Transition from child to adult health services for young people with cerebral palsy in Ireland; implications from a mixed-methods study

Introduction

Cerebral palsy (CP) is one of the most prevalent disabling conditions among children worldwide¹. Children with CP require transfer from child-centred to adult-oriented healthcare. In Ireland, this typically occurs between the age of 16 and 18 years. Transfer is the event when responsibility for healthcare shifts from a child to adult healthcare provider. After transfer to adult-oriented healthcare, many adults with CP experience difficulty navigating services², reduced access and visits to specialist services, and a decrease in use of rehabilitation services^2,3. Poor preparation for this transfer may contribute to the deterioration in function and development of chronic conditions^4,5.

While the term ‘transfer’ refers to the actual event of moving from child-centred to adult oriented healthcare, transition is defined as ‘the purposeful, planned process that addresses the medical, psychosocial, educational and vocational needs of adolescents and young adults with chronic medical and physical conditions as they move from child-centred to adult-oriented healthcare systems’⁶. Although guidance to support transition has been published in several countries^7–9, there is still evidence that young people with CP and parents are not prepared for transition, lack support and information, and struggle to navigate the adult healthcare system³.

To facilitate the implementation of successful transition for young people with CP, it is important to understand the experience of transition from multiple perspectives of young people with CP, parents and health professionals. In this paper, we report the integrated findings from the Ignition study, a mixed-methods study which aimed to examine how transition care is provided to and experienced by young people with cerebral palsy (CP) in Ireland¹⁰. The Ignition study firstly assessed gaps in the current management of transition against best practice from the perspective of service users (young people and parents) and service providers through a quantitative survey. Quantitative findings indicated that many young people did not experience practices that may improve transition experiences and outcomes¹¹. Secondly, the Ignition study explored transition experiences from the perspective of young people with CP, their families and health professionals using semi-structured interviews. Qualitative findings suggested that the experience of transition is impacted by a number of diverse factors at several ecological levels¹². In-depth findings from quantitative and qualitative studies are separately reported elsewhere^11–13. The purpose of this paper is to enhance the value of the study by integrating quantitative and qualitative findings and to provide implications for practice based on the totality of knowledge generated.

Methods

We conducted a convergent parallel mixed methods study¹⁰ and collected quantitative and qualitative data from young people with CP aged 16–22, parents, and health professionals. We used convenience and snowball sampling. We shared information about the quantitative and qualitative components of the study through three national organisations that provide health and social care services to people with CP, disability officers in higher education, special education needs schools, professional bodies, and social media. Participants could choose to take part in either the quantitative or qualitative components of the study, or both. As recruitment occurred through multiple streams, anonymised data was collected via online surveys, and consent was sought separately for each data collection method, it is not possible to identify whether there was overlap between those who took part in the quantitative and qualitative components. Data collection and analysis are described in detail elsewhere^10–13. Data collection and analysis were informed by a framework of nine key transition practices representing best practices, which is published in the study protocol¹⁰ and presented in Box 1. These transition practices were based on a research programme conducted in the UK¹⁴. the UK NICE guideline on transition⁷ and national strategies relating to children and neurodisability in Ireland^15–18.

Quantitative data were collected using two questionnaires completed by 75 young people and/or parents and 108 health professionals¹¹. Young people had the option of completing the questionnaire alone, or with support from a parent. In instances where it was not possible to obtain the young person’s perspectives, parents could complete the questionnaire on their behalf. This was explained in the introduction to the questionnaire where it stated young people were encouraged to complete it with a parent, family member or carer, but could complete it alone or a parent or carer could complete it behalf of the young person if they were unable to. The questionnaire were developed by the research team in collaboration with and piloted by young people, parents, and health professionals. Minor changes were made to the in response to feedback from the piloting phase. We collected data on socio-demographic and condition-specific data, and the experience or provision of transition practices. Questions regarding the transition process were based on the framework of nine key transition practices representing best practices¹⁰. Questions in the young person/ parent questionnaire mirrored those in the health professional questionnaire. For example, young people were asked if they had a named worker to help with the transition process, while health professionals were asked whether their service ensured that a named worker was provided. Descriptive statistics (e.g. mean, SD) were used to describe data as appropriate.

Qualitative data were collected using in-depth semi-structured interviews with 13 young people, 14 parents, and 27 health professionals¹². Young people and parents had the option to participate in interviews together or separately. For young people over 18 years, interviews were organised based on the young person’s preference to participate alone or with a parent. All young people under 18 years were interviewed together with a parent or guardian. The framework of key transition practices was used to develop the interview topic guides, and interview questions were thus similar to those asked in the questionnaires. Separate topic guides were developed for service users and service providers. These were piloted by young people, parents, and health professionals. Indicative questions are included in Table S1. Data were analysed using the Framework Method.

This study obtained approval from the research ethics committee of the Principal Investigator’s host institution and the research ethics committees of two organisations that supported recruitment (Royal College of Surgeons in Ireland's Research Ethics Committee, REC201911010, 6^th February 2020; Enable Ireland Research, Ethics and Quality Committee, RA72JR2020, 16^th September 2020; Central Remedial Clinic Research Ethics Committee, 73001, 9^th July 2020. Written informed consent was not obtained from those who completed the online questionnaire because it was anonymous and completion indicated consent. However, before completing the anonymous online questionnaire, participants confirmed they had read and understood the information leaflet, knew they could withdraw at any time, and were happy to complete the questionnaire. Where a person completed a paper questionnaire, they provided written informed consent. Where the person was under the age of 18 years, their parent or guardian additionally provided written consent for their child to participate. Written or verbal informed consent was obtained from young people, parents, and health professional to participate in interviews, as approved by the ethics committees.

Results

We outline integrated findings for each transition practice according to whether there was complementarity, dissonance, or silence between quantitative and qualitative findings.

Implications

Quantitative and qualitative findings from the Ignition Study primarily offered complementary information that frequently indicated transition practices were not adequately met. Qualitative findings served to elucidate the reasons why young people’s experience of transition was poor and provide some explanation for differences between young people’s and health professionals’ perceptions of transition. The integrated findings lead to some overarching implications, which were further informed by details of individual studies and discussions with our advisory groups of health professionals, young people, and parents. Implications are discussed in the context of current research. Based on the implications identified, a number of recommendations have been developed. Recommendations are presented alongside relevant implications in Table 2.

     1. Tailoring parent involvement requires intentional actions to enable parents and young people to adapt to changing roles.

Involving parents in their child’s healthcare at a level that is appropriate to young people and parents should be a priority during adolescence because it is associated with improved outcomes in adulthood²⁰. This requires organisations to adopt developmentally appropriate healthcare (DAH), which meets the biopsychosocial needs of young people, and a change in how health professionals engage both young people and parents²¹. Our findings indicate that parents need education and support from professionals and peers throughout their child's life to understand strengths-based ideas about health and development, and to learn how to adapt communication and collaboration with health professionals according to their child’s development. Parents and health professionals also have to concurrently provide young people with the time and space to build confidence to self-advocate, learn about their condition and healthcare, and practice communicating with health professionals. The mind and skill set of staff who are not ready to introduce DAH in both child and adult-oriented healthcare settings acts as a barrier²¹. Organisations must create opportunities and develop capability among health professionals to tailor how they involve parents by supporting changes in organisational processes such as longer appointment times and introducing protocols and training. An organisational-wide change to support DAH requires formal support at senior executive and board level, planning that engages senior managers in both child and adult services, and an organisation wide strategy for training²².

     2. A standardised transition pathway is required for all young people with CP across organisations, developed in collaboration with young people and parents.

A transition pathway should consist of a phased and systematic approach to transition, begin at a consistent age across organisations, include assessment of transition readiness and signposting, and be tailored to meet the needs of individuals, while ensuring that all young people and parents are automatically included in the pathway regardless of their perceived need. Each young person should be assigned a named worker with knowledge of the transition pathway to oversee the process. This person or people require access to standardised information and training to ensure consistency in young people’s and parents’ experience. Lack of standardised national practice regarding the age of transfer and an absence of written transition protocols is not unique to CP in Ireland²³. Variation in practice, resulting in inequity of care to young people, has also been reported for young people with diabetes, cystic fibrosis and congenital heart disease in Ireland²⁴.

Organisations need to create a dedicated role or provide several health professionals with dedicated time within their existing role to ensure that all young people have a named worker for transition and that this role is sustainable. In addition, one person at a senior level within organisations should have responsibility for implementing the transition pathway, advocating for evidence-based transition practices, and identifying and developing organisational processes, supports and training required to deliver organisation-wide DAH for young people. However, transition is often not a priority in health systems, which translates to resource limitations and lack of organisation-wide implementation of best-practice²⁵. This may be a significant barrier to the development and implementation of a standardised transition pathway. Without formal support from management, a few enthusiastic and experienced individuals will continue to provide transitional care in isolation, perpetuating inequities in skills and experience across staff and inequity of care to young people²⁴.

     3. Provision of information to all young people, families and health professionals should be provided in a collaborative and phased approach that starts well before transfer.

A standardised approach to providing information that addresses a range of topics is needed to ensure that all young people receive information, not just those who ask for it or have a particular type or severity of impairment. Although health professionals may be concerned about the practicality of finding time to complete and discuss transition plans²⁶, a lack of standardised approach results in young people and parents having to advocate for support and source information, which may or may not be accurate. Parents of young people with cystic fibrosis in Ireland reported similar unmet information needs²⁷. In the UK, only 6% of young people with CP received a written transition plan compared to 26% with diabetes, suggesting unmet information needs is higher for people with CP than those with long-term medical conditions²⁶. Young people and parents identified the need for information about transition, CP, services and supports available in adulthood, education options, living options, mental health supports, managing finances, personal assistants, and rights and entitlements. These were similar to the information needs of people with CP identified in other countries^28,29. Health professionals identified the need for information about a transition pathway and a directory of services that are available to adults with CP. To facilitate information sharing, one organisation needs to take responsibility for producing a reliable and updateable resource to share information and signpost individuals to trusted information.

     4. A common understanding of self-management is needed between young people, health professionals and parents.

Self-management encompasses diverse knowledge and skills that young people want and need to manage their physical and mental health and navigate health services as adults. To effectively support young people to develop self-management behaviours, they must be given knowledge and opportunities to practise skills within routine care in a phased but direct approach to ensure they recognise it as self-management support. When young people are given space to explore their boundaries to develop independence, they must be told that this approach is being taken and when and where to seek support if needed. This may be supplemented by structured programmes to develop skills and knowledge that young people want to manage their physical and mental health in adulthood. Such programmes have shown some evidence in improving self-management, condition knowledge and transition readiness^30,31. To effectively self-manage, some young people may need additional support with self-advocacy, self-esteem and management of emotions. Further, promoting health self-efficacy should be prioritised as a distinct intervention that supports self-management but is not interchangeable with it, given the association between the promotion of health self-efficacy and improved outcomes in adulthood²⁰.

     5. In the absence of health services for adults with CP that take a lifespan approach, there is a need for joint working between child services, adult services, and GP’s to optimise transition.

Meeting the adult team before transfer is associated with improved outcomes in adulthood²⁰. A gradual handover to adult services would facilitate nuanced information transfer about the young person, reassure families of support in adulthood, and allow the young person to connect to their new adult team. At a minimum, young people and parents should receive a joint meeting with professionals from child and adult health services. This requires child and adult health service professionals taking responsibility for transition. In the current context, providing a period of joint working is not possible for most young people with CP because health services available to adults with CP provide a limited service to address acute needs and have different, often narrower, eligibility criteria than children’s services. A study from the UK similarly identified that adult services generally have higher thresholds for accessing services²⁵. Although CP is a lifelong condition, it has often historically been considered to be a paediatric condition, which may also impact the development of policy and provision of services for adults. For example, CP is included in the National Model of Care for Paediatric Healthcare Services in Ireland¹⁸ but not in the National Policy and Strategy for the Provision of Neuro-Rehabilitation Services¹⁶. In Ireland, disability services for children are typically provided by multidisciplinary teams, consisting of professionals with various areas of expertise who thus have the capability to provide support for a broad range of needs. In contrast, there are less available multidisciplinary services for adults with disability, limiting the scope of the support provided³². This lack of equivalent services for people with CP in adulthood may explain why fewer young people with CP have the opportunity to meet an adult team than young people with long-term medical conditions²⁶. Expanding the provision of multidisciplinary supports for adults would require significant dedicated funding. However, this has been identified as a key objective in increasing service capacity in the Irish disability sector³².

Creating health services for adults with CP that take a lifespan approach is essential to enable joint working and optimise transition. In the absence of this, a systematic approach to involving GP’s in transition is required. Further research on the best approach to involving GP’s is required as there is currently limited evidence underpinning primary care interventions to improve transition outcomes³³. Although the GP is the most frequently visited health professional among adults with CP⁴, young people and parents described challenges with obtaining satisfactory care from GP’s following discharge from children’s services. To adequately support adults with CP, GP’s need support to develop a basic knowledge of CP and the ability to work in partnership with adults. Adults with CP must also have sufficient knowledge and skills to work in partnership with GP’s and other professionals in adult health services to optimise the services currently available to them.

Conclusion

Findings from the Ignition study provide insights into how transition may be improved for young people with CP. While findings highlight challenges that are unique to young people with CP or other child-onset physical disabilities, other challenges are applicable to people with different long-term conditions. Although provision of discrete supports such as standardised information, peer support and educational programmes about transition may partly improve the transition experience, systemic changes are required to facilitate the implementation of transitional care that adequately meets the needs of young people with CP. Transition will only be optimal for people with CP if multidisciplinary teams are created to provide proactive rather than reactive healthcare to adults and enable continuity of care from childhood to adulthood. This requires a fundamental shift in the perception of CP from a childhood condition to a child-onset, lifelong condition.

Ethics and consent

This study obtained approval from the research ethics committee of the Principal Investigator’s host institution and the research ethics committees of two organisations that supported recruitment (Royal College of Surgeons in Ireland's Research Ethics Committee, REC201911010, 6^th February 2020; Enable Ireland Research, Ethics and Quality Committee, RA72JR2020, 16^th September 2020; Central Remedial Clinic Research Ethics Committee, 73001, 9^th July 2020. Written informed consent was not obtained from those who completed the online questionnaire because it was anonymous and completion indicated consent. However, before completing the anonymous online questionnaire, participants confirmed they had read and understood the information leaflet, knew they could withdraw at any time, and were happy to complete the questionnaire. Where a person completed a paper questionnaire, they provided written informed consent. Where the person was under the age of 18 years, their parent or guardian additionally provided written consent for their child to participate. Written or verbal informed consent was obtained from young people, parents, and health professional to participate in interviews, as approved by the ethics committees.