The Journal of Rheumatology Scleroderma Patient-centered Intervention Network Cohort Study Physical or Occupational Therapy Utilization in Systemic Sclerosis: A

Background: Systemic sclerosis (SSc) is characterized by significant disability due to musculoskeletal involvement. Physical and occupational therapy (PT/OT) have been suggested to improve function. However, the rate of PT/OT utilization has been shown to be low in SSc. We aimed to identify demographic, medical, and psychological variables associated with PT/OT use in SSc. Methods: Participants were patients with SSc enrolled in the Scleroderma Patient-centered Intervention Network Cohort. We determined the rate and indication of PT/OT use in the 3 months prior to enrollment. Multivariable logistic regression was used to identify variables independently associated with PT/OT utilization. Results: Of the 1,627 SSc patients included in the analysis, 23% used PT/OT in the preceding 3 months. PT/OT use was independently associated with higher education (odds ratio [OR] 1.08, 95% confidence interval [CI] 1.04-1.12), having moderately severe small joint contractures (OR 2.09, 95% CI 1.45 – 3.03), severe large joint contractures (OR 2.33, 95% CI 1.14 – 4.74), fewer digital ulcerations (OR 0.70, 95% CI 0.51 – 0.95), and higher disability (OR 1.54, 95% CI 1.18 – 2.02) and pain (OR 1.04, 95% CI 1.02 – 1.06) scores. The highest rate of PT/OT utilization was reported in France (43%) and lowest in the USA (17%). Conclusions : Despite the potential of PT/OT interventions to improve function, less than 1 in 4 SSc patients enrolled in a large international cohort used PT/OT services in the last 3 months. Patients who utilized PT/OT had more severe musculoskeletal manifestations and higher pain and disability.


Introduction
Systemic sclerosis (SSc, or scleroderma) is a chronic systemic disease characterized by dysregulated fibrosis, autoimmunity, inflammation and vasculopathy (1). Musculoskeletal involvement is nearly universal in SSc. Skin fibrosis, joint and muscle pain, arthritis, hand deformities, joint contractures and reduced range of motion are common manifestations and result in significant disability (2). Hand pain and joint stiffness are amongst the five highest rated symptoms and are described by more than 80% of patients with SSc (3). Skin fibrosis of the face and oral tissues causes difficulties with eating, speaking, dental care and oral hygiene (4).
Musculoskeletal rehabilitation and physical and occupational therapy (PT/OT) are recommended for the management of musculoskeletal impairment in SSc (5). Multiple rehabilitation techniques including range of motion exercises, connective tissue massages, joint manipulation, splinting, heat/paraffin wax baths, and generalized PT have been suggested to improve pain and joint motion in small randomized control trials, case reports and case series (5). Similarly, hand and orofacial exercises have been suggested to improve gingival health (6,7).
A trial of 220 patients with SSc found that a 4-week PT program significantly reduced disability one-month post-randomization, although there was not an effect on disability at 12-month follow up (8).
Variable rates of PT/OT utilization have been reported in the literature with most studies showing less than 50% of patients with SSc using PT/OT despite it being one of the primary available interventions to address musculoskeletal manifestations (9)(10)(11).
In this study, we aimed to determine the rates and indications of PT/OT utilization among patients with SSc enrolled in one of the largest SSc cohorts worldwide, the Scleroderma Patientcentered Intervention Network (SPIN) Cohort. We also aimed to identify demographic, medical and psychological variables associated with the use of these services.

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Patients and Procedures:
The present study included a convenience sample of patients enrolled in the SPIN Cohort.
As previously described (12), this large international cohort is composed of patients recruited from more than 40 centers in Canada, the USA, and Europe. To be eligible for enrollment,

Measures:
PT/OT Utilization: Patients were asked to answer three questions on PT/OT utilization: "In the last 3 months, have you seen a physical therapist, physiotherapist, occupational therapist, ergotherapist, kinesiotherapist or other health care professional for rehabilitation services?"; "If yes, was this for?" (patients could pick one or more options from hands, feet, mouth/face, wound care, activities of daily living, other); and "If yes, how many times in the last 3 months did you receive rehabilitation services?".

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Rheumatology
The Journal of on July 9, 2020 -Published by www.jrheum.org Downloaded from involving the limbs proximal to the elbows and knees and/or the chest and/or trunk at any time, whereas limited SSc was defined as skin sclerosis confined to the limbs distal to the elbows.
MRSS is a clinical measure of skin thickness from 0 to 51, with higher scores indicating more severe thickness. The presence of overlap syndromes with rheumatoid arthritis (RA), systemic lupus erythematosus (SLE), and/or idiopathic inflammatory myositis was also determined.

Disability Measures:
Patients completed the Scleroderma Health Assessment Questionnaire (SHAQ) and Cochin Hand Function Scale (CHFS-II). The SHAQ assesses physical disability with a composite score from 0 to 3, with higher scores indicating greater disability (14,15). The CHFS-II assesses hand disability with a score from 0 to 90, with higher scores indicating higher disability (15,16). The SHAQ and CHFS-II have been validated in patients with SSc (14-16).
Psychological Measures:

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The Journal of on July 9, 2020 -Published by www.jrheum.org Downloaded from To assess symptoms of depression, patients completed the Patient Health Questionnaire (PHQ-8). Scores range from 0 to 24, with higher scores indicating more depressive symptoms (17). Patients also completed the Patient Reported Outcomes Measurement Information System (PROMIS-29) measure version 2, which assessed 7 patient-reported outcome domains including the anxiety, fatigue, and pain intensity domains used in this study. Scores are standardized with a mean of 50 and a standard deviation of 10 with the mean score representing the average of a general US population and higher scores reflecting more of the measured domain (18). The Satisfaction with Appearance (SWAP) scale was used to measure body image distress. Scores can range from 0 to 84 with higher scores indicating greater dissatisfaction with appearance (19).
The Social Interaction Anxiety Scale-6 (SIAS-6) was used to assess distress due to social interactions by rating patients' experience in social situations from 0 to 24 with higher scores indicating higher social anxiety symptoms (20). Patients also completed the Self-Efficacy for Managing Chronic Disease (SEMCD) scale to assess their confidence in self-managing diseasespecific symptoms. Scores ranged from 1 to 10, with higher scores indicating higher selfefficacy. These scores have been validated in SSc (18,19,(21)(22)(23)(24)(25).

Statistical Analysis:
Descriptive analyses included means, standard deviations (SD), minimum and maximum for continuous variables and frequencies and percentages for discrete variables. Prior to initiation of inferential analyses, data completeness and normality for continuous measures was evaluated.
Demographic, psychological, disability and medical variables were compared between patients who participated in PT/OT in the prior 3 months and those who didn't using a chi square test and independent samples t-test. Multivariable binary logistic regression was subsequently used to identify variables independently associated with PT/OT utilization using an a priori defined model that included age, gender, education level, employment status, disease subset, disease

Factors associated with PT/OT utilization:
In bivariate analyses, no differences were observed in age, sex, race, education, or housing location among patients who used PT/OT in the 3 months prior to enrollment ( Table 1).
Compared to employed patients, we observed a higher rate of unemployed (28% vs 20%, p = 0.01) and disabled (36% vs 20%, p < 0.01) patients in the PT/OT utilization group. While all other sites reported a higher percentage of PT/OT utilization compared to the USA, only Canada (23% vs 17%, p = 0.02) and France (43% vs 17%, p < 0.01) had statistically significant higher utilization of PT/OT services.

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The a priori defined and final multivariable regression models are described in Table 3.
Using a backward stepwise technique, higher education, more small joint contractures, fewer DUs, higher disability, and more pain were significantly associated with PT/OT utilization after adjusting for age and sex. There was an 8% increase in the likelihood of PT/OT utilization for every additional year of education (OR 1.08, 95% CI 1.04 -1.12). The likelihood of PT/OT utilization was significantly higher in the presence of moderately severe small joint contractures (OR 2.09, 95% CI 1.45 -3.03) and severe large joint contractures (OR 2.33, 95% CI 1.14 -4.74). Higher disability (OR 1.54, 95% CI 1.18 -2.02) and pain (OR 1.04, 95% CI 1.02 -1.06) scores were associated with more PT/OT use. On the other hand, the presence of DUs was found to decrease the odds of PT/OT utilization (OR 0.70, 95% CI 0.51 -0.95). The association between PT/OT use and country of enrollment was analyzed after a priori variables were defined and included in the final model for analysis. Compared to patients from the USA, patients from Canada were 62% more likely to use PT/OT services (OR 1.62, 95% CI 1.12 -2.32) while those from France were 4 times more likely to use PT/OT (OR 4.38, 95% CI 2.99 -6.42).

Discussion
In the present cross-sectional study, we found that 23% of SSc patients in a large international cohort used PT/OT services in the 3 months prior to enrollment and that hand PT/OT was the most common rehabilitation service used by these patients. Our findings also demonstrated that PT/OT utilization was significantly and independently associated with higher

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Rheumatology
The Journal of on July 9, 2020 -Published by www.jrheum.org Downloaded from education, more severe musculoskeletal involvement, and higher disability and pain scores. We showed that geographical differences existed in the rate of PT/OT use. an OT since SSc diagnosis (9). A higher rate of PT/OT utilization was shown in Western Europe.
Among 198 Dutch patients with SSc surveyed from June and August of 2011, 75% reported contact with a PT and 36% with an OT since SSc onset. Of these patients, 53% and 13% reported contact with a PT and an OT, respectively, in the 12 months prior to study survey (11). Betweenstudy differences in sampling and methodology can explain some of the variability in the rates of PT/OT utilization. For example, in our study, the evaluation was restricted to the 3 months prior to enrollment while other studies looked at use over longer periods of time. However, this variability is in accordance with our study findings of different rates of PT/OT use in the different countries evaluated, which reflects regional variations in SSc management, access to rehabilitation services, and health care costs.
Rehabilitation services use has been more extensively studied in RA, a more common rheumatic disease with a predominance of musculoskeletal manifestations. In RA, the rates of PT and OT use among 8,001 German RA patients were estimated at 44% and 15%, respectively, from 1993 to 1998 (26). Similar utilization rates were observed among 1,200 patients with RA from Amsterdam in 1997 (40% PT and 17% OT use in the preceding year) (27). In the US, a 2011 study of 772 patients with RA showed that 15% of patients used PT in the preceding 6 months (28). The variability can be explained by differences in the management and prognosis of

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The Journal of on July 9, 2020 -Published by www.jrheum.org Downloaded from RA between the pre-and post-biologics eras but might also reflect country differences in PT/OT utilization as seen in our study. Similar to our findings, disease activity, disability, and higher education were also predictive of PT use in RA (28). In both RA and SSc, patients with these characteristics are in more need for (and thus more likely to be referred to) PT/OT and are possibly more likely to ask for/participate in these services due to their higher education and/or better access.
In the SPIN Cohort, hand PT/OT was the most commonly used rehabilitation service, reflecting the importance of hand symptoms in SSc. Hand pain and stiffness were previously shown to be frequently reported by SSc patients, with more than 50% associating them with moderate to severe impact on quality of life (3). In our study, patients who received PT/OT had more severe small joint contractures, tendon friction rubs, and higher hand dysfunction. Of interest, the presence of DUs was independently associated with less PT/OT use. Wound care is recommended for the management of DUs (29) and is offered by different specialists including specially trained physical therapists. However, wound care was an indication for PT/OT for about 6% of the SPIN cohort only, limiting conclusions on its association with DUs. Based on limited studies, exercise had no direct effect on healing ulcers (30). In contrary, since avoiding trauma is one of the recommended non-pharmacological interventions in the management of DUs (29), it is conceivable that concerns regarding exacerbation of ulcers due to the trauma of PT/OT activities result in hesitancy to refer to or participate in therapy when DUs are active.
No association was seen between PT/OT use and age, sex, race/ethnicity and overlap syndromes with other rheumatic diseases with significant musculoskeletal involvement.
Although patients who used PT/OT were slightly more likely to have interstitial lung disease than not, no independent association was found between PT/OT use and SSc-associated cardiopulmonary disease.
Patients who used PT/OT were found to have higher psychological distress and lower

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The Journal of on July 9, 2020 -Published by www.jrheum.org Downloaded from self-efficacy than those who did not. Since all measures were obtained at the same time point, conclusions on a cause-effect relationship between distress, self-efficacy, and PT/OT use are not possible. The higher distress and lower self-efficacy may reflect a more severe disease and higher disease impact in those who were more likely to be referred to, and thus more likely to use, these services. In SSc, lower self-efficacy has been found to correlate with greater physical limitation, increased pain and fatigue, and more depression (31). Moreover, better self-efficacy has been associated with better health outcomes, improved adherence to home exercise programs, and reduced health services utilization and cost in patients with chronic diseases including arthritis (32)(33)(34). Most of these studies focused on hospitalizations and emergency room and physician visits.
The large sample size, number of variables, and a priori selected variables are important strengths. We included demographic variables, disease-specific variables including a detailed assessment of musculoskeletal manifestations, disability measures, and a number of psychological variables that were thought and/or previously shown to affect health services utilization. SSc diagnosis and medical variables were ascertained by physicians and all variables were provided at one time-point allowing for a real-time assessment of their association with PT/OT use.
There are important limitations to consider when interpreting the findings of this study.
The SPIN Cohort is a convenience sample of patients recruited from specialized SSc centers, which might limit the generalizability of our findings. These patients have access to tertiary healthcare systems and are likely different from those seen in other settings. The rate of PT/OT use might, therefore, be lower in patients receiving care in generalized rheumatology practices.
As previously stated, we were only able to look at PT/OT utilization in the 3 months prior to enrollment and the evaluation of PT/OT referrals was limited. A previous study showed differences between the rates of PT/OT referrals and utilization indicating possible differences in

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Rheumatology
The Journal of on July 9, 2020 -Published by www.jrheum.org Downloaded from the perceived need for PT/OT between physicians and patients among other factors worth exploring. In SPIN, questions on PT/OT utilization were restricted to supervised rehabilitation services only and might not have captured home-based physical therapy and exercising which have been reported by more than half of SSc patients in the SPIN cohort in a study by Azar et al (35). In addition, it is important to recognize that the lack of data on income, medical insurance, and access to rehabilitation services are important limitations of this study given the critical impact these variables have been shown to have on PT/OT and other health services use. In adult patients with low back pain in the USA, out-of-pocket expenditure and type of insurance were shown to be predictive of PT visits, with privately and Medicare insured people being more likely to have more visits compared to those publically insured and uninsured (36). Similarly, SSc patients with private insurance were almost twice as likely to be referred to PT/OT in the study by Bassel et al (10). Because of the complexity and variability of insurance coverage within each healthcare system, looking at the rate of PT/OT use across the different countries does not fully overcome this limitation.
In conclusion, we showed that fewer than 25% of patients with SSc in a large international cohort used PT/OT services in the 3 months prior to enrollment and that geographical differences exist in the rate of utilization. Future research evaluating the impact of rehabilitation services on functional outcomes in SSc is needed to fully understand the importance of these interventions in SSc and strengthen the evidence based on which management guidelines are developed. Furthermore, in addition to identifying barriers to PT/OT in different healthcare settings, interventions are needed to improve the utilization of these services among patients with SSc. Such interventions include developing effective SSc-specific online and home-based physical therapy and exercise programs, which is one of the primary aims of SPIN. These programs could help overcome some of the barriers to PT/OT use, including cost and access.

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