Transition from child-centred to adult-oriented healthcare systems for young people with neurodisability: a scoping review protocol

Background: The transition from child-centred to adult-oriented healthcare is a challenging time for young people with neurodisability. As the prevalence of neurodisability increases, greater numbers of young people will eventually transfer to the adult healthcare system. While there is a growing recognition of the importance of providing quality, transitional care, little is known about how to manage and optimise this process for young people with neurodisability. The objective of this scoping review is to examine and map existing literature related to the transition from child-centred to adult-oriented healthcare systems for young people with neurodisability. Methods: Systematic literature searches of OVID MEDLINE, EMBASE, PsycINFO, CINAHL, Cochrane Library and Web of Science will be conducted from inception to present. A structured iterative search of grey literature will be conducted. This review will consider all study designs examining the transition from child to adult health services in neurodisability. Two reviewers will independently screen each retrieved title and abstract and assess full-text articles against the inclusion criteria to determine eligibility. Data will be extracted and synthesised quantitatively and qualitatively. The process and reporting will follow PRISMA-ScR guidelines. Conclusion: This review will provide a broad and systematically mapped synthesis of the extent and nature of the available published and unpublished literature on transition from child-centred to adult-oriented healthcare systems in neurodisability. The results will be used to determine gaps in the current evidence base in order to prioritise areas for future research.


Introduction
Neurodisability describes congenital or acquired long-term conditions that are attributed to impairment of the brain and/or neuromuscular system and create functional limitations 1 . Neurodisability encompasses conditions characterised by physical, intellectual, behavioural or sensory impairments, such as cerebral palsy, autism spectrum disorders and epilepsy 2 . The prevalence of neurodisability is increasing 3 . Individually, many conditions in this heterogeneous group are rare but when grouped together they are common 4 . Children and young people (CYP) with neurodisability represent between 6 and 9% of the general population 5-7 . Advances in medical management and treatment mean many CYP with neurodisability are surviving to need care as adults 8-10 .
The transition from adolescence to adulthood is associated with profound physiological, psychological, and social changes 11 as young people orientate towards a greater degree of independence in many concurrent areas including relationships, housing, education and employment 12 . The complexity of this process is amplified for CYP with long-term conditions who are simultaneously negotiating the developmental process of adolescence and independence in managing a long-term condition 13 . Adaptation from a supported environment to one of individual responsibility and independence in self-advocacy and management during this developmental phase is particularly challenging for individuals with neurodisability who may have communication, physical or learning difficulties, higher rates of comorbid health problems 1 , and who utilise healthcare services more intensively than their peers 14,15 .
The term transition has been adopted to describe the complex and continuous process of preparing CYP with long-term conditions to move from child-centred to adult-oriented healthcare systems 16 . Transition is therefore distinct from transfer, the latter being considered as a one-time event when the young person moves from a child-centred to adult-oriented healthcare setting 17 . The transition process aims to maximise potential and lifelong functioning through the provision of cohesive and continuous, developmentally appropriate healthcare as the individual moves to adult health services 18 . Ideally, this process is coordinated, comprehensive, and patient-centred spanning adolescence to adulthood 18 , preferably beginning when the individual is 13-14 years of age and continuing until they are capable of taking full responsibility for their health 19 . However, the transition process is frequently turbulent and experienced negatively by CYP, their families and caregivers 20 . Obstacles to successfully implementing transition include funding limitations, lack of continuity and coordination between child and adult services 21-23 and limited training opportunities and specialist expertise in the adult system 24-27 . In addition, there are significant changes to healthcare provider relationship and reduced support levels in the adult environment 28 .
Suboptimal transition to adult health care is associated with diminished treatment adherence and interruption or loss to follow-up 29 . This discontinuity leaves CYP vulnerable to adverse health consequences including functional decrements, medical complications and a heightened risk of hospital admissions 30-32 as well as poor psychological, social and vocational outcomes 12,33-36 . Preventing such declines for CYP with neurodisability is a healthcare priority 19 . This is reflected in the increasing volume of research examining transition and transfer of care in neurodisability in recent years.
To date, literature has explored effective and efficient features of transition practices and programmes 37-40 , explored the perspectives of healthcare professionals 25,26 and experiences of young people and their families 34,41-44 and the impact of transitional care on measurable outcomes 45-47 . A number of scoping reviews have explored transition among CYP with physical 48 , mental health 49,50 and endocrine, neurological and gastrointestinal conditions 51,52 . Evidence from these reviews may not be generalizable to neurodisability due to the range and complexity of these conditions and the greater need for service coordination 53 . A scoping review protocol by Bogassian and colleagues is the only one available looking specifically at transition and neurodisability 54 . However, their review focus considers the ethical issues encountered in transition programmes only. Young people with neurodisability constitute a unique and growing population to whom a well-managed and executed transition process can be valuable. Therefore, a comprehensive synthesis of the literature on transition for CYP with neurodisability is needed to consider what is known in order to guide future research and improve transition care.
A scoping review will be undertaken to explore existing literature relating to transition for CYP with neurodisability. This methodology is appropriate as it will provide a comprehensive map of key concepts underpinning the research area and a substantial overview of the types and sources of evidence available in the current body of literature 55 . It will clarify the aspects of transition which have been the focus of research initiatives to date and identify any knowledge gaps or research deficits that exist within the field that require further research 55,56 .

Design
The methodology for this review draws on the five-stage framework outlined by Arksey and O'Malley 55 and more recent refinements to the methodology proposed by Levac et al. 56 and the Joanna Briggs Institute (JBI) 57 . The optional sixth stage, 'consultation with relevant stakeholders' will not be included as part of this review. This protocol follows the Preferred Reporting Items for Systematic reviews and Meta-Analyses extension for Scoping Reviews (PRISMA-ScR) guidelines 58 to ensure rigour in reporting.

Stage 1: Identifying the research questions
The primary aim of this review is to determine and describe the extent and nature of available evidence addressing transition for CYP with neurodisability and to identify gaps in the existing literature.
Several secondary questions will guide the subsequent stages of the scoping review. 7. Which theories, models or frameworks have been used to inform transition in neurodisability?

Inclusion criteria
The inclusion criteria for this scoping review will be guided by the population, concepts and context (PCC) approach 56 . Inclusion and exclusion criteria are summarised in Table 1.

Participants.
A multitude of definitions for neurodisability exist in the literature. Historically the group of conditions encompassed by the term neurodisability were interchangeably referred to as neurodevelopmental disorders 59 or neurodevelopmental disabilities 60 . For uniformity, the term used throughout will be neurodisability. In the context of this review, we will define neurodisability as "A group of congenital or acquired long-term conditions that are attributed to impairment of the brain and/or neuromuscular system and create functional limitations. A specific diagnosis may or may not be identified. Conditions may vary over time, occur alone or in combination, and include a vast range of severity and complexity" 1 .
For the purpose of this review we will include people who experience disturbances of movement, cognition, communication or emotion and behaviour. Studies including male and female CYP with neurodisability will be included. Studies that focus on families, caregivers, health care providers, programme managers and policymakers involved in the transition process will be included.

Concept.
The concept examined by this scoping review is the transition from child-centred to adult-orientated healthcare systems. In the context of this review we will define transition as "a purposeful, planned process that addresses the medical, psychosocial and educational/vocational needs of adolescents and young adults with long-term physical and medical conditions as they move from child-centred to adult oriented healthcare systems" 16 .
Context. This scoping review will consider studies on transition that have been conducted in any setting such as hospitals, healthcare settings, acute care, primary care, special care, home-care or the community. The context will not be limited to specific geographic location.
Types of evidence sources. The review will consider studies of any design that address transition including qualitative, quantitative and mixed-methods methodology. Quantitative studies will include both experimental (e.g., randomised trials, nonrandomised trials) and observational (e.g., cohort, crosssectional) study designs. Case series and individual case reports will also be included. Qualitative studies will include designs such as grounded theory, ethnography, phenomenology, action research and qualitative descriptive. Text and opinion papers will be considered for inclusion if they are published in peerreviewed journals. In addition, all types of reviews (e.g., systematic reviews, narrative reviews) will be included. Grey literature will also be considered for inclusion in the review.

Stage 2: Identifying relevant studies
A comprehensive search strategy was developed in consultation with an information specialist. To develop the search strategy, an initial limited search was conducted in OVID MEDLINE and CINAHL to identify articles relevant to the topic area. Key words and index terms were identified from the title and abstract of relevant articles and used to inform the search strategy. Search terms included key words and index terms relating to neurodisability, transition and young people. The search strategy for OVID MEDLINE can be found in the online supplementary material (see Extended data 61 ). It will be modified as necessary for the other databases. We will search the following electronic databases from inception to the present date: OVID MEDLINE, EMBASE, PsycINFO, CINAHL, Cochrane Library and Web of Science. We will also perform targeted searches for grey literature through OpenGrey, BASE (Bielefeld Academic Search Engine) and Google. Finally, the literature search will be supplemented by hand searching reference lists of included reports. Only reports published in English will be included. Literature searches will be completed by an information specialist.

Stage 3: Study selection
All identified citations will be collated and uploaded into End-Note (Clarivate Analytics, PA, USA), and duplicates removed.
Two reviewers will independently screen the titles and abstracts of the literature search results considering the eligibility criteria for the review using Rayyan QCRI 62 . Full texts of potentially eligible studies will be obtained and reviewed by two reviewers independently. Prior to commencing the screening process, two reviewers will conduct a calibration exercise to ensure reliability in correctly screening for inclusion. It will entail independently screening a random sample of the included citations by each reviewer. If low agreement is observed between the reviewers, eligibility criteria will be modified. Discrepancies will be resolved by discussion between reviewers. A third reviewer will be consulted if consensus is not achieved between reviewers.
Stage 4: Charting the data Data charting will be conducted using a standardised form, developed from the JBI data extraction tool 57 . Two reviewers will independently pilot the form on a random sample of included reports. If poor agreement is found, the data extraction form will be revised iteratively and the training exercise will be repeated 55 . The data charted will include specific details about the population, concept, context, study methods and key findings of significance to the scoping review objective and questions. Authors of papers will be contacted to request missing or additional data, where required. A quality appraisal will not be undertaken in keeping with guidance on scoping review conduct 63 .
Stage 5: Collating, summarising and reporting the results Results of the literature search and study screening process will be presented in a PRISMA-ScR flow diagram 58 . Charted data will be synthesised quantitatively and qualitatively. For example, summary statistics will be used to describe the current volume, yearly distribution, countries of origin, sample characteristics and methodological design. Key concepts will be summarised using descriptive content analysis. Results will be presented in tabular, graphic or diagrammatical formats according to key findings and knowledge gaps.

Study status
At the time of publication of this protocol, database searches have been completed.

Discussion
Given the challenges experienced by CYP with neurodisability during the transition to adult health care, there is an urgent need to better understand this process. This scoping review will broadly and systematically explore what is known about transition in neurodisability. Findings will be used to identify knowledge gaps to direct future research and provide a foundation for developing research priorities. The findings of the review will be published in an open-source journal, presented at national and international conferences, and shared with clinicians, young people and families through organisations for people with disability.

Data availability
Underlying data No underlying data are associated with this article.

5.
Thanks for the opportunity to comment. I wonder what issues you will find in the search to pin down the research question? There are many unique circumstances that ought to be addressed. There is variability between the neurodisabilities in the health related comorbidities and the earlier these occur, the better they are described and the more dire the outcomes in the event of poor management, the more likely that these conditions will have a transition process in place e.g. spina bifida. Where serious health related problems occur later in the trajectory of a condition e.g. cerebral palsy, the more lacklustre the transition program in my experience.
Success or otherwise is also dependent on the agency of the child which is a subject I have not seen much research about in the disability literature.
Children in state care are a unique problem in transition programs as medical transition can be accompanied by a transition in housing and guardianship.
The adult perspective of graduates of childhood conditions is also unpredictable and influences the willingness of adult engagement. Severe disability can be met with the automatic presumption of "palliation" and with it an assumption that death is "imminent" despite the child already having lived for 17 or so years, once services are sought in the adult sector and levels of care can be set on the basis of these value judgments. While it may not come into the scope of your questions, I wonder too whether CYP with congenital disabilities are not differentiated from adults with acquired disabilities who have lost skills they once had. I wonder whether this too affects the interest level in adult engagement with paediatric neurodisability graduates. There is a need to look into these clinically relevant scenarios witnessed in practice but not (yet?) in the literature.

Is the rationale for, and objectives of, the study clearly described? Yes
Is the study design appropriate for the research question?