Association of COVID-19 vaccination and anxiety symptoms: A Scleroderma Patient-centered Intervention Network (SPIN) Cohort longitudinal study.

OBJECTIVE: Symptoms of anxiety increased early in the COVID-19 pandemic among people with systemic sclerosis (SSc) then returned to pre-pandemic levels, but this was an aggregate finding and did not evaluate whether vaccination may have contributed to reduced anxiety symptom levels. We investigated whether being vaccinated for COVID-19 was associated with reduced anxiety symptoms among people with SSc. METHODS: The longitudinal Scleroderma Patient-centered Intervention Network (SPIN) COVID-19 Cohort was launched in April 2020 and included participants from the ongoing SPIN Cohort and external enrollees. Participants completed measures bi-weekly through July 2020, then every 4 weeks afterwards through August 2022 (32 assessments). We used linear mixed models to evaluate longitudinal trends of PROMIS Anxiety 4a v1.0 anxiety domain scores and their association with vaccination. RESULTS: Among 517 participants included in analyses, 489 (95%) were vaccinated by September 2021, and no participants were vaccinated subsequently. Except for briefly at the beginning, when few had received a vaccine, and end, when only 28 participants remained unvaccinated, anxiety symptom trajectories were largely overlapping. Participants who were never vaccinated had higher anxiety symptoms by August 2022, but there were no other differences, and receiving a vaccination did not appear to change anxiety symptom trajectories meaningfully. CONCLUSION: Vaccination did not appear to influence changes in anxiety symptoms among vulnerable people with SSc during the COVID-19 pandemic. This may be due to people restricting their behavior when they were unvaccinated and returning to more normal social engagement once vaccinated to maintain a steady level of anxiety symptoms.

COVID-19 vaccinations and infections among individuals with systemic sclerosis: A Scleroderma Patient-centered Intervention Network (SPIN) Cohort study.

BACKGROUND/PURPOSE: We previously surveyed adults with systemic sclerosis (SSc) regarding COVID-19 vaccination in April-May 2021. The objective of the present study was to update through June-July 2022 and assess self-reported (1) COVID-19 vaccination rates, including boosters; (2) vaccine-related adverse events; (3) peri­vaccination immunosuppressive medication management; (4) vaccine hesitancy; and (5) prevalence and severity of COVID-19 infections. METHODS: In April-May 2021 and June-July 2022, SPIN Cohort participants completed surveys on COVID-19 vaccination and infection. Primary vaccine series was defined according to the standard for each COVID-19 vaccine; additional vaccine administrations were considered booster doses. Fully vaccinated was defined as having completed a primary vaccine series and at least one booster dose. RESULTS: 544 participants completed the 2021 survey only, 101 the 2022 survey only, and 388 both surveys. Among 489 participants with 2022 data, 437 (89 %) had received both primary and booster vaccines. Among all 1,033 participants, 960 (93 %) received at least one dose. At least one adverse reaction was reported by 34 % (330 of 960 participants) following first, 48 % (314 of 657 participants) following second, and 34 % (147 of 437 participants) following booster vaccine doses (primarily sore arm and fatigue); no severe adverse reactions were reported. SSc symptom worsening was reported in 6 % (53 of 960) after the first, 6 % after the second (39 of 657), and 4 % (17 of 437) after the booster dose. Of participants taking methotrexate or mycophenolate (including Cellcept or Myfortic), 34 of 266 (13 %) reported that they temporarily stopped or decreased their medication at the first dose, 32 of 215 (15 %) at the second dose, and 28 of 148 (19 %) for booster vaccination. Of 52 individuals not fully vaccinated with primary and booster doses in 2022, 29 (56 %) reported worry about vaccine related SSc flares. 172 of 489 (35 %) 2022 participants reported a history of at least one COVID-19 infection; 114 (66 %) occurred after receiving at least a primary vaccine series. Among initial COVID-19 infections, 9 (5 %) were asymptomatic, 66 (38 %) involved mild symptoms, 82 (48 %) moderate symptoms, and 15 (9 %) required hospitalization. CONCLUSION: Most people with SSc in the study were fully vaccinated, and most continued their methotrexate or mycophenolate post-primary and booster vaccinations. Over half of vaccine-hesitant participants were concerned regarding risk of SSc flare; however, few vaccinated participants reported this. These data may be useful for counselling people with SSc regarding COVID-19 vaccine safety and outcomes.

Minimal Detectable Changes of the HAQ-DI, PROMIS-29v2.0 Domains, and PHQ-8 in Systemic Sclerosis: A SPIN Cohort Cross-Sectional Study.

OBJECTIVE: Systemic sclerosis (SSc) is a rare, chronic, autoimmune disorder associated with disability, diminished physical function, fatigue, pain, and mental health concerns. We assessed minimal detectable changes (MDCs) of the Health Assessment Questionnaire-Disability Index (HAQ-DI), Patient Reported Outcomes Measurement Information System-29 Profile version 2.0 (PROMIS-29v2.0) domains, and Patient Health Questionnaire-8 (PHQ-8) in SSc. METHODS: Scleroderma Patient-centered Intervention Network Cohort participants completed the HAQ-DI, PROMIS-29v2.0 domains, and PHQ-8 at baseline assessments from April 2014 until August 2023. We estimated MDC95 and MDC90 with 95% confidence intervals (CI) generated via the percentile bootstrapping method resampling 1000 times. We compared MDC estimates by age, sex and SSc subtype. RESULTS: A total of 2,571 participants were included. Most were female (N = 2,241; 87%), and 38% (N = 976) had diffuse SSc. Mean (SD) age was 54.9 (12.7) years and duration since onset of first non-Raynaud phenomenon symptom 10.8 (8.7) years. MDC95 estimate was 0.41 points (95% CI: 0.40 to 0.42) for the HAQ-DI, between 4.88 points (95% CI: 4.72 to 5.05) and 9.02 points (95% CI: 8.80 to 9.23) for the 7 PROMIS-29v2.0 domains, and 5.16 points (95% CI: 5.06 to 5.26) for the PHQ-8. MDC95 estimates were not materially different across subgroups. CONCLUSION: MDC95 and MDC90 estimates were precise and similar across age, sex and SSc subtype groups. HAQ-DI MDC95 and MDC90 were substantially larger than previous estimates of HAQ-DI minimal important difference from several small studies. Minimally important differences of all measures should be evaluated in large studies using anchor-based methods.

Protocol for the development of a tool to map systemic sclerosis pain sources, patterns, and management experiences: a Scleroderma Patient-centered Intervention Network patient-researcher partnership.

INTRODUCTION: Systemic sclerosis (SSc) is a rare, complex autoimmune rheumatic disease with multiple factors that contribute to pain. People with SSc emphasize the effect pain has on their quality of life, but no studies have systematically examined the frequency and relative importance of different SSc pain sources, patterns of pain from different sources, and pain management experiences. Our objectives are to (1) develop a tool, jointly with researchers, health care providers, and patients, to map sources of pain in SSc, determine patterns of pain from different sources, and understand pain management experiences; and (2) administer the final tool version to participants in the large multinational Scleroderma Patient-centered Intervention Network (SPIN) Cohort. METHODS: First, we will use validated pain assessment tools as templates to develop an initial version of our pain assessment tool, and we will obtain input from patient advisors to adapt it for SSc. The tool will include questions on pain sources, pain patterns, pain intensity, pain management techniques, and barriers to pain management in SSc. Second, we will conduct nominal group technique sessions with people living with SSc and health care providers who care for people with SSc to further refine the tool. Third, we will conduct individual usability testing sessions with SPIN Cohort participants. Once the tool has been finalized, we will administer it to individuals in the multinational SPIN Cohort, which currently includes over 1,300 active participants from 54 sites in 7 countries. We will perform unsupervised clustering using the KAy-Means for MIxed LArge data (KAMILA) method to identify participant subgroups with similar profiles of pain sources (present or absent) and to evaluate predictors of subgroup membership. We will use latent profile analysis to identify subgroups of participants with similar profiles based on pain intensity scores for each pain source and evaluate predictors. DISCUSSION: Once completed, our pain assessment tool will allow our team and other researchers to map sources of pain in SSc and to understand pain management experiences of people living with SSc. This knowledge will provide avenues for studies on the pathophysiology of pain in SSc and studies of interventions to improve pain management.

Physical activity during COVID-19 in people with systemic sclerosis: A Scleroderma Patient-centred Intervention Network COVID-19 Cohort longitudinal study.

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.

Use and perceptions of nutrition information resources in systemic sclerosis: a Scleroderma Patient-centred Intervention Network (SPIN) cohort study.

OBJECTIVES: People with systemic sclerosis (SSc) may find it challenging to obtain high-quality nutrition and diet information. Objectives were to evaluate (i) how commonly different information resources are used and (ii) perceived trustworthiness, accessibility, comprehensibility, and individualisation of resources. METHODS: We administered the Scleroderma Patient-centred Intervention Network Nutrition Information Resources Survey to participants in an international cohort. Participants were asked if they had used 26 informational resources in four categories, including (i) health care providers, (ii) websites or social media, (iii) print materials, and (iv) events, and to rate each resource on trustworthiness, accessibility, comprehensibility, and individualisation (0 = not at all to 10 = completely). RESULTS: 727 participants completed the survey. Most (94%) had sought nutrition or diet information from at least one resource. The most-used category was health care providers (86%), followed by print materials (68%), websites or social media (66%), and events (43%). People who had used a resource generally rated it more favourably across all domains than those who had not. The highest-rated resources across domains were conventional health care providers (doctors, registered dieticians, nurses), SSc patient organisations, SSc support groups, and university or research institution websites. CONCLUSIONS: Respondents used many different diet and nutrition information resources. They preferred resources from conventional health care providers, affiliated with credible institutions (e.g., SSc patient organisations), or with personal connections (e.g., SSc support groups). Future research should address the limited evidence base on nutrition in SSc and assess the quality of information provided by different information resources.

Factors associated with physical function among people with systemic sclerosis: a SPIN cohort cross-sectional study.

OBJECTIVES: To compare physical function in systemic sclerosis (SSc, scleroderma) to general population normative data and identify associated factors. METHODS: Scleroderma Patient-centered Intervention Network Cohort participants completed the Physical Function domain of the Patient-Reported Outcomes Measurement Information System Version 2 upon enrolment. Multivariable linear regression was used to assess associations of sociodemographic, lifestyle, and disease-related variables. RESULTS: Among 2,385 participants, mean physical function T-score (43.7, SD = 8.9) was ∼2/3 of a standard deviation (SD) below the US general population (mean = 50, SD = 10). Factors associated in multivariable analysis included older age (-0.74 points per SD years, 95% CI -0.78 to -1.08), female sex (-1.35, -2.37 to -0.34), fewer years of education (-0.41 points per SD in years, -0.75 to -0.07), being single, divorced, or widowed (-0.76, -1.48 to -0.03), smoking (-3.14, -4.42 to -1.85), alcohol consumption (0.79 points per SD drinks per week, 0.45-1.14), BMI (-1.41 points per SD, -1.75 to -1.07), diffuse subtype (-1.43, -2.23 to -0.62), gastrointestinal involvement (-2.58, -3.53 to -1.62), digital ulcers (-1.96, -2.94 to -0.98), moderate (-1.94, -2.94 to -0.93) and severe (-1.76, -3.24 to -0.28) small joint contractures, moderate (-2.10, -3.44 to -0.76) and severe (-2.54, -4.64 to -0.44) large joint contractures, interstitial lung disease (-1.52, -2.27 to -0.77), pulmonary arterial hypertension (-3.72, -4.91 to -2.52), rheumatoid arthritis (-2.10, -3.64 to -0.56) and idiopathic inflammatory myositis (-2.10, -3.63 to -0.56). CONCLUSION: Physical function is impaired for many individuals with SSc and associated with multiple disease factors.

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.

Factors associated with satisfaction with social roles and activities among people with systemic sclerosis: a Scleroderma Patient-centered Intervention Network (SPIN) cohort cross-sectional study.

OBJECTIVE: The objectives were to (1) compare satisfaction with social roles and activities in a large multinational systemic sclerosis (SSc) cohort to general population normative data and (2) identify sociodemographic, lifestyle and SSc disease factors associated with satisfaction with social roles and activities. METHODS: Participants in the Scleroderma Patient-centered Intervention Network Cohort completed the Patient Reported Outcomes Information System Version 2 satisfaction with social roles and activities domain questionnaire. Multivariable regression was used to assess associations with sociodemographic, lifestyle and disease factors. RESULTS: Among 2385 participants, mean satisfaction with social roles and activities T-score (48.1, SD=9.9) was slightly lower than the US general population (mean=50, SD=10). Factors independently associated with satisfaction were years of education (0.54 per SD, 95% CI 0.14 to 0.93); non-White race or ethnicity (-1.13, 95% CI -2.18 to -0.08); living in Canada (-1.33, 95% CI -2.40 to -0.26 (reference USA)) or the UK (-2.49, 95% CI -3.92 to -1.06); body mass index (-1.08 per SD, 95% CI -1.47 to -0.69); gastrointestinal involvement (-3.16, 95% CI -4.27 to -2.05); digital ulcers (-1.90, 95% CI -3.05 to -0.76); moderate (-1.62, 95% CI -2.78 to -0.45) or severe (-2.26, 95% CI -3.99 to -0.52) small joint contractures; interstitial lung disease (-1.11, 95% CI -1.97 to -0.25); pulmonary arterial hypertension (-2.69, 95% CI -4.08 to -1.30); rheumatoid arthritis (-2.51, 95% CI -4.28 to -0.73); and Sjogren's syndrome (-2.42, 95% CI -3.96 to -0.88). CONCLUSION: Mean satisfaction with social roles and activities is slightly lower in SSc than the general population and associated with multiple sociodemographic and disease factors.

Measurement equivalence of the English and French versions of the self-efficacy to manage chronic disease scale: a Scleroderma Patient-Centered Intervention Network (SPIN) study.

PURPOSE: The Self-Efficacy to Manage Chronic Disease (SEMCD) scale is widely used, including in systemic sclerosis (SSc). The SEMCD has been validated in SSc, but the metric equivalence of the English and French versions has not been assessed (i.e., whether psychometric properties are equivalent across English and French). METHODS: Participants were adults from the Scleroderma Patient-Centered Intervention Network (SPIN) Cohort (N = 2159) who completed baseline measures in English (n = 1473) or French (n = 686) between May 2014 to July 2020. Analyses assessed internal consistency reliability via Cronbach's alpha and McDonald's omega, convergent validity via Pearson's correlations, structural validity via confirmatory factor analysis (CFA), and differential item functioning via the Multiple-Indicator Multiple-Cause (MIMIC) model. RESULTS: Internal consistency reliability was high in English (α = .93, ω = .93) and French (α = .92, ω = .93). All correlations between the SEMCD and measures of health outcomes were moderate to large, statistically significant, and in the hypothesized direction in both languages. The CFA demonstrated that the one-factor model of self-efficacy, overall, fit reasonably well (CFI = .96, TLI = .93, SRMR = .03, RMSEA = .14). Standardized factor loadings were large (.76 to .88). Three items displayed statistically significant uniform DIF and all six displayed nonuniform DIF; all DIF was of minimal magnitude. Comparison of unadjusted and DIF-adjusted models indicated that DIF did not meaningfully impact total score (ICC = 0.999, r = 0.999). CONCLUSION: Scores from English- and French-speaking adults with SSc can be combined for analysis or compared.

Moderators of Loneliness Trajectories in People with Systemic Sclerosis During the COVID-19 Pandemic: A SPIN COVID-19 Cohort Longitudinal Study.

BACKGROUND: Many individuals with systemic sclerosis (SSc) are at heightened risk for COVID-19 related morbidity and isolation due to interstitial lung disease, frailty, and immunosuppressant use. Minimal research has explored loneliness predictors in individuals with chronic illnesses during COVID-19. This study evaluated moderators of loneliness trajectories in individuals with SSc during COVID-19. METHODS: Longitudinal data were analyzed across 30 timepoints from April 2020 to May 2022 from 775 adults in the Scleroderma Patient-centered Intervention Network (SPIN) COVID-19 Cohort. Hierarchical linear modeling evaluated cross-level moderators of loneliness trajectories, including marital status, baseline number of household members, number of virtual or telephone one-on-one or virtual group conversations, number of hours spent enjoying in-person household conversations or activities, and satisfaction with quality of in-person household conversations (all in the past week). Level-1 moderation analyses assessed effects of conversation, activity, and satisfaction means and slopes over time. RESULTS: Baseline values were not statistically significant moderators of loneliness trajectories. Higher mean (averaged over time) virtual or telephone one-on-one and in-person household conversations, in-person household activity, and in-person household conversation satisfaction were associated with lower loneliness trajectories (ps < .05). The relationship between in-person household conversation satisfaction and loneliness trajectory was statistically significantly but minimally attenuated over time (p < .001). CONCLUSIONS: For people with SSc, higher mean conversation, activity, and satisfaction variables were associated with lower levels of loneliness during the pandemic, but changes in these social variables were generally not predictive of changes in loneliness.

Researcher and patient experiences of co-presenting research to people living with systemic sclerosis at a patient conference: content analysis of interviews.

BACKGROUND: Patient engagement in research is important to ensure research questions address problems important to patients, that research is designed in a way that can effectively answer those questions, and that findings are applicable, relevant, and credible. Yet, patients are rarely involved in the dissemination stage of research. This study explored one way to engage patients in dissemination, through co-presenting research. METHODS: Semi-structured, one-on-one, audio-recorded interviews were conducted with researchers and patients who co-presented research at one patient conference (the 2022 Canadian National Scleroderma Conference) in Canada. A pragmatic orientation was adopted, and following verbatim transcription, data were analyzed using conventional content analysis. RESULTS: Of 8 researchers who were paired with 7 patients, 5 researchers (mean age = 28 years, SD = 3.6 years) and 5 patients (mean age = 45 years, SD = 14.2 years) participated. Researcher and patient perspectives about their experiences co-presenting and how to improve the experience were captured across 4 main categories: (1) Reasons for accepting the invitation to co-present; (2) Degree that co-presenting expectations were met; (3) The process of co-presenting; and (4) Lessons learned: recommendations for co-presenting. CONCLUSIONS: Findings from this study suggest that the co-presenting experience was a rewarding and enjoyable way to tailor research dissemination to patients. We identified a patient-centred approach and meaningful and prolonged patient engagement as essential elements underlying co-presenting success.

Involving patients throughout the entire research process is important to ensure research effectively addresses problems important to patients and that findings are applicable, relevant, and credible. Yet, patients are rarely involved in the dissemination of research. We explored one way to engage patients in dissemination, through co-presenting research. We conducted one-on-one interviews with 5 researchers and 5 patients who co-presented research at a patient conference in Canada. Both researchers and patients indicated that the co-presenting experience was rewarding and enjoyable and a useful way to tailor dissemination to patients. We found that a patient-centred approach and meaningful and prolonged patient engagement were essential elements underlying co-presenting success.

A qualitative interview study exploring the psychological health impacts of the SPIN-CHAT program among people with systemic sclerosis at the onset of COVID-19: perceptions of trial participants and research team members.

PURPOSE: Explore trial participants' and research team members' perceptions of the impact of the videoconference-based, supportive care program (SPIN-CHAT Program) during early COVID-19 for individuals with systemic sclerosis (SSc). METHODS: Data were collected cross-sectionally. A social constructivist paradigm was adopted, and one-on-one videoconference-based, semi-structured interviews were conducted with SPIN-CHAT Trial participants and research team members. A hybrid inductive-deductive approach and reflexive thematic analysis were used. RESULTS: Of the 40 SPIN-CHAT Trial participants and 28 research team members approached, 30 trial participants (Mean age = 54.9; SD = 13.0 years) and 22 research team members agreed to participate. Those who took part in interviews had similar characteristics to those who declined. Five themes were identified: (1) The SPIN-CHAT Program conferred a range of positive psychological health outcomes, (2) People who don't have SSc don't get it: The importance of SSc-specific programming, (3) The group-based format of the SPIN-CHAT Program created a safe space to connect and meet similar others, (4) The structure and schedule of the SPIN-CHAT Program reduced feelings of boredom and contributed to enhanced psychological health, (5) The necessity of knowledge, skills, and tools to self-manage SSc and navigate COVID-19. CONCLUSION: Participants' and research team members' perspectives elucidated SPIN-CHAT Program benefits and how these benefits may have been realized. Results underscore the importance of social support from similar others, structure, and self-management to enhance psychological health during COVID-19. TRIAL REGISTRATION: clinicaltrials.gov (NCT04335279)IMPLICATIONS FOR REHABILITATIONThe videoconference-based, supportive care SPIN-CHAT Program enhanced psychological health amongst individuals affected by systemic sclerosis.SPIN-CHAT Program participants and research team members shared that being around similar others, program structure, and self-management support were important and may have contributed to enhanced psychological health.Further efforts are required to explore experiences within supportive care programs to better understand if and how psychological health is impacted.

Support and information needs of people with systemic sclerosis by time since diagnosis: A cross-sectional study.

Background: How support and informational needs of people with systemic sclerosis (SSc) may differ by time since diagnosis is not known. Our objective was to determine if informational and support needs of recently diagnosed individuals with systemic sclerosis differ from people diagnosed for longer periods of time. Methods: The North American Scleroderma Support Group Members survey included 30 items on reasons for attending support groups. Respondents were classified by time since diagnosis of 0-3 years, 4-9 years or 10+ years. Survey item responses were dichotomized into Not Important or Somewhat Important versus Important or Very Important. We conducted Chi-square tests with Hochberg's Sequential Method to identify item differences by time since diagnosis. Results: A total of 175 respondents completed the survey. Most support needs were rated as Important or Very Important by respondents, regardless of disease duration, particularly needs related to interpersonal and social support (10 items; median 81%) and learning about disease treatment and management strategies (11 items; median 82%). Discussing other aspects of living with systemic sclerosis (e.g. spirituality, discussing disease with family and friends) was rated lower (9 items; 44%). Respondents with 0-3 years since diagnosis were the highest on 29 of 30 items. Respondents with 0-3 years since diagnosis were significantly higher on items related to discussing medical care and 4 items on other aspects (spirituality, talking with family and friends, financial issues, sexual issues). Conclusion: People with systemic sclerosis have a wide range of information and support needs, regardless of their disease duration, but people with recent diagnoses have greater needs.

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.

Differential Item Functioning on the Cochin Hand Function Scale Among People With Systemic Sclerosis by Language, Sex, and Disease Subtype: A Scleroderma Patient-Centered Intervention Network (SPIN) Cohort Study.

OBJECTIVE: To evaluate the degree that the Cochin Hand Function Scale (CHFS) generates scores that are comparable across language, sex, and disease subtype. METHODS: We included participants enrolled in the Scleroderma Patient-centered Intervention Network (SPIN) Cohort who completed the CHFS at their baseline assessment between April 2014 and September 2020. Confirmatory factor analysis (CFA) was used to test unidimensionality, and multiple indicator multiple cause (MIMIC) models were used for differential item functioning (DIF) analysis based on language, sex, and disease subtype. Both intraclass correlation coefficient (ICC) and Pearson's correlation were calculated using factor scores obtained from unadjusted and DIF-adjusted MIMIC models to evaluate agreement and correlation between scores. RESULTS: A total of 2,155 participants were included. CFA with covarying error terms supported a good fit of the model (χ2 [127] = 1,754.671; P < 0.001; Tucker-Lewis index = 0.985; comparative fit index = 0.987; root mean square error of approximation = 0.077). Nine items displayed statistically significant DIF for language of administration, 10 items for sex, and 10 items for disease subtype. However, the overall impact of DIF was negligible when comparing factor scores that did and did not account for DIF (ICC = 0.999; r = 0.999). CONCLUSION: The CHFS has score comparability in systemic sclerosis regardless of participants' language, sex, and disease subtype.

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.

Mental health symptoms in scleroderma during COVID-19: a Scleroderma Patient-centred Intervention Network (SPIN) cohort longitudinal study.

OBJECTIVES: People with systemic sclerosis (SSc) are vulnerable in COVID-19 and face challenges related to shifting COVID-19 risk and protective restrictions. We evaluated mental health symptom trajectories in people with SSc through March 2022. METHODS: The longitudinal Scleroderma Patient-centred Intervention Network (SPIN) COVID-19 cohort was launched in April 2020 and included participants from the ongoing SPIN Cohort and external enrolees. Analyses included estimated means with 95% CIs for anxiety and depression symptoms pre-COVID-19 for ongoing SPIN Cohort participants and anxiety, depression, loneliness, and fear of COVID-19 for all participants across 28 COVID-19 assessments up to March 2022. We conducted sensitivity analyse including estimating trajectories using only responses from participants who completed >90% of items for ≥21 of 28 possible assessments ("completers") and stratified analyses for all outcomes by sex, age, country, and SSc subtype. RESULTS: Anxiety symptoms increased in early 2020 but returned to pre-COVID-19 levels by mid-2020 and remained stable through March 2022. Depression symptoms did not initially change but were slightly lower by mid-2020 compared to pre-COVID-19 and were stable through March 2022. COVID-19 fear started high and decreased. Loneliness did not change across the pandemic. Results were similar for completers and for all subgroups. CONCLUSIONS: People with SSc continue to face COVID-19 challenges related to ongoing risk, the opening of societies, and removal of protective restrictions. People with SSc, in aggregate, appear to be weathering the pandemic well, but health care providers should be mindful that some individuals may benefit from mental health support.

Evaluation of Measurement Properties and Differential Item Functioning in the English and French Versions of the University of California, Los Angeles, Loneliness Scale-6: A Scleroderma Patient-Centered Intervention Network (SPIN) Study.

OBJECTIVE: Loneliness has been associated with poorer health-related quality of life but has not been studied in patients with systemic sclerosis (SSc). The current study was undertaken to examine and compare the psychometric properties of the English and French versions of the University of California, Los Angeles, Loneliness Scale-6 (ULS-6) in patients with SSc during the COVID-19 pandemic. METHODS: This study used baseline cross-sectional data from 775 adults enrolled in the Scleroderma Patient-Centered Intervention Network (SPIN) COVID-19 Cohort. Reliability and validity of ULS-6 scores overall and between languages were evaluated using confirmatory factor analysis (CFA), differential item functioning (DIF) through the multiple-indicator multiple-cause (MIMIC) model, omega/alpha calculation, and correlations of hypothesized convergent relationships. RESULTS: CFA for the total sample supported the single-factor structure (comparative fit index [CFI] 0.96, standardized root mean residual [SRMR] 0.03), and all standardized factor loadings for items were large (0.60-0.86). The overall MIMIC model with language as a covariate fit well (CFI 0.94, SRMR 0.04, root mean square error of approximation 0.11). Statistically significant DIF was found for 3 items across language (ßitem2  = 0.14, P < 0.001; ßitem4  = -0.07, P = 0.01; ßitem6  = 0.13, P < 0.001), but these small differences were without practical measurement implications. Analyses demonstrated high internal consistency with no language-based convergent validity differences. CONCLUSION: Analyses demonstrated evidence of acceptable reliability and validity of ULS-6 scores in English- and French-speaking adults with SSc. DIF analysis supported use of the ULS-6 to examine comparative experiences of loneliness without adjusting for language.