Priority setting for children and young people with chronic conditions and disabilities

Abstract Background The aim of this project was to identify the top 10 priorities for childhood chronic conditions and disability (CCD) research from the perspectives of children and young people with lived experience, their parents and caregivers and the professionals who work with them. Methods We conducted a three‐stage study based on the James Lind Alliance priority‐setting partnership methods. It comprised two online surveys (n = 200; n = 201) and a consensus workshop (n = 21) with these three stakeholder groups in Australia. Results In the first stage, 456 responses were submitted, which were coded and collapsed into 40 overarching themes. In the second stage, 20 themes were shortlisted, which were further refined in stage 3, before the top 10 priorities being selected. Of these, the top three priorities were improving awareness and inclusion in all aspects of their life (school, work and social relationships), improving access to treatments and support and improving the process of diagnosis. Conclusions The top 10 priorities identified reflect the need to focus on the individual, health systems and social aspects of the CCD experience when conducting research in this area. Patient or Public Contribution This study was guided by three Advisory Groups, comprising (1) young people living with CCD; (2) parents and caregivers of a child or young person with CCD and (3) professionals working with children and young people with CCD. These groups met several times across the course of the project and provided input into study aims, materials, methods and data interpretation and reporting. Additionally, the lead author and seven members of the author group have lived and experienced CCD.


| BACKGROUND
Chronic conditions and disabilities (CCD) affect at least 10% of children and young people worldwide, although some studies estimate a much higher prevalence, 1,2 with prevalence for specific conditions varying across age bands. 3 Living with a CCD can have profound and pervasive impacts on children and their families, including 2-3 times greater odds of academic underperformance, 4 double the likelihood of experiencing psychological distress 5 and disruptions across social, family and financial domains. 6 Further, children and young people with CCD often experience complicated and lengthy pathways to diagnosis and support due to a lack of health literacy and limited health provider understanding coupled with stigmatising attitudes and beliefs. 7 Given the increasing prevalence of these conditions and their pervasive impacts, it is critical that more research is directed towards promoting meaningful outcomes amongst these individuals and their families.
A starting point to achieving this is asking people with lived experience of CCD which research topics are most meaningful to them. Researchers and decision-makers are increasingly valuing stakeholder involvement in defining research priorities due to the many benefits presented. [8][9][10] Stakeholder involvement promotes positive impacts across the entire research and translation pipeline, including the development of user-relevant research questions, greater engagement of research participants and more efficient and consumer-relevant implementation, dissemination and translation of evidence into policy and practice. 8,11 Despite these benefits, research questions and directions defined by researchers do not always reflect the priorities of patients, their families and service providers. [12][13][14] A previous review of 258 research priority-setting studies found that only 19% of studies involved both patients and clinicians, 15 while a review of 83 paediatric chronic disease priority-setting studies found that only 4% involved patients. 16 Odgers et al. 16 recommended that priority-setting for paediatric chronic disease follow a systematic and explicit process to develop a translational research agenda that is meaningful for patients, parents, clinicians and researchers alike. A methodology known as a 'priority-setting partnership' (PSP) has been developed by the James Lind Alliance (JLA) for identifying and prioritising health research questions that matter to patients, their families and clinicians. 17 PSPs bring key stakeholders together to determine the most important unanswered research questions concerning a particular patient group or groups. PSPs have been used to identify stakeholder priorities for research on specific long-term conditions in childhood, including children with neurodisability, 18 cancer, 19,20 asthma, 21 cleft lip and palate, 22 eczema, 23 cystic fibrosis 24 and chronic pain. 25 Prior PSPs have revealed the need for research agendas to encompass a more holistic approach to supporting outcomes for children, young people and families, rather than being restricted to biological and drug intervention research.
These recommendations were echoed in recent work with paediatric chronic conditions groups in Australia 26 and Canada. 27 These studies were valuable in their transdiagnostic approach: many young people with CCD experience significant comorbidity and identifying common priorities can help to improve efficiency and collaboration in research. However, one study 26 only involved a small number of children and young people (n = 3) and parents (n = 19) relative to professionals (n = 51), while in the other, 27 only 22%-29% of the sample had lived experience.
Accordingly, the aim of the current research was to build on this work by seeking input from a larger proportion of young people and parents/caregivers from across Australia. Our goal was to identify the top 10 priorities for research on child and adolescent CCD, from the perspective of young people with CCD, their parents and caregivers and the professionals who work with them.

| Participants
This study recruited Australian participants from three groups: (1) Young people aged 14-25 years with CCD; (2) parents and caregivers with a child under the age of 25 years with CCD and (3) professionals working with young people aged 0-25 years of age with CCD. There were no exclusion criteria regarding types of CCD.

| Procedure
The WA Health Child and Adolescent Health Service Ethics

| Advisory group formation
We recruited three Advisory Groups, comprising (1) 11 young people with CCD; (2) seven parents and caregivers of young people with CCD and (3) five professionals in clinical, advocacy or decisionmaking roles in the child and adolescent CCD field. Advisory Groups met at least twice across the course of the project to discuss the project scope, provide feedback on project methods and support recruitment and translation strategies. Additionally, the research team was led by a researcher with CCD, supported by two research assistants and three workshop facilitators with lived experience of CCD.

| Recruitment
At each stage, participants were recruited via social media and through mailing lists of CCD advocacy agencies and community support groups. Participants who consented to be contacted about the project were emailed as each stage became active. Participants were not required to have been involved in previous stages to join, with additional participants recruited in each stage. Following this open-ended question, participants were prompted to consider whether they had specific questions related to school and learning; relationships; well-being and mental health; promoting acceptance and understanding; patient and family education;

| Elicitation survey
F I G U R E 1 Overview of the child and adolescent chronic conditions and disability priority-setting process. condition management; transition out of child and adolescent services; supporting parents and families; accessing or navigating treatment or support and the future. These prompts were developed in consultation with our Advisory Groups. Participants were then asked to list any other questions that did not fit under a previous heading.
We conducted a content analysis of responses using NVivo. 30 After familiarisation with the data, we conducted primary coding of each question to identify manifest content. Secondary coding was conducted to identify higher-level meanings that could be extracted from each question. This enabled questions to be combined into themes that could be summarised by a single overarching question.
Input from the Advisory Groups was sought regarding the phrasing of the themes and to determine whether these adequately captured the content provided from stage 1. Where survey responses were not framed as questions, they were reframed following dialogue between two of the researchers (A. F.-J-and B. S.) and the Advisory Groups.

| Ranking survey
A second online survey was conducted to establish a shortlist of themes to take to the consensus workshop. Participants were presented with the list of themes following stage 1 (40 in total) and asked to select as many themes as they felt were important to them.
Their selections were carried through to the second step, in which participants were asked to rank questions in order from most to least important. Data were downloaded to SPSS to identify the top 20 most frequently selected and highly ranked questions.

| Online consensus workshop
The final stage of the project involved a 2.5-h online workshop. A summary of the previous stages of the project and an explanation of the aim of the workshop were provided to the group. Participants were divided into smaller 'breakout' groups, comprising a mix of young people, professionals and parents. Each group was facilitated by a member of the research team with experience in the JLA priority-setting methodology and small-group facilitation. Groups had access to their own Google Jamboard, which displayed the top 20 themes on digital sticky notes that could be modified and moved during the discussion.
In the first breakout group session, facilitators led the groups through each theme sequentially, asking for input regarding the wording of the theme, whether themes could be combined further or if any important themes were missing. Following this, all participants returned to the main room, and facilitators were asked to present the outcome of these discussions on behalf of their group. As a whole group, themes were reworded, and a consensus was reached on themes that could be combined. The consensus was considered to have occurred when, following a period of discussion, no further comments or requests for the amendment were put forward after appropriate prompts. In a second breakout group session, facilitators led each group in arranging themes from most to least important. The top 10 priorities were then determined by scoring each question according to its ranking in each group's list. Participants were also asked to provide feedback on the workshop process and top 10 priorities. Specifically, they were asked to identify whether there were any groups that the priorities may not be relevant to and whether there were any ideas for how research might address the top-ranked priority.  Table 3. Professionals were from a range of backgrounds, including occupational therapists, psychologists, rare disease advocates, general paediatricians, paediatric neurologists, paediatric oncologists, nurses, physiotherapists, in-hospital children's entertainers and representatives from CCD advocacy groups.

| Stages 1 and 2
In stage 1, 456 questions were submitted, which were coded and collapsed into 40 themes. A list of the collapsed themes from stage 1 is shown in Table 4; the 20 highest-ranked themes following stage 2 are marked with an asterisk. Following discussion at the workshop, several of these themes were collapsed further into 12 themes, and based on participant feedback, an additional theme was added for a total of 13 themes. The additional theme was 'How can we promote and respect young people's agency and self-advocacy?' At the workshop, participants discussed at length the language used to capture their experience appropriately and respectfully.

| Consensus workshop
The top 10 priorities following the workshop are shown in Table 5.  Abbreviations: CFS, chronic fatigue syndrome; IBD, inflammatory bowel disease; ME, myalgic encephalomyelitis. a Note that some respondents reported more than one condition; proportions are calculated based on the total n for each participant category, but cumulative proportions do not sum to 100. quickly and easily was the second-ranked priority, with improving the diagnostic process ranked third. Individual questions and reflections on these priorities revealed many barriers to access, including lack of availability of specialist services, limited understanding of specific conditions within the available services, long wait times for specialist T A B L E 3 List of 'other' conditions reported by young people and parents/caregivers at stages 1 and 2.

| Workshop feedback
In reflecting on the workshop, participants reported feeling satisfied with the outcomes and enjoying the process. One professional said 'I Condition S1 S2 Condition S1 S2 Functional gut disorder -1 Vision loss -1 Functional neurological disorder -1 Abbreviations: ARFID, avoidant restrictive food intake disorder; POTS, postural orthostatic tachycardia syndrome; S1, stage 1; S2, stage 2.
T A B L E 4 Collapsed themes from stage 1 that were presented for ranking in stage 2 (not listed in order of priority).

No. Theme
A* How can young people with chronic conditions and disabilities access treatment and support quickly and easily? E.g., Reducing waiting times, choosing from a range of options and specialists and having access to services before diagnosis.

B
How can we support young people to communicate about their chronic conditions and disabilities? E.g., Communicating in relationships, when and how to disclose diagnoses and managing online communication.
C How can we support the identity development of young people with chronic conditions and disabilities? E.g., Increasing feelings of acceptance amongst young people and their families, acknowledging young people's feelings of being different from their peers.
D* How can we increase access to research-informed evidence and clinical trials for young people with chronic conditions and disabilities and their families? E.g., Techniques to help health practitioners communicate health information, tailoring information to the young person and family and clinical trial support tools.
E* How can we support the transition of young people with chronic conditions and disabilities from paediatric to adult care? E.g., Knowing which age is best for transition, providing information to prepare young people and their families, providing mental health support and making adult hospitals less scary.
F How can we help parents reduce career disturbance when they have a child with chronic conditions and disabilities? E.g., Due to work missed to attend medical appointments, the need to work part-time or loss of income.
G* How can the social exclusion of young people with chronic conditions and disabilities be minimised? E.g., Education to reduce stigma, programmes to reduce bullying.
H* How can we help young people with chronic conditions and disabilities plan for the future? E.g., Supporting the transition to adulthood.
I* How can young people manage the uncertainty surrounding their chronic conditions and disabilities? E.g., Sustaining hope when the future is unpredictable, managing fluctuating needs and responding to worsening or possible complications of their chronic conditions and disabilities.
J* How can we minimise the impact of chronic conditions and disability on young people's work and study? E.g., Career options and workplace support.
K* How can we support young people to manage their chronic conditions and disabilities at school and university? E.g., Education and training for school staff, increasing flexibility and providing support for catchup.
L How can we reduce the impact of a young person's chronic conditions and disabilities on other children in the family? E.g., the Impact on family dynamics and functioning, the impact on relationships between parents and the balancing of time and resources between children.
M* How can we raise awareness and community acceptance of chronic conditions and disabilities? E.g., Educating the public, teachers, family members, professionals and peers; including education about chronic conditions and disabilities in schools; identifying the best language to use in education.
N How can families and young people find chronic conditions and disabilities advocacy opportunities and organisations to work with?
O* How can we improve the effectiveness of treatments for chronic conditions and disabilities? E.g., Developing our understanding of best management and treatment, working to find a cure and focusing on promoting recovery. while all priorities were relevant to people living with CCD, the order of importance may vary depending on one's age, cultural background or the type of CCD experienced.
To improve the workshop, participants recommended allowing more time and suggested that the workshop be conducted across two separate sessions, given that the target group is prone to fatigue and other symptoms that may limit their participation. It was Reflections on the workshop process also provide valuable guidance for how online priority-setting processes can be improved for young people living with CCD.

| Awareness and inclusion
Prior work with young people living with CCD has highlighted the role of stigma, prejudice and ableist expectations in experiences of disability, inclusion and exclusion. 31,32 Moreover, prior research has found that in some cases, children (including those with disability) actively participate in ableist discourses, 33 including comparing the severity of impairment across different types of illness and disability. 33 years, the link between disability and subjective well-being was almost entirely accounted for by the experience of adversity and relative lack of access to social and economic resources. These findings further indicate the need for future research to consider the interaction between individual and social characteristics of CCD when exploring mental health impacts.

| Strengths and limitations
Promoting children's voices, agency and competency and protecting their rights are critical in childhood CCD research. 45 The current study had both strengths and weaknesses in this regard. The strengths of the research include the involvement of children, young people and family members with lived experience of a broad range of CCD. This brings a diversity of perspectives to the process and helped us to identify priorities that are likely relevant for many children, young people and family members. Relative to prior CCD priority-setting work, we also included a large proportion of young people and parents/caregivers in our sample. This helps to increase confidence that the priorities identified are reflective of those with lived experience. Confirmation of these priorities with an independent sample would help to determine their generalisability.
We also acknowledge that there are many young people who would not have been able to participate in the research activities outlined in this study without additional support. Because we used a convenience sample, we are unable to determine how representative our sample is, or which young people were excluded from the process. Further, intersectionality was not measured in our study and young people with intersectional identities likely face additional challenges and may have different priorities. For example, prior work emphasises the need for specific consideration to be given to promoting access for Aboriginal and Torres Strait Islander families, who frequently experience intersectional disadvantage. 46 Based on recommendations from our Advisory Groups, future research should focus specifically on the priorities of Aboriginal and Torres Strait Islander children and young people with chronic conditions to ensure that their perspectives are appropriately represented. We did not seek to achieve this in the current study, based on advice from our Aboriginal Research and Development Unit that such engagement could not be appropriately achieved within our budget and timeframe.

| CONCLUSIONS
The current priorities, derived from a robust sample of children and young people with lived experience, their parents and caregivers and the professionals who work with them, highlight the interplay between social and individual facets of the CCD experience. They demonstrate the need to consider multiple aspects of CCD impact when designing research that is meaningful to those with CCD and their support networks. Future work should focus on the involvement of those with lived experience in designing research to address these priorities, as well as confirmation of these priorities with independent samples, including specific subgroups.

AUTHOR CONTRIBUTIONS
Amy Finlay-Jones conceived the study with the support of Belinda