Significance and Innovations

This study highlights a need for information, communication and decision support about pain management options.

There is a need for assessing pain in an accurate manner and for sharing evidence-based information for pain management in JIA, especially for non-pharmacological treatment options.

There is a need for clarifying and discussing young people’s and families’ values and preferences about pain management in JIA and for a joint decision with HCPs.

Families’ values and preferences were similar to those of HCPs and included the importance of effectiveness, safety and ease of use of treatments.

Decision support interventions may enable HCPs to work with youth and families to address decision-making needs for pain management among young people with JIA.

Background

Musculoskeletalpain is an important symptom of juvenile idiopathic arthritis (JIA) [1,2,3], with most young people experiencing some pain [1], and about 17% developing chronic pain [2]. Pain is associated with difficulties in physical, emotional, social and school functioning, thus affecting health-related quality of life and activity participation [4,5,6,7,8,9,10]. To effectively manage pain in JIA, a multi-disciplinary approach, including pharmacological, physical and psychological interventions, is required [11]. Unfortunately, pain in JIA is often under-recognized and communication about it is not optimal [12, 13].

An optimal way to make decisions, especially when there is no perfect treatment option and the choice depends on what families value most, is to engage in shared decision making (SDM). SDM is a process by which patients and health care providers (HCPs) make a joint decision by considering the best available evidence for treatment options as well as the patient’s and family’s values and preferences [14]. SDM is recommended as part of JIA treatment-to-target recommendations [15]. When supported by decision support interventions, SDM can lead to improved knowledge of treatment options, decisions which are consistent with patients’ values, and increased patient participation in decision-making [16].

Studies in JIA have shown that decision-making is not optimal [17, 18], but most studies focused on pharmacological disease management and not pain management, which often involves both pharmacological and non-pharmacological options [17]. Some studies revealed a lack of information-sharing with families on pain management options [17, 18], but other aspects of SDM have not been assessed thoroughly [17]. No study has assessed decision-making needs for pain management in the context of JIA using a SDM conceptual framework. We sought to explore families’ decision-making needs with respect to pain management among young people with JIA, parents/caregivers and HCPs.

Methods

Reporting is based on the Standards for Reporting Qualitative Research (SRQR) reporting guidelines [19].

Study design

We performed a decisional needs assessment based on the Ottawa Decision Support Framework (ODSF) [20] and on the workbook “Decisional Needs Assessment in Populations” [21]. Following a qualitative descriptive study design [22,23,24], we conducted semi-structured individual interviews with young people with JIA, their parents/caregivers and HCPs. Upon consent/assent, we conducted one interview per participant either face-to-face or online between October 2017 and August 2018. Interviewers audio-recorded interviews and took notes. The Children’s Hospital of Eastern Ontario (CHEO) Research Ethics Board approved this study (REB#16/100X) and participants signed consent/assent forms.

The research team comprised of women and men who are patient partners (LP, ASirois, ESirotich, NT, NA), clinicians from six professions (medicine, nursing, occupational therapy, physical therapy, psychology, social work), researchers, research coordinators and trainees, with varying level of familiarity with JIA pain management. Interviewers were experienced and trained in qualitative research. Interviewers explained the research goals to participants, their role and that there were no wrong answers. Interviewers had met some of the participants beforehand in other research projects.

Sample

Young people and parents/caregivers

We recruited a purposive sample of young people aged 8 to 18 years old with JIA and parents/caregivers at the CHEO rheumatology clinic, as well as through the newsletter and social media of the Pediatric Rheumatology Care and Outcomes Improvement Network (PR-COIN), a learning health network of parents and clinicians in the United States and Canada (see selection criteria in Table 1). Purposive sampling was chosen so that participants’ characteristics varied in age, gender, disease severity and experience with pain management options. Eligible young people and parents/caregivers participated in face-to-face or online interviews with an interviewer (KTA, scientist, TEH or MG, research coordinators) and completed socio-demographic and medical information forms.

Table 1 Inclusion and exclusion criteria
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HCPs

We invited a purposive sample of pediatric rheumatology HCPs who were practicing at CHEO and/or were part of PR-COIN via e-mail and newsletter to ask them to participate in an interview in person or online with an interviewer (KTA). We included HCPs from various professions and different experiences with chronic pain management (Table 1).

Interview guides and questionnaires

Interview guides (Tables 2 and 3) were based on the ODSF[20] and included questions about the pain experience, and questions modified from the Personal Interview Questions for Client Key Informants [21]. We pilot-tested interview guides with two patients with JIA and a rheumatology colleague. Socio-demographic and medical information forms asked about children’s age and gender, family income, parents’ level of education and cultural background, as well as disease-related information. Another form asked about HCPs experience, location and type of practice.

Table 2 Interview guide for parents/caregivers (same interview guide with simpler language for young people)
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Table 3 Interview guide for HCPs
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Data analyses

Audiotapes from the interviews and notes were transcribed verbatim. We analyzed the data using thematic analysis [25] with the help of the NVivo 11 software. Research trainees (DC, HS, ASivakumar, TEH, MR) coded and analyzed transcripts in pairs, and codes were discussed with KTA. We developed initial codes based on elements of the ODSF, and SDM process and outcomes, as per the Outcome Measures in Rheumatology (OMERACT) SDM Working Group work [26, 27] (Fig. 1). We added new emerging codes. We regrouped codes into overarching themes. We held team meetings and discussed findings to ensure these truly reflected participants’ experience rather than our own assumptions. We used an audit trail. We conducted interviews until data saturation was reached for each participant group, meaning that no new themes emerged as we conducted additional interviews, and we compared findings between groups. We analysed quantitative data using descriptive statistics in Statistical Package for the Social Sciences (SPSS;version 28).

Fig. 1
figure 1

SDM process and outcomes to guide the analyses Fig. 1 shows the elements of the SDM process and outcomes used to guide the analyses

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Results

Participant characteristics

We approached 27 families. An additional 9 parents contacted the research team via social media. A total of 12 young people (n = 6 children and n = 6 adolescents) and 13 parents/caregivers (6 related to youths and 7 related to adolescents) participated in interviews. There were 12 young people/parent dyads and one additional parent/caregiver (Table 4).

Table 4 Young people and families’ disease-related and socio-demographic information
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Some families declined participation (n = 3) or initially accepted but later declined (n = 20) because of their schedule. Out of 13 young people, 11 were girls and eight had inactive disease. Young people and parents reported a median pain value over the past week of 30 mm and 25 mm out of 100 mm, respectively. Participants reported currently using both medication and non-pharmacological options to manage pain (Table 5).

Table 5 Interventions currently used by young people
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Eleven HCPs, comprising of pediatric rheumatologists (n = 4), physical therapists (n = 3), pediatric rheumatology nurses (n = 2) and occupational therapists (n = 2) participated (see Table 6).

Table 6 Health care providers’ clinical practice information
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Themes

Findings revealed six themes which will be discussed. Quotes are also shown (see Table 7).

Table 7 Quotes illustrating decision-making needs
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(1) Need to assess pain in an accurate manner

Young people and parents/caregivers

Some parents mentioned it was difficult for them to assess their child’s pain since they were relying on their child’s self-report which they did not always feel was accurate. Parents did not feel that their own perceptions of their child’s pain were accurate, and thus needed a better means to assess their child’s self-reported pain. One parent shared that a HCP did not think that their child’s pain was real. Parents felt that assessing pain was easier as youths became older.

HCPs

Consistent with parents’ reports, HCPs described difficulty assessing youths’ pain. HCPs indicated this was due partly to discrepancies between youths’ and parents’ report of pain, and between reported pain and participation in their daily activities, as well as the youths’ difficulty in remembering pain over time, especially among younger children and those who do not experience a flare. A few HCPs mentioned not always assessing pain if families do not raise the issue.

A few HCPs reported that pain is not the predominant issue that families bring forward. HCPs thought that some young people may be reluctant to mention their pain to avoid escalating treatment. A few HCPs stated that even when young people report pain, they do not volunteer a lot of information about it, especially younger youths. Similar to parents, HCPs mentioned that youths, especially younger ones, have difficulty identifying and describing pain. A few HCPs voiced that pain is very subjective and cannot be predicted by physical findings. HCPs felt the need to assess the child’s self-report of pain and its functional impact in an accurate manner.

(2) Need to address pain in pediatric rheumatology consultations

Young people and parents/caregivers

Young people and parents stated that discussions with pediatric rheumatologists and nurses revolved mostly around disease activity and arthritis medication (medication targeting disease remission). They had less thorough discussions specifically about pain and its treatments, including non-pharmacological options. Families mentioned that they discussed pain management options in more depth with HCPs, including a wider range of non-pharmacological options, when pain was the main disease feature and when they were referred to allied HCPs or a chronic pain team. Most felt that there is a need to address pain management in pediatric rheumatology.

HCPs

Similar to young people’s and parents’ reports, rheumatologists mentioned that consultations do not usually focus on pain since it is usually not the dominant problem in JIA. They also mentioned having more thorough discussions about pain when youth have persistent pain with low disease activity.

Consistent with families’ responses, rheumatologists and nurses reported that their first goal was to control disease activity, which they felt should address the pain. Thus, their interventions focused on medication to control disease activity, followed by medication and non-pharmacological options to reduce residual pain. Allied HCPs and rheumatologists working in chronic pain clinics tended to focus more on symptom management (including pain) and improving function, which they achieved by discussing and recommending various options including a wider range of non-pharmacological options.

(3) Need for information on pain management options, especially non-pharmacological approaches

Young people and parents/caregivers

Although most parents and young people mentioned receiving enough information on treatments targeting disease activity, most mentioned a need for more information on treatments specifically targeting pain, especially for non-pharmacological options and complementary health approaches (CHAs). They wished to receive information in a clear, concise and honest manner.

Participants reported that HCPs only presented a few pain management options when pain was an issue. They felt that some evidence-based information was presented on benefits and risks of medications but much less for non-pharmacological approaches. When asked, participants could identify a few potential pain management options, but were often unsure about benefits and risks, especially for non-pharmacological options.

Parents mentioned that information on pain management was provided at the time of diagnosis, but it was overwhelming. Also, they received information mostly in the consultation and were given little information between appointments. Parents mentioned they needed information throughout the disease course, as needed, with time to digest information between appointments.

Some parents and young people mentioned that HCPs provided information on pain management from pamphlets, books and health organisation websites. Many participants said they had to search for information on websites and social media or ask others, especially for non-pharmacological options. They found it useful to obtain information on various options and knowing what other parents tried but acknowledged that the information was not always reliable.

Overall, parents and young people wished to know more about a wide variety of pain management options, and scientific evidence of potential benefits and risks (short and long term), how well each worked for others, as well as logistics (e.g., time, cost), especially for non-pharmacological treatments. A few said that they wished to get probabilities of benefits and risks of treatments. They mentioned HCPs should be more educated on pain management, especially physicians and nurses concerning non-pharmacological options.

HCPs

HCPs, especially physicians and nurses, voiced a lack of knowledge about available options for pain management and their evidence, mostly for non-pharmacologic options. Some also mentioned limited scientific evidence for these options, even though they did not search for it. They reported that this lack of knowledge makes it difficult to discuss the options with families, which is consistent with families’ perceptions.

Consistent with young people's and parents’ perspectives, most HCPs said they present benefits and risks with some evidence-based information to families but mostly for medications and less for non-pharmacological options. Some say they give a few options for non-pharmacological options that they have experience with, either professionally or personally. They mentioned that the information they provide on non-pharmacological options is not consistent and depends on each family’s needs and concerns.

HCPs mentioned sharing links to trusted websites to help guide families to manage pain. HCPs who treat more youth with chronic pain reported providing more resources such as handouts, websites and apps. HCPs acknowledged that families seek information online.

As with young people’s and parents’ wishes, HCPs suggested that families should receive information on a wider range of pain management options, especially non-pharmacologic options, and their benefits and risks. Information should be thorough but not overwhelm families.

(4) Importance of effectiveness, safety, and ease of use of treatments

Young people's and parents/caregivers’ characteristics

Young people and parents reported that the most important consideration when choosing pain management options was treatment effectiveness. Most adolescents also wished to avoid injections because they are uncomfortable and splints because of stigma. It was also important for them and their caregivers to use treatments that were easy to use and did not disrupt their life.

Parents wished to avoid the potential short- and long-term side effects of pain medication. They preferred non-pharmacological options that would encourage their children to learn to manage their condition rather than relying on pain medication such as NSAIDs. Many parents felt that non-pharmacological options such as rest, heat, cold, stretching and deep breathing were helpful. However, some families mentioned lack of agreement between them and their child regarding the use of CHAs, with youths sometimes not interested in using these. Parents voiced practice variation among HCPs with some providers recommending non-pharmacologic interventions and others less so, with a lack of knowledge, belief or comfort with these interventions, especially CHAs.

HCPs

Consistent with families’ perspective, HCPs acknowledged that families wished to avoid injections and preferred non-pharmacological treatments for fear of overmedicating; a sentiment echoed by most HCPs. Some HCPs preferred not to add pain medications because they have limited effectiveness and possible risks. Some mentioned that they did not always support the use of some non-pharmacological options because of their lack of knowledge. They mentioned that families should not be overwhelmed with a complex treatment regimen.

(5) Need to discuss young people's/families’ values and preferences for pain management options

Young people and parents/caregivers

Young people and parents mentioned that HCPs listened to them and adjusted arthritis medications and additional pain medications based on their values. However, families reported that HCPs did not always actively ask about their values and preferences. HCPs usually recommended medications but changed them if families had issues. Some families said they had accepted the medical regimen but then discussed it at home and decided not to adopt the regimen.

For non-pharmacological approaches to help alleviate pain, there were few discussions with HCPs. When they happened, young people and parents mentioned that HCPs told them to use these options and then sometimes were asked if they wished to use these treatments and if it fitted in their life. A few parents and young people reported that HCPs do not always take into account their preferences for non-pharmacological options such as CHAs, but usually let them use these. Some mentioned that they had to bring new options and convince their HCPs. Parents and youths wished to discuss their values and preferences with HCPs.

HCPs

Consistent with families’ perspective, some HCPs mentioned that they do not ask directly about patient values and preferences. They said that it usually emerges in the conversation over time as families ask questions about treatment options.

Some HCPs reported that they discuss patient preferences for pain management in the consultation but do not spend enough time on it. They tried to tailor information based on families’ concerns and willingness to use treatments. However, some mentioned that they often reached a decision with families but were then disappointed to see that they had not used the treatment.

(6) Need for decision support

Young people and parents/caregivers

Need for families and HCPs engagement in decision-making

Many families mentioned that HCPs often told them which treatment options to use and then families decided which treatments to use daily. Thus, many decisions to manage pain were made by families outside the clinical consultation. Parents and youths wished to be involved in decision-making by having a discussion with HCPs and felt current engagement in clinical settings is not optimal. Most parents mentioned that their youths, especially older ones, were actively engaged in choosing pain management options. They felt that this engagement is crucial since youths know their pain best and which options they could follow. Parents also wanted their youth to learn how to make decisions and have discussions with HCPs.

Barriers to optimal decisions and their impact

Some parents and young people mentioned feeling pressured by their physician to use pain medication such as NSAIDs and felt they could not speak up. Parents sometimes felt pressured and criticized by friends for their treatment choices. Parents mentioned fear when their child was diagnosed and they had to choose medications.

Some young people felt stressed, confused and worried when choosing how to manage pain, especially at school, and unable to make a decision, especially regarding medications because of potential risks. Some youths felt upset about having to take pain medications.

Most participants mentioned being sure of their decision to manage pain but some acknowledged uncertainty because of their lack of knowledge, especially for non-pharmacological options. Most said they used the chosen options, but they sometimes stopped using treatments because of side effects they were not aware of. Families said that choosing non-pharmacological options was not based as much on their values as they wished. Some felt a lack of support to choose pain management options but mentioned that trusting HCPs facilitated decision-making.

Need for decision support interventions and their potential impact

Parents and young people mentioned that having access to a tool to help assess pain and provide evidence-based information on options that match with their values would help them make decisions and engage in discussions with HCPs. Most families mentioned that the tool should be accessible on a computer/tablet/smart phone for easy access, and some wished to print the information. Most participants would like to use this tool between consultations and discuss information with HCPs using a printout or their phone.

Families felt that using an app or website would help ensure they have enough information and time to review and discuss with HCPs. Parents mentioned that giving information via an app developed by experts would help adolescents trust parents’ advice if recommendations are similar. Participants felt that using SDM would lead to more informed and personalized decisions, and youth empowerment.

HCPs

Need for families and HCPs engagement in decision-making

HCPs reported that they typically recommended pain management options and let families determine how to use treatments on a daily basis. They mentioned engaging families and young people in discussions for pain management (with older youths being more engaged) but felt that families and young people should be more engaged in decision-making, especially for non-pharmacological options.

Barriers to optimal decisions and their impact

HCPs mentioned that the lack of information and discussion about pain management led to uncertainty about which pain management options to use, especially non-pharmacological options. They also mentioned pressure on families from HCPs and others to use certain treatments. HCPs mentioned facilitators to decision-making such as the fact that families can voice their opinion. HCPs felt that families had more difficulty making decisions about arthritis medication because of potential risks. They felt that choosing how to deal with pain using non-pharmacological options was less difficult. A few HCPs mentioned that families were unsure which treatments to choose for pain after trying a few options.

Need for decision support interventions and their potential impact

HCPs reported that they would benefit from a tool to help describe young people’s pain and present evidence-based information on a range of treatments that match each young people’s values to engage in a collaborative approach to pain management. HCPs felt that an app or website that is easily accessible along with discussions with HCPs would be optimal to meet families’ needs. HCPs mentioned that some families may prefer an electronic version while others may prefer a paper version. HCPs also mentioned that using this tool could help them learn from families which options may be effective in real life.

Discussion

This study identified families’ and HCPs’ mutually agreed upon decision-making needs related to JIA pain management. Findings reveal a need for assessing pain in an accurate manner and for sharing evidence-based information for pain management, especially on non-pharmacological treatment options. Participants also voiced a need for clarifying and discussing families’ values and preferences about pain management and for a joint decision with HCPs. Participants felt that a decision support intervention may enable HCPs to work with youth and families to better describe their pain and identify evidence-based treatment options to make informed and value-based decisions.

Findings emphasize a previously demonstrated need for assessing pain in an accurate manner [13, 18, 28]. Indeed, learning how to best recognize and treat pain is a research priority among youth with JIA [29]. However, studies have shown that it is difficult for parents and HCPs to accurately assess youths’ pain [13, 30] and HCPs are reluctant to assess pain, in part because of lack of training and confidence to do so [13]. Using validated tools, as suggested by our study and others [13, 18, 31], may address these barriers.

Findings reveal that consultations with HCPs do not often focus on pain management but mostly on disease activity, which is consistent with studies showing the low priority of addressing JIA pain [13]. This may explain the need for information on a wide range of pain management options for families [12, 32]. Allied HCPs and HCPs working in chronic pain clinics tend to discuss pain more often, its impact and a wide range of options, demonstrating the importance of specialized pain training [13]. Furthermore, information provided by HCPs varies and may not meet each family’s needs, thus showing the need to assess each family’s information needs [33]. Families must often resort to finding information online which, although not necessarily evidence-based, can be helpful [12, 34].

Several families’ values and preferences reflected those of HCPs and included the importance of effectiveness, safety and ease of use of treatments, which are similar to other studies [35, 36]. Families mentioned that HCPs wished to reduce pain medication and use non-pharmacological options, but that sometimes HCPs were not open to some options such as CHAs, showing that HCPs and families may not have the same preferences [33]. Parents also mentioned that their values and preferences are sometimes different from their children, which is consistent with studies on biologics in JIA and Crohn’s disease [37]. These differences among the youth-parent-HCP triad should be assessed and discussed to promote personalized decisions. Unfortunately, the current study shows that families and HCPs do not always discuss values and preferences which is similar to another study in JIA [38] and shows the need for a formal assessment of values and preferences.

Participants felt that families and young people should be engaged in decision-making about pain management and that current engagement is not optimal. There is a need to ensure optimal information-sharing and decision support to allow for decisions based on families’ and youths’ values and preferences. Participants felt a need for a tool to assess young people’s pain, provide evidence-based information on a range of pain management options and discuss families’ values and preferences in a joint decision with HCPs. Decision support interventions, such as patient decision aids (PDAs), decision coaching and HCP training in SDM, may be helpful in meeting these needs [16, 39,40,41,42].

Study limitations

Most of the young people in our sample were girls, lived in Canada and had oligoarthritis or polyarthritis, which may preclude us from fully understanding the experience of boys, young people from the United States and those who have other JIA subtypes where pain is very common such as enthesitis-related arthritis. Also, we were not able to recruit all relevant HCPs, such as psychologists. However, we included psychologists in our research team and will engage them in developing decision support interventions.

Conclusion

Patients with JIA, their caregivers and HCPs all identify a need to assess pain in an accurate manner and for sharing evidence-based information for pain management, especially on non-pharmacological treatment options. There is also a need for clarifying and discussing families’ values and preferences about pain management and for a joint decision with HCPs. A decision support intervention that addresses these needs, as well as HCP training about pain management and SDM, may enable HCPs to work with youth and families to better describe their pain and identify evidence-based treatment options to make informed and values-based decisions about pain management options. Work is underway to develop such interventions and implement them into practice to improve pain management in JIA and in turn lead to better health outcomes.