Introduction
With a disease prevalence of 3 in 1000 Canadian children, juvenile idiopathic arthritis (JIA) is the most common type of arthritis among children and adolescents and is a great cause of pain and discomfort [
1]. Studies in the US have shown that within the first year of diagnosis, being non-White and having lower household income are characteristics associated with higher disease activity in children with JIA as well as a longer “time to first appointment” [
2,
3]. Furthermore, previous studies have also shown that there is a notable economic burden of having a child with JIA due to costly medications and specialist treatments such as physiotherapy [
4,
5]. This is burdensome as specialist treatments are typically not covered by the Canadian healthcare system and there is a lack of consistency in medication coverage across Canadian provinces as it is dependent on the degree of provincial and private insurance coverage.
There are recent calls for consideration of health inequities, that is, differences in health status or differences in the distribution of health resources between groups in society caused by dissimilarity in the social conditions they live [
6], in rheumatology research. In a 2019 systematic review of US-based randomized controlled trials (RCTs) in rheumatoid arthritis (RA), Strait et al. characterized participants as mostly females and noted underrepresentation of minority racial and ethnic groups, males, and younger and older individuals [
7]. In 2022, we conducted a scoping review of Canadian RCTs in RA patients and similarly found that participants were largely middle-aged, female, and White. In addition, we assessed reporting of determinants of health inequities and found that most frequently reported were sex and age, and to a lesser extent race, education, and socioeconomic status (SES) [
8]. To our knowledge, there are no syntheses on how health inequities have been considered in research among patients with JIA. As such, we conducted a scoping review of Canadian RCTs of interventions among JIA patients to characterize participants and assess how determinants of health inequities are reported. We used the Campbell and Cochrane Equity Methods Group’s PROGRESS-Plus Framework which represents factors (“PROGRSS factors”) that lead to inequities in health (i.e., (i.e.,
Place of residence;
Place,
Culture,
Ethnicity,
Language;
Occupation;
Gender/sex;
Religion;
Education;
Socioeconomic status; and
Social capital)
Plus additional factors (i.e., Personal characteristics associated with discrimination; Features of relationships; and Time-dependent relationships) [
9].
Discussion
Guided by the Campbell and Cochrane Equity Methods Group’s PROGRESS-Plus framework [
9], we conducted a scoping review of five RCTs of interventions for individuals with JIA in Canada to improve our understanding of how health inequities are reported. Among PROGRESS-Plus factors, only sex and age were reported in all RCTs. Race and education were reported once by different studies [
16,
17]. Socioeconomic status and features of relationships were reported once by the same study [
18]. From this limited data, we characterized participants of JIA RCTs in Canada as primarily early-adolescents and female. In showing the very limited reporting of determinants of health inequities in RCTs of JIA in Canada our study has implications for raising awareness of the need of increased representation and diversity in research among this unique patient population.
A key to our scoping review was assessing how PROGRESS-Plus factors were considered in studies that reported them. Although sex was reported in all studies, there were some inconsistencies with the use of sex and gender in some studies [
15,
19]. For example, the terms “male” and female” were used when reporting overall patient characteristics however the terms “girl” and “boy” were used when describing participants through the body of the text as equivalent terms, with one study explaining they compared different sexes and used boys and girls as their description. However, conflation of sex and gender terms were not seen in studies published in the 2010s and onwards [
16‐
18]. This is interesting as this pattern is not consistent with reporting trends in studies on RA [
8], where more studies published before the year 2000 correctly used the terms sex and gender compared to those after 2000. Age was the other commonly reported PROGRESS-Plus factor, and it was reported as a mean age in years in most studies. JIA is diagnosed following a period of arthritis (joint inflammation) lasting longer than six weeks with an onset age of less than 16 years [
24], however depending on the type of JIA (systemic arthritis, oligoarthritis, rheumatic factor (RF) positive arthritis, RF negative arthritis, enthesitis-related arthritis or psoriatic arthritis) the age at onset differs. The age at onset tends to fall under either the ages of 2 to 4 years or between 9 and 12 years, with only RF positive arthritis and enthesitis-related arthritis having an onset during adolescence [
25]. The average age of JIA patients is thus well captured in the trials, as the mean age of participants across all studies was 12.6 years, especially as most trial interventions had a focus on patients coping with JIA independently rather than treatment at diagnosis [
16‐
18].
It is also important to assess less frequently reported PROGRESS-Plus factors. Looking at the factor of SES, this was reported in one study as annual household income, according to five categories ranging from <$25,000 CAD to between $100,000 and $150,000 CAD, with an option to not report (22.3% of participants) [
18]. The majority of participants who chose to report (77.7%) had average household incomes upwards of $100,000 CAD (the highest income category option). This study was conducted in eight Canadian provinces (Ontario, Alberta, Nova Scotia, Saskatchewan, Quebec, British Colombia, Newfoundland and Labrador, and Manitoba) and patient responses fall in line with epidemiological data found on JIA patients in Manitoba where 65.5% of patients were categorized in the highest income quintiles (Q3-5, no information was given regarding the range of incomes each quintile fell under) [
26]. However as average household income is self-reported with an option to decline answering, the reliability of this data is limited as it may be subject to reporting bias. Furthermore, given the age of the JIA patient population, it is likely that parents/guardians are providing this information. Understanding individual patients’ ability to afford treatment is important as it has been shown that there is a substantial cost related to JIA treatment [
4,
5]. An accurate awareness of how much of an economic burden JIA care is on families could help pave the way to increased financial support from the healthcare system.
It is difficult to speak on the degree of representation of determinants of health inequity within JIA trials due to the limited published data. The ratio of male to female participants is reflective of the demographic characteristics of JIA, with most trials having females account for about 70% of total participants as the majority of JIA patients tend to be female [
26,
27]. Looking at the single study that reported on race [
16], there is a significantly higher percentage of White participants (70%) compared to all other races which could indicate a need for increased participant diversity across the board, but without reported data from other trials definitive conclusions cannot be made. Interestingly, no studies reported on the place of residence of participants. Some research has shown that 43% of patients live in rural areas which is particularly relevant at it has been found that individuals in rural Canada tend to have a poorer access to healthcare [
28,
29] and JIA is an illness that requires individualized and ongoing treatment. Because of this, it is important to know the place of residence of participants as it may have a direct effect on the quality and effectiveness of interventions due to lack of resources.
Our scoping review has demonstrated the lack of health inequity reporting, as in current conditions, there is insufficient data to determine if RCTs in JIA are being conducted in a manner that is reflective of equity, diversity, and inclusion factors such as race, sex, and socioeconomic status, as well as other PROGRESS-Plus factors. It is important to ensure that RCTs (and other health research studies) are being designed in a manner that would have a benefit to patients from all backgrounds rather than those that are typically represented, such as White and wealthy populations [
30,
31]. This raises the question of why there has been such a drastic limit to the number of determinants of inequity being reported in JIA RCTs. It could in part be due to the increased workload of asking what could be considered “invasive” questions such as religion and SES. Factors such as occupation, SES, and time-dependent relationships may seem irrelevant to ask paediatric patients, and it may not be clear that these questions could be asked of the guardians and/or household (for example, asking average household income or marital status of parents/guardians). Due to the impact of parents/guardians as primary caregivers on the management and maintenance of JIA treatment it is important to gain an understanding of the general demographics of these caregivers. Caregiver characteristics were only reported in one study [
18] where parent/guardian education level was asked, most likely as caregivers were also asked to participate in the trial. This indicates that clearer guidance should be developed to better encompass the relevant characteristics of paediatric patients and their households.
This scoping review has several limitations. As only RCTs were considered, we may have included a more biased sample of studies that may be considered of higher rigor. This may suggest that our findings underestimate the extent of issues of underreporting of determinants of inequity. However, RCTs may be limited with respect to inclusion criteria, measurement of determinants of inequity (e.g., have comprehensive questionnaires) and time for participants to complete such measures. Future research could consider expanding to expanding to observational studies and point-of-care trials to address determinants of equity. We used the PROGRESS-Plus framework to guide extraction of reporting of determinants of inequities; however, this framework may not be exhaustive, or as previously discussed, entirely aligned with paediatric patient characteristics.
Publisher’s Note
Springer Nature remains neutral with regard to jurisdictional claims in published maps and institutional affiliations.