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Considering the changing landscape of internet use and rising ownership of digital technology by young people, new methods could be considered to improve the current model of juvenile idiopathic arthritis (JIA) management.
This systematic review aims to evaluate the usability of eHealth and mobile health (mHealth) interventions currently available for young people living with JIA.
The PRISMA (Preferred Reporting Items for Systematic Reviews and Meta-Analyses) guidelines were used to oversee this review. We systematically searched 15 databases for 252 potential studies; 2 authors independently screened all quantitative studies reporting the use of eHealth and mHealth interventions for young people (aged 1-18 years) diagnosed with JIA. Studies were excluded if they did not report outcome measures or were reviews, commentaries, or qualitative studies. Study methodological quality was scored using the Down and Black (modified) checklist. A narrative descriptive methodology was used to quantify the data because of heterogeneity across the studies.
A total of 11 studies were included in this review, reporting 7 eHealth and mHealth interventions for young people (aged 4-18 years) living with JIA, targeting health issues such as pain, health-related quality of life, physical activity, and chronic disease self-management. The usability of the interventions was facilitated through training and ongoing support. The engagement was promoted by a combination of persuasive influences, and barriers preventing adherence were removed through personal reminders and flexible program schedules to cater to JIA and non-JIA illnesses or other commonly seen activities in childhood. The feedback obtained was that most young people and their parents liked the interventions.
The results of this review need to be considered cautiously because of the lack of rigorous testing and heterogeneity, which limits the detailed descriptions of data synthesis. Further research is needed to consider gender differences, associated costs, and the effectiveness of the interventions on health outcomes to better support young people living with JIA.
Juvenile idiopathic arthritis (JIA) is the most common type of arthritis in young people [
Responding to the needs of young people living with JIA is a challenge because of the problems in the current model of care [
Feedback from parent and carer surveys also suggests that pediatric rheumatology services need to improve the quality of their service and the patients’ experience. Responses suggest they need to optimize their efficiency, improve information exchange [
Good self-management behavior has a positive effect on health outcomes [
An innovative way to improve the current care model and foster self-management skills could be through eHealth or mobile health (mHealth) interventions [
Critical to the success of any digital intervention is the manner in which young people accept and interact with the technology [
eHealth is described by the World Health Organization (WHO) as an activity that delivers health-related information, resources, and services through electronic technology and internet connectivity [
This systematic review presents the first of 2 steps in evaluating the clinical use of eHealth and mHealth interventions for young people (aged 1-18 years) living with JIA. This review aims to evaluate quantitative studies examining the usability of eHealth and mHealth interventions to understand how young people interact with the technology. The following 3 areas were considered:
Identification of the digital health intervention.
Usability (delivery of the intervention) [
Costs associated with the intervention [
It is anticipated that such information will improve our understanding of the mechanisms that support the use of these interventions by young people living with JIA and inform future development. The second step of this review aims to evaluate the effectiveness of interventions. These results will be published subsequently in another review.
The PRISMA (Preferred Reporting Items for Systematic Reviews and Meta-Analyses) statement [
All young people (aged 1-18 years) diagnosed with JIA using the International League of Associations for Rheumatology criteria [
Any eHealth or mHealth interventions (see definition in the
No comparator was used.
We considered an outcome as any quantifiable measure specifically targeting the pediatric population or pediatric rheumatology.
All quantitative studies reporting the use of eHealth and mHealth interventions for young people (aged 1-18 years) diagnosed with JIA were included. Studies were excluded if they did not report outcome measures or were reviews, commentaries, or qualitative studies.
To develop search terms, MEDLINE and CINAHL were initially searched by SB to identify keywords in the titles, abstracts, and indexed terms. In October 2018, the search terms were adapted to suit the controlled vocabulary, Boolean operators, truncation, and wildcards in MEDLINE/PubMed, the Cochrane Library, Joanna Briggs Institute, AMED (Allied and Complementary Medicine Database), CINAHL complete, EMBASE, JAMA (Journal of the American Medical Association), Informit Health, ProQuest database, PsycINFO, IEEE (Institute of Electrical and Electronics Engineers and Institution of Engineering and Technology) Xplore, SAGE Publishing, ScienceDirect, Scopus, and Web of Science. Further studies were retrieved from Google Scholar and arthritis-related organizations (Arthritis Australia, Arthritis Foundation, and Childhood Arthritis and Rheumatology Research Alliance funded projects and conferences) and by hand searching reference lists. The search strategy was not restricted by language or year of publication. The database search was repeated in November 2019 (
Two authors (SB and AC) independently reviewed all studies retrieved by the search strategy via individual log-in systems on the web-based platform Covidence [
A data extraction Excel (Microsoft) form was designed to collect all relevant information from the studies, including participant demographics, eHealth or mHealth characteristics, study design, study outcomes, and costs. Conclusions were drawn from the outcomes reported by study authors. Data extraction was completed by SB and checked by all reviewers.
Using the Down and Black (modified) checklist for randomized and nonrandomized studies [
To summarize the participants’ characteristics, the mean scores reported by the study authors were averaged. The range of data was determined by the reported highest and lowest values. Individual JIA subtypes were combined and expressed as the total number (n) and the proportion of each subtype as a percentage (%). The total number of studies including the information required was stated (ie, “4 studies reported…”) to account for missing data.
Meta-analysis was considered unsuitable for this systematic review because of the heterogeneity across the studies and the different intervention development stages. Instead, a narrative synthesis methodology was used to allow the data to be organized, explored, and presented in a logical way [
Develop a theoretical model to understand how the intervention works.
Conduct a preliminary synthesis to:
identify factors supporting implementation and barriers;
consider relationships among studies.
Perform a content analysis (translation of data) to:
report characteristics among studies;
identify moderator variables;
develop numerical/statistical rubrics.
Draw a conclusion by critically reflecting on methodology synthesis.
A total of 252 studies were identified using the search strategy. After removing 70 duplicates, 127 studies that did not meet the inclusion criteria based on their title or abstract and 44 based on the full-text screening, a total of 11 studies met the inclusion criteria for this review (
Summary of the study selection process using the PRISMA (Preferred Reporting Items for Systematic Reviews and Meta-Analyses) diagram.
This review included 634 participants [
Juvenile idiopathic arthritis subtypes, based on the International League of Associations for Rheumatology criteria.
Juvenile idiopathic arthritis subtypes | Value |
Oligoarthritisa, n (%) | 195 (30.8) |
Polyarthritisb, n (%) | 172 (27.1) |
Polyarthritis (rheumatoid factor positive), n (%) | 41 (6) |
Enthesitis related, n (%) | 54 (8) |
Systemic, n (%) | 46 (7) |
Psoriatic, n (%) | 33 (5) |
Undifferentiated, n (%) | 7 (1) |
Unknown or not yet diagnosed or other, n (%) | 39 (6) |
Chronic arthritis with other/or other forms of rheumatic diseasec, n (%) | 13 (2) |
Not recorded, n (%)d | 34 (5) |
Disease activity (cm), mean (range)e | 1.8 (0.2-3.7) |
Disease duration, mean (range)f | 5 years (<1 month-15.65 years) |
aIncludes the subcategories of oligoarthritis: persistent and extended [
bIncludes 2 studies not reporting positive or negative rheumatoid factor. [
cJuvenile dermatomyositis, systemic lupus erythematosus [
dReasons: drop-out, loss of follow-up (n=32) [
eA total of 8 studies reported disease activity [
fA total of 7 studies reported disease duration [
The 11 studies included in this review reported 7 interventions, describing varying stages of development (preprototype to maturity); 4 interventions were web-based programs [
Custom-designed programs designed by the research team were used in 5 interventions [
Study outcomes considered feasibility, usability, efficiency, and/or effectiveness. To align usability outcomes with research questions 2 and 3, usability outcomes were synthesized to form 4 themes: (1) user engagement (promotional activities and adherence), (2) barriers preventing usability (personal, technical, and device barriers), (3) user feedback (acceptability and satisfaction), and (4) cost assessment (basic financial costs). Themes were based on 5 of 16 areas of reporting by Agarwal et al [
Formation of themes, evaluation criteria, and main outcomes supporting the delivery of the eHealth and mobile health interventions for juvenile idiopathic arthritis.
Research question, theme, and evaluation criteria | Outcomes | ||
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Promotional activities | Adoption inputs [ |
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Adherence | Activity log, dropout [ |
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Personal barriers, technical barriers, or device barriers | Device malfunction rate [ |
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Acceptability | Questionaries [ |
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Satisfaction | Questionaries [ |
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Basic financial cost (owing to no comparator) | Cost (adding together development of the program, staff costs, financial consequences ie, traveling expenses and babysitters) [ |
The study design included 4 descriptive studies, 3 pilot randomized controlled trials (RCTs), 1 multicenter RCT, 1 pre- and postintervention design, 1 correlational research design, and 1 sequential cohort study—studies reported from 2 high-infrastructure countries, Canada [
The methodological quality of study scores ranged from 15 to 21 out of 28 (mean score 18.6, SD 1.9), a fair to good score [
All studies reported promotional activities to support engagement with the intervention. The top 2 included training [
Promotional activities used to support engagement with the intervention for juvenile idiopathic arthritis.
Promotional activity | Misfit flash | Rheumates@Work | ePROfile | iPeer2Peer | eOuch | SUPER-KIDZ | Teens taking charge |
Training | Ya | Y | Y | Y | Y | Y | Y |
Instruction manual | Y | —b | — | — | Y | — | — |
Goals set by users | Y | Y | — | — | — | — | Y |
Ongoing technical support | — | Y | — | — | Y | — | Y |
Personal reminders | — | — | — | — | Y | — | — |
Ongoing human communication | — | Y | — | Y | Y | — | Y |
Reactive feedback loop | — | Y | — | — |
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— | — |
Linear design | — | Y | — | — | Y | — | Y |
Interactive content | — | Y | — | — |
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— | Y |
Flexibility in length of the program | — | Y | — | Y |
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— | Y |
aY: yes.
bThe promotional activity listed was not used.
All interventions provided participants and/or parents with training (range <5-20 min) [
Throughout the study period, 4 interventions provided ongoing human communication [
A total of 3 fair to good–quality studies monitored participants’ safety [
All interventions had an expected level of engagement, ranging from minutes to 17 weeks [
A significant difference was seen in adherence in pain reporting, using the eOuch pain diary, by 2 fair to good quality studies, week to week and according to the time of day (
The impact of gender and/or age on adherence was considered by 2 high-quality studies [
The most common topics raised/learning modules visited by participants in self-management programs were reported by 3 high-quality studies. The most common were
Personal barriers preventing adherence were reported qualitatively by 6 fair to good–quality studies. The main barrier was illness, both JIA- and non-JIA-related [
Technical barriers preventing adherence were reported by 7 fair to good–quality studies [
Overall, 3 fair to good–quality studies compared electronic
In another fair-quality study, 3 different devices were compared by children (aged 4-7 years), adolescents (aged 8-18 years), and parents when completing the web-based pain assessment
In the same study, children preferred the computer to paper or iPod because it was simple and fun to use (
All 7 interventions gained positive feedback from young people [
Parental feedback was also considered by 2 interventions [
Only 1 of the 7 interventions reported the cost of establishing and maintaining the intervention [
Overall, 4 studies reported that they provided participants with a device to enable access to the intervention [
To the best of our knowledge, this is the first systematic review to evaluate the usability of eHealth and mHealth interventions targeting young people living with JIA (aged 4-18 years). Guided by our 3 research questions, this review identified 7 interventions:
Of the interventions, 3 allowed participants to self-report pain [
In total, 3 interventions aimed to improve self-management behavior [
A range of promotional activities was used to facilitate the engagement of the participants with the interventions. These promotional activities are referred to in the literature as persuasive influences [
The interventions in this review used, on average, 4 persuasive influences (range 1-7), the 2 most common being training [
However, not all forms of human communication identified in this review were supported
In this review, to improve the young person’s experience and ensure that the interventions were achieving the intended interactions, personal barriers were removed [
All interventions in this review gained positive feedback from young people [
Only 1 study in this review reported costs related to the implementation of their intervention [
Only 3 studies in this review considered gender differences [
The findings of this systematic review need to be considered cautiously because of the limited number of studies included. Our methodology could have been improved by including qualitative studies in our selection criteria and individually handing searching journals relevant to digital health to ensure no usability issues were omitted and reduce publication bias from the database search.
Meta-analysis was also not possible in this review because all interventions considered different outcomes measures, depending on their stage of development. Instead, this systematic review largely relied on descriptive summaries to organize and clarify the data from formal and informal assessments. This form of analysis can be subjective; participants may have been influenced by the novelty of the intervention, boosting their engagement and feedback. There is also a risk of reporting bias by the author. To reduce this risk and improve transparency, all authors reviewed each stage of the data analysis.
The generalizability of our findings may also be limited. Participants were included in this review with other forms of rheumatic disease, or their diagnosis was unknown or not recorded (86/634, 13.6%;
This review also only focused on 3 of our 4 protocol questions: (1) identification of the intervention, (2) usability, and (4) cost, rather than (3) effectiveness, to allow us to capture a more detailed description of the interventions and usability problems faced by participants. The effectiveness of the interventions will be covered in a follow-up publication.
Using a narrative, descriptive methodology, our review identified 7 interventions for JIA, targeting health issues such as pain, HRQoL, physical activity, and chronic disease self-management. The usability of the interventions was facilitated through training and ongoing human communication. Engagement was promoted by a combination of persuasive influences, and barriers preventing adherence were removed through personal reminders and flexible program schedules to cater to JIA- and non-JIA illness or other activities commonly seen in childhood. The feedback obtained was that most young people and their parents liked the interventions. Although too premature to support the effectiveness of our claims, this review will add to the growing body of evidence influencing the development of future eHealth and mHealth interventions. Further research is needed to consider gender differences, associated costs, and the effectiveness of interventions on health outcomes to better support young people living with JIA.
PRISMA (Preferred Reporting Items for Systematic Reviews and Meta-Analyses) checklist.
Search terms and database search strategy.
Overview of the seven eHealth and mHealth interventions for JIA.
Overview of the eleven eHealth and mHealth studies targeting Juvenile Idiopathic Arthritis.
Methodological scores of the eleven studies using the Down and Black.
Intervention adherence rates, including week to week, time of day and content adherence by young people with JIA.
Summary of the technical problems experienced by users with JIA, obstructing the adoption of the intervention.
User feedback: Results of Acceptability and Satisfaction questionnaires.
brief pain inventory
control group
health-related quality of life
intervention group
juvenile idiopathic arthritis
mobile health
personal digital assistant
pediatric rheumatologists
randomized controlled trial
World Health Organization
All authors contributed to the conceptual design, methodology, data extraction, manuscript development, and final manuscript approval.
None declared.