Parent and patient knowledge and attitudes about cancer predisposition syndrome genetic testing in pediatric oncology: Understanding sociodemographic and parent-child differences.

BACKGROUND: Cancer predisposition syndromes (CPS) impact about 10% of patients with pediatric cancer. Genetic testing (CPS-GT) has multiple benefits, but few studies have described parent and child knowledge and attitudes regarding CPS-GT decision-making. This study examined parent and patient CPS-GT decision-making knowledge and attitudes. PROCEDURE: English- or Spanish-speaking parents of children with pediatric cancer and patients with pediatric cancer ages 15-18 within 12 months of diagnosis or relapse were eligible to participate. Seventy-five parents and 19 parent-patient dyads (N = 94 parents, 77.7% female, 43.6% Latino/a/Hispanic; 19 patients, 31.6% female) completed surveys measuring CPS-GT-related beliefs. Independent samples t-tests compared parent responses across sociodemographic characteristics and parent-patient responses within dyads. RESULTS: Spanish-speaking parents were significantly more likely than English-speaking parents to believe that CPS-GT not being helpful (p < .001) and possibly causing personal distress (p = .002) were important considerations for deciding whether to obtain CPS-GT. Parents with less than four-year university education, income less than $75,000, or Medicaid (vs. private insurance) were significantly more likely to endorse that CPS-GT not being helpful was an important consideration for deciding whether to obtain CPS-GT (p < .001). Parents felt more strongly than patients that they understood what CPS-GT was (p = .01) and that parents should decide whether patients under 18 should receive CPS-GT (p = .002). CONCLUSIONS: Spanish-speaking parents and parents with lower socioeconomic statuses were more strongly influenced by the potential disadvantages of CPS-GT in CPS-GT decision-making. Parents felt more strongly than patients that parents should make CPS-GT decisions. Future studies should investigate mechanisms behind these differences and how to best support CPS-GT knowledge and decision-making.

The association of outdoor temperature and self-reported Raynaud's phenomenon severity among people with systemic sclerosis: a Scleroderma Patient-centered Intervention Network Cohort study.

BACKGROUND: Raynaud's phenomenon is the earliest and most common systemic sclerosis manifestation. Episodes can be triggered by cold exposure and ambient temperature changes. Small studies have found that Raynaud's phenomenon outcomes were associated with season. We aimed to map the degree that differences in ambient temperature are associated with Raynaud's phenomenon outcomes across the temperature spectrum. METHODS: People with Raynaud's phenomenon secondary to systemic sclerosis in the Scleroderma Patient-centered Intervention Network Cohort completed past-week Raynaud's phenomenon severity assessments (0-10 numerical rating scale) at enrolment and longitudinally at 3-month intervals. Mean daily temperature and feels like temperature, which incorporates wind chill and humidity, for the week before each assessment were extracted for each participant from a weather site close to the participant's recruiting centre via the Iowa Environmental Mesonet. We used linear mixed models with basis splines to flexibly model non-linear changes in Raynaud's phenomenon severity across the temperature spectrum. People with lived experience of systemic sclerosis contributed to the study design and interpretation. FINDINGS: Between April 15, 2014 and Aug 1, 2023, we included data on 20 233 Raynaud's phenomenon severity assessments from 2243 participants. 1964 (88%) of 2243 participants were women, 279 (12%) were men, and 1813 (82%) were White. Mean age was 54·8 (SD 12·7) years. The maximum predicted Raynaud's phenomenon severity score was 6·8 points (95% CI 5·6-8·1), which occurred at -25°C. Severity scores decreased minimally from -15°C to 5°C (0·05-0·21 points per 5°C difference), then decreased in larger steps between 5°C and 25°C (0·37-0·54 points per 5°C difference). The minimum predicted score was at 25°C (2·6 points [95% CI 2·5-2·7]). Scores increased at temperatures above 25°C to 3·5 points (3·0-4·1) at 35°C and 5·6 points (4·5-6·8) at 40°C. Results were similar for feels like temperature. INTERPRETATION: Raynaud's phenomenon severity is worst at very cold temperatures but also increases with very warm temperatures, presumably due to air conditioning. Clinical management and Raynaud's phenomenon intervention trial designs should consider temperature patterns. FUNDING: 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, the Canadian Institutes of Health Research, the Arthritis Society, the Lady Davis Institute for Medical Research of the Jewish General Hospital, the Jewish General Hospital Foundation, and McGill University.

Minimal Detectable Changes of the Health Assessment Questionnaire-Disability Index, Patient-Reported Outcomes Measurement Information System-29 Profile Version 2.0 Domains, and Patient Health Questionnaire-8 in People With Systemic Sclerosis: A Scleroderma Patient-Centered Intervention Network 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 (PHQ)-8 in people with 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 (smallest change that can be detected with 95% certainty) and MDC90 (smallest change that can be detected with 90% certainty) with 95% confidence intervals (CIs) generated via the percentile bootstrapping method resampling 1,000 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 was 10.8 (±8.7) years. MDC95 estimate was 0.41 points (95% CI 0.40-0.42) for the HAQ-DI, between 4.88 points (95% CI 4.72-5.05) and 9.02 points (95% CI 8.80-9.23) for the seven PROMIS-29v2.0 domains, and 5.16 points (95% CI 5.06-5.26) 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.

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 2385 participants, the 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 the 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 is 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.

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.

Social and built neighborhood environments and sleep health: The Hispanic Community Health Study/Study of Latinos Community and Surrounding Areas and Sueño Ancillary Studies.

STUDY OBJECTIVES: To test associations between neighborhood social, built, and ambient environment characteristics and multidimensional sleep health in Hispanic/Latino adults. METHODS: Data were from San Diego-based Hispanic/Latino adults mostly of Mexican heritage enrolled in the Hispanic Community Health Study/Study of Latinos (N = 342). Home addresses were geocoded to ascertain neighborhood characteristics of greenness, walkability (density of intersections, retail spaces, and residences), socioeconomic deprivation (e.g. lower income, lower education), social disorder (e.g. vacant buildings, crime), traffic density, and air pollution (PM 2.5) in the Study of Latinos Communities and Surrounding Areas Study. Sleep dimensions of regularity, satisfaction, alertness, timing, efficiency, and duration were measured by self-report or actigraphy approximately 2 years later. Multivariable regression models accounting for study design (stratification and clustering) were used to examine associations of neighborhood variables with individual sleep dimensions and a multidimensional sleep health composite score. RESULTS: Neighborhood characteristics were not significantly associated with the multidimensional sleep health composite, and there were few significant associations with individual sleep dimensions. Greater levels of air pollution (B = 9.03, 95% CI: 1.16, 16.91) were associated with later sleep midpoint, while greater social disorder (B = -6.90, 95% CI: -13.12, -0.67) was associated with earlier sleep midpoint. Lower walkability was associated with more wake after sleep onset (B = -3.58, 95% CI: -7.07, -0.09). CONCLUSIONS: Living in neighborhoods with lower walkability and greater air pollution was associated with worse sleep health, but otherwise findings were largely null. Future research should test these hypotheses in settings with greater variability and investigate mechanisms of these associations.

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.

To Operate or Not to Operate? Reconstructive Surgical Burden and Quality of Life of Pediatric Patients with Facial Differences.

OBJECTIVE: The Craniofacial Condition Quality of Life Scale (CFC-QoL) was used to evaluate the relationship between surgical burden and quality of life (QoL). DESIGN: Patient-parent dyads completed the CFC-QoL which queries the following QoL domains: Bullying, Peer Problems, Psychological Impact, Family Support, Appearance Satisfaction, and Desire for Appearance Change. Stepwise multivariate linear regressions were performed for each QoL domain. SETTING: Urban tertiary care center. PATIENTS, PARTICIPANTS: Pediatric patients with facial differences, and their parents. INTERVENTION: Survey study. MAIN OUTCOME MEASURE(S): Demographic, diagnostic, and surgical characteristics were collected. Surgical burden was calculated as the standard deviation from the mean number of surgeries per diagnostic cohort. RESULT: Patients (N = 168) were majority female (57.1%) and Hispanic (64.3%). Diagnoses were cleft lip and/or palate (CLP,n = 99) or other craniofacial conditions (CFC,n = 69). Average patient age was 2.3 ± 5.6 years at first reconstructive surgery and 12.3 ± 3.4 years at study enrollment. Patients received an average of 4.3 ± 4.1 reconstructive surgeries.Worse Bullying was associated with higher surgical burden. Worse Peer Problems was associated with higher surgical burden, but only for children with non-CLP CFCs. Worse Family Support was associated with CFC diagnosis, female sex, and higher surgical burden. Worse Psychological Impact was associated with higher surgical burden. Worse Appearance Satisfaction was associated with younger age and with lower surgical burden. Greater Desire for Appearance Change was associated with older age, higher surgical burden, CLP diagnosis, female sex, and non-Hispanic ethnicity. Socioeconomic status did not predict QoL per patient self- or parent-proxy report. CONCLUSIONS: Higher surgical burden was associated with worse QoL outcomes in multiple domains.

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.

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.

Effects of a support group leader education program jointly developed by health professionals and patients on peer leader self-efficacy among leaders of scleroderma support groups: a two-arm parallel partially nested randomised controlled trial.

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-efficacy was higher post-intervention [SGLSS; 16.7 points, 95% CI 11.0-22.3; standardized mean difference (SMD) 0.84] and 3 months later (15.6 points, 95% CI 10.2-21.0; SMD 0.73); leader volunteer satisfaction was significantly higher at both assessments, emotional distress was lower post-intervention but not 3 months later, and leader burnout was not significantly different at either assessment. CONCLUSIONS: Peer support group leader education improved leader self-efficacy substantially. The program could be easily adapted for support group leaders in other rare diseases. TRIAL REGISTRATION: NCT03965780 ; registered on May 29, 2019.

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.

Mental Health Care Use and Associated Factors in Systemic Sclerosis: A Scleroderma Patient-Centered Intervention Network Cohort Study.

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.

Clinical trials-related knowledge, attitudes, and behaviors among Black and Latina women: A randomized controlled trial of the Women United: Clinical Trials and the Fight Against Breast Cancer Program.

Black and Latino adult cancer patients are underrepresented in cancer clinical trials, which limits generalizability of findings and amplifies disparities in healthcare access and outcomes. Community-level education programs designed to address barriers to participation could improve representation in cancer clinical trials. Through a community-campus partner framework, this study evaluated the Women United: Clinical Trials and the Fight Against Breast Cancer Program in Spanish and English. Participants were 422 women (141 Black, 140 Latina Spanish preference, 141 Latina English preference) who were randomized to view either the intervention (n = 215) or a control (n = 207) program. Assessments of clinical trials knowledge and barriers to clinical trials participation were taken before and after viewing. Results suggested that clinical trials knowledge increased and perceived barriers to participation decreased for those who viewed the educational program. More specifically, those in the intervention condition perceived fewer barriers related to personal benefits, mistrust, and familiarity of clinical trials. As expected, there were no differences in perceived barriers related to community support for either condition. Participants in both conditions were equally likely to join a subsequent study or a clinical trials community ambassador program. There were no differences in any of the outcomes across ethnicity or language, suggesting the program works equivalently across groups. This program is easy to administer and can be recommended for use among Black and Latina women to address factors related to clinical trials participation.

Pathways from sociocultural and objectification constructs to body satisfaction among women: The U.S. Body Project I.

Objectification theory proposes that widespread sexualization causes women to engage in surveillance of their appearance. We integrated this concept into a model with constructs from the tripartite influence model, which proposes that body dissatisfaction is a result of internalizing cultural notions of thin ideal beauty that stem from family, peer, and media appearance-related pressures. We tested this model with an online sample of 6327 adult women. Specifically, we tested whether these pressures predicted increased thin-ideal and muscular-ideal internalization, leading to greater body surveillance, and in turn lower appearance evaluation and body image quality of life. Structural equation modeling supported many aspects of the model. Family, peer, and media pressures related to higher thin-ideal internalization, which related to higher body surveillance and lower appearance evaluation. Peer and media pressures related to higher muscular-ideal internalization, which related to lower appearance evaluation. However, muscular-ideal internalization was not related to body image quality of life. An indirect relationship emerged between thin-ideal internalization and body image outcomes via body surveillance. Body mass index (BMI) moderated several of these model paths. Findings highlight the value of this integrated sociocultural model, and of BMI as an important moderating factor when examining objectification and tripartite influence models.