The association of resilience and positive mental health in systemic sclerosis: A Scleroderma Patient-centered Intervention Network (SPIN) cohort cross-sectional study.

OBJECTIVE: A previous study using Scleroderma Patient-centered Intervention Network (SPIN) Cohort data identified five classes of people with systemic sclerosis (also known as scleroderma) based on patient-reported somatic (fatigue, pain, sleep) and mental health (anxiety, depression) symptoms and compared indicators of disease severity between classes. Across four classes ("low", "normal", "high", "very high"), there were progressively worse somatic and mental health outcomes and greater disease severity. The fifth ("high/low") class, however, was characterized by high disease severity, fatigue, pain, and sleep but low mental health symptoms. We evaluated resilience across classes and compared resilience between classes. METHODS: Cross-sectional study. SPIN Cohort participants completed the 10-item Connor-Davidson-Resilience Scale (CD-RISC) and PROMIS v2.0 domains between August 2022 and January 2023. We used latent profile modeling to identify five classes as in the previous study and multiple linear regression to compare resilience levels across classes, controlling for sociodemographic and disease variables. RESULTS: Mean CD-RISC score (N = 1054 participants) was 27.7 (standard deviation = 7.3). Resilience decreased progressively across "low" to "normal" to "high" to "very high" classes (mean 4.7 points per step). Based on multiple regression, the "high/low" class exhibited higher resilience scores than the "high" class (6.0 points, 95% confidence interval [CI] 4.9 to 7.1 points; standardized mean difference = 0.83, 95% CI 0.67 to 0.98). CONCLUSIONS: People with worse disease severity and patient-reported outcomes reported substantially lower resilience, except a class of people with high disease severity, fatigue, pain, and sleep disturbance but positive mental health and high resilience.

The Activity Management Inventory for Pain (AMI-P): Initial Development and Validation of a Questionnaire Based on Operant Learning and Energy Conservation Models of Activity Management.

OBJECTIVES: Activity management is an important treatment component in chronic pain programs. However, there are shortcomings in measures of this construct, leading to inconsistencies in research findings. Here, we describe the development of the Activity Management Inventory for Pain (AMI-P). MATERIALS AND METHODS: The AMI-P was developed by a group of international researchers with extensive expertise in both chronic pain and activity management. The initial evaluation of the AMI-P items included 2 studies that were both conducted in Canadian tertiary pain care centers. RESULTS: The resulting 20-item measure has 3 behavior scales (Rest, Alternating Activity, and Planned Activity), and 4 goal scales (Feel Less Pain, Get More Done, Complete the Task, and Save Energy). The behavior scales evidenced marginal to good internal consistency and test-retest reliability, and a moderate positive association with an existing pacing measure. The Rest and Alternating Activity scales were associated with greater pain interference, the Alternating Activity and Planned Activity scales were associated with less satisfaction with social roles, and the Planned Activity scale was associated with fewer depressive symptoms. The Alternating Activity scale increased significantly from pretreatment to posttreatment. All goal scales were positively associated with all behavior scales. The Feel Less Pain goal scale was positively associated with measures of avoidance and pain interference, while the Get More Done goal scale was negatively associated with measures of depressive symptoms and overdoing. DISCUSSION: The findings support the reliability and validity of the AMI-P scales, while also highlighting the complexity and multidimensional aspects of activity management.

Patterns of patient-reported symptoms and association with sociodemographic and systemic sclerosis disease characteristics: a scleroderma Patient-centered Intervention Network (SPIN) Cohort cross-sectional study.

Background: Systemic sclerosis is a heterogenous disease in which little is known about patterns of patient-reported symptom clusters. We aimed to identify classes of individuals with similar anxiety, depression, fatigue, sleep disturbance, and pain symptoms and to evaluate associated sociodemographic and disease-related characteristics. Methods: This multi-centre cross-sectional study used baseline data from Scleroderma Patient-centered Intervention Network Cohort participants enrolled from 2014 to 2020. Eligible participants completed the PROMIS-29 v2.0 measure. Latent profile analysis was used to identify homogeneous classes of participants based on patterns of anxiety, depression, fatigue, sleep disturbance, and pain scores. Sociodemographic and disease-related characteristics were compared across classes. Findings: Among 2212 participants, we identified five classes, including four classes with "Low" (565 participants, 26%), "Normal" (651 participants, 29%), "High" (569 participants, 26%), or "Very High" (193 participants, 9%) symptom levels across all symptoms. Participants in a fifth class, "High Fatigue/Sleep/Pain and Low Anxiety/Depression" (234 participants, 11%) had similar levels of fatigue, sleep disturbance, and pain as in the "High" class but low anxiety and depression symptoms. There were significant and substantive trends in sociodemographic characteristics (age, education, race or ethnicity, marital or partner status) and increasing disease severity (diffuse disease, tendon friction rubs, joint contractures, gastrointestinal symptoms) across severity-based classes. Disease severity and sociodemographic characteristics of "High Fatigue/Sleep/Pain and Low Anxiety/Depression" class participants were similar to the "High" severity class. Interpretation: Most people with systemic sclerosis can be classified by levels of patient-reported symptoms, which are consistent across symptoms and highly associated with sociodemographic and disease-related variables, except for one group which reports low mental health symptoms despite high levels of other symptoms and substantial disease burden. Studies are needed to better understand resilience in systemic sclerosis and to identify and facilitate implementation of cognitive and behavioural strategies to improve coping and overall quality of life. Funding: National Institute of Nursing Research (F31NR019007), Canadian Institutes of Health Research, Arthritis Society Canada, the Lady Davis Institute for Medical Research, the Jewish General Hospital Foundation, McGill University, Scleroderma Society of Ontario, Scleroderma Canada, Sclérodermie Québec, Scleroderma Manitoba, Scleroderma Atlantic, Scleroderma Association of BC, Scleroderma SASK, Scleroderma Australia, Scleroderma New South Wales, Scleroderma Victoria, and Scleroderma Queensland.

Validity, Reliability, and Differential Item Functioning of English and French Versions of the 10-Item Connor-Davidson Resilience Scale in Systemic Sclerosis: A Scleroderma Patient-Centered Intervention Network Cohort Study.

OBJECTIVE: Some individuals with systemic sclerosis (SSc) report positive mental health, despite severe disease manifestations, which may be associated with resilience, but no resilience measure has been validated in SSc. This study was undertaken to assess the validity, reliability, and differential item functioning (DIF) between English- and French-language versions of the 10-item Connor-Davidson Resilience Scale (CD-RISC-10) in SSc. METHODS: Eligible participants were enrolled in the Scleroderma Patient-centered Intervention Network Cohort and completed the CD-RISC-10 between August 2022 and January 2023. We used confirmatory factor analysis (CFA) to evaluate the CD-RISC-10 factor structure and conducted DIF analysis across languages with Multiple Indicators Multiple Causes models. We tested convergent validity with another measure of resilience and measures of self-esteem and depression and anxiety symptoms. We assessed internal consistency and test-retest reliability using Cronbach's alpha and intraclass correlation coefficient (ICC). RESULTS: A total of 962 participants were included in this analysis. CFA supported a single-factor structure (Tucker-Lewis index = 0.99, comparative fit index = 0.99, root mean square error of approximation = 0.08 [90% confidence interval (90% CI) 0.07, 0.09]). We found no meaningful DIF. Internal consistency was high (α = 0.93 [95% CI 0.92, 0.94]), and we found that correlations with other measures of psychological functioning were moderate to large (|r| = 0.57-0.78) and confirmed study hypotheses. The scale showed good 1-2-week test-retest reliability (ICC 0.80 [95% CI 0.75, 0.85]) in a subsample of 230 participants. CONCLUSION: The CD-RISC-10 is a valid and reliable measure of resilience in SSc, with score comparability across English and French versions.

Randomized feasibility trial of the Scleroderma Patient-centered Intervention Network hand exercise program (SPIN-HAND).

Purpose: The Scleroderma Patient-centered Intervention Network (SPIN) online hand exercise program (SPIN-HAND), is an online self-help program of hand exercises designed to improve hand function for people with scleroderma. The objective of this feasibility trial was to evaluate aspects of feasibility for conducting a full-scale randomized controlled trial of the SPIN-HAND program. Materials and Methods: The feasibility trial was embedded in the SPIN cohort and utilized the cohort multiple randomized controlled trial (cmRCT) design. In the cmRCT design, at the time of cohort enrollment, cohort participants consent to be assessed for trial eligibility and randomized prior to being informed about trials conducted using the cohort. When trials were conducted in the cohort, participants randomized to the intervention were informed and consented to access the intervention. Participants randomized to control were not informed that they have not received an intervention. All participants eligible and randomized to participate in the trial were included in analyses on an intent-to-treat basis. Cohort participants with a Cochin Hand Function Scale score ≥ 3/90 and an interest in using an online hand-exercise intervention were randomized (1:1 ratio) to be offered as usual care plus the SPIN-HAND Program or usual care for 3 months. User satisfaction was assessed with semi-structured interviews. Results: Of the 40 randomized participants, 24 were allocated to SPIN-HAND and 16 to usual care. Of 24 participants randomized to be offered SPIN-HAND, 15 (63%) consented to use the program. Usage of SPIN-HAND content among the 15 participants who consented to use the program was low; only five (33%) logged in more than twice. Participants found the content relevant and easy to understand (satisfaction rating 8.5/10, N = 6). Automated eligibility and randomization procedures via the SPIN Cohort platform functioned properly. The required technical support was minimal. Conclusions: Trial methodology functioned as designed, and the SPIN-HAND Program was feasibly delivered; however, the acceptance of the offer and use of program content among accepters were low. Adjustments to information provided to potential participants will be implemented in the full-scale SPIN-HAND trial to attempt to increase offer acceptance.

Randomized feasibility trial of the Scleroderma Patient-centered Intervention Network Self-Management (SPIN-SELF) Program.

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). CONCLUSIONS: The results of this study will lead to substantial changes for the planned full-scale RCT of the SPIN-SELF Program that we will incorporate into a planned additional feasibility trial with progression to a full-scale trial. These changes include transitioning to a conventional RCT design with pre-randomization consent and supplementing the online self-help with peer-facilitated videoconference-based groups to enhance engagement. TRIAL REGISTRATION: clinicaltrials.gov , NCT03914781 . Registered 16 April 2019.

The Scleroderma Patient-centered Intervention Network Self-Management (SPIN-SELF) Program: protocol for a two-arm parallel partially nested randomized controlled feasibility trial with progression to full-scale trial.

BACKGROUND: Systemic sclerosis (scleroderma; SSc) is a rare autoimmune connective tissue disease. We completed an initial feasibility trial of an online self-administered version of the Scleroderma Patient-centered Intervention Network Self-Management (SPIN-SELF) Program using the cohort multiple randomized controlled trial (RCT) design. Due to low intervention offer uptake, we will conduct a new feasibility trial with progression to full-scale trial, using a two-arm parallel, partially nested RCT design. The SPIN-SELF Program has also been revised to include facilitator-led videoconference group sessions in addition to online material. We will test the group-based intervention delivery format, then evaluate the effect of the SPIN-SELF Program on disease management self-efficacy (primary) and patient activation, social appearance anxiety, and functional health outcomes (secondary). METHODS: This study is a feasibility trial with progression to full-scale RCT, pending meeting pre-defined criteria, of the SPIN-SELF Program. Participants will be recruited from the ongoing SPIN Cohort ( http://www.spinsclero.com/en/cohort ) and via social media and partner patient organizations. Eligible participants must have SSc and low to moderate disease management self-efficacy (Self-Efficacy for Managing Chronic Disease (SEMCD) Scale score ≤ 7.0). Participants will be randomized (1:1 allocation) to the group-based SPIN-SELF Program or usual care for 3 months. The primary outcome in the full-scale trial will be disease management self-efficacy based on SEMCD Scale scores at 3 months post-randomization. Secondary outcomes include SEMCD scores 6 months post-randomization plus patient activation, social appearance anxiety, and functional health outcomes at 3 and 6 months post-randomization. We will include 40 participants to assess feasibility. At the end of the feasibility portion, stoppage criteria will be used to determine if the trial procedures or SPIN-SELF Program need important modifications, thereby requiring a re-set for the full-scale trial. Otherwise, the full-scale RCT will proceed, and outcome data from the feasibility portion will be utilized in the full-scale trial. In the full-scale RCT, 524 participants will be recruited. DISCUSSION: The SPIN-SELF Program may improve disease management self-efficacy, patient activation, social appearance anxiety, and functional health outcomes in people with SSc. SPIN works with partner patient organizations around the world to disseminate its programs free-of-charge. TRIAL REGISTRATION: ClinicalTrials.gov NCT04246528 . Registered on 27 January 2020.

Pain and Self-Efficacy Among Patients With Systemic Sclerosis: A Scleroderma Patient-Centered Intervention Network Cohort Study.

BACKGROUND: Pain is one of the most common symptoms affecting patients with systemic sclerosis; however, little is known about the relationship between self-efficacy and pain and changes in pain over time. OBJECTIVES: The purpose of this study was to describe the relationships between self-efficacy and pain in patients with systemic sclerosis, as well as determine whether changes in self-efficacy mediate changes in pain. METHODS: A prospective longitudinal study was conducted using data from the Scleroderma Patient-Centered Intervention Network Cohort. The baseline sample included 1,903 adults, with a trajectory subsample of 427 who completed 3-month assessments across 3 years. Hierarchical (sequential) forward multivariable regression, covarying for participant characteristics, was conducted to determine the association between self-efficacy and patient characteristics on pain outcomes. Trajectory models, covarying for participant characteristics, were used to examine changes in self-efficacy and pain outcomes across time and whether self-efficacy mediated the pain trajectories. RESULTS: Mean time since diagnosis was 9.5 years, with 39.2% diagnosed with diffuse cutaneous systemic sclerosis. Greater self-efficacy was associated with less pain interference and intensity. Increasing age, female gender, finger ulcers, and small joint contractures were related to greater pain interference and intensity. Esophageal gastrointestinal symptoms were associated with more pain interference. Self-efficacy and pain trajectories remained stable across time, and self-efficacy did not mediate the pain trajectories. DISCUSSION: This study identified self-efficacy, age, gender, finger ulcers, small joint contractures, and esophageal gastrointestinal symptoms as important correlates associated with pain in patients with systemic sclerosis. In addition, this study found that self-efficacy and pain outcomes remained stable over time, providing important insights into the longitudinal pain experiences of patients with systemic sclerosis.

Identifying barriers and facilitators to physical activity for people with scleroderma: a nominal group technique study.

PURPOSE: People with systemic sclerosis (scleroderma) face difficulties being physically active. This study identified physical activity barriers and facilitators experienced by people with scleroderma. MATERIALS AND METHODS: We conducted nominal group technique sessions with scleroderma patients who shared physical activity barriers, barrier-specific facilitators, and general facilitators. Participants rated importance of barriers and likelihood of using facilitators from 0 to 10, and indicated whether they had tried facilitators. Barriers and facilitators across sessions were merged to eliminate overlap; edited by investigators, patient advisors, and clinicians; and categorized using qualitative content analysis. RESULTS: We conducted 9 sessions (n = 41 participants) and initially generated 181 barriers, 457 barrier-specific facilitators, and 20 general facilitators. The number of consolidated barriers (barrier-specific facilitators in parentheses) per category were: 14 (61) for health and medical; 4 (23) for social and personal; 1 (3) for time, work, and lifestyle; and 1 (4) for environmental. There were 12 consolidated general facilitators. The consolidated items with ≥1/3 of participants' ratings ≥8 were: 15 barriers, 69 barrier-specific facilitators, and 9 general facilitators. CONCLUSIONS: Scleroderma patients reported many barriers related to health and medical aspects of scleroderma and several barriers in other categories. They reported facilitators to remain physically active despite the barriers.Implications for RehabilitationPeople with scleroderma experience difficulty being physically active due to the diverse and often severe manifestations of the disease, including involvement of the skin, musculoskeletal system, and internal organs.In addition to regular care of scleroderma-related symptoms, patients overcome many exercise challenges by selecting physical activities that are comfortable for them, adjusting the intensity and duration of activities, adapting activities, and using adapted equipment or other materials to reduce discomfort.Rehabilitation professionals should help people with scleroderma to tailor activity options to their capacity and needs when providing care and advice to promote physical activity.

The Scleroderma Patient-Centered Intervention Network Self-Management Program: Protocol for a Randomized Feasibility Trial.

BACKGROUND: Systemic sclerosis (SSc), or scleroderma, is a rare disease that often results in significant disruptions to activities of daily living and can negatively affect physical and psychological well-being. Because there is no known cure, SSc treatment focuses on reducing symptoms and disability and improving health-related quality of life (HRQoL). Self-management programs are known to increase self-efficacy for disease management in many chronic diseases. The Scleroderma Patient-centered Intervention Network (SPIN) developed a Web-based self-management program (SPIN self-management; SPIN-SELF) to increase self-efficacy for disease management and to improve HRQoL for patients with SSc. OBJECTIVE: The proposed study aims to assess the feasibility of conducting a full-scale randomized controlled trial (RCT) of the SPIN-SELF program by evaluating the trial implementation processes, required resources and management, scientific aspects, and participant acceptability and usage of the SPIN-SELF program. METHODS: The SPIN-SELF feasibility trial will be conducted via the SPIN Cohort. The SPIN Cohort was developed as a framework for embedded pragmatic trials using the cohort multiple RCT design. In total, 40 English-speaking SPIN Cohort participants with low disease management self-efficacy (Self-Efficacy for Managing Chronic Disease Scale score ≤7), who have indicated interest in using a Web-based self-management program, will be randomized with a 3:2 ratio into the SPIN-SELF program or usual care for 3 months. Feasibility outcomes include trial implementation processes, required resources and management, scientific aspects, and patient acceptability and usage of the SPIN-SELF program. RESULTS: Enrollment of the 40 participants occurred between July 5, 2019, and July 27, 2019. By November 25, 2019, data collection of trial outcomes was completed. Data analysis is underway, and results are expected to be published in 2020. CONCLUSIONS: The SPIN-SELF program is a self-help tool that may improve disease-management self-efficacy and improve HRQoL in patients with SSc. The SPIN-SELF feasibility trial will ensure that trial methodology is robust, feasible, and consistent with trial participant expectations. The results will guide adjustments that need to be implemented before undertaking a full-scale RCT of the SPIN-SELF program. INTERNATIONAL REGISTERED REPORT IDENTIFIER (IRRID): DERR1-10.2196/16799.

Pain-Related Activity Management Patterns as Predictors of Treatment Outcomes in Patients with Fibromyalgia Syndrome.

OBJECTIVES: This study sought to determine if pre- to post-treatment changes in pain-related activity patterns (i.e., overdoing, avoidance, and pacing) were associated with pre- to post-treatment changes in function (i.e., pain interference, psychological function, and physical function) in patients with fibromyalgia syndrome who participated in either an operant learning- or an energy conservation-based training in activity management. METHODS: Sixty-nine patients with fibromyalgia syndrome participated in an activity management treatment (32 in an operant learning group and 37 in an energy conservation group). Outcomes were assessed at pre- and post-treatment, and patients provided demographic information and completed measures assessing pain intensity, pain interference, psychological function, physical function, and pain management activity patterns. Three linear hierarchical regression analyses predicting changes in pain outcomes from changes in pacing, overdoing, and avoidant activity patterns were performed. RESULTS: Changes in pain-related activity patterns made significant contributions to the prediction of changes in patients' function. Specifically: (a) increases in overdoing predicted reductions in pain interference; (b) decreases in avoidance predicted improvements in psychological function; and (c) increases in pacing predicted improvements in physical function. CONCLUSIONS: This study provides support for a role of activity management treatments in improved adjustment to chronic pain. Research is needed to replicate and extend these findings in order to build an empirical basis for developing more effective chronic pain treatments for facilitating improved physical and psychological function in individuals with chronic pain.

Factors associated with patient-reported likelihood of using online self-care interventions: a Scleroderma Patient-centered Intervention Network (SPIN) cohort study.

OBJECTIVES: The Scleroderma Patient-centered Intervention Network (SPIN) Cohort uses the cohort multiple randomised controlled trial design to embed trials of online self-care interventions for people living with systemic sclerosis (SSc; scleroderma). To offer interventions to patients interested in using them, participants complete signalling items that query about the likelihood that patients would agree to participate in nine different hypothetical online programmes addressing common SSc-related problems. It is not known what factors influence patient-reported interest in participating in a particular online intervention and if intervention-specific signalling questions provide unique information or replicate broader characteristics, such as overall willingness to participate or self-efficacy. This study assessed factors that explain responses to intervention-specific signalling items. DESIGN: Cross-sectional survey. SETTING: SPIN Cohort participants enrolled at 42 centres from Canada, the USA, the UK, France, Spain and Mexico who completed study questionnaires from March 2014 to January 2018 were included. MEASURES: Demographic and disease characteristics, self-efficacy and symptoms related to each specific intervention were completed in addition to signalling items. General likelihood of using interventions was calculating by taking the mean score of the remaining signalling questions. PARTICIPANTS: 1060 participants with complete baseline data were included in the analyses. RESULTS: For all individual signalling questions, controlling for other variables, the mean of the remaining signalling questions was the strongest predictor (standardised regression coefficient ß from 0.61 (sleep) to 0.80 (self-management)). Smaller, but statistically significant, associations were found with the symptom associated with the respective signalling question and with general self-efficacy for 7 of 9 signalling questions. CONCLUSIONS: The main factor associated with patients' interest in participating in a disease-specific online self-care intervention is their general interest in participating in online interventions. Factors that may influence this general interest should be explored and taken into consideration when inviting patients to try online interventions.

Pain and affective distress in arthritis: relationship to immunity and inflammation.

INTRODUCTION: Most arthritides are associated with pain and psychological distress (clinically significant depression and anxiety). Pain and depression are mutually exacerbating; both may continue even when joint involvement appears well controlled. Area covered: There is strong evidence that arthritis-related stress impacts the central nervous system and, together with peripheral inflammatory changes, can cause central sensitization that can lead to chronic pain and worsening of affective distress. Cytokines and chemokines participate both in joint inflammation and in central sensitization. We review evidence of these relationships in five arthritides, namely rheumatoid arthritis, ankylosing spondylitis, psoriatic arthritis, gout, and in osteoarthritis of the hips and knees. Central sensitization in these conditions results in long-lasting pain and psychological distress. Expert commentary: Chronic pain and depression are important but often neglected in the clinical assessment and treatment of arthritis. The potential role of biologic cytokines and Janus kinase inhibitors in dealing with these symptoms needs further study.

Challenges and strategies for coping with scleroderma: implications for a scleroderma-specific self-management program.

Purpose: The purpose of this study was to explore challenges faced by patients with systemic sclerosis, also called scleroderma, in coping with their disease and the strategies they used to face those challenges. Method: Five focus groups were held with scleroderma patients (4 groups, n = 34) and health care professionals who have experience treating scleroderma (1 group, n = 8). Participants' discussions were recorded, transcribed and analyzed using thematic analysis. Results: Participants reported challenges accessing information (e.g., knowledgeable specialists), dealing with negative emotions (e.g., stress due to misunderstandings with loved ones), and accessing resources (e.g., helpful products or devices). Strategies for overcoming challenges were also discussed (e.g., advocating for own needs). Conclusion: When faced with significant challenges while coping with scleroderma, patients develop strategies to manage better and improve their quality of life. To help them cope, patients would benefit from easier access to supportive interventions, including tailored scleroderma self-management programs. Although the challenges experienced by patients with scleroderma are unique, findings from this study might help better understand patients' perspectives regarding coping and disease management for other chronic diseases as well. Implications for Rehabilitation People living with rare diseases, including the rare autoimmune disease scleroderma, face unique challenges and often do not have access to disease-specific educational or other support resources. People with scleroderma report that they face challenges in accessing information, including knowledgeable healthcare providers; managing difficult social interactions and negative emotions; and accessing resources. Strategies employed by scleroderma patients to overcome these challenges include seeking connections to other people with scleroderma or scleroderma patient organizations, actively seeking out local resources, and learning to communicate and advocate more effectively. Rehabilitation professionals can support people with scleroderma by providing them with information on connecting with scleroderma patient organizations or by facilitating local patient support networks.

Operant Learning Versus Energy Conservation Activity Pacing Treatments in a Sample of Patients With Fibromyalgia Syndrome: A Pilot Randomized Controlled Trial.

This study's aim was to assess the efficacy of 2 forms of activity pacing in patients with fibromyalgia syndrome (FMS). Treatment-related changes in activity management patterns were also examined. Patients with FMS (n = 178) were randomly assigned to an operant learning (OL; delayed [n = 36] or immediate [n = 54] groups) or an energy conservation (EC; delayed [n = 35] or immediate [n = 53] groups) treatment condition. Of these, 32 OL and 37 EC patients completed treatment. Forty-three patients were allocated to the delayed treatment condition (control group). Repeated measures analyses of variance were used to examine the effects of OL and EC treatments on primary (average pain and usual fatigue), secondary (pain and fatigue interference, physical and psychological function, sleep quality, depressive symptoms, and anxiety symptoms), and tertiary (pain-related activity patterns) outcomes. Neither treatment was effective in reducing average pain or usual fatigue symptoms. Relative to EC, OL patients showed greater improvements in depressive symptoms, whereas nonsignificant trends (P values ranging between .05 and .06) were observed for pain interference, fatigue interference, and psychological function. Both treatments were associated with improvements in sleep quality and physical function, increases in pacing, and decreases in overdoing activity patterns. Reductions in activity avoidance were only found in OL. These findings suggest that OL may be more beneficial than EC and that it could potentially be viewed as an effective stand-alone activity pacing treatment for patients with FMS. Research to determine the extent to which these preliminary findings replicate is warranted. PERSPECTIVE: This article examines the efficacy of 2 forms of activity pacing in patients with fibromyalgia syndrome. The results suggest the possibility that operant learning may be more beneficial than energy conservation and could potentially be viewed as an effective stand-alone activity pacing treatment for patients with fibromyalgia syndrome.

Patterns of pain-related activity: replicability, treatment-related changes, and relationship to functioning.

Changes in activity patterns frequently accompany the experience of chronic pain. Two activity patterns, avoidance and overdoing, are hypothesized to contribute to the development of ongoing pain and pain-related disability, while activity pacing is frequently introduced to enhance pain management and functioning. Two studies were conducted to assess whether reliable subgroups with differing activity patterns could be identified in different pain populations and to evaluate changes in these subgroups after a group format, pain management program. In study 1, individuals with ongoing pain being assessed for treatment at 2 different tertiary care pain centres completed a measure of pain-related activity. Separate cluster analyses of these samples produced highly similar cluster solutions. For each sample, a 2-cluster solution was obtained with clusters corresponding to the activity patterns described by the avoidance-endurance model of pain. In study 2, a subset of individuals completing a 12-session, group format, pain management program completed measures of pain-related activity, pain intensity, and physical and psychological functioning at the beginning and end of the program. At the conclusion of the program, 4 clusters of pain-related activity were identified. Individuals who used high levels of activity pacing and low levels of avoidance consistently reported significantly better functioning relative to all other individuals. Observed changes in activity patterns from pre-treatment to post-treatment suggested that decreasing the association between activity pacing and avoidance was associated with better functioning. These results have implications for both the assessment of activity pacing and for its use as an intervention in the management of ongoing pain.

The Scleroderma Patient-Centered Intervention Network Cohort: baseline clinical features and comparison with other large scleroderma cohorts.

Objectives: The Scleroderma Patient-centered Intervention Network (SPIN) Cohort is a web-based cohort designed to collect patient-reported outcomes at regular intervals as a framework for conducting trials of psychosocial, educational, self-management and rehabilitation interventions for patients with SSc. The aim of this study was to present baseline demographic, medical and patient-reported outcome data of the SPIN Cohort and to compare it with other large SSc cohorts. Methods: Descriptive statistics were used to summarize SPIN Cohort characteristics; these were compared with published data of the European Scleroderma Trials and Research (EUSTAR) and Canadian Scleroderma Research Group (CSRG) cohorts. Results: Demographic, organ involvement and antibody profile data for SPIN (N = 1125) were generally comparable with that of the EUSTAR (N = 7319) and CSRG (N = 1390) cohorts. There was a high proportion of women and White patients in all cohorts, though relative proportions differed. Scl70 antibody frequency was highest in EUSTAR, somewhat lower in SPIN, and lowest in CSRG, consistent with the higher proportion of interstitial lung disease among dcSSc patients in SPIN compared with in CSRG (48.5 vs 40.3%). RNA polymerase III antibody frequency was highest in SPIN and remarkably lower in EUSTAR (21.1 vs 2.4%), in line with the higher prevalence of SSc renal crisis (4.5 vs 2.1%) in SPIN. Conclusion: Although there are some differences, the SPIN Cohort is broadly comparable with other large prevalent SSc cohorts, increasing confidence that insights gained from the SPIN Cohort should be generalizable, although it should be noted that all three cohorts include primarily White participants.

Pain-related Activity Management Patterns and Function in Patients With Fibromyalgia Syndrome.

OBJECTIVES: To clarify the importance of avoidance, pacing, and overdoing pain-related activity management patterns as predictors of adjustment in patients with fibromyalgia syndrome. METHODS: A total of 119 tertiary care patients with fibromyalgia syndrome who agreed to be part of an activity management pain program completed a survey, which requested information about demographics, pain intensity and pain interference, psychological and physical function, and pain-related activity management patterns. Hierarchical regression analyses were used to identify the unique contributions of the 3 different pain-related activity management patterns (avoidance, pacing, and overdoing) to the prediction of pain interference, psychological function, and physical function. RESULTS: The avoidance pattern was a significant and unique predictor of worse psychological and physical function as well as greater pain interference. Pacing was significantly associated with less pain interference and better psychological function, whereas overdoing was not found to predict patient functioning. DISCUSSION: The findings confirm the importance of pain-related activity management patterns as predictors of patient function, and support the necessity of addressing these factors in chronic pain treatment. In addition, the results suggest that targeting increases in activity pacing and decreases in pain avoidance, specifically, might yield the best patient outcomes. However, further research to evaluate this possibility is necessary.

Lower Functional Connectivity of the Periaqueductal Gray Is Related to Negative Affect and Clinical Manifestations of Fibromyalgia.

Fibromyalgia (FM) syndrome is characterized by chronic widespread pain, muscle tenderness and emotional distress. Previous studies found reduced endogenous pain modulation in FM. This deficiency of pain modulation may be related to the attributes of chronic pain and other clinical symptoms experienced in patients with FM. Thus, we tested whether there is a link between the clinical symptoms of FM and functional connectivity (FC) of the periaqueductal gray (PAG), a key node of pain modulation. We acquired resting state 3T functional MRI (rsfMRI) data from 23 female patients with FM and 16 age- and sex- matched healthy controls (HC) and assessed FM symptoms with the Brief Pain Inventory (BPI), Fibromyalgia Impact Questionnaire (FIQ), Hospital Anxiety and Depression Scale (HADS) and Pain Catastrophizing Scale (PCS). We found that patients with FM exhibit statistically significant disruptions in PAG FC, particularly with brain regions implicated in negative affect, self-awareness and saliency. Specifically, we found that, compared to HCs, the FM patients had stronger PAG FC with the lingual gyrus and hippocampus but weaker PAG FC with regions associated with motor/executive functions, the salience (SN) and default mode networks (DMN). The attenuated PAG FC was also negatively correlated with FIQ scores, and positively correlated with the magnification subscale of the PCS. These alterations were correlated with emotional and behavioral symptoms of FM. Our study implicates the PAG as a site of dysfunction contributing to the clinical manifestations and pain in FM.