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Systemic sclerosis
Original research
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
  1. Tiffany Dal Santo1,2,
  2. Danielle Rice3,4,
  3. Marie-Eve Carrier1,
  4. Gabrielle Virgili-Gervais1,
  5. Brooke Levis1,
  6. Linda Kwakkenbos5,6,7,
  7. Susan J Bartlett8,9,
  8. Amy Gietzen10,
  9. Karen Gottesman11,
  10. Genevieve Guillot12,
  11. Marie Hudson1,8,
  12. Laura K Hummers13,
  13. Vanessa Malcarne14,15,
  14. Maureen Mayes16,
  15. Luc Mouthon17,18,
  16. Michelle Richard19,20,
  17. Maureen Sauve20,21,
  18. Robyn Wojeck22,
  19. Marie-Claude Geoffroy2,23,
  20. Andrea Benedetti8,9,24,25 and
  21. Brett Thombs1,2,8,25,26,27
  22. on behalf of the SPIN investigators
    1. 1Lady Davis Institute for Medical Research, Montreal, Québec, Canada
    2. 2Department of Psychiatry, McGill University, Montreal, Québec, Canada
    3. 3Department of Psychology, St Joseph's Healthcare Hamilton, Hamilton, Ontario, Canada
    4. 4Department of Psychiatry and Behavioural Neurosciences, McMaster University, Hamilton, Ontario, Canada
    5. 5Department of Clinical Psychology, Radboud Universiteit, Nijmegen, The Netherlands
    6. 6Department of IQ Healthcare, Radboud University Medical Center, Nijmegen, The Netherlands
    7. 7Department of Psychiatry, Radboud University Medical Center, Nijmegen, The Netherlands
    8. 8Department of Medicine, McGill University, Montreal, Québec, Canada
    9. 9Research Institute of the McGill University Health Centre, Montreal, Québec, Canada
    10. 10National Scleroderma Foundation Tri-State Chapter, Buffalo, New York, USA
    11. 11National Scleroderma Foundation, Los Angeles, California, USA
    12. 12Sclérodermie Québec, Longueuil, Québec, Canada
    13. 13The Johns Hopkins University School of Medicine, Baltimore, Maryland, USA
    14. 14Department of Psychology, San Diego State University, San Diego, California, USA
    15. 15San Diego Joint Doctoral Program in Clinical Psychology, San Diego State University/Univeristy of California, San Diego, California, USA
    16. 16University of Texas McGovern Medical School, Houston, Texas, USA
    17. 17Service de Médecine Interne, Centre de Référence Maladies Autoimmunes Systémiques Rares d'Ile de France, Hôpital Cochin, Paris, France
    18. 18Assistance Publique Hôpitaux de Paris-Centre, Hôpital Cochin, Université Paris Cité, Paris, France
    19. 19Scleroderma Atlantic, Halifax, Nova Scotia, Canada
    20. 20Slceroderma Canada, Hamilton, Ontario, Canada
    21. 21Scleroderma Society of Ontario, Hamilton, Ontario, Canada
    22. 22University of Rhode Island, Kingston, Rhode Island, USA
    23. 23McGill Group for Suicide Studies, Douglas Research Centre, Montreal, Québec, Canada
    24. 24Respiratory Epidemiology and Clinical Research Unit, McGill University Health Centre, Montreal, Québec, Canada
    25. 25Department of Epidemiology, Biostatistics, and Occupational Health, McGill University, Montreal, Québec, Canada
    26. 26Biomedical Ethics Unit, McGill University, Montreal, Québec, Canada
    27. 27Department of Psychology, McGill University, Montreal, Québec, Canada
    1. Correspondence to Dr Brett Thombs; brett.thombs{at}mcgill.ca

    Abstract

    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.

    • Patient Reported Outcome Measures
    • Scleroderma, Systemic
    • Risk Factors

    Data availability statement

    Data are available upon reasonable request. De-identified individual participant data with a data dictionary and analysis codes that were used to generate the results reported in this article will be made available upon request to the corresponding author and presentation of a methodologically sound proposal that is approved by the Scleroderma Patient-centered Intervention Network Data Access and Publications Committee. Data requesters will need to sign a data transfer agreement.

    http://creativecommons.org/licenses/by-nc/4.0/

    This is an open access article distributed in accordance with the Creative Commons Attribution Non Commercial (CC BY-NC 4.0) license, which permits others to distribute, remix, adapt, build upon this work non-commercially, and license their derivative works on different terms, provided the original work is properly cited, appropriate credit is given, any changes made indicated, and the use is non-commercial. See: http://creativecommons.org/licenses/by-nc/4.0/.

    https://doi.org/10.1136/rmdopen-2023-003876

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      WHAT IS ALREADY KNOWN ON THIS TOPIC

      • Individuals with systemic sclerosis (SSc) face multiple challenges that can negatively affect their ability to fulfil social roles and participate in social activities.

      WHAT THIS STUDY ADDS

      • On average, satisfaction was only minimally lower than the US general population.

      • Many factors are significantly associated with satisfaction with social roles and activities in SSc.

      HOW THIS STUDY MIGHT AFFECT RESEARCH, PRACTICE OR POLICY

      • Our study suggests that many individuals with SSc may have learnt how to adapt and cope with their limited capacities leading to a minimal impact on their satisfaction with their social roles and activities.

      • More research is needed to better understand strategies that may be used to support coping.

      Introduction

      Systemic sclerosis (SSc, scleroderma) is a chronic autoimmune disease characterised by microvascular damage and fibrosis of the skin and multiple other organs including the lungs, gastrointestinal tract, kidneys and heart.1 Associated challenges include pain, fatigue, disability and diminished physical function, dissatisfaction with appearance, social discomfort, fear of disease and symptom progression, and reduced mental health,1–6 all of which may negatively influence the ability to successfully fulfil social roles and participate in activities.7

      There is limited data on how satisfied people with SSc are with their ability to successfully carry out social roles and participate in activities, but individuals with SSc may experience substantial impairment in social functioning, including limitations in the ability to work, complete personal and household responsibilities, and perform typical daily routines.8–11 A 2018 systematic review10 (7 studies; 795 people with SSc and 1154 healthy controls) found a mean difference in scores on the Short Form Survey-36 social functioning subscale, which measures limitations in social activities due to physical or emotional problems, between SSc patients and healthy controls of 13 points (95% CI 5 to 21 points),10 an approximately 0.5 standardised mean difference.12 13 Only relatively small studies with <300 participants have examined factors associated with satisfaction with social function or participation in social roles in SSc, and these studies have included only small numbers of general SSc disease characteristics as possible factors (eg, diffuse or limited subtype, disease duration) but not specific physician-assessed disease manifestations.14 15

      A better understanding of satisfaction with social roles and activities among people with SSc and factors associated with satisfaction would support development of tools to improve social function and adjustment to changes in social functioning. The objectives of this study were to (1) compare satisfaction with social roles and activities in a large multinational 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

      This was a cross-sectional study that evaluated baseline data from the Scleroderma Patient-centered Intervention Network (SPIN) cohort.16–18 It was reported based on guidance in the Strengthening the Reporting of Observational Studies in Epidemiology statement.19 Methods from studies that use data from the SPIN cohort are similar. Thus, we followed reporting guidance from the Text Recycling Research Project.20

      Participants and procedures

      The SPIN cohort is a convenience sample of participants from seven countries: Australia, Canada, France, Mexico, the UK, the USA and Spain.16–18 Eligible participants are recruited by the attending physician or a nurse coordinator during regular physician visits. Participants included in the SPIN cohort must be ≥18 years of age; fluent in English, French or Spanish; and classified as having SSc based on the 2013 American College of Rheumatology/European League Against Rheumatism classification criteria for SSc21 as verified by a SPIN site physician. After obtaining written informed consent from eligible participants, onsite staff submit an online medical data form and participants receive an automated email with instructions on how to activate their online SPIN account and complete their baseline measures. SPIN cohort participants are invited to complete subsequent online assessments every 3 months. This study used baseline assessment data from participants enrolled in the SPIN cohort from April 2014, the date of inception, until March 2023. SPIN cohort participants were included in this study if they completed all Patient Reported Outcomes Information System (PROMIS-29) version 2.0 domains at their baseline assessment.

      Measures

      SPIN cohort participants provided sociodemographic (race or ethnicity, education level, marital status) and lifestyle (eg, smoking status, alcohol consumption) information and completed patient-reported outcome measures. Physicians reported participants’ age; sex; height; weight; years since initial onset of non-Raynaud phenomenon symptoms; SSc subtype (limited, diffuse, sine); modified Rodnan skin score (mRSS); presence of gastrointestinal symptoms (upper; lower; or no gastrointestinal involvement); presence of digital ulcers anywhere on the fingers; presence of tendon friction rubs (currently; in the past; never); presence of small or large joint contractures (none; mild (≤25% range of motion limitation); moderate to severe (>25%)); presence of pulmonary arterial hypertension; presence of interstitial lung disease; existing history of SSc renal crisis; presence of current or past overlap syndromes (systemic lupus erythematosus, rheumatoid arthritis, Sjogren’s syndrome, autoimmune thyroid disease, idiopathic inflammatory myositis and primary biliary cirrhosis); and presence of SSc-related antibodies (antinuclear antibody, anticentromere, antitopoisomerase I and anti-RNA polymerase III).

      Satisfaction with social roles and activities

      Satisfaction with social roles and activities was evaluated in the SPIN cohort using the 4a Short Form of the PROMIS-29 V.2.0 satisfaction with social roles and activities domain, which assesses patient-reported health status over the past 7 days.22 This domain measures the level of satisfaction individuals have with their ability to perform different social roles and activities such as work inside and outside the home, personal and household responsibilities, and daily routines.23 Satisfaction with social roles and activities is assessed with four items, each rated on a 5-point Likert scale. Item scores are summed to give a satisfaction with social roles and activities domain score that is converted into a T-score standardised to the US general population (mean=50, SD=10).24 Normal levels of satisfaction with one’s social roles and activities is represented by a T-score over 45.0, mild impairment by a T-score that ranges between 40.0 and 45.0, moderate impairment between 30.0 and 39.9 and severe impairment in satisfaction with social roles and activities by a T-score less than 30.0.25 The PROMIS-29 V.2.0 has been validated within the SPIN cohort, with a satisfactory Cronbach’s α ranging from 0.86 to 0.96 for all PROMIS-29 V.2.0 domains and a good convergent validity.26

      Pruritus

      Pruritus severity was evaluated with a single item: ‘In the past week, how severe was your itch?’, with patients using an 11-point numeric rating scale (0=not severe at all to 11=unbearable). Similar numerical rating scales have been shown to be valid for assessing pruritus severity.27

      Pain

      Pain intensity in the last week was assessed with the PROMIS-29 V.2.0 using a single-item: ‘In the past 7 days, how would you rate your pain on average?’.28 29 This item is rated on a 10-point numerical rating scale (0=no pain to 10=worst imaginable pain). Single-level and multi-level item measurements of pain intensity have been shown to perform equivalently in individuals with SSc.30 Pain interference in the last week was assessed with the PROMIS-29 V.2.0 using four items, each rated on a 5-point Likert scale (1=‘not at all’ and 5=‘very much’).

      Statistical analysis

      We computed descriptive statistics for all variables for the entire sample and separately for those with diffuse and limited SSc (including sine) and by sex. Unadjusted outcomes were generated with simple linear regressions used to assess bivariate associations of sociodemographic, lifestyle and disease-related variables with satisfaction with social roles and activities. Adjusted outcomes were generated via multivariable linear regression used to assess the independent association of each variable with satisfaction with social roles and activities. We identified items to be included in the model a priori based on factors associated with psychosocial outcomes in SSc4 7 31–33 and based on the experience of research team members who either have or provide healthcare for individuals with SSc. We did not include psychosocial or functional variables that are outcomes of SSc (depression symptoms, anxiety symptoms, pain, fatigue, self-efficacy) as predictors in the main model as they are likely to have bidirectional causal associations with social functioning. We did this to avoid reverse causality where outcome variables may be causally associated to predictor variables, which can lead to (1) biased model coefficients, potentially masking important associations between disease variables and social functioning; (2) spuriously inflated goodness-of-fit estimates (R2); and (3) inability to determine the relative causal influence between the variables for which reverse causation is likely.34

      Variables included in the main analysis were age (years standardised); male sex (reference=female); years of education (years standardised); single, divorced or separated, or widowed (reference=married or living as married); non-White (reference=White); Canada, UK, France, other (Australia, Mexico, Spain) (reference=USA); body mass index (BMI) (standardised); years since first non-Raynaud’s symptoms (years standardised); diffuse subtype (reference=limited or sine); gastrointestinal involvement (reference=no); digital ulcers (reference=no); current or past tendon friction rubs (reference=never); moderate or severe small joint contractures (reference=none or mild); moderate or severe large joint contractures (reference=none or mild); history of SSc renal crisis (reference=no); interstitial lung disease (reference=no); pulmonary arterial hypertension (reference=no); systemic lupus erythematosus (reference=no); rheumatoid arthritis (reference=no); Sjogren’s syndrome (reference=no); autoimmune thyroid disease (reference=no); idiopathic inflammatory myositis (reference=no); primary biliary cirrhosis (reference=no). We did not include variables such as smoking status or alcohol consumption as social functioning may influence these. See online supplemental material 1 for variable specifications.

      We accounted for missing data by using multiple imputations via chained equations, using the mice package in R.35 We generated 20 imputed datasets, using 15 cycles per dataset. Variables included in the mice procedure included: all variables in the main regression model, all variables considered in sensitivity analyses, alcohol consumption and smoking status, and anxiety, depression, pain intensity and interference, fatigue, sleep, and physical function domain scores on the PROMIS-29 V.2.0.

      We conducted four multivariable sensitivity analyses. We (1) conducted a complete case analysis of the main model; (2) added pruritus and pain to the main model; (3) replaced disease subtype with continuous mRSS; and (4) added SSc-related antibodies (antinuclear antibodies (reference=negative); anticentromere (reference=negative); antitopoisomerase I (Scl70) (reference=negative); and anti-RNA polymerase III (reference=negative)) to the main model. See online supplemental material 1.

      We standardised continuous predictor variables after imputation and prior to entering them into the models. We reported unstandardised regression coefficients with 95% CIs and total explained variance for each model (adjusted R2). All regression analyses were conducted in R (R V.3.6.3, RStudio V.1.2.5042).

      Patient involvement

      Patient members of the SPIN Steering Committee play a role in developing SPIN research priorities, including identifying the need for the present study. Five patient members of the Steering Committee reviewed and provided comments on the study protocol and manuscript and are coauthors.

      Results

      Our sample consisted of 2385 participants from 53 sites with baseline SPIN cohort PROMIS-29 V.2.0 satisfaction with social roles and activities domain data. Participants were predominantly female (N=2079; 87%) and White (N=1970; 83%). Mean (SD) age was 54.9 (12.6) years, mean (SD) education was 15.0 (3.7) years and mean (SD) BMI was 25.3 (5.6). Most participants were from the USA (N=813; 34%), France (N=713; 30%) or Canada (N=515; 22%). Mean (SD) years since onset of first non-Raynaud’s symptoms was 10.9 (8.8), and 904 (38%) participants had diffuse SSc. Table 1 shows participant sociodemographic and disease characteristics, including the number with data for each variable, for the full sample and by disease subtype. See online supplemental material 2 for participant characteristics by sex.

      View this table:
      Table 1

      Sample sociodemographic and disease characteristic

      As shown in table 2, the mean (SD) satisfaction with social roles and activities score in the full sample was 48.1 (9.9), which is slightly lower than the USA general population mean (SD) of 50.0 (10.0). Among all participants, 1307 (55%) had social functioning scores within normal limits; 590 (25%) reported mildly impaired scores, 387 (16%) reported moderately impaired scores; and 101 (4%) reported severe impairment. By country, mean (SD) scores ranged from 45.5 (10.2) among 241 participants from the UK to 50.7 (8.5) in 101 participants from Australia, Mexico or Spain. Participants with diffuse SSc reported a lower mean (SD) of satisfaction with social roles and activities 46.8 (9.8) than those with limited or sine SSc 49.0 (9.8). Scores were similar by sex.

      View this table:
      Table 2

      Satisfaction with social roles and activities by country, disease subtype and sex

      In the main multivariable analysis (table 3), among sociodemographic variables, fewer years of education (−0.54 points per SD in years, 95% CI −0.93 to −0.14); self-reported race or ethnicity other than White (−1.13 points, 95% CI −2.18 to −0.08); and living in Canada (−1.33 points, 95% CI −2.40 to −0.26) or UK (−2.49 points, 95% CI −3.92 to −1.06) were associated with reduced satisfaction with social roles and activities. Higher BMI (−1.08 points per SD in BMI, 95% CI −1.47 to −0.69) was also significantly associated. Among disease variables, there were significant associations with gastrointestinal involvement (−3.16 points, 95% CI −4.27 to −2.05); digital ulcers (−1.90 points, 95% CI −3.05 to −0.76); moderate (−1.62 points, 95% CI −2.78 to −0.45) or severe (−2.26 points, 95% CI −3.99 to −0.52) small joint contractures; interstitial lung disease (−1.11 points, 95% CI −1.97 to −0.25); and pulmonary arterial hypertension (−2.69 points, 95% CI −4.08 to −1.30). Among overlap syndromes, rheumatoid arthritis (−2.51 points, 95% CI −4.28 to −0.73); and Sjogren’s syndrome (−2.42 points, 95% CI −3.96 to −0.88) were significantly associated. Variables not significantly associated were age; sex; marital status; living in France, Australia, Mexico or Spain; years since first non-Raynaud’s symptoms; disease subtype; presence of current or past tendon friction rubs; moderate or severe large joint contractures; history of SSc renal crisis; systemic lupus erythematosus; autoimmune thyroid disease; idiopathic inflammatory myositis; and primary biliary cirrhosis. Adjusted R2 for the final model was 0.10.

      View this table:
      Table 3

      Linear regression analysis of sociodemographic and disease characteristic associations with satisfaction with social roles and activities

      Complete case analysis results, which included 1664 participants, were similar to those of the main analyses (see online supplemental material 3). When adding pruritus and pain intensity to the model in a sensitivity analysis, both pruritus (−0.83 points per SD in pruritus, 95% CI −1.20 to −0.47); and pain intensity (−5.33 points per SD in pain intensity, 95% CI −5.69 to −4.98) were associated with reduced satisfaction with social roles and activities. The sensitivity analysis replacing disease subtype with mRSS found that mRSS was significantly associated with satisfaction with social roles and activities (−0.69 points per SD in mRSS score, 95% CI −1.18 to −0.21). No antibodies had a significant association. See online supplemental materials 4–6.

      Discussion

      Among 2385 participants with SSc from 7 countries, the mean T-score for satisfaction with social roles and activities was 48.1 (9.9), which is approximately 0.2 SD below the US general population mean. Over half (55%) of participants reported satisfaction with social roles and activities within normal limits. We found that disease variables associated with reduced satisfaction with social roles and activities included gastrointestinal involvement, digital ulcers, the presence of moderate or severe small joint contractures, interstitial lung disease, pulmonary arterial hypertension, mRSS, pruritus, pain intensity, and the presence of overlap syndromes including rheumatoid arthritis and Sjogren’s syndrome. We also found that fewer years of education, self-reported race or ethnicity other than White, living in Canada or the UK, having a higher BMI, pruritus, pain intensity and mRSS were associated with greater impairment in satisfaction with one’s social functioning. Among those with large associations, gastrointestinal involvement may reduce one’s ability to carry out social roles due to symptoms such as nausea, abdominal pain or faecal incontinence36; pulmonary arterial hypertension can cause breathlessness, fatigue and dizziness, which can impact the ability to perform everyday tasks such as travelling to work or taking care of household chores37 38; hand and joint involvement can decrease one’s the ability to carry out many tasks necessary for work or household roles8; overlap syndromes, including rheumatoid arthritis or Sjogren’s syndrome, present their own challenges and can exacerbate other symptom-related barriers from SSc.

      Our findings on satisfaction with ability to participate in social roles and activities are generally consistent with the next largest study of people with SSc and another large study of people with rheumatoid arthritis or systemic lupus erythematosus.39 40 One study assessed 477 Australian patients with SSc and reported a mean (SD) on the PROMIS-29v1 social interaction domain of 46.5 (9.7).39 The other study assessed 4346 participants with rheumatoid arthritis and 240 with systemic lupus erythematosus, and reported PROMIS-29 satisfaction with social role T-score means (SD) of 48.9 (9.7) for rheumatoid arthritis, and 49.2 (10.0) for systemic lupus erythematosus.40

      Our findings on satisfaction with social roles and activities, which were close to the US general population mean, differed from our findings on physical function using data from the same participants from the SPIN cohort.41 In that study, we found a mean T-score (SD) for the PROMIS-29 V.2.0 physical function domain of 43.7 (8.9), equivalent to approximately 2/3 of an SD below the US general population mean. The finding of substantially lower physical function but only minimally impaired satisfaction with social roles and activities may reflect the nature of each construct. The physical function domain is a relatively objective measure, though self-reported, of one’s ability to complete concrete tasks, such as going up and down stairs, whereas the satisfaction with social roles and activities domain measures one’s satisfaction with their ability to fulfil social roles and participate in activities. This is also seen in other chronic diseases, where people adapt their expectations and satisfaction with what they can do, despite objectively decreasing abilities. This phenomenon is known as a response shift42 and suggests that people may be coping with the changes they are experiencing by adapting their expectations and, thus, their level of satisfaction with what they are able to do despite their adverse circumstances.43 44 The degree to which people may be able to participate in social roles and activities may be reduced but their satisfaction with their ability to participate is not.

      The adjusted R2 for the main multivariable model was 0.10. Low R2 measures are expected in samples of people with a chronic condition as all experience similar symptoms and side effects of the condition. High R2 values are an important consideration in predictive modelling but are not as valuable when models are used for testing hypotheses about the possible effects of variables of interest. In such cases, including in the present study, having a large enough sample size to generate reasonably precise parameter estimates is a more important consideration.34

      More studies of this nature are needed to better understand the course of satisfaction with social roles and activities in SSc. Future studies might focus on pinpointing characteristics of individuals who successfully cope and adapt and potential reasons as why differences occur. We found that a wide variety of factors may impact an individual’s satisfaction with their social abilities, some of which are modifiable (eg, BMI), but some of which are not (eg, co-occurring diseases such as rheumatoid arthritis). Considering this, future studies are needed to help patients who are less satisfied with their social roles and activities cope with and accept the challenges related to living with SSc. One potential strategy may be acceptance and commitment therapy (ACT), which works on verbal behaviour to influence and change one’s thoughts and feelings.45 A 2016 systematic review (N=18 studies) on the use of ACT in chronic and long-term conditions concluded that evidence is promising, although limited.46

      Strengths of our study include its large international sample with participants from 53 SPIN sites across seven countries, our analysis of a wide range of sociodemographic, lifestyle and physician-assessed disease-related factors, and the involvement of people with lived SSc experience in the project via leadership in SPIN and participation in the study. There are some limitations that also need to be considered. First, the SPIN cohort is a convenience sample. However, a comparison with the European Scleroderma Trials and Research and Canadian Scleroderma Research Group cohorts indicated broad comparability of participant characteristics, which supports generalisability in SSc.17 Second, participants were required to answer questions via online questionnaires, which could potentially reduce generalisability of our results. Third, this was a cross-sectional study and we cannot infer causality based on our results. Fourth, our sample may have been older or differed in other ways compared with the US general population sample to which we compared levels of satisfaction with social roles. Fifth, we did not assess the number of organs involved or the presence of end of stage organ dysfunction.

      To summarise, we assessed the association of several factors with satisfaction with social roles and activities in 2385 patients with SSc. We found many variables statistically significantly associated, including fewer years of education, self-reported race or ethnicity other than White, living in Canada or the UK, having a higher BMI, gastrointestinal involvement, presence of digital ulcers, moderate or severe small joint contractures, interstitial lung disease, pulmonary arterial hypertension, pruritus, pain intensity, mRSS, rheumatoid arthritis and Sjogren’s syndrome. However, overall, there was only a minimal impairment in satisfaction with social roles and activities, despite evident physical limitations. This may suggest that many individuals with SSc have learnt how to adapt and cope with limited physical capacity. More research is needed to better understand strategies that may be used to support coping. For the time being, healthcare providers can work to help patients adapt and cope with their current symptoms, side effects, and levels of satisfaction with their social roles and activities.

      Data availability statement

      Data are available upon reasonable request. De-identified individual participant data with a data dictionary and analysis codes that were used to generate the results reported in this article will be made available upon request to the corresponding author and presentation of a methodologically sound proposal that is approved by the Scleroderma Patient-centered Intervention Network Data Access and Publications Committee. Data requesters will need to sign a data transfer agreement.

      Ethics statements

      Patient consent for publication

      Ethics approval

      This study involves human participants and the SPIN cohort study was approved by Research Ethics Committee of the Centre intégré universitaire de santé et de services sociaux du Centre-Ouest-de-l’Île-de-Montréal (#MP-05-2013-150) and by the ethics committees of all recruiting sites. Participants gave informed consent to participate in the study before taking part.

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      Supplementary materials

      • Supplementary Data

        This web only file has been produced by the BMJ Publishing Group from an electronic file supplied by the author(s) and has not been edited for content.

      Footnotes

      • Collaborators Catherine Fortuné, Ottawa Scleroderma Support Group, Ottawa, Ontario, Canada; Claire E. Adams, Lady Davis Institute for Medical Research, Jewish General Hospital, Montreal, Quebec, Canada; Ghassan El-Baalbaki, Université du Québec à Montréal, Montreal, Quebec, Canada; Kim Fligelstone, Scleroderma & Raynaud’s UK, London, UK; Tracy Frech, Vanderbilt University, Nashville, Tennessee, USA; Daphna Harel, New York University, New York, New York, USA; Richard S. Henry, Lady Davis Institute for Medical Research, Jewish General Hospital, Montreal, Quebec, Canada; Monique Hinchcliff, Yale School of Medicine, New Haven, Connecticut, USA; Sindhu R. Johnson, Toronto Scleroderma Program, Mount Sinai Hospital, Toronto Western Hospital, and University of Toronto, Toronto, Ontario, Canada; Maggie Larche, McMaster University, Hamilton, Ontario, Canada; Catarina Leite, University of Minho, Braga, Portugal; Christelle Nguyen, Université Paris Descartes, Université de Paris, Paris, France, and Assistance Publique - Hôpitaux de Paris, Paris, France; Karen Nielsen, Scleroderma Society of Ontario, Hamilton, Ontario, Canada; Janet Pope, University of Western Ontario, London, Ontario, Canada; François Rannou, Université Paris Descartes, Université de Paris, Paris, France, and Assistance Publique - Hôpitaux de Paris, Paris, France; Tatiana Sofia Rodriguez-Reyna, Instituto Nacional de Ciencias Médicas y Nutrición Salvador Zubirán, Mexico City, Mexico; Anne A. Schouffoer, Leiden University Medical Center, Leiden, the Netherlands; Maria E. Suarez-Almazor, University of Texas MD Anderson Cancer Center, Houston, Texas, USA; Christian Agard, Centre Hospitalier Universitaire - Hôtel-Dieu de Nantes, Nantes, France; Laurent Alric, CHU Rangueil, Toulouse, France; Marc André, Centre Hospitalier Universitaire Gabriel-Montpied, Clermont-Ferrand, France; Floryan Beaslay, CHU La Réunion, Saint-Denis, La Réunion, France; Elana J. Bernstein, Columbia University, New York, New York, USA; Sabine Berthier, Centre Hospitalier Universitaire Dijon Bourgogne, Dijon, France; Lyne Bissonnette, Université de Sherbrooke, Sherbrooke, Quebec, Canada; Sophie Blaise, Grenoble Alpes University Hospital, France; Eva Bories, CHU Rangueil, Toulouse, France; Alessandra Bruns, Université de Sherbrooke, Sherbrooke, Quebec, Canada; Carlotta Cacciatore, Assistance Publique – Hôpitaux de Paris, Hôpital St-Louis, Paris, France; Patricia Carreira, Servicio de Reumatologia del Hospital 12 de Octubre, Madrid, Spain; Marion Casadevall, Assistance Publique - Hôpitaux de Paris, Hôpital Cochin, Paris, France; Benjamin Chaigne, Assistance Publique - Hôpitaux de Paris, Hôpital Cochin, Paris, France; Lorinda Chung, Stanford University, Stanford, California, USA; Benjamin Crichi, Assistance Publique - Hôpitaux de Paris, Hôpital St-Louis, Paris, France; Thylbert Deltombe, CHU La Réunion, Saint-Denis, La Réunion, France; Christopher Denton, Royal Free London Hospital, London, UK; Tannvir Desroche, CHU La Réunion, Saint-Denis, La Réunion, France; Robyn Domsic, University of Pittsburgh, Pittsburgh, Pennsylvania, USA; James V. Dunne, St. Paul's Hospital and University of British Columbia, Vancouver, British Columbia, Canada; Bertrand Dunogue, Assistance Publique - Hôpitaux de Paris, Hôpital Cochin, Paris, France; Regina Fare, Servicio de Reumatologia del Hospital 12 de Octubre, Madrid, Spain; Dominique Farge-Bancel, Assistance Publique - Hôpitaux de Paris, Hôpital St-Louis, Paris, France; Paul R. Fortin, CHU de Québec - Université Laval, Quebec, Quebec, Canada; Loraine Gauzère, CHU La Réunion, Saint-Denis, La Réunion, France; Anne Gerber, CHU La Réunion, Saint-Denis, La Réunion, France; Jessica Gordon, Hospital for Special Surgery, New York City, New York, USA; Brigitte Granel-Rey, Université, and Assistance Publique - Hôpitaux de Marseille, Hôpital Nord, Marseille, France; Aurélien Guffroy, Les Hôpitaux Universitaires de Strasbourg, Nouvel Hôpital Civil, Strasbourg, France; Geneviève Gyger, Jewish General Hospital and McGill University, Montreal, Quebec, Canada; Eric Hachulla, Centre Hospitalier Régional Universitaire de Lille, Hôpital Claude Huriez, Lille, France; Sabrina Hoa, Centre hospitalier de l’Université de Montréal – CHUM, Montreal, Quebec, Canada; Michael Hughes, Salford Royal NHS Foundation Trust, Salford, UK; Alena Ikic, CHU de Québec - Université Laval, Quebec, Quebec; Niall Jones, University of Alberta, Edmonton, Alberta, Canada; Suzanne Kafaja, University of California, Los Angeles, California, USA; Nader Khalidi, McMaster University, Hamilton, Ontario, Canada; Kimberly Lakin, Hospital for Special Surgery, New York City, New York, USA; Marc Lambert, Centre Hospitalier Régional Universitaire de Lille, Hôpital Claude Huriez, Lille, France; David Launay, Centre Hospitalier Régional Universitaire de Lille, Hôpital Claude Huriez, Lille, France; Yvonne C. Lee, Northwestern University, Chicago, Illinois, USA; Paul Legendre, Centre Hospitalier du Mans, Le Mans, France; Hélène Maillard, Centre Hospitalier Régional Universitaire de Lille, Hôpital Claude Huriez, Lille, France; Nancy Maltez, University of Ottawa, Ottawa, Ontario, Canada; Joanne Manning, Salford Royal NHS Foundation Trust, Salford, UK; Isabelle Marie, CHU Rouen, Hôpital de Bois-Guillaume, Rouen, France; Maria Martin Lopez, Servicio de Reumatologia del Hospital 12 de Octubre, Madrid, Spain; Thierry Martin, Les Hôpitaux Universitaires de Strasbourg, Nouvel Hôpital Civil, Strasbourg, France; Ariel Masetto, Université de Sherbrooke, Sherbrooke, Quebec, Canada; François Maurier, Uneos - Groupe hospitalier associatif, Metz, France; Arsène Mekinian, Assistance Publique - Hôpitaux de Paris, Hôpital St-Antoine, Paris, France; Sheila Melchor Díaz, Servicio de Reumatologia del Hospital 12 de Octubre, Madrid, Spain; Morgane Mourguet, CHU Rangueil, Toulouse, France; Mandana Nikpour, St Vincent’s Hospital and University of Melbourne, Melbourne, Victoria, Australia; Louis Olagne, Centre Hospitalier Universitaire Gabriel-Montpied, Clermont-Ferrand, France; Vincent Poindron, Les Hôpitaux Universitaires de Strasbourg, Nouvel Hôpital Civil, Strasbourg, France; Susanna Proudman, Royal Adelaide Hospital and University of Adelaide, Adelaide, South Australia, Australia; Grégory Pugnet, CHU Rangueil, Toulouse, France; Loïc Raffray, CHU La Réunion, Saint-Denis, La Réunion, France; Alexis Régent, Assistance Publique - Hôpitaux de Paris, Hôpital Cochin, Paris, France; Frederic Renou, CHU La Réunion, Saint-Denis, La Réunion, France; Sébastien Rivière, Assistance Publique - Hôpitaux de Paris, Hôpital St-Antoine, Paris, France; David Robinson, University of Manitoba, Winnipeg, Manitoba, Canada; Esther Rodríguez Almazar, Servicio de Reumatologia del Hospital 12 de Octubre, Madrid, Spain; Sophie Roux, Université de Sherbrooke, Sherbrooke, Quebec, Canada; Perrine Smets, Centre Hospitalier Universitaire Gabriel-Montpied, Clermont-Ferrand, France; Vincent Sobanski, Centre Hospitalier Régional Universitaire de Lille, Hôpital Claude Huriez, Lille, France; Robert Spiera, Hospital for Special Surgery, New York City, New York, USA; Virginia Steen, Georgetown University, Washington, DC, USA; Evelyn Sutton, Dalhousie University, Halifax, Nova Scotia, Canada; Carter Thorne, Southlake Regional Health Centre, Newmarket, Ontario, Canada; Damien Vagner, CHU La Réunion, Saint-Denis, La Réunion, France; John Varga, University of Michigan, Ann Arbor, Michigan, USA; Pearce Wilcox, St. Paul's Hospital and University of British Columbia, Vancouver, British Columbia, Canada; Mara Cañedo Ayala, Lady Davis Institute for Medical Research, Jewish General Hospital, Montreal, Quebec, Canada; Vanessa Cook, Lady Davis Institute for Medical Research, Jewish General Hospital, Montreal, Quebec, Canada; Sophie Hu, Lady Davis Institute for Medical Research, Jewish General Hospital, Montreal, Quebec, Canada; Elsa-Lynn Nassar, Lady Davis Institute for Medical Research, Jewish General Hospital, Montreal, Quebec, Canada; Marieke Alexandra Neyer, Lady Davis Institute for Medical Research, Jewish General Hospital, Montreal, Quebec, Canada; and Sabrina Provencher, Lady Davis Institute for Medical Research, Jewish General Hospital, Montreal, Quebec, Canada.

      • Contributors TDS, DR, M-EC, BL, LK, M-CG, AB, BT contributed to study conceptualisation; GV-G, BL to data curation; TDS, GV-G, BL to formal analysis; TDS, M-EC, LK, BT to funding acquisition; TDS, DR, M-EC, LK, SJB, AG, KG, GG, MH, LKH, VM, MM, LM, MR, MS, RW, BT to investigation; TDS, DR, M-EC, BL, LK, BT to methodology; M-EC to project administration; DR, M-CG, AB, BT to supervision; TDS to visualisation; TDS, BT to writing the original draft; and all authors reviewing and editing the final draft. BT is the guarantor.

      • Funding The Scleroderma Patient-centered Intervention Network (SPIN) cohort has received funding from the Canadian Institutes of Health Research (CIHR); the Lady Davis Institute for Medical Research of the Jewish General Hospital, Montreal, Quebec, Canada; the Jewish General Hospital Foundation, Montreal, Quebec, Canada; and McGill University, Montreal, Quebec, Canada. SPIN has also received support from the Scleroderma Society of Ontario; Scleroderma Canada; Sclérodermie Québec; Scleroderma Manitoba; Scleroderma Atlantic; the Scleroderma Association of BC; Scleroderma SASK; Scleroderma Australia; Scleroderma New South Wales; Scleroderma Victoria; and Scleroderma Queensland. TDS was supported by a CIHR Masters Award and M-CG and BT by Canada Research Chairs, all outside of the present work.

      • Competing interests None declared.

      • Provenance and peer review Not commissioned; externally peer reviewed.

      • Supplemental material This content has been supplied by the author(s). It has not been vetted by BMJ Publishing Group Limited (BMJ) and may not have been peer-reviewed. Any opinions or recommendations discussed are solely those of the author(s) and are not endorsed by BMJ. BMJ disclaims all liability and responsibility arising from any reliance placed on the content. Where the content includes any translated material, BMJ does not warrant the accuracy and reliability of the translations (including but not limited to local regulations, clinical guidelines, terminology, drug names and drug dosages), and is not responsible for any error and/or omissions arising from translation and adaptation or otherwise.