Keywords
neurodisability, neurodevelopmental disorders, transition to adult care, transitional care, young adult, adolescent, scoping review
neurodisability, neurodevelopmental disorders, transition to adult care, transitional care, young adult, adolescent, scoping review
Neurodisability describes congenital or acquired long-term conditions that are attributed to impairment of the brain and/or neuromuscular system and create functional limitations1. Neurodisability encompasses conditions characterised by physical, intellectual, behavioural or sensory impairments, such as cerebral palsy, autism spectrum disorders and epilepsy2. The prevalence of neurodisability is increasing3. Individually, many conditions in this heterogeneous group are rare but when grouped together they are common4. Children and young people (CYP) with neurodisability represent between 6 and 9% of the general population5–7. Advances in medical management and treatment mean many CYP with neurodisability are surviving to need care as adults8–10.
The transition from adolescence to adulthood is associated with profound physiological, psychological, and social changes11 as young people orientate towards a greater degree of independence in many concurrent areas including relationships, housing, education and employment12. 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 condition13. 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 problems1, and who utilise healthcare services more intensively than their peers14,15.
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 systems16. 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 setting17. 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 services18. Ideally, this process is coordinated, comprehensive, and patient-centred spanning adolescence to adulthood18, preferably beginning when the individual is 13–14 years of age and continuing until they are capable of taking full responsibility for their health19. However, the transition process is frequently turbulent and experienced negatively by CYP, their families and caregivers20. Obstacles to successfully implementing transition include funding limitations, lack of continuity and coordination between child and adult services21–23 and limited training opportunities and specialist expertise in the adult system24–27. In addition, there are significant changes to healthcare provider relationship and reduced support levels in the adult environment28.
Suboptimal transition to adult health care is associated with diminished treatment adherence and interruption or loss to follow-up29. This discontinuity leaves CYP vulnerable to adverse health consequences including functional decrements, medical complications and a heightened risk of hospital admissions30–32 as well as poor psychological, social and vocational outcomes12,33–36. Preventing such declines for CYP with neurodisability is a healthcare priority19. 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 programmes37–40, explored the perspectives of healthcare professionals25,26 and experiences of young people and their families34,41–44 and the impact of transitional care on measurable outcomes45–47. A number of scoping reviews have explored transition among CYP with physical48, mental health49,50 and endocrine, neurological and gastrointestinal conditions51,52. 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 coordination53. A scoping review protocol by Bogassian and colleagues is the only one available looking specifically at transition and neurodisability54. 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 literature55. 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 research55,56.
The methodology for this review draws on the five-stage framework outlined by Arksey and O’Malley55 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) guidelines58 to ensure rigour in reporting.
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.
1. What is the current volume and yearly distribution of evidence on transition in CYP with neurodisability?
2. What types of studies on transition in CYP with neurodisability have been conducted (e.g. quantitative, qualitative or mixed-method methodologies)?
3. In which settings and geographical contexts have previous transition studies in neurodisability been conducted?
4. Which conditions have been included in previous studies?
5. How has the concept of ‘transition’ been defined, operationalised and measured in the literature in relation to people with neurodisability?
6. What involvement did CYP and their family/caregivers have in the design, conduct and dissemination of the research?
7. Which theories, models or frameworks have been used to inform transition in neurodisability?
The inclusion criteria for this scoping review will be guided by the population, concepts and context (PCC) approach56. 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 disorders59 or neurodevelopmental disabilities60. 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, non-randomised trials) and observational (e.g., cohort, cross-sectional) 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 peer-reviewed 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.
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 data61). 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.
All identified citations will be collated and uploaded into EndNote (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 QCRI62. 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.
Data charting will be conducted using a standardised form, developed from the JBI data extraction tool57. 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 repeated55. 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 conduct63.
Results of the literature search and study screening process will be presented in a PRISMA-ScR flow diagram58. 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.
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.
Open Science Framework: Transition from child-centred to adult-oriented healthcare systems for young people with neurodisability: a scoping review protocol. https://doi.org/10.17605/OSF.IO/DX8ZF61.
This project contains the following extended data:
OVID MEDLINE search strategy.pdf
Draft data charting_data extraction template.pdf
PRISMA-P checklist.pdf
Extended data are available under the terms of the Creative Commons Zero "No rights reserved" data waiver (CC0 1.0 Public domain dedication).
Is the rationale for, and objectives of, the study clearly described?
Yes
Is the study design appropriate for the research question?
Yes
Are sufficient details of the methods provided to allow replication by others?
Yes
Are the datasets clearly presented in a useable and accessible format?
Not applicable
Competing Interests: No competing interests were disclosed.
Reviewer Expertise: paediatric cerebral palsy, spinal cord pathology, acquired brain injury
Is the rationale for, and objectives of, the study clearly described?
Yes
Is the study design appropriate for the research question?
Yes
Are sufficient details of the methods provided to allow replication by others?
Yes
Are the datasets clearly presented in a useable and accessible format?
Yes
Competing Interests: No competing interests were disclosed.
Reviewer Expertise: Education, health education, educational and life transitions, systematic literature review, creativity, inclusion, voice
Is the rationale for, and objectives of, the study clearly described?
Yes
Is the study design appropriate for the research question?
Yes
Are sufficient details of the methods provided to allow replication by others?
Yes
Are the datasets clearly presented in a useable and accessible format?
Not applicable
Competing Interests: No competing interests were disclosed.
Reviewer Expertise: Health Services Research, Implementation, CYP
Alongside their report, reviewers assign a status to the article:
Invited Reviewers | |||
---|---|---|---|
1 | 2 | 3 | |
Version 1 04 Sep 20 |
read | read | read |
Provide sufficient details of any financial or non-financial competing interests to enable users to assess whether your comments might lead a reasonable person to question your impartiality. Consider the following examples, but note that this is not an exhaustive list:
Sign up for content alerts and receive a weekly or monthly email with all newly published articles
Register with HRB Open Research
Already registered? Sign in
Submission to HRB Open Research is open to all HRB grantholders or people working on a HRB-funded/co-funded grant on or since 1 January 2017. Sign up for information about developments, publishing and publications from HRB Open Research.
We'll keep you updated on any major new updates to HRB Open Research
The email address should be the one you originally registered with F1000.
You registered with F1000 via Google, so we cannot reset your password.
To sign in, please click here.
If you still need help with your Google account password, please click here.
You registered with F1000 via Facebook, so we cannot reset your password.
To sign in, please click here.
If you still need help with your Facebook account password, please click here.
If your email address is registered with us, we will email you instructions to reset your password.
If you think you should have received this email but it has not arrived, please check your spam filters and/or contact for further assistance.
Comments on this article Comments (0)