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

Robyn K Wojeck; Mitchell R Knisely; Donald E Bailey; Tamara J Somers; Linda Kwakkenbos; Marie-Eve Carrier; Warren R Nielson; Susan J Bartlett; Vanessa L Malcarne; Marie Hudson; Brooke Levis; Andrea Benedetti; Luc Mouthon; Brett D Thombs; Susan G Silva; SPIN Investigators

doi:10.1016/j.eclinm.2023.102104

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

EClinicalMedicine. 2023 Jul 20:62:102104. doi: 10.1016/j.eclinm.2023.102104. eCollection 2023 Aug.

Authors

Robyn K Wojeck¹, Mitchell R Knisely², Donald E Bailey², Tamara J Somers³, Linda Kwakkenbos^{4

5

6

7}, Marie-Eve Carrier⁸, Warren R Nielson⁹, Susan J Bartlett^{10

11}, Vanessa L Malcarne^{12

13}, Marie Hudson^{8

10}, Brooke Levis⁸, Andrea Benedetti^{10

14

15}, Luc Mouthon^{16

17}, Brett D Thombs^{8

10

14

18

19

20}, Susan G Silva²; SPIN Investigators

Collaborators

SPIN Investigators:
Claire E Adams, Richard S Henry, Catherine Fortuné, Karen Gottesman, Geneviève Guillot, Laura K Hummers, Amanda Lawrie-Jones, Maureen D Mayes, Michelle Richard, Maureen Sauvé, Shervin Assassi, Ghassan El-Baalbaki, Kim Fligelstone, Tracy Frech, Amy Gietzen, Daphna Harel, Monique Hinchcliff, Sindhu R Johnson, Maggie Larche, Catarina Leite, Christelle Nguyen, Karen Nielsen, Janet Pope, François Rannou, Tatiana Sofia Rodriguez-Reyna, Anne A Schouffoer, Maria E Suarez-Almazor, Christian Agard, Nassim Ait Abdallah, Marc André, Elana J Bernstein, Sabine Berthier, Lyne Bissonnette, Alessandra Bruns, Patricia Carreira, Marion Casadevall, Benjamin Chaigne, Lorinda Chung, Benjamin Crichi, Christopher Denton, Robyn Domsic, James V Dunne, Bertrand Dunogue, Regina Fare, Dominique Farge-Bancel, Paul R Fortin, Jessica Gordon, Brigitte Granel-Rey, Aurélien Guffroy, Genevieve Gyger, Eric Hachulla, Sabrina Hoa, Alena Ikic, Suzanne Kafaja, Nader Khalidi, Kimberly Lakin, Marc Lambert, David Launay, Yvonne C Lee, Hélène Maillard, Nancy Maltez, Joanne Manning, Isabelle Marie, Maria Martin Lopez, Thierry Martin, Ariel Masetto, François Maurier, Arsene Mekinian, Sheila Melchor Díaz, Mandana Nikpour, Louis Olagne, Vincent Poindron, Susanna Proudman, Alexis Régent, Sébastien Rivière, David Robinson, Esther Rodríguez Almazar, Sophie Roux, Perrine Smets, Vincent Sobanski, Robert Spiera, Virginia Steen, Evelyn Sutton, Carter Thorne, John Varga, Pearce Wilcox, Mara Cañedo Ayala, Vanessa Cook, Sophie Hu, Bianca Matthews, Elsa-Lynn Nassar, Marieke Alexandra Neyer, Julia Nordlund, Sabrina Provencher

Affiliations

¹ University of Rhode Island, Kingston, Rhode Island, USA.
² Duke University School of Nursing, Durham, North Carolina, USA.
³ Duke University School of Medicine, Durham, North Carolina, USA.
⁴ Department of Clinical Psychology, Radboud University, Nijmegen, the Netherlands.
⁵ Department of Medical Psychology, Radboud Institute for Health Sciences, Radboud University Medical Center, Nijmegen, the Netherlands.
⁶ IQ Healthcare, Radboud Institute for Health Sciences, Radboud University Medical Center, Nijmegen, the Netherlands.
⁷ Radboudumc Center for Mindfulness, Department of Psychiatry, Radboud Institute for Health Sciences, Radboud University Medical Center, Nijmegen, the Netherlands.
⁸ Lady Davis Institute for Medical Research, Jewish General Hospital, Montreal, Quebec, Canada.
⁹ Lawson Health Research Institute, London, Ontario, Canada.
¹⁰ Department of Medicine, McGill University, Montreal, Quebec, Canada.
¹¹ Research Institute of the McGill University Health Centre, Montreal, Quebec, Canada.
¹² Department of Psychology, San Diego State University, California, USA.
¹³ San Diego State University/University of California, San Diego Joint Doctoral Program in Clinical Psychology, San Diego, California, USA.
¹⁴ Department of Epidemiology, Biostatistics, and Occupational Health, McGill University, Montreal, Quebec, Canada.
¹⁵ Respiratory Epidemiology and Clinical Research Unit, McGill University Health Centre, Montreal, Quebec, Canada.
¹⁶ Service de Médecine Interne, Centre de Référence Maladies Autoimmunes Systémiques Rares d'Ile de France, Hôpital Cochin, Assistance Publique-Hôpitaux de Paris (AP-HP), Paris, France.
¹⁷ APHP-CUP, Hôpital Cochin, Université de Paris, Paris, France.
¹⁸ Department of Psychiatry, McGill University, Montreal, Quebec, Canada.
¹⁹ Department of Psychology, McGill University, Montreal, Quebec, Canada.
²⁰ Biomedical Ethics Unit, McGill University, Montreal, Quebec, Canada.

Abstract

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.

Keywords: Patient-reported symptoms; Symptom cluster; Systemic sclerosis.