Development of a tool to map SSc pain sources, characteristics and management experiences: the SPIN Pain Assessment Tool

Abstract

Objective

People with SSc report pain from multiple sources. Objectives were to develop a tool to map sources of pain in SSc, determine characteristics of pain from different sources and understand pain management experiences.

Methods

We referred to existing research and clinical pain assessment tools and partnered with a 4-member Patient Advisory Team to develop an initial tool version. The tool included questions on SSc pain sources and, for each source, questions on pain source intensity and interference, temporality and predictability, sensory experience and attempted pain management techniques. To further refine the tool, we conducted nominal group technique (NGT) sessions with people living with SSc and received feedback from healthcare providers who care for people with SSc. Finally, we conducted individual usability testing sessions with people living with SSc.

Results

We conducted six NGT sessions (five English, one French; 22 total participants). We received feedback from 18 healthcare providers, and conducted five individual usability testing sessions. The final version of our Scleroderma Patient-centered Intervention Network (SPIN) pain assessment tool included 18 items on pain sources (plus an ‘other’ category), and for each pain source, 12 questions on pain intensity and interference, temporality and predictability, sensory experience and management.

Conclusion

The SPIN pain assessment tool includes critical information on pain sources important to people with SSc. It provides a useful tool to study the epidemiology of the complex pain experience in SSc, including pain sources and how pain is experienced.

Rheumatology key messages.

• Pain in SSc can come from many different sources and manifests differently within and across individuals.

• We developed a detailed assessment tool to map sources of pain in SSc.

• This tool will help characterize pain from different sources in SSc and understand pain management experiences.

Introduction

SSc (scleroderma) is a rare, chronic, autoimmune disease characterized by thickening and fibrosis of the skin and involvement of internal organs [1, 2]. Common symptoms that impact the ability to carry out daily activities include hand function and mobility limitations, difficulty breathing, fatigue, gastrointestinal symptoms and pain [3–11].

A study of over 2000 participants with SSc from seven countries found that 38% reported moderate or severe pain, defined as ≥5 on a 0–10 scale. Mean pain intensity was 3.5 out of 10 [12], which is similar to levels in RA cohorts [12–14]. Pain management was identified as a top priority in a survey of 100 people with SSc [15], and pain is the most common reason people with SSc seek physical or occupational therapy [16]. Yet, pain has not been the primary outcome in any SSc clinical trials and is rarely included as an outcome at all [12]. Reviews of SSc pathology, disease manifestations and clinical management mention pain only briefly [17, 18].

SSc is a complex disorder, and multiple factors may contribute to pain, including skin and joint inflammation, RP, digital ulcers, calcinosis, gastrointestinal manifestations and overlapping conditions such as RA and SS [12, 19, 20]. Most existing pain measures [21–23] are global pain scales that generate overall pain intensity or interference scores. None provide a ‘roadmap’ of the pain experience from each of multiple sources or address features of pain that may be critical in SSc (e.g. intensity and interference, temporality and predictability, sensory experience) or assess experiences with pain management.

The objective was to develop an assessment tool to map sources of pain in SSc, determine characteristics of pain from different sources and understand pain management experiences.

Methods

Scleroderma Patient-centered Intervention Network (SPIN) investigators partnered with a Patient Advisory Team of people with SSc to develop a preliminary pain assessment tool. We sought input from people with SSc via nominal group technique (NGT) sessions [24] and from SPIN healthcare providers. We revised the tool based on this input and further review by Patient Advisory Team members. We then conducted individual usability sessions with additional people with SSc. We published a protocol prior to initiating the study [25]. This study was approved as an amendment of the SPIN Cohort Study (#MP-05–2013-150–12-123) by the Research Ethics Committee of the Centre intégré universitaire de santé et de services sociaux du Centre-Ouest-de-l’Île-de-Montréal. All participants in NGT sessions and usability testing provided informed consent electronically.

Development of the initial version of the scleroderma pain assessment tool

SPIN researchers and a 4-member SPIN Pain Patient Advisory Team of people with SSc together reviewed an initial list of possible pain sources developed based on previous studies of possible SSc pain sources [12, 19, 26]. Patient Advisory Team members offered suggestions on listed sources and additional pain sources they believed should be included, as well as pain characteristics to capture from each possible source (e.g. intensity and interference, temporality and predictability, sensory experience, management strategies) [27–29].

To create the initial tool version, we referred to the structure of an existing multi-dimensional tool developed for general chronic pain, the Mainz Pain Staging System [30–32]. Pain dimensions include frequency, duration, chronic or episodic nature, fluctuations in intensity, bodily distribution and healthcare. We adapted the tool for SSc based on Patient Advisory Team input.

NGT sessions

The NGT is a consensus technique used to develop or review assessment tool items directly with stakeholders [24], as we have done previously [33–35]. We aimed to conduct 4–6 NGT sessions of 60–90 min via the videoconferencing platform Zoom with 4–8 people with SSc per session, separately in English and French. The final number of sessions was determined based on data redundancy and consistency.

Eligibility and recruitment

Eligible participants had to have SSc, confirmed by a physician; be fluent in English or French; have a Patient-Reported Outcomes Measurement Information System (PROMIS)-29 Version 2.0 Pain Interference domain T-score ≥55 (mild to severe pain interference) and have a PROMIS-29 Pain Intensity domain score ≥1 (numerical rating scale 0–10) [21, 36, 37]. See Supplementary Data S1 for descriptions of the PROMIS-29 Pain Intensity and Pain Interference measures.

Participants were recruited via posts on SPIN’s Facebook page and invitation emails sent to SPIN Cohort participants. Cohort participants are recruited to the SPIN Cohort during medical visits at 54 SPIN recruitment sites in seven countries (Australia, Canada, France, Mexico, Spain, UK, USA) [38, 39]. They complete patient-reported outcomes every 3 months and provide written informed consent at enrolment to be contacted about future studies. Participants complete these patient-reported outcomes in their preferred language (English, French, Spanish).

Consent and pre-NGT survey

Social media and email recruitment material provided a link to a Qualtrics survey page where we obtained informed consent and assessed eligibility. The online consent form included a detailed description of the study and NGT process. Individuals with questions about the study were encouraged to contact the study coordinator by email or phone prior to providing consent.

Individuals who consented proceeded to a questionnaire that included PROMIS-29 Version 2.0 Pain Interference and Pain Intensity domain items to assess eligibility and questions about their experience with important sources of pain (e.g. ulcer pain, joint contractures, gastrointestinal pain). SPIN Cohort participants provided the email address linked with their SPIN Cohort account to allow us to access sociodemographic and medical information from the cohort database. Individuals who were not SPIN Cohort participants also provided this information via Qualtrics. Eligible respondents provided their availability for NGT sessions.

The study coordinator assigned participants to a session, if possible, based on availability and attempted to form groups that were as diverse as possible based on pain experience and sociodemographic and disease characteristics. Invitation emails were sent to confirm the date and time of each participant’s session and to confirm participation. If necessary, a follow-up email was sent to potential participants who did not respond. Participants received a pre-session survey via email once they confirmed attendance for a session. See Supplementary Data S2 and S3 for English- and French-language pre-NGT surveys.

In the first part of the pre-NGT survey, participants were presented with a list of pain sources included in the initial assessment tool version and asked to rate the importance of including each listed source in the final tool (0 = not at all important to 10 = extremely important). For each pain source, participants were instructed to indicate if they believed any revisions were warranted and to provide a brief explanation for each suggested revision. Participants were then asked to list any pain sources they believed were missing from the list. In the second part of the pre-NGT survey, participants were presented with a set of items about each pain source, including questions on pain intensity and interference, temporality and predictability, sensory experience and management. They were similarly asked to rate the importance of each item and provide recommendations for revisions.

Prior to each NGT session, the study coordinator emailed each participant with the feedback they provided so they could share it during their NGT session. Participants received a reminder email informing them to [1] log into Zoom 15 min early [2], have their feedback available and [3] have materials for brainstorming during the session (e.g. pen and paper).

NGT sessions

Each NGT session was led by two bilingual SPIN members with SSc knowledge and experience in discussion-based research. Each session began with an introduction by moderators and followed a round-robin format. Participants first took turns presenting their list of potential new pain sources, and the group discussed the importance of each. Next, participants were presented with a list of pain sources that were rated as having low importance or had modification suggestions from participants in the pre-NGT survey. Participants discussed their low ratings of sources and their perspective of the importance and relevance of each. Participants then shared their input and discussed items on pain intensity and interference, temporality and predictability, sensory experience and management.

Once each group developed a final list of new pain sources and previously low-rated sources, as well as new and previously low-rated items on pain intensity and interference, temporality and predictability, sensory experience and management, the study coordinator transferred the list into a survey template in Qualtrics. This was sent to participants via email following the session to allow them to evaluate the discussed items. In the post-NGT survey, participants rated each item from 0 = not at all important to 10 = extremely important. Across sessions, as possible, team researchers revised the pain assessment tool as new or previously low-rated items emerged, using the updated version as a template in subsequent NGT sessions. We could not provide an updated item list in two NGT sessions because they occurred too soon after the previous session to do this.

Following completion of all NGT sessions, we sent the post-NGT version of the tool via email to experienced healthcare providers from SPIN’s network for review. Then, we met with SPIN Pain Patient Advisory Team members in a group discussion format to review NGT session and healthcare provider input and make decisions about final tool revisions.

Individual usability testing

We invited individuals who completed the consent form for the NGT sessions but could not be scheduled for a session to participate in usability testing. Each participant met on Zoom with two researchers who introduced the participant to the tool and took field notes. Participants were instructed to review the tool and its items via a ‘Think Aloud’ process, a common strategy for patient tool usability testing [40–42]. Unlike the brainstorming process used in the NGT sessions, which was a structured group activity to generate new ideas, this was a process by which participants articulated their experience completing the tool as it was unfolding. Once participants completed their review, brief open-ended questions were asked (‘Would you change anything about the tool?’ and ‘Do you have any other comments or suggestions for us?’). After each interview, the researchers debriefed and compared field notes. Usability testing session data was analysed using conventional content analysis [43] and adapted constant comparative analyses [43, 44]. In this way, data analysis began after the first usability session to inform subsequent sessions. We determined the number of participants based on consistency and redundancy of input, as well as the absence of reporting on any major issues with the tool.

Data processing, revision and analysis

Sociodemographic characteristics and pain interference and intensity levels were presented descriptively for NGT and usability session participants.

Development of the pain assessment tool was done in increments with modifications incorporated following each NGT session. During each NGT session, new and modified items were reworded until a consensus was reached. Suggested changes were reviewed by study investigators for clarity and then integrated into the tool. Once we completed all NGT sessions, the updated version received three additional stages of review by (1) 18 healthcare providers who care for people with SSc (eight rheumatologists, seven internists, two physical medicine and rehabilitation physicians, one psychologist), (2) members of the SPIN Pain Patient Advisory Team and (3) five people with SSc via usability testing sessions. Healthcare providers reviewed the tool and provided input on items that might be confusing or generate difficult-to-interpret responses. They provided a rationale for any potentially problematic items and, if possible, a suggestion for improving the item. We then compiled a list of all comments received from healthcare providers and discussed the implementation of these suggestions during a Patient Advisory Team meeting. We included modifications agreed upon during this meeting and used this version of the tool for usability testing sessions.

Results

Six NGT sessions (five English, one French) were held between April and June 2024. The number of participants per session ranged from 3 to 5. Five individual usability testing sessions were conducted in English in July 2024.

Participant characteristics and pain scores

Of the 44 individuals with SSc who expressed interest, 22 were scheduled and participated in an NGT session. Participants were aged 23–72 years (median 59 years), 16 (73%) participants were female and 14 (of 21 who reported, 67%) were White. Mean (S.D.) education years was 17.2 (3.1). Mean time (S.D.) since diagnosis was 15.7 (10.5) years, and 43% had limited SSc (N = 9 of 21 who reported). Mean PROMIS-29 Pain Interference T-score was 61.1 (range 55.6–66.6), and mean PROMIS-29 Pain Intensity score was 5.3 (range 2.0–8.0 out of 10).

Five individuals who shared interest in participating but were not selected for an NGT session participated in an individual usability testing session. Participants were aged 46–72 years old (median 55 years), all five were female and three were White. Mean (S.D.) education years was 15.6 (2.5), mean time (S.D.) since diagnosis was 6.3 (6.7) years, and three had limited SSc. Participant characteristics are shown in Table 1.

Table 1.

Participant characteristics.

	Nominal group technique participants (N = 22)		Individual usability testing participants (N = 5)
	N	Mean (SD) or N (%)	N	Mean (SD) or N (%)
Age (years)	22	57.5 (11.3)	5	57.4 (9.8)
Female sex	22	16 (73%)	5	5 (100%)
Education (years)	21	17.2 (3.1)	5	15.6 (2.5)
Marital status	21		5
Married or living as married (vs single, divorced, separated, widowed)		14 (67%)		2 (40%)
Race or ethnicity	21		5
Aboriginal		1 (5%)		0 (0%)
Asian		4 (19%)		1 (20%)
Latin American		1 (5%)		1 (20%)
Mixed		1 (5%)		0 (0%)
White		14 (67%)		3 (60%)
Country	22		5
Canada		11 (50%)		1 (20%)
France		3 (14%)		0 (0%)
UK		2 (9%)		0 (0%)
USA		6 (27%)		4 (80%)
Occupational status	21		5
Full-time employed		2 (10%)		2 (40%)
Part-time employed		3 (14%)		1 (20%)
Full-time homemaker		2 (10%)		1 (20%)
Full-time student		1 (5%)		0 (0%)
On disability		2 (10%)		0 (0%)
Unemployed		1 (5%)		0 (0%)
Retired		7 (33%)		0 (0%)
Other		3 (14%)		1 (20%)
SSc subtype	21		5
Limited		9 (43%)		3 (60%)
Diffuse		12 (57%)		1 (20%)
Did not know		0 (0%)		1 (20%)
Years since SSc diagnosis	22	15.7 (10.5)	4	6.3 (6.7)
Pain Interference T-score	22	61.1 (4.0)	5	55.2 (9.6)
Pain intensity score (0–10 numerical rating scale)	22	5.3 (1.9)	5	6.0 (2.3)

NGT session, healthcare provider and patient partner feedback

Pain source items

The six NGT groups generated 3, 2, 3, 2, 2 and 2 new pain source items although there were duplicate new items across some groups. Following discussions among investigators, a total of eight new pain sources were added to the initial tool version. Some suggested items were excluded due to repetition or low SSc-specific relevance (e.g. back pain). Several items were modified throughout NGT sessions, including rewording and adding or removing examples for some pain sources and their associated body locations. A total of 24 items on pain sources were sent to healthcare providers for review. Following discussions with our Patient Advisory Team on healthcare provider feedback, a total of 18 items on specific pain sources (plus an ‘other’ category) were included in the final version of our pain assessment tool (see Fig. 1).

Figure 1.

Flow diagram from initial items to final items on pain sources

Items on pain intensity and interference, temporality and predictability, sensory experience and management

The six NGT groups generated 2, 1, 1, 0, 0 and 1 new items. Following the NGT sessions, a total of three new items were added to the initial tool version. Modifications made to items in the sessions mainly consisted of rewording, providing more detail and including more response options. One item was removed. A total of 13 items were sent to healthcare providers for review. Following discussions with our SPIN Pain Patient Advisory Team on healthcare provider feedback, a total of 12 items to assess characteristics of each source of pain respondents endorsed were included in the final pain assessment tool (see Fig. 2).

Figure 2.

Flow diagram from initial items to final items on pain intensity and interference, temporality and predictability, sensory experience and management

Individual usability testing feedback

The five individual usability testing sessions with the online Qualtrics version of the assessment tool lasted ∼1 h each. Participants completed as many of the items as possible, and were asked to share their input and ask clarification questions, if they had any. Participants helped us identify errors with the tool programming (e.g. item repetition) and wording, but no clarification of items was requested. Therefore, no items were removed, added or modified following usability testing sessions.

The final SPIN pain assessment tool is shown in Table 2. Participants are first asked whether they have ever experienced SSc-related pain from each of the 18 specific sources (31 including sub-categories), including ulcers on the skin; mouth ulcers or ulcers in the nose; RP; gangrene in fingers or toes; surgical or auto-amputation; phantom pain; tight, thick, hard or irritated skin; calcinosis; gastrointestinal symptoms (swallowing, choking or sensation of choking, nausea, regurgitation, acid reflux or heartburn, bloating, constipation, diarrhoea, other); oro-facial symptoms (tightness around the eyes, reduced mouth opening, thinned lips with rigidity, tooth sensitivity, dryness from reduced saliva production, tissue loss around teeth or inflammation of gums, trigeminal neuralgia, other); dry eyes; chest; joints; muscles; tendons; points on the body that are hypersensitive to touch; carpal tunnel syndrome; plus other sources. Then, for each source where pain is reported, participants are asked to answer 12 follow-up questions on the intensity and interference, temporality and predictability, sensory experience and management strategies of that pain source.

Table 2.

Elements included in the SPIN pain assessment tool.

Sources of pain (N = 19 items; 31 items including subcategories) Response options in italics for each item
1. Ulcers on the skin (sores or breakthrough on skin) Yes, No Please specify in which part(s) of the body pain from ulcers on the skin is felt. Select all that apply. Fingers, toes, other (specify location)
2. Mouth ulcers or ulcers in the nose Yes, No Please specify in which part(s) of the body pain from mouth or nose ulcers is felt. Select all that apply. Mouth, nose
3. RP (decreased blood flow and colour change in certain part(s) of the body) Yes, No Please specify in which part(s) of the body pain from RP is felt. Select all that apply. Fingers, toes, ears, nose, other (specify location)
4. Gangrene in fingers or toes (lack of blood flow causing death of body tissue which turns black) Yes, No
5. Process of auto-amputation and related complications Yes, No Please specify in which part(s) of the body pain from the process of auto-amputation and related complications is felt. Select all that apply. Fingers, toes, other (specify location)
6. Surgical amputation and related complications Yes, No Please specify in which part(s) of the body pain from surgical amputation and related complications is felt. Select all that apply. Fingers, toes, other (specify location)
7. Phantom pain (ongoing painful sensations that seem to be coming from a part of the body that is no longer there) Yes, No Please specify in which part(s) of the body phantom pain is felt. Select all that apply. fingers, toes, other (specify location)
8. Tight, thick, hard skin, or irritated skin yes, no Please specify in which part(s) of the body pain from tight, thick, hard skin or irritated skin is felt. Select all that apply. Face, hands, arms, legs, fingers or toes, other (specify location)
9. Calcinosis Yes, No Please specify in which part(s) of the body pain from calcinosis is felt. Select all that apply. Neck, upper back, shoulders, elbows, arms, hands, hips, knees, feet, other (specify location)
10. Gastrointestinal symptoms. Please specify which, if any, cause pain. Select all that apply. Swallowing, choking or sensation of choking, nausea, regurgitation, acid reflux or heartburn, bloating, constipation, diarrhoea, other (specify), I have never experienced painful gastrointestinal symptoms.
11. Oro-facial symptoms. Please specify which, if any, cause pain. Select all that apply. Tightness around the eyes, reduced mouth opening, thinned lips with rigidity, tooth sensitivity, dryness from reduced saliva production, tissue loss around the teeth or inflammation of the gums, trigeminal neuralgia, other (specify), I have never experienced painful oro-facial symptoms
12. Dry eyes Yes, No
13. Chest Yes, No
14. Joints Yes, No Please specify in which part(s) of the body joint pain is felt. Select all that apply. Fingers, wrists, toes, shoulders, elbows, hips, knees, ankles, other (specify)
15. Muscles Yes, No Please specify in which part(s) of the body muscle pain is felt. Select all that apply. Hands, arms, legs, other (specify)
16. Tendons Yes, No Please specify in which part(s) of the body tendon pain is felt. Select all that apply. Wrists, knees, ankles, other (specify location)
17. Points on the body that are hypersensitive to touch Yes, No Please specify in which part(s) of the body pain from points that are hypersensitive to touch is felt. Select all that apply. Hands, arms, legs, feet, other (specify location)
18. Carpal tunnel syndrome (neurological disorder involving the nerve which runs from your forearm into the palm of the hand. Symptoms include numbness, swollen fingers, tingling, loss of movement) Yes, No
19. Do you experience any other source(s) of pain that has/have not been mentioned in the list above? If so, please describe this pain and specify in which part(s) of the body the pain is felt. Yes, No

Items on pain intensity and interference, temporality and predictability, sensory experience and management (N = 12) Response options in italics for each item
1. Since your first experience with [pain source], recurrences have been:    infrequent and predictable, infrequent and unpredictable, frequent and predictable, frequent and unpredictable, I have only experienced one occurrence of pain from ulcers on the skin with no recurrence
2. In the month when you most recently experience [pain source] pain, how often was the pain present? Every day, most days, some days but not others, only a few days, one day only
3. When you experience [pain source], how would you describe the pattern of pain? Continuous, steady or constant; rhythmic, periodic or intermittent; brief, sharp or transient
4. On days when you experience [pain source], what part(s) of the day is the pain present? Morning, afternoon, evening, overnight, not related to the time of day
5. In the month when you most recently experience [pain source] pain, how would you rate the intensity of the pain at its worse? 0 (no pain) to 10 (worst imaginable pain)
6. In the month when you most recently experienced [pain source] pain, how would you rate the average intensity of the pain? 0 (no pain) to 10 (worst imaginable pain)
7. In the month when you most recently experienced [pain source] pain, how similar was the pain intensity across different episodes? It was very similar or the same, it differed a little bit across episodes, it differed a lot across episodes
8. During an average or typical day when you experience [pain source] pain, how much does it interfere with your ability to carry out your normal day to day activities (e.g. bathing and grooming, preparing meals, household chores)? Not at all, very little, somewhat, to a great extent, completely
9. To what extent do your pain intensity levels and patterns related to [pain source] depend on outdoor temperatures/seasons? Not at all, very little, somewhat, to a great extent, completely, I’m not sure
10. Have you ever received and used medical care from your doctor or another health professional for [pain source] pain? How effective was the treatment? Non-opioid analgesics, weak or strong opioids, prescribed marijuana, rehabilitation, physiotherapy/physical therapy, occupational therapy, exercise, surgery, psychological or behavioural therapy, other, I have not received medical care from my doctor or another health professional for this source of pain 0 (not at all effective in reducing my pain) to 10 (completely effective in reducing my pain)
11. Have you ever used any of the alternative approaches listed below to reduce [pain source] pain? How effective was the treatment? Vitamins, supplements, minerals, or herbs; unprescribed marijuana; CBD; natural intravenous or injection therapy; medicated patches; heat pad; mind-body exercises; acupuncture; massage; TENS; specific diet/dietary change; exercise; other; I have not used any alternative approach for this source of pain 0 (not at all effective in reducing my pain) to 10 (completely effective in reducing my pain)
12. Are there currently other factors that may be influencing your pain level now (e.g. recent surgery, winter in a northern climate)? If so, please specify.

Respondents complete each item. For each source that respondents have experienced, they complete the 12 items on pain intensity and interference, temporality and predictability, sensory experience and management.

Discussion

We developed a novel tool that will facilitate assessment and better understanding of pain sources in SSc. The tool was co-developed with the SPIN Pain Patient Advisory Team, refined through six NGT sessions with 22 individuals with SSc, and informed by 18 healthcare providers with experience in SSc. The tool includes 18 specific pain source items (plus an ‘other’ category) (31 with sub-categories) and, for each pain source, 12 follow-up items on intensity and interference, temporality and predictability, sensory experience and management.

To our knowledge, this is the first assessment tool designed to comprehensively assess the diverse pain-related experiences among people with SSc. The SPIN pain assessment tool differs from existing pain assessment tools [21–23], which provide overall scores for intensity or pain interference but do not identify the sources of pain or other aspects that are critical for understanding the pain experience. Unravelling why pain occurs, what are the most important sources of pain and characteristics of each pain source is crucial if we are to better understand and develop effective approaches to address pain in SSc.

Prior to the start of our study and throughout our work with people with SSc during the study, Patient Advisory Team members and study participants shared that their pain was not addressed in their SSc healthcare or noted that, rarely, pain medication was offered. Consistent with this, SSc management guidelines and articles on treating SSc either do not mention pain or mention it only briefly [45–49]; none provide guidance on addressing pain from multiple sources or include a dedicated section on pain management.

The SPIN pain assessment tool will be administered in the SPIN Cohort, which includes over 1300 active participants from 48 centres in seven countries who complete patient-reported outcomes in English or French every 3 months via the internet and participate in additional sub-studies, including our planned sub-study on pain sources and characteristics. The tool can also be administered in other research settings. This will allow SPIN and other researchers to describe the prevalence of pain sources from SSc, as well as, for each source, pain intensity and interference, temporality and predictability, sensory experience and management strategies that are attempted. This information will inform future research on the most important pain sources in SSc and the development and dissemination of effective management strategies.

The SPIN pain assessment tool has several strengths. First, the tool assesses pain intensity and interference, temporality and predictability, sensory experience and management for each pain source, and data from the tool will provide a comprehensive view of the pain experience in SSc. Second, the tool and its items were developed together by researchers and people living with SSc with additional input from other patients and physicians and other health professionals experienced in SSc management. Third, the tool collects information on strategies that people with SSc have attempted to manage pain, as well as their perceptions of the effectiveness of these strategies. There are also limitations. While many items were patient-generated, not all pain sources that were recommended by one or more patients could be included. As a result, some pain sources may not be explicitly captured, although we believe that these are likely not very common in SSc or not specific to SSc. The tool includes an ‘other’ category to capture additional potentially helpful information. Second, the tool is lengthy, particularly for people with multiple pain sources, which could lead to respondent fatigue and difficulty completing it in a single sitting; some people may need to complete it in multiple sessions.

Study strengths and limitations

Our study to develop the SPIN pain assessment tool has important strengths. First, SPIN Pain Patient Advisory Team members, who are people with SSc who live with pain, were engaged at each stage of the research, beginning with developing a funding proposal, conceptualizing the project and providing input on how data were collected, making decisions on items to include in the tool, and interpretation of results. They are co-authors of this report and will be involved in dissemination. Second, NGT sessions in English and French allowed us to obtain input from other people living with SSc and pain, including identifying additional pain sources and questions regarding those pain sources that we would have not otherwise identified. This improved the tool’s content and format. Third, 18 expert healthcare providers provided feedback on the tool. There are also limitations to consider. First, most participants were recruited from the SPIN Cohort, which is a convenience sample of people who receive care at specialty SSc treatment sites. In addition, we used online recruitment and had a limited representation of participants from low- and middle-income countries. Second, among countries with SPIN Cohort participants, Australian participants who expressed interest could not be included due to scheduling restrictions, and language was a barrier to recruiting participants from Spain and Mexico. Only one group of French-speaking participants was included. Third, only 22 individuals with SSc participated in the NGT sessions. These factors could affect the generalizability of our item pool; however, we believe that our comprehensive patient engagement plan and consulting via NGT sessions and with healthcare providers minimized this possibility. Future studies should evaluate the tool in other settings.

Conclusion

SPIN researchers and patients worked together to develop the SPIN pain assessment tool. We used NGT sessions with people with SSc to solicit feedback on the tool and its items and consulted with healthcare providers who care for people with SSc. The tool includes 18 sources of possible pain in SSc (31 with sub-categories). For each source that is present, 12 items assess intensity and interference, temporality and predictability, sensory experience and management experiences. The tool will be administered to people with SSc to generate a ‘roadmap’ of what pain looks and feels like for them. This information will support research on pain in SSc and lead to better individualized treatment for pain in SSc.

Supplementary material

Supplementary material is available at Rheumatology online.

Data availability

De-identified data 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 will be available beginning 12 months after publication. Data requesters will need to sign a data transfer agreement.

Author contributions

T.D.S., M.E.C., E.N., L.K., S.J.B., R.S.F., Y.C.L., J.V., B.D.T., and members of the Scleroderma Patient-centered Intervention Network Pain Patient Advisory Team contributed to study conceptualization; T.D.S., E.N., and B.D.T. to methodology; T.D.S., M.E.C., C.D.S., and S.H. to data collection; T.D.S., B.D.T., and members of the Patient Advisory Team to data analysis; T.D.S. and B.D.T. to writing the original draft; all authors to reviewing and editing the draft; B.D.T. to supervision; T.D.S. and M.E.C. to project administration and E.N., M.E.C., B.D.T. to funding acquisition.

Funding

This study was funded by an Arthritis Society Canada Ignite Innovation Grant (22–0000000130). 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, Montréal, Québec, Canada; the Jewish General Hospital Foundation, Montréal, Québec, Canada and McGill University, Montréal, Québec, 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 and Scleroderma Victoria and the Scleroderma Foundation of California. T.D.S. was supported by a Fonds de Recherche du Québec—Santé (FRQS) Master’s Training Scholarship, E.N. by an FRQS Doctoral Training Scholarship Award, Y.C.L. by a National Institutes of Health Grant (K24AR080840) and B.D.T. by a Canada Research Chair, all outside of the present work. No sponsor had any role in the study design; in writing the protocol or in the decision to submit it for publication.

Disclosure statement: All authors declare no conflicts of interest.