionnaires) according to the study the session’s topic and final comments regarding each session timeline (Figure 4). Except for participants and EP experience with development. The EP will answer all questions collected as unanswered the program, which will be evaluated after the intervention, during each session in the next. Furthermore, the EP will clarify any questionnaires will be self-administered prior to the intervention inappropriate or inaccurate information shared during sessions in the (Weeks 1–3) and after the intervention (Week 9, Month 6, and next session, with the support of the research team. Month12). Pre-intervention questionnaires will be distributed within the first 3 weeks considering the contents of each session. Hence, Quality of life questionnaires assessing fatigue and lifestyle will be administered in The Spanish validated translation (20) of the MSQoL-54 (19) is a session 2 (Week 2) and the corresponding contents will be presented self-administered questionnaire with a completion time of in sessions 3 and 5, respectively. The Research Electronic Data Capture approximately 15 min and a Cronbach alpha ranging 0.75–0.96 for the (REDCap) platform will automatically send the questionnaires and different scales. It consists of 54 items, of which 36 correspond to the instructions for completion, and participants will complete them generic Short Form-36 Health Survey questionnaire and 18 are online on the platform’s site. The platform automatically resends the specific to MS. 52 items are classified in 12 dimensions. The remaining questionnaires weekly during three consecutive weeks in case answers two individual items measure changes in health status (comparing are not received. Moreover, it allows real-time monitoring of the current health and health during the previous year) and satisfaction questionnaires received and the PI may resend the questionnaires with sexual function. In addition to the scores of the dimensions and manually and contact participants to solve any issues regarding individual items, two composite scores corresponding to mental and questionnaire completion on the platform’s site. physical health subscales are obtained. The MSQoL-54 questionnaire Frontiers in Neurology 07 frontiersin.org Robles-Sanchez et al. 10.3389/fneur.2023.1172640 is widely used and is, therefore, a relevant tool for assessing the impact Participant-reported experience and expert of the EPPC-MS on participants’ QoL. patient-reported experience The EPPC coordinating panel developed a specific questionnaire Emotional impact to assess satisfaction related to their participation and experience with The HADS (16) and the Spanish version elaborated by Herrero in the intervention. This questionnaire has already been used in other 2003 (23) specifically assess the level of anxiety or depression of an variants of the program, demonstrating its usefulness in assessing individual in the last 7 days. Several studies have validated the participants’ experience in this program (4, 17, 26). translated version in the Spanish population, yielding Cronbach alphas of 0.87–0.90 (24). It consists of a 14-item self-administered questionnaire, including an anxiety and a depression subscales, with Data management seven items each. For each subscale, the scores range from 0 to 3 (0 being the minimum affectation and 3 the maximum) for each item, Study data will be collected and managed using REDCap whereby a 0–7 score indicates no disorder, 8–10 is a questionable case, electronic data capture tools hosted at Vall d’Hebron Research and a score ≥ 11 indicates the presence of anxiety or depressive Institute (VHIR). The REDCap (Research Electronic Data Capture) is disorder. HADS is a useful tool for detecting individuals with a a secure, web-based software platform designed to support data neuropsychological condition requiring referral to a specialist for capture fro research studies. The REDCap provides (1) an intuitive assessment and care (exclusion criteria) and evaluating the program’s interface for validated data capture; (2) audit trail for tracking data effectiveness on MS-related emotional impact. manipulation and export procedures; (3) automated export procedures for seamless data downloads to common statistical packages; and (4) Fatigue procedures for data integration and interoperability with external The FSS (21) and its Spanish translation by Tola in 1998 (25) is a sources (27, 28). Each participant will be assigned a code for 9-item self-administered questionnaire, rated on a 1–7 scale (1 being de-identification. No data will be shared. Each site will send the results “strongly agree” and 7, “strongly disagree”) assessing the impact of to the coordinating center, and the PI will be responsible for guarding fatigue on a person’s daily life. The Cronbach alpha was 0.81 for its the database, which will be saved until the analysis is complete. English version. This tool will enable the evaluation of the program’s impact on fatigue self-management. Contact details of participants Patient activation (engagement) The PAM-13 and its validated Spanish version by Moreno-Chico Each unit will provide minimum and essential contact details to in 2017 (22) is an instrument assessing knowledge, skills and people’s allow communication between the program coordinating team and confidence in self-management of their health and medical care. The EP or participant candidates. Data will be stored by the coordinating validation of the Spanish version showed an Item Separation Index team in a different database than the study’s database, and will (i.e., comparable to Cronbach alpha, whereby values ≥0.7 show a be destroyed after the intervention is completed. Only the PIs at each differentiated scale between individuals and items along the latent participating unit will have access to the database. These data must trait) of 6.64 (22). PAM-13 consists of a self-administered include the code assigned to each individual, name and surname, questionnaire of 13 items, rated on a 1–4 scale (1 being “strongly telephone number, and e-mail address to send them the program agree” and 4 being “strongly disagree”). PAM-13 is a useful tool to questionnaires and instructions to connect to the Patient Expert assess patients’ confidence regarding disease self-management and Catalonia Program platform. will enable evaluating the program’s impact on the activation and involvement of participants. Variables and questionnaires Knowledge of MS Given the lack of validated questionnaires in Spanish assessing The REDCap is an electronic data capture software and workflow MS-related knowledge, the expert panel of the EPPC developed a methodology we will use to design the research database (27, 28). specific questionnaire to assess MS-related knowledge. This REDCap, developed by Vanderbilt University, provides institutional questionnaire assesses disease-related knowledge and specific aspects licenses to various clinical research institutes worldwide. In this study, essential to empower MS patients identified as the target population the Basic Statistics Unit (UBE) associated with the Vall d’Hebron through a qualitative study of focus groups (12). This questionnaire, Research Institute (VHIR) of the Vall d’Hebron Barcelona Hospital which has not been validated yet, was designed based on the previous Campus will manage the institutional license provided and guarantee pilot qualitative study (12) and will assess the impact of the program security of the data and the computer support for databases created on the acquired knowledge. with this system. Specifically, in this study, the UBE will support the study’s PI to create the database and, through a collaboration Habits and lifestyles agreement, will perform the statistical analysis of this study’s The EPPC coordinating panel developed an MS-specific outcomes. The coordinating team will enter all data related to the questionnaire to assess habits and lifestyles. This questionnaire awaits questionnaires into the study database using REDCap as participants validation, but has already been used in other program variants, complete the questionnaires. The centers’ research teams will provide demonstrating its usefulness in assessing changes due to participation all clinical or disease-related variables with a code for each participant, in this program (4, 26). without any sensitive data allowing the identification of study Frontiers in Neurology 08 frontiersin.org Robles-Sanchez et al. 10.3389/fneur.2023.1172640 participants. Therefore, their identity will remain blind to the research Furthermore, a negative impact of the intervention may lead to team of each center, who will have access to the REDCap platform for missing sessions, and we will contact participants with missing data entry. As mentioned above, each center will send the results to sessions to provide support. If the investigators detect any issues, the the coordinating center through the platform. The nurse principal participant will be referred to the relevant specialist. investigator will keep the database and save it until the end of the analysis related to the research project. Ethics statement Statistical analysis The study protocol (version 2.0) was approved by the Clinical Research Ethics Committee (CEIM, Comité de Ética de la Investigación Statistical analyses will be performed on the intention-to-treat con medicamentos) of the Hospital Universitario de la Vall d’Hebron (ITT) population, defined as the population of patients starting the with protocol number PR(AG)334/2021 on June 26, 2021. It was intervention, irrespective of program completion. Continuous registered at clinicaltrials.gov (NCT04988880) on September 22, 2021. variables will be described as mean and standard deviation (SD) or, in This study will be conducted following the foundations of the case of non-compliance with normality criteria, median and Declaration of Helsinki and the current legislation regarding interquartile range (IQR), and categorical variables as absolute confidentiality of personal data in clinical studies: the Spanish frequency and percentages. The non-parametric U Mann–Whitney Personal data protection regulations, the Organic Law 3/2018, of 5 test for independent samples and the Wilcoxon test for dependent December, on the Protection of Personal Data and the guarantee of samples will be used to analyze the relationship between a categorical digital rights (BOE 6 December 2018, applicable from 7 variable and a continuous variable. The Chi-squared test or the Fisher’s December 2018). exact test will be used to analyze the relationship between two The CEIm that approved the protocol performs a scheduled categorical variables. For the secondary outcomes, changes in the follow-up of the studies by requesting information regarding study variables throughout time (i.e., before and after the intervention) recruitment, study development, and modifications to the will be analyzed using an ANCOVA or linear mixed model to etimate PI. Furthermore, this study’s PI is committed to update the study changes in effect size. No methods to handle missing data (i.e., information (i.e., protocol and status) on the registry imputation) will be applied. A significance level of 5% will (Clinicaltrials.gov). be considered. Data will be analyzed using the latest version of the Participants will be informed of the voluntary nature of the study, Statistical Package for the Social Sciences (SPSS) software (IBM, and their participation will not be compensated. Participants will have Armonk, New York, United States). time to decide whether they will take part in the study. Informed consent forms will assure anonymity and confidentiality regarding adaption, presentation, and data publication. Participants will also Monitoring be informed that they can withdraw from the study without having to give a reason. All participants will sign informed consent forms to A study monitor will visit the participating centers before starting confirm their acceptance to participate in the study. They will also recruitment to ensure that they have the human and material be informed verbally by the study coordinator and researchers before resources and the necessary training for their satisfactory participation any procedure. The authors confirm that no patient/personal in the study and in accordance with the protocol. The research staff of identifiers will appear in any publication, ensuring the anonymity of each unit will attend a training session before the recruitment starts the study participants. and a monthly meeting to verify uniformity in data collection. The role as observer of the study’s nurse PI during the program will facilitate the prompt detection of protocol deviations and data Data access collected in the field notebook will allow the research team to implement the necessary correcting actions. For example, unanswered Study investigators will have access to their local study data during questions during each group session will be collected in a field the research. After data collection is completed, only the manuscript notebook by the research team and answered in the next session. authors and the study’s PI will have access to the data, stored in a Furthermore, inappropriate or inaccurate information shared during trusted digital repository. After study completion, the anonymized sessions will be clarified by the EP in the next session, with the support data will be made available for future research. Patients provided of the research team. informed consent regarding the use of data obtained in this study in A data monitoring committee was deemed unnecessary in this future research. study, as the intervention was anticipated to cause no harm to any of the participants. The EPPC has been implemented for other chronic conditions, such as heart failure, chronic obstructive pulmonary Dissemination planning disease, Chagas disease, and intestinal inflammatory bowel disease, demonstrating the safety of peer-led educational interventions (17, 26, The results of the research study will be published, within the 29). In this regard, this study will not include a safety assessment, as framework of the requirements of the doctoral program, in journals the intervention is not expected to generate any related adverse events. indexed in the JCR. The research team will disseminate the results of Nevertheless, changes in the HADS (23) subscales scores may the study at national and international scientific conferences in the indirectly provide valuable safety information and will be monitored. fields of neurology, MS, and health promotion. Importantly, the Frontiers in Neurology 09 frontiersin.org Robles-Sanchez et al. 10.3389/fneur.2023.1172640 EPPC-MS implemented in Catalonia will become available to the provides knowledge on the disease, symptoms, and care that each target population (MS patients) through disseminating the nurse-led patient needs and favors treatment adherence. Consequently, health intervention to other MS units in Catalonia. Moreover, any literacy entails empowerment over your health, making patients go opportunities to improve its effectiveness identified in this study may from a passive role to a much more active one, who will thus take care be implemented before the dissemination of the intervention. The of getting the best medical care possible, managing their disease and, study protocol, statistical codes, and datasets will be available upon ultimately, will be much more committed to their health condition reasonable request. (10). Health-related empowerment directly impacts QoL, symptom management, emotional status, and the person activation. Regarding the healthcare system, higher empowerment can also reduce the use Discussion of health services (33). Health literacy is beneficial for healthcare professionals as well. This multicenter clinical study aims to implement an intervention Knowing the degree of health literacy in their patients may help the in patients with MS and evaluate its effects on QoL, disease-related nursing team to identify those at a higher risk of suffering knowledge, and self-management related to the health process of the misunderstandings or lack of treatment adherence (34). participants. The intervention consists of educational discussion This study is aligned with the current Health Plan for Catalonia sessions led by an EP and coordinated by a nurse within the Expert 2021–2025 (35) and with the WHO global strategy on integrated Patient Program of Catalonia framework. people-centered health services (36). These especially emphasize the Interventions involving EPPs have proven to be useful before, as need to promote autonomy and adaptation of healthcare systems to well as other patient support programs with similar goals (4–6). provide the best possible care to the elderly or people with However, they have all been based on in-person strategies, where all chronic conditions. participants meet and interact simultaneously and in the same place. This study assessing the intervention effectiveness and EPs’ and The leadership of the nursing coordinating team has facilitated the participants’ satisfaction, will enable to identify opportunities to adaptation of the study to the current COVID-19 pandemic situation, improve the EPPC-MS in its future nurse-led implementation with sessions that will be carried out using a telematic format. This throughout the Catalan territory. Nevertheless, the program may format is new compared with previous studies. At the same time, it benefit from further research aiming to develop validated instruments minimizes potential risks associated with personal contact and there to evaluate the suitability of candidate EPs regarding knowledge and is less use of material resources to carry out the program (rooms, leadership skills, and the disease-related knowledge acquired by spaces, etc.). Despite the decreased COVID-19 restrictions and risks participants compared to non-participants with MS. since this intervention was envisioned, the telematic format is still an advantage for health centers owing to the low economic cost for participant sites, and offers many advantages for program participants Limitations as well. First, it avoids unnecessary travel, facilitating family conciliation. On the other hand, meeting attendance is made easier for Health regulations and recommendations owing to the COVID-19 those people with reduced mobility, some type of difficulty of access, pandemic may alter the study’s schedule due to the restrictions at the or those who live farther away from the site. For working people, the MS units on participants’ attendance to assess inclusion and exclusion potential impact of their attendance on their work life is minimized. criteria. Being a study lasting more than 12 months, the elimination of Because of all this, the program will have a direct influence on the knowledge-induced response of studied participants is guaranteed individuals and their lifestyles, on their education and knowledge of (37). With respect to session attendance and missing data, the health, and on the possible inequities in access to the health system, program will ensure attendance by sending reminders to confirm it which, in turn, will have a direct impact on social health determinants and, if necessary, re-scheduling sessions. Furthermore, the affecting the individual. questionnaires’ receipt from each participant will be monitored and However, it must be considered that there may be a digital gap in reminders will be sent to ensure collection of data at all timepoints. certain groups of patients; therefore, not everybody will potentially Regarding evaluation instruments, a Spanish version of the BICAMS benefit from a remote strategy, and some patient groups may (15) battery has been developed for the Argentine population (15). To be unintentionally excluded due to the intervention telematic format. date, we do not have scales for the population of Spain. Nevertheless, Upon completion of this study, the EPPC-MS will become available to the demographic characteristics of the sample used in Vanotti’s study all MS patients in Catalonia. Future editions of the EPPC-MS may are likely extrapolable to the population’s demographic characteristics consider other intervention formats, including face-to-face and hybrid in the present study. Likewise, instruments evaluating MS-related formats, to reach all the target population. In this regard, the EPPC for knowledge and habits and lifestyle are still missing. Therefore, this other chronic diseases, such as heart failure and chronic obstructive study will evaluate these variables using questionnaires developed by pulmonary disease, was implemented in a face-to-face format and the EPPC and awaiting validation. In this regard, an objective therefore, we anticipate that adapting the EPPC-MS to this format will instrument to evaluate EPs’ skills and knowledge is missing and be feasible (30, 31). therefore, existing differences among EPs may influence this study’s The EP figure favors health literacy. This term encompasses all results. However, we believe that EP planned training before starting health-related knowledge, skills, and experiences that make a the learning sessions may partly even any potential differences. non-health professional individual an expert in their state of health Regarding the study’s design, an approach to the study and how to take care of themselves (32). Health literacy is effective in phenomenon through a pragmatic clinical trial was considered promoting health in patients with chronic diseases, such as MS. It in the planning phase. However, a larger sample size would Frontiers in Neurology 10 frontiersin.org Robles-Sanchez et al. 10.3389/fneur.2023.1172640 be needed to establish comparability, which we deemed difficult Funding to achieve. Of the 7,000 people with MS in Catalonia, the population with severe cognitive or psycho-emotional This study was promoted by the MS unit at Vall d’Hebron impairment must be ruled out, considerably decreasing the Hospital Campus and had no external sponsor. The PhD candidate/ eligible population. Furthermore, this study’s main purpose is to first author was supported through the Strategic Plan for Research implement the innovative intervention, and therefore, a pre-post and Innovation in Health 2016–2020 (PERIS) (ref. BDNS 542793) intervention study design was chosen. In addition, the main goal funded by the Health Department of Catalonia. This study had been is to evaluate an innovative practice rather than to establish partially funded by the Official College of Nurses of Barcelona (www. causal relationships. The intervention aims to improve QoL and coib.cat) as part of the Nurse Research Projects Grants (PRN- self-management skills regarding the health process and the 475/2021). None of the funders were involved in the design of the implementation of a feasible health intervention within the study, manuscript writing or data collection, and will not be involved services catalog of the respective units. Regarding the telematic in data analysis or interpretation and manuscript writing in the nature of the intervention, the research team will provide future. The only funders’ requirement is that any publications informatic support, but will not provide electronic devices, associate with this study must be open access and deposited in an hampering the participation of eligible people with MS who do institutional repository. not own a suitable electronic device. This limitation, together with limited digital skills, may be particularly relevant for elderly candidate participants. Moreover, it may bias patient selection Acknowledgments and consequently, the results from this study may not reflect the outcomes obtained in a face-to-face implementation modality. The research team would like to thank all the study centers and The study will train the units to provide standardized and local investigators for their willingness, support, and participation in programmed peer support groups to people with MS. the study. In turn, the research team would like to express special thanks to Maria Rosario González Gómez and Emilio Cortegoso Lobato for their collaboration and involvement in the development Conclusion of this study protocol as expert patients in multiple sclerosis. The authors would like to thank Sara Cervantes for providing medical This protocol describes a pre-post intervention multicenter writing assistance during the preparation of the final manuscript draft. clinical study in patients with MS, which will be carried out in different MS reference units of Catalonia through an expert patient program with remote working sessions. The study will allow the Conflict of interest evaluation of the impact of the program on participants’ QoL, disease- related knowledge, and self-management of MS. MR-S received speaking or consulting honoraria, participated in scientific activities organized by Merck, Teva, Biogen, Novartis, Sanofi- Genzyme, Celgene, EXCEMED, and Roche, and was awarded the Ethics statement ECTRIMS MS Nurse Training Fellowship Programme and the Strategic Plan for Research and Innovation in Health 2016–2020 (PERIS). CC-M The studies involving human participants were reviewed and has received support for attending congresses from sanofi, merk, teva approved by Clinical Research Ethics Committee of the Hospital and novartis and is funded by a fellowship from the Departament de Universitario de la Vall d’Hebron, PR(AG)334/2021. The patients/ Salut de la Generalitat de Catalunya [SLT017/20/000115, 2021]. MA participants provided their written informed consent to participate in received speaking or consulting honoraria, participated in scientific this study. activities in the last 2 years organized by Bristol-Myers Squibb/Celgene. XM received speaking honoraria and travel expenses for participation in scientific meetings, has been a steering committee member of clinical Author contributions trials or participated in advisory boards of clinical trials in the past years with Abbvie, Actelion, Alexion, Biogen, Bristol-Myers Squibb/Celgene, MR-S: conceptualization, data curation, funding acquisition, EMD Serono, Genzyme, Hoffmann-La Roche, Immunic, Janssen investigation, methodology, project administration, resources, Pharmaceuticals, Medday, Merck, Mylan, Nervgen, Novartis, Sandoz, software, visualization, and roles/writing – original draft. PA-B: Sanofi-Genzyme, Teva Pharmaceutical, TG Therapeutics, Excemed, conceptualization, funding acquisition, investigation, methodology, MSIF and NMSS. JS-G received speaking or consulting honoraria and resources, software, supervision, validation, visualization, writing – attended scientific activities in the last 2 years organized by Merck, Teva, review, and editing. CB-F: conceptualization, data curation, Bial, EXCEMED, Biogen, Celgene, Novartis, Sanofi-Genzyme, and investigation, methodology, software, supervision, validation, Roche; and is the director of the “Revista de Neurologia” (Neurology visualization, roles/writing – original draft, writing – review, and Journal) and an editorial board member of the Multiple Sclerosis Journal. editing. CC-M, MA, EA, RM-D, XM, JS-G, LR-T, and CB-N: LR-T has received speaking or consulting honoraria, attended scientific conceptualization, data curation, investigation, methodology, activities organized by Merck, Teva, Biogen, Novartis, Sanofi, Roche, supervision, validation, visualization, roles/writing – original draft, Bristol-Myers-Squibb, Almirall, and Mylan, and participated in advisory writing – review, and editing. All authors contributed to the article and boards organized by Sanofi, Merck, Roche, Biogen, Novartis, Bristol- approved the submitted version. Myers-Squibb, and Almirall. PA-B, the coordinator of the Expert Patient Frontiers in Neurology 11 frontiersin.org Robles-Sanchez et al. 10.3389/fneur.2023.1172640 Program Catalunya. CC-M has received support for attending congresses Publisher’s note from Bristol Myers Squibb, Sanofi, Merck, and Novartis and is funded by a fellowship from the Departament de Salut de la Generalitat de All claims expressed in this article are solely those of the authors Catalunya [SLT017/20/000115, 2021]. and do not necessarily represent those of their affiliated organizations, The remaining authors declare that the research was conducted in or those of the publisher, the editors and the reviewers. Any product the absence of any commercial or financial relationships that could be that may be evaluated in this article, or claim that may be made by its construed as a potential conflict of interest. manufacturer, is not guaranteed or endorsed by the publisher. References 1. MacLurg K, Reilly P, Hawkins S, Gray O, Evason E, Whittington D. A primary 20. Aymerich M, Guillamón I, Perkal H, Nos C, Porcel J, Berra S, et al. 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