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Contains fulltext : 305294.pdf (Publisher’s version ) (Closed access), Introduction/Objective: People with systemic sclerosis (SSc) face barriers to physical activity. Few studies have described physical activity in SSc, and none have explored physical activity longitudinally during COVID-19. We evaluated physical activity from April 2020 to March 2022 among people with SSc. Methods: The Scleroderma Patient-centred Intervention Network (SPIN) COVID-19 Cohort was launched in April 2020 and included participants from the ongoing SPIN Cohort plus external enrolees. Participants completed measures bi-weekly through July 2020, then every 4 weeks afterwards (28 assessments). Physical activity was assessed via the self-reported International Physical Activity Questionnaire-Elderly. Analyses included estimated means with 95% confidence intervals for physical activity across assessments. Missing data were imputed for main analyses. Sensitivity analyses included evaluating only participants who completed >90% of items for >21 of 28 possible assessments ('completers') and stratified analyses by sex, age, country and SSc subtype. Results: A total of 800 people with SSc enrolled. Mean age was 55.6 (standard deviation (SD) = 12.6) years. Physical activity significantly decreased from April 2020 to March 2021 (standardized mean difference (SMD) = -0.17, 95% confidence interval (CI) = -0.26 to -0.07) and was stable from March 2021 to March 2022 (SMD = -0.05, 95% CI = -0.15 to 0.05). Results were similar for completers and subgroups. The proportion of participants who met World Health Organization minimum physical activity recommendations of at least 150 min of moderate-to-vigorous activity per week ranged from 63% to 82% across assessments. Conclusion: Physical activity decreased by a relatively small amount, on average, across the pandemic. Most participants met recommended physical activity levels., 02 april 2024, 7 p.

Item does not contain fulltext, Objectives: Objectives were to evaluate the sociodemographic and disease-related factors, and coping style associated with social interaction and social appearance anxiety in people with unilateral facial palsy. Methods: Medical data were extracted from electronic health records, and participants completed the Social Interaction Anxiety Scale (SIAS), Social Appearance Anxiety Scale (SAAS), and Coping Orientation to Problems Experienced inventory. Associations of SIAS and SAAS scores with sociodemographic and disease variables, and coping were assessed with multiple linear regression. Results: Among 111 participants (mean age 58.6 years; 59% women), higher age and greater use of emotion-focused coping were associated with lower SIAS scores, whereas greater use of avoidant coping was associated with higher SIAS scores. Higher age, male sex, and greater use of emotion-focused coping were associated with lower SAAS scores, whereas greater use of avoidant coping was associated with higher SAAS scores. Conclusions: Healthcare providers should understand that women and younger people are more likely to have social appearance concerns and that this is not predicted by the objective severity of facial palsy.

Contains fulltext : 237435.pdf (Publisher’s version ) (Open Access), Objective: We evaluated reporting completeness and transparency in randomised controlled trials (RCTs) conducted using administrative data based on 2021 CONSORT Extension for Trials Conducted Using Cohorts and Routinely Collected Data (CONSORT-ROUTINE) criteria. Study Design and Setting: MEDLINE and the Cochrane Methodology Register were searched (2011 and 2018). Eligible RCTs used administrative databases for identifying eligible participants or collecting outcomes. We evaluated reporting based on CONSORT-ROUTINE, which modified eight items from CONSORT 2010 and added five new items. Results: Of 33 included trials (76% used administrative databases for outcomes, 3% for identifying participants, 21% both), most were conducted in the United States (55%), Canada (18%), or the United Kingdom (12%). Of eight items modified in the extension; six were adequately reported in a majority (>50%) of trials. For the CONSORT-ROUTINE modification portion of those items, three items were reported adequately in >50% of trials, two in <50%, two only applied to some trials, and one only had wording modifications and was not evaluated. For five new items, four that address use of routine data in trials were reported inadequately in most trials. Conclusion: How administrative data are used in trials is often sub-optimally reported. CONSORT-ROUTINE uptake may improve reporting.

Contains fulltext : 241655.pdf (Publisher’s version ) (Open Access)

Item does not contain fulltext, Background: Systemic sclerosis (scleroderma; SSc) is a rare autoimmune connective tissue disease. Functional impairment of hands is common. The Scleroderma Patient-centered Intervention Network (SPIN)-HAND trial compared effects of offering access to an online self-guided hand exercise program to usual care on hand function (primary) and functional health outcomes (secondary) in people with SSc with at least mild hand function limitations. Methods: The pragmatic, two-arm, parallel-group cohort multiple randomized controlled trial was embedded in the SPIN Cohort. Cohort participants with Cochin Hand Function Scale (CHFS) scores ≥ 3 and who indicated interest in using the SPIN-HAND Program were randomized (3:2 ratio) to an offer of program access or to usual care (targeted N = 586). The SPIN-HAND program consists of 4 modules that address (1) thumb flexibility and strength; (2) finger bending; (3) finger extension; and (4) wrist flexibility and strength. The primary outcome analysis compared CHFS scores 3 months post-randomization between participants offered versus not offered the program. Secondary outcomes were CHFS scores 6 months post-randomization and functional health outcomes (Patient-Reported Outcomes Measurement Information System profile version 2.0 domain scores) 3 and 6 months post-randomization. Results: In total, 466 participants were randomized to intervention offer (N = 280) or usual care (N = 186). Of 280 participants offered the intervention, 170 (61%) consented to access the program. Of these, 117 (69%) viewed at least one hand exercise instruction video and 77 (45%) logged into the program website at least 3 times. In intent-to-treat analyses, CHFS scores were 1.2 points lower (95% CI - 2.8 to 0.3) for intervention compared to usual care 3 months post-randomization and 0.1 points lower (95% CI - 1.8 to 1.6 points) 6 months post-randomization. There were no statistically significant differences in other outcomes. Conclusion: The offer to use the SP

Contains fulltext : 237218.pdf (Publisher’s version ) (Open Access), Objective: Registries are important data sources for randomised controlled trials (RCTs), but reporting of how they are used may be inadequate. The objective was to describe the current adequacy of reporting of RCTs using registries. Study Design and Setting: We used a database of trials using registries from a scoping review supporting the development of the 2021 CONSORT extension for Trials Conducted Using Cohorts and Routinely Collected Data (CONSORT-ROUTINE). Reporting completeness of 13 CONSORT-ROUTINE items was assessed. Results: We assessed reports of 47 RCTs that used a registry, published between 2011 and 2018. Of the 13 CONSORT-ROUTINE items, 6 were adequately reported in at least half of reports (2 in at least 80%). The 7 other items were related to routinely collected data source eligibility (32% adequate), data linkage (8% adequate), validation and completeness of data used for outcome assessment (8% adequate), validation and completeness of data used for participant recruitment (0% adequate), participant flow (9% adequate), registry funding (6% adequate) and interpretation of results in consideration of registry use (25% adequate). Conclusion: Reporting of trials using registries was often poor, particularly details on data linkage and quality. Better reporting is needed for appropriate interpretation of the results of these trials.

Contains fulltext : 284227.pdf (Publisher’s version ) (Open Access), Background: More people with rare diseases likely receive disease education and emotional and practical support from peer-led support groups than any other way. Most rare-disease support groups are delivered outside of the health care system by untrained leaders. Potential benefits may not be achieved and harms, such as dissemination of inaccurate information, may occur. Our primary objective was to evaluate the effects of a rare-disease support group leader education program, which was developed collaboratively by researchers, peer support group leaders, and patient organization leaders, compared to waitlist control, on peer leader self-efficacy among scleroderma support group leaders. Methods: The trial was a pragmatic, two-arm partially nested randomised controlled trial with 1:1 allocation into intervention or waitlist control. Eligible participants were existing or candidate peer support group leaders affiliated with a scleroderma patient organization. Leader training was delivered in groups of 5-6 participants weekly for 13 weeks in 60-90 min sessions via the GoToMeeting® videoconferencing platform. The program included 12 general leader training modules and one module specific to scleroderma. Primary outcome was leader self-efficacy, measured by the Support Group Leader Self-efficacy Scale (SGLSS) immediately post-intervention. Secondary outcomes were leader self-efficacy 3 months post-intervention; emotional distress, leader burnout, and volunteer satisfaction post-intervention and 3 months post-intervention; and program satisfaction among intervention participants post-intervention. Results: One hundred forty-eight participants were randomised to intervention (N = 74) or waitlist (N = 74). Primary outcome data were provided by 146 (99%) participants. Mean number of sessions attended was 11.4 (standard deviation = 2.6). Mean program satisfaction score (CSQ-8) was 30.3 (standard deviation = 3.0; possible range 8-32). Compared to waitlist control, leader self

Contains fulltext : 237219.pdf (Publisher’s version ) (Open Access), Objective: To describe characteristics of randomised controlled trials (RCTs) conducted using electronic health records (EHRs), including completeness and transparency of reporting assessed against the 2021 CONSORT Extension for RCTs Conducted Using Cohorts and Routinely Collected Data (CONSORT-ROUTINE) criteria. Study design: MEDLINE and Cochrane Methodology Register were searched for a sample of RCTs published from 2011–2018. Completeness of reporting was assessed in a random sample using a pre-defined coding form. Results 183 RCT publications were identified; 122 (67%) used EHRs to identify eligible participants, 139 (76%) used the EHR as part of the intervention and 137 (75%) to ascertain outcomes. When 60 publications were evaluated against the CONSORT 2010 item and the corresponding extension for the 8 modified items, four items were 'adequately reported' for the majority of trials. Five new reporting items were identified for the CONSORT-ROUTINE extension; when evaluated, one was 'adequately reported', three were reported 'inadequately or not at all', the other 'partially'. There were, however, some encouraging signs with adequate and partial reporting of many important items, including descriptions of trial design, the consent process, outcome ascertainment and interpretation. Conclusion: Aspects of RCTs using EHRs are sub-optimally reported. Uptake of the CONSORT-ROUTINE Extension may improve reporting.

Item does not contain fulltext, Objective: To support physical activity among people with systemic sclerosis (SSc; scleroderma), we sought to determine the (1) prevalence and importance of barriers and (2) likelihood of using possible facilitators. Methods: We invited 1,707 participants from an international SSc cohort to rate the (1) importance of 20 barriers (14 medical; 4 social or personal; 1 lifestyle; 1 environmental); and (2) likelihood of using 91 corresponding barrier-specific and 12 general facilitators. Results: Among 721 respondents, 13 barriers were experienced by ≥25% of participants, including 2 (fatigue, Raynaud's) rated 'important' or 'very important' by ≥50% of participants, 7 (joint stiffness and contractures, shortness of breath, gastrointestinal problems, difficulty grasping, pain, muscle weakness and mobility limitations, low motivation) by 26-50%, and 4 by <26%. Overall, 23 (18 medical-related) of 103 facilitators were rated by ≥75% as 'likely' or 'very likely' to use among those who experienced corresponding barriers; these facilitators focused on adapting exercise (e.g., using controlled, slow movement), taking care of one’s body (e.g., stretching), keeping warm (e.g., wearing gloves), and protecting skin (e.g., covering ulcers). Among those who had previously tried the facilitator, all facilitators were rated by ≥50% as 'likely' or 'very likely' to use; among those with the barrier who had not tried the facilitator, only 12 of 103 were rated by >50% as 'likely' or 'very likely'. Conclusion: Medical-related physical activity barriers were common and considered important. Facilitators considered as most likely to be used involved adapting exercise, taking care of one’s body, keeping warm, and protecting skin.

Contains fulltext : 250298.pdf (Publisher’s version ) (Open Access), Objective: Systemic sclerosis (SSc) has significant psychosocial implications. We aimed to evaluate the proportion of participants in a large international SSc cohort who used mental health services in a 3-month period and to evaluate demographic, psychological, and disease-specific factors associated with use. Methods: Baseline data of participants enrolled in the Scleroderma Patient-Centered Intervention Network Cohort were analyzed. We determined the proportion that used mental health services and the source of services in the 3 months prior to enrollment. Multivariable logistic regression was used to identify variables associated with service use. Results: Of the 2319 participants included in the analysis, 417 (18%) used mental health services in the 3 months prior to enrollment. General practitioners were the most common mental health service providers (59%), followed by psychologists (25%) and psychiatrists (19%). In multivariable analysis, mental health service use was independently associated with higher education (odds ratio [OR] 1.07, 95% confidence interval [CI] 1.03-1.11), smoking (OR 1.06, 95% CI 1.02-1.11), being retired (OR 0.60, 95% CI 0.38-0.93), having limited SSc (OR 1.39, 95% CI 1.02-1.89), and having higher anxiety symptom scores (OR 1.04, 95% CI 1.03-1.06) and lower self-efficacy scores (OR 0.90, 95% CI 0.83-0.97). Variables not significantly associated included age, race, disease manifestations, depression symptom scores, and body image distress. Conclusion: About 18% of participants in a large international cohort received mental health services in a 3-month period, of whom the majority received these services from a general practitioner.

Contains fulltext : 247598.pdf (Publisher’s version ) (Open Access), Background: The Scleroderma Patient-centered Intervention Network (SPIN) developed an online self-management program (SPIN-SELF) designed to improve disease-management self-efficacy in people with systemic sclerosis (SSc, or scleroderma). The aim of this study was to evaluate feasibility aspects for conducting a full-scale randomized controlled trial (RCT) of the SPIN-SELF Program. Methods: This feasibility trial was embedded in the SPIN Cohort and utilized the cohort multiple RCT design. In this design, at the time of cohort enrollment, cohort participants consent to be assessed for trial eligibility and randomized prior to being informed about the trial. Participants in the intervention arm are informed and provide consent, but not the control group. Forty English-speaking SPIN Cohort participants from Canada, the USA, or the UK with low disease-management self-efficacy (Self-Efficacy for Managing Chronic Disease Scale [SEMCD] score <= 7) who were interested in using an online self-management program were randomized (3:2 ratio) to be offered the SPIN-SELF Program or usual care for 3 months. Program usage was examined via automated usage logs. User satisfaction was assessed with semi-structured interviews. Trial personnel time requirements and implementation challenges were logged. Results: Of 40 SPIN Cohort participants randomized, 26 were allocated to SPIN-SELF and 14 to usual care. Automated eligibility and randomization procedures via the SPIN Cohort platform functioned properly, except that two participants with SEMCD scores > 7 (scores of 7.2 and 7.3, respectively) were included, which was caused by a system programming error that rounded SEMCD scores. Of 26 SPIN Cohort participants offered the SPIN-SELF Program, only 9 (35%) consented to use the program. Usage logs showed that use of the SPIN-SELF Program was low: 2 of 9 users (22%) logged into the program only once (median = 3), and 4 of 9 (44%) accessed none or only 1 of the 9 program’s modules (median = 2

Item does not contain fulltext, Objective People with systemic sclerosis (SSc) often report substantial burden from appearance changes. We aimed to estimate the patient acceptable symptom state (PASS) for burden from appearance changes in people with SSc. Methods We conducted a secondary analysis of the SCISCIF II study, a cross-sectional survey of 113 patients with SSc from France enrolled in the Scleroderma Patient-centered Intervention Network Cohort. Burden from appearance changes was assessed with a self-administered numeric rating scale (0, no burden to 10, maximal burden). Acceptability of the symptom state was assessed with a specific anchoring question. Participants who answered "yes" were in the group of patients who considered their symptom state as acceptable. The PASS for the burden from appearance changes was estimated with the 75th percentile method. Results Assessments of burden from appearance changes and answers to the anchoring question were available in 82/113 (73%) participants from the SCISCIF II study. Mean age was 55.9 (14.3) years, disease duration 9.6 (6.5) years and 32/82 (40%) participants had diffuse cutaneous SSc. The PASS estimate for the burden from appearance changes was 4.8 (95% CI from 1.0 to 7.0) of 10 points. Conclusion Our study provides a PASS estimate for burden from appearance changes. Our estimate could serve as a binary response criterion to assess the efficacy of treatments targeting burden from appearance changes.

Radboud Universiteit Nijmegen, 01 juli 2015, Promotores : Hoogen, F.H.J. van den, Vliet Vlieland, T.P.M. Co-promotores : Ende, C.H.M. van den, Kwakkenbos, C.M.C., Contains fulltext : 141377.pdf (publisher's version ) (Open Access)

Radboud Universiteit Nijmegen, 16 januari 2013, Promotor : Becker, E.S. Co-promotores : Ende, C.H.M. van den, Hoogen, F.H.J. van den, Contains fulltext : 101023.pdf (publisher's version ) (Open Access)