Sarah D. Mills

The Scleroderma Patient-centered Intervention Network (SPIN) Cohort: protocol for a cohort multiple randomised controlled trial (cmRCT) design to support trials of psychosocial and rehabilitation interventions in a rare disease context

Introduction Psychosocial and rehabilitation interventions are increasingly used to attenuate disability and improve health-related quality of life (HRQL) in chronic diseases, but are typically not available for patients with rare diseases. Conducting rigorous, adequately powered trials of these interventions for patients with rare diseases is difficult. The Scleroderma Patient-centered Intervention Network (SPIN) is an international collaboration of patient organisations, clinicians and researchers. The aim of SPIN is to develop a research infrastructure to test accessible, low-cost self-guided online interventions to reduce disability and improve HRQL for people living with the rare disease systemic sclerosis (SSc or scleroderma). Once tested, effective interventions will be made accessible through patient organisations partnering with SPIN. Methods and analysis SPIN will employ the cohort multiple randomised controlled trial (cmRCT) design, in which patients consent to participate in a cohort for ongoing data collection. The aim is to recruit 1500–2000 patients from centres across the world within a period of 5 years (2013–2018). Eligible participants are persons ≥18 years of age with a diagnosis of SSc. In addition to baseline medical data, participants will complete patient-reported outcome measures every 3 months. Upon enrolment in the cohort, patients will consent to be contacted in the future to participate in intervention research and to allow their data to be used for comparison purposes for interventions tested with other cohort participants. Once interventions are developed, patients from the cohort will be randomly selected and offered interventions as part of pragmatic RCTs. Outcomes from patients offered interventions will be compared with outcomes from trial-eligible patients who are not offered the interventions. Ethics and dissemination The use of the cmRCT design, the development of self-guided online interventions and partnerships with patient organisations will allow SPIN to develop, rigourously test and effectively disseminate psychosocial and rehabilitation interventions for people with SSc.

Psychosocial Aspects of Scleroderma.

Patients with systemic sclerosis (SSc; also called scleroderma) have to cope with not only the physical impacts of the disease but also the emotional and social consequences of living with the condition. Because there is no cure for SSc, improving quality of life is a primary focus of treatment and an important clinical challenge. This article summarizes significant problems faced by patients with SSc, including depression, anxiety, fatigue, sleep disruption, pain, pruritus, body image dissatisfaction, and sexual dysfunction, and describes options to help patients cope with the consequences of the disease.

Psychosocial aspects of systemic sclerosis.

Purpose of reviewIn 2010, an international consortium of researchers published a consensus agenda for research on psychosocial aspects of systemic sclerosis (SSc; scleroderma). The present review summarizes recent research on SSc-related depression and anxiety, fatigue and sleep, pain, pruritus, body image distress, sexual function, work disability, healthcare needs, psychosocial interventions, and psychoneuroimmunology. Recent findingsResearchers have used structured interviews to establish prevalence rates for clinical mood disorders in SSc, although anxiety remains understudied and distress may be a useful outcome to consider. Longitudinal research has identified predictors of fatigue. Research on body image distress suggests the importance of changes in the facial skin and hands. Studies have identified sexual function problems in women and men with SSc. A prospective study found that breathing problems and fatigue predicted workplace disability. A randomized controlled trial evaluating multidisciplinary care showed benefits for health-related quality of life. SummaryThere has been a recent expansion in studies of psychosocial aspects of SSc, and in the validation of psychosocial measures that can be used in clinics to identify problems and track outcomes; however, prospective studies remain scarce. To better address the psychosocial needs of persons with SSc, interventions need to be developed and tested via randomized controlled trials with power to detect clinically meaningful changes.

Psychometric Evaluation of the Brief Acculturation Scale for Hispanics.

This study examined the psychometric properties of the Brief Acculturation Scale for Hispanics (BASH), a four-item, language-based measure of acculturation. Participants in the study were 435 Hispanic Americans from a large metropolitan area with English or Spanish language preference. Internal consistency reliability was strong in both language-preference groups. Multiple-group confirmatory factor analysis was used to evaluate the structural validity of the measure. A unidimensional factor structure was found for both English and Spanish language-preference groups and items loaded equivalently across groups, demonstrating measurement invariance. The BASH had good convergent validity and incremental validity. Overall, this study provides further evidence that the BASH offers a brief, reliable, and valid measure of acculturation to be used among Hispanic Americans.

The psychometric properties of the generalized anxiety disorder-7 scale in Hispanic Americans with English or Spanish language preference.

The Generalized Anxiety Disorder-7 scale (GAD-7) is a self-report questionnaire that is widely used to screen for anxiety. The GAD-7 has been translated into numerous languages, including Spanish. Previous studies evaluating the structural validity of the English and Spanish versions indicate a unidimensional factor structure in both languages. However, the psychometric properties of the Spanish language version have yet to be evaluated in samples outside of Spain, and the measure has not been tested for use among Hispanic Americans. This study evaluated the reliability, structural validity, and convergent validity of the English and Spanish language versions of the GAD-7 for Hispanic Americans in the United States. A community sample of 436 Hispanic Americans with an English (n = 210) or Spanish (n = 226) language preference completed the GAD-7. Multiple-group confirmatory factor analysis (CFA) was used to examine the goodness-of-fit of the unidimensional factor structure of the GAD-7 across language-preference groups. Results from the multiple-group CFA indicated a similar unidimensional factor structure with equivalent response patterns and item intercepts, but different variances, across language-preference groups. Internal consistency was good for both English and Spanish language-preference groups. The GAD-7 also evidenced good convergent validity as demonstrated by significant correlations in expected directions with the Perceived Stress Scale, the Patient Health Questionnaire-9, and the Physical Health domain of the World Health Organization Quality of Life-BREF assessment. The unidimensional GAD-7 is suitable for use among Hispanic Americans with an English or Spanish language preference.

Evaluation of the Satisfaction with Appearance Scale and Its Short Form in Systemic Sclerosis: Analysis from the UCLA Scleroderma Quality of Life Study

Objective. Changes in appearance are common in patients with systemic sclerosis (SSc) and can significantly affect well-being. The Satisfaction with Appearance Scale (SWAP) measures body image dissatisfaction in persons with visible disfigurement; the Brief-Satisfaction with Appearance Scale (Brief-SWAP) is its short form. The present study evaluated the reliability and validity of SWAP and Brief-SWAP scores in SSc. Methods. A sample of 207 patients with SSc participating in the University of California, Los Angeles Scleroderma Quality of Life Study completed the SWAP. Brief-SWAP scores were derived from the SWAP. The structural validity of both measures was investigated using confirmatory factor analysis. Internal consistency reliability of total and subscale scores was assessed with Cronbach’s alpha coefficients. Convergent and divergent validity was evaluated using the Center for Epidemiological Studies Depression Scale, the Health Assessment Questionnaire-Disability Index, and the Medical Outcomes Study Short Form-36 questionnaire. Results. SWAP and Brief-SWAP total scores were highly correlated (r = 0.97). The 4-factor structure of the SWAP fit well descriptively; the 2-factor structure of the Brief-SWAP fit well descriptively and statistically. Internal consistencies for total and subscale scores were good, and results supported convergent and divergent validity. Conclusion. Both versions are suitable for use in patients with SSc. The Brief-SWAP is most efficient; the full SWAP yields additional subscales that may be informative in understanding body image issues in patients with SSc.

Multidimensional profiles of health locus of control in Hispanic Americans.

Latent profile analysis identified health locus of control profiles among 436 Hispanic Americans who completed the Multidimensional Health Locus of Control scales. Results revealed four profiles: Internally Oriented-Weak, -Moderate, -Strong, and Externally Oriented. The profile groups were compared on sociocultural and demographic characteristics, health beliefs and behaviors, and physical and mental health outcomes. The Internally Oriented-Strong group had less cancer fatalism, religiosity, and equity health attributions, and more alcohol consumption than the other three groups; the Externally Oriented group had stronger equity health attributions and less alcohol consumption. Deriving multidimensional health locus of control profiles through latent profile analysis allows examination of the relationships of health locus of control subtypes to health variables.

Psychometric Evaluation of the Patient Health Questionnaire–4 in Hispanic Americans

The present study evaluated the psychometric properties of the Patient Health Questionnaire–4 (PHQ-4), a screener of psychological distress, in English- and Spanish-speaking Hispanic Americans. Hispanic American adults (N = 436) completed the PHQ-4, which yields two subscales (Anxiety and Depression) that can be summed to create a total score. Multiple-group confirmatory factor analysis was used to evaluate structural validity. The two-factor structure was the best fit to the data for both English- and Spanish-speaking Hispanic Americans, and items loaded equivalently across groups, demonstrating measurement invariance. Internal consistency reliability was good as measured by coefficient alpha. Construct validity was evidenced by significant expected relationships with perceived stress. These findings provide support for the reliability and validity of the PHQ-4 as a brief measure of psychological distress for English- or Spanish-speaking Hispanic Americans.

Perceptions of Cancer Risk/Efficacy and Cancer-Related Risk Behaviors: Results From the HCHS/SOL Sociocultural Ancillary Study

This study evaluated the associations among perceived risk, perceived efficacy, and engagement in six cancer-related risk behaviors in a population-based Hispanic/Latino sample. Interviews were conducted with 5,313 Hispanic/Latino adults as part of the Hispanic Community Health Study/Study of Latinos (HCHS/SOL) Sociocultural Ancillary Study. Participants were recruited from the study’s four field centers (Bronx, NY; Chicago, IL; Miami, FL; San Diego, CA) between February 2010 and June 2011. Perceived risk and perceived efficacy were assessed with questions drawn from the Health Interview National Trends Survey. More than half of the sample endorsed perceived risk of cancer associated with the six evaluated behaviors, as well as general perceived efficacy for preventing cancer. Adjusted logistic regression analyses demonstrated significant differences across Hispanic/Latino background groups for perceived risk associated with high consumption of alcohol and saturated fat, low consumption of fruits and vegetables, and insufficient exercise but not with smoking or low consumption of fiber. Differences were also found for the belief, “It seems like everything causes cancer” but not for other perceived efficacy items. Perceived cancer risk and perceived efficacy for preventing cancer were neither independently nor interactively associated with engagement in cancer-related risk behaviors after controlling for sociodemographic covariates. Results suggest that perceptions of risk and efficacy with regard to cancer vary across Hispanic/Latino background groups, and therefore background group differences should be considered in prevention efforts. Perceived risk and perceived efficacy were not related to cancer-related risk behaviors among Hispanics/Latinos. Further work is needed to evaluate determinants of cancer-related risk in this population.

Validation of the Social Appearance Anxiety Scale in Patients With Systemic Sclerosis: A Scleroderma Patient-Centered Intervention Network Cohort Study

Systemic sclerosis (SSc) is an autoimmune disease that can cause disfiguring changes in appearance. This study examined the structural validity, internal consistency reliability, convergent validity, and measurement equivalence of the Social Appearance Anxiety Scale (SAAS) across SSc disease subtypes.