How can we support children, adolescents and young adults in managing chronic health challenges? A scoping review on the effects of patient education interventions

Abstract Objectives This scoping review aims to give a comprehensive and systematic overview of published evaluations and the potential impact of patient education interventions for children, adolescents and young adults who are living with chronic illness and/or impairment loss. Methods Relevant literature published between 2008 and 2018 has been comprehensively reviewed, with attention paid to variations in study, intervention and patient characteristics. Arksey and O'Malley's framework for scoping studies guided the review process, and thematic analysis was undertaken to synthesize extracted data. Results Of the 7214 titles identified, 69 studies were included in this scoping review. Participant‐reported benefits of the interventions included less distress from symptoms, improved medical adherence and/or less use of medication, and improved knowledge. The majority of studies measuring physical activity and/or physiologic outcomes found beneficial effects. Interventions were also beneficial in terms of decreased use of urgent health care, hospitalization, visits to general practitioner and absence from school. By sharing experiences, participants had learned from each other and attained new insight on how they could manage illness‐related challenges. Discussion Study results corroborate previous research suggesting that different types of patient education interventions have a positive impact on children, adolescents and young adults, but research on this field is still in a starting phase. The results summed up in the current review supports the utility of patient education interventions that employ behavioural strategies tailored to the developmental needs of children, adolescents and young adults with different cultural backgrounds.


| INTRODUC TI ON
Children living with chronic illness are less able to participate in social activities. The daily management of their illness often requires that the whole family adjust to a new way of life. 1 Adolescents encounter difficulties due to experiences of physical and psychosocial changes, social pressure from their peers and adolescent health-care transition. Moreover, adolescents tend to be afraid, anxious and shameful of their illness. 2 Because chronic condition in childhood is one of the major health challenges of this century, gaining skills in self-management becomes increasingly important. 3 The challenges are particular worrisome in low-and middle-income countries who experience an increase in the number of young people developing long-term conditions. 4 The process where patients are enabled to become actively involved in finding out what is important to them, in making decisions about factors that affect their lives and in taking action to achieve change, is often described as patient engagement. 5 More recently, the concept of patient engagement has been envisaged as an crucial factor impacting on patients' ability to self-manage and as an important goal for medical communication and relationships. 6 Patient education is a key patient engagement intervention for supporting and enabling children, adolescents and young adults to manage their lives with illness challenges. [7][8][9] As others have argued, 10,11 children and adolescents who are living with long-term health conditions want to gain more knowledge about their illness and its consequences for their everyday life. Many studies also report that young people do not have sufficient knowledge of the transition from child to adult health care. 10,12 There is a great variety in how patient education interventions are being offered to children, adolescents and young people, and they are often described as complex interventions. 13 They can be given to groups or to individuals alone, and they can be led by health-care providers or laypersons. 14 Group-based patient education programme, both disease-specific and general approaches, has been considered an important part of health promotion politics in several Western countries and as being essential for chronic illness self-management. [15][16][17] Describing and evaluating the content and impact of how patient education interventions can help to pave the way towards more efficient interventions. A few reviews provide evidence that patient education interventions have been beneficial for children and adolescents with asthma, 2,18,19 diabetes, 9,20 cancer, 11 physical disabilities 3 or across diagnoses (general paediatric care). 10 To capture the health economic aspects, one separate scoping review on the health economic impact of patient education interventions has been conducted and published in 2018. 21 These four scoping reviews on impact of patient education interventions have followed the same methodological framework [22][23][24] and are reporting on similar research questions regarding evaluation of patient education interventions targeting different kinds of participants.
As described earlier in the two published reviews 14,21 in this project, research on the effects of patient education interventions is a relatively new field. To gain a comprehensive overview of the published literature, the research questions were best answered by including different study designs. Thus, scoping review was considered appropriate, also for the current review. Scoping reviews "aim to rapidly identify the key concepts underpinning a research area and the main sources and types of evidence available, and can be undertaken as stand-alone projects in their own right, especially where an area is complex or has not been reviewed comprehensively before." 25 This review followed the five-stage framework proposed by Arksey and O`Malley 22 and further refined by Levac et al 23,24 The following specifications were considered relevant for this scoping review: • Population: target population includes children, adolescents or young adults between the age of 0 and 25 who are living and coping with any type of chronic illness and/or impairment loss.
• Intervention: any kind of face-to-face patient education intervention aimed at supporting self-management, and optimizing health and well-being, led by health-care professionals and/or lay participants.
• Comparisons: usual care/treatment, different types of interventions or no comparisons.
• Outcomes: any of a range of different types of impacts and outcomes related to social, health, psychological, health economic or behavioural aspects.
We have conducted systematic searches in the following electronic databases from 01 January 2008 to 01 February 2018: MEDLINE, EMBASE, PsychINFO, AMED, CINAHL, SweMed+, ERIC and Cochrane Library Online. The literature searches have followed the PICO principles combined with and "OR" within group and subsequently combined with an "AND" between groups. We have used a wide variety of terms in the database thesaurus in addition to free text/key word method: • Participants: children, adolescent, youth, paediatric, young people, young adults.
• Intervention: self-management programme/education/group, group support programme, learning and mastery course, patient education, patient education course/programme/intervention, patient engagement, peer support, group intervention, groupbased education/programme.
We only included studies published in English, Norwegian, Swedish or Danish in peer-reviewed journals. The studies were required to have investigated: the impact or effects over time (a) of individual-and/or group-based patient education interventions (b) for children, adolescents and/or young adults living with any type of chronic illness challenges (c). Interventions based mainly on the use of technology were excluded. A different search strategy would have been required in order to capture the full scope of such studies.
All the members of the study group were involved in the discussions of the search strategy, and our discussions helped clarify the inclusion and exclusion criteria for this review. A broad search in all the relevant databases was conducted, with no restrictions. The search of the online databases yielded 7216 articles, and 7049 of these articles were excluded because they did not meet the inclusion criteria ( Figure 1). The remaining 167 articles were obtained in full text and read by the first author and one co-author. Of these, 98 articles were excluded, because inclusion criteria were not met. There were few disagreements about article inclusion, and these were resolved by discussion in the study group to reach consensus. As is frequently seen in research on patient education interventions tailored to adult patients, 14 the interventions for children, adolescents and young people were often poorly described. In addition, interventions with similar-sounding names could be very different in content. Therefore, every intervention was screened before inclusion, and 59 were excluded because the aim or content of the patient education intervention did not meet the criteria. A final total of 69 articles were included for analysis in this review.
Information about study characteristics, participant characteristics, descriptions of interventions, methods and results was collected on data extraction forms and reported separately for each study in an evidence summary The research included in this study showed significant variation in type of intervention, design and outcome measures. The 69 study results comprising the research material under scrutiny were compared according to the type of patient education intervention, diagnosis and type of outcome measured in order to find patterns and similarities. The data summarization was mainly done by two of the authors (US and MH) and subsequently validated by all co-authors.

| RE SULTS
In this scoping review, 69 research-based studies have been included. The presentation of the results is organized according to the main questions addressed in this review.

| Participant characteristics
A total of 15 124 participants were included in the studies for this scoping review ( Table 2). The mean age of children and adolescents was 12.1 years (17 studies did not report mean age); 31/69 studies reported ethnicity, with the mean of 46.5% white participants.

| Classification of chronic condition
A breakdown of the 69 studies by chronic condition is provided in Table 3. The largest number of studies included in this review focused on asthma (30/69), followed by diabetes (15/69).

| Characteristics of the patient education interventions
The interventions had diverse origins, aims, target groups, settings and number of modules and were delivered by different health-care personnel and/or peers. This is described in detail in Supporting Information Table S1. The interventions in these studies were faceto-face patient engagement interventions aimed at helping children,

| Characteristics of methods for evaluation
The studies included in this review have used a wide range of different outcome measures. Outcomes concerning disease management and coping, knowledge about conditions and treatments, symptom severity, self-efficacy, self-management behaviours, empowerment, self-esteem and health economy were frequently measured. The health economic evaluations were measured in terms of hospitalization, use of urgent and preventive health services and number of days absent from school/ college or work. Table 4 presents all the validated outcome measures and gives references to the primary source and/or validation studies.
The table also shows whether the outcome measure is typically associated with a specific diagnose or is used across diagnoses. Only one study had empowerment as one of several outcome measures, and this study found no changes in empowerment. 49 Four studies measured strengths and difficulties by using a behavioural screening tool for psychopathology and adaptation. One study found that, when compared to the control group, participants in the intervention group became more aware of their strengths as well as the difficulties connected to their diagnosis. 77 One study was unfortunately underpowered and found no differences. 73 One of the two studies that compared cognitive behavioural therapy-based intervention with psycho-education failed to find significant differences between the groups. 82 The second study, however, found significant changes in the cognitive behavioural therapy group controlled for baseline scores. 44 Self-esteem was investigated in three studies. Two of these studies found increased self-esteem after participation in group programmes for adolescents with type 1 diabetes 78 or mental health challenges. 83 However, the third study, investigating a one-on-one intervention for young people diagnosed with psychosis, did not detect statistically significant changes. 35 Two studies had measured the level of patient engagement in their health. The interventions investigated in these studies did not affect activation over time. 69,72 One study had measured differences in social support and found that intervention participants gained significantly more social support when compared to participants receiving ordinary care. 78 One study investigated mindfulness changes and found significant higher mindfulness scores among adolescents in the intervention group compared to those in the control group. 83

| Health-care utilization
Of the studies dealing with patient education interventions, 14/17 studies resulted in beneficial effects as measured by one or several health economic outcomes. 31

| Perceptions of participation-results from qualitative studies
The five studies with a qualitative approach had explored how children and adolescents who were living with diabetes, asthma, stressrelated problems or cancer experienced participating in patient education interventions. 1,40,54,93,94 Overall, the studies showed that by sharing experiences, participants had learned from each other and attained new insight. They also learned through interaction with educational material and from health-care personnel. Adolescents in one study described that the most challenging part was deciding to sign up for the course because it required them to admit to themselves that they needed the course. 94 The studies showed that sharing experiences with peers spurred meaningful learning experiences and an empowering process. Children and adolescents found it easier to talk about their diseases and to share their thoughts and feelings about living with health challenges with family members and others after participating in patient education interventions. 1,54,93,94 In general, children and adolescents experienced alleviation, comfort and a feeling of hope when realizing that there are others struggling with the same issues. 1,94 And that's what it's about, meeting in a group: where everyone has the same problem, then everybody dares to raise issues. That's really great. Nobody judges anybody, no way. 94 p. 8 Another important result from these five studies was that participants gained insight and concrete knowledge about the disease, its symptoms and potential causes, and how they could manage all the daily illness-related challenges. For example, they learned how to manage symptoms and to be more aware of triggers and stress responses, how to take medication and cope with side-effects, or other difficult situations and problems. Adolescents taking part in a stress management course found it useful to learn and understand how physical discomfort is highly related to stress in daily life. 94 Participants in all five studies had learned concrete problem-solving skills. How these skills and knowledge could be used in everyday life and activities was verbalized in the groups. Despite the perceived benefits of participating, after completing the courses some found it hard to remember what they learned, making it difficult to sustain a change in behaviour over a long period of time. These participants suggested including re-education as an additional component in forthcoming courses, also because certain topics would become more significant when they got older. 93

| Strengths and limitations
This study shares the limitations that are inherent to scoping reviews in general. 14,21 Balancing breadth and depth of analysis is challenging, and a further complication lies in synthesizing studies with different designs and methods in the same review. 95 In this review, we have included studies on patient education interventions for children, adolescents and young adults with any type of long-term illness challenges. We wanted to capture as many relevant studies as possible; therefore, we used a large number of synonyms in our searches in the databases. Nonetheless, the list of search terms was neither complete nor exhaustive. Since we had a broad definition of patient education interventions, we could include a wide range of interventions. Similar to our earlier reviews on patient education, 13,20 the interventions in this review also varied in terms of setting, theoretical basis, target groups, modules, duration and personnel/lay participants. In addition, the components of "ordinary care" or "waitlist controls" were often not described.
Chronic illness in children affects daily functioning in the whole family. Some of the interventions in the included studies involved parents, but parental outcomes are not included in this review.
Another limitation we also found in our previous reviews on patient

| Recommendations for future research
Although progress has been made in understanding the effects that can be achieved from patient education intervention for children, adolescents and young adults, much is yet to be learned. We need more knowledge on the effect of participation over time, This review highlights the need for a comprehensive approach in evaluating patient education interventions tailored to children, adolescents and young adults. As is evident from Table 4, the ways of measuring outcomes differ greatly. We need more knowledge about how we can evaluate impact, both for outcomes with standardized measurements, and on how we can evaluate process and subjective experiences from participating in patient education interventions.
More consistent use of standardized measurements would also facilitate comparing interventions internationally.
There is a paucity of research on psychological and emotional experiences of children, adolescents and young adults becoming more actively involved in improving their own health. Based on the results from this review, we need more insight into the psychosocial and subjective experience for children, adolescents and young adults, and more knowledge about factors that may sustain or hinder engagement.

| Implications for practice
The findings from the present scoping review give important input to political decision makers and health administrators. Most importantly, patient education interventions targeting children, adolescents and young adults can reduce the cost of care and improve the levels of physical activity, BMI and blood glucose control. Moreover, the participants experience beneficial effects owing to less symptom distress and improved knowledge.
This review supports the utility of patient education interventions that employ behavioural strategies tailored to the developmental needs of children, adolescents and young adults with different cultural backgrounds. Such interventions should be made available to a broader range of children, adolescents and young adults who are living with health challenges.

ACK N OWLED G EM ENTS
Thanks to Camilla Lyngen for useful input in the planning of this study and to Janet Holmén for language editing. We would also like to thank all our colleagues for their support in this work, with special thanks to Siw-Anita Bratli, Ann Britt Sandvin Olsson, Ann-Helen Henriksen, Kari Hvinden, Cecilia Sønstebø, Siw Merete Paulsen and Hilde Blindheim Børve.

CO N FLI C T O F I NTE R E S T
There are no financial or other ties involved in the present work that might lead to a conflict of interest.