Background
Effective transition of adolescents from pediatric to adult health services has become a prominent issue in health care research. As a result of unprecedented advances in medical practice over the past three decades, as many as 50 - 90% of children with congenital or acquired physical disabilities now reach adolescence and live into adulthood [
1‐
6]. For example, in 1995, twice as many newborns with spina bifida survived in the US as compared to 1975 [
3]. Consequently, a new cohort of adults with childhood-onset disabilities and complex chronic conditions has emerged, requiring appropriate transitional care upon their “discharge” from pediatric facilities.
Transition from the pediatric to adult health care system is a complex process that must be addressed in a holistic manner inclusive of medical, psychosocial, educational, and vocational components [
7,
8]. Ideally, health care during the period of transition should be delivered in a coordinated and uninterrupted manner through the provision of developmentally appropriate and comprehensive services [
7]. However, finding appropriate adult care is challenging [
9], as many adult programs have eligibility criteria that focus on adults with new onset disability issues [
10], and there is a general lack of expertise among most adult health care providers related to aging with a childhood-onset disability [
4,
11,
12]. In the absence of an adequate system of care, adolescents with disabilities are often significantly under-serviced as young adults, and many receive no care whatsoever [
6,
13,
14].
Even when links to adult care are established, the process of transition remains difficult. First, there are important differences between the pediatric and adult health care environments, such as decreased family involvement and highly specialized, fragmented care that characterize the adult health care system [
15‐
18]. To succeed in this system, young people need to learn how to manage their own health [
19]. Unfortunately, the pediatric system has not emphasized developing the skills of children with disabilities to enable them to navigate the adult system that requires more self-advocacy and self-determination skills than the pediatric sector, where parents often play this role [
20,
21]. Second, adolescence is a critical developmental phase between childhood and adulthood, characterized by increased socio-cultural turbulence and vulnerability for all adolescents, regardless of disability [
19,
22]. Two of its hallmarks are a search for an identity separate from that of the family of origin and re-defining relationships with adults in parental and caring roles [
23]. These may contribute to tension in the relationships of adolescents with their families and health care providers.
This difficulty in establishing continuity of care from the pediatric to adult system and inadequate transition preparation often results in unfavourable outcomes for young adults with disabilities. Gaps in transition to the adult health system result in poor health outcomes and diminished opportunities to participate as productive members of the community [
6,
24]. Lack of continuity is especially detrimental to populations with demanding and complex health care needs. For example, Young et al. [
14] reported a significant drop in self-reported health status from youth to adulthood in young people with acquired brain injury (ABI), cerebral palsy (CP), and spina bifida (SB). In the absence of community and primary care services, health issues go unmonitored, putting these individuals at further risk for developing preventable secondary complications [
25]. This paradoxically has led to increased utilization of health services (e.g., inpatient hospitalizations) and inappropriate reliance on emergency health services (e.g., use of walk-in clinics and emergency departments) [
10,
14,
26].
Despite much pressure to identify best practices in transition, research has only recently started to move from consensus reports to formal evaluations. The research to-date has focused largely on the need for transitional care [
19] and articulating challenges to implementing effective models of care [
9,
10,
13,
17]. Empirical evidence addressing processes and outcomes of transition remains limited [
27,
28]. The majority of recent papers on transition focus on generating recommendations for supporting transitions, while few qualitative studies and one prospective evaluation provided empirical data [
29]. To our knowledge, only seven studies described in the transition literature on physical disability (SB [
29]) and chronic illness (type 1 diabetes, cystic fibrosis, juvenile arthritis, epilepsy [
30‐
35]) were formal evaluations of transition care that included prospective data collection. Of these, no studies were longitudinal or followed the participants significantly past the transitional period, and three studies were qualitative and thus lacked a comparison group and dealt with patient perceptions rather than provided measures of transition outcomes [
31,
32,
34]. Only four studies [
29,
30,
33,
35] examined health status, health care utilization, quality of life or drop-out rates using objective measures. It is difficult to draw conclusions from these studies because of the variability in the sample sizes, diagnoses, measures, types of comparison groups, and international differences in models of health care provision affecting access to services (e.g., health care insurance differences in the US and Canada). In addition, participants in the majority of these studies had a chronic illness as their primary diagnosis [
30‐
35], rather than a physical disability. Although these chronic conditions can be quite physically disabling, transition preparation for these youth may differ from that for youth with disabilities in having a much more specific self-management focus. Thus, research in transition care for these clients is rooted in adult literature that does not take into account the shift of responsibility from parent to youth [
36,
37]. Finally, the optimal window for measuring continuity of care is not yet known, and cross-sectional studies might not have reflected the outcomes of youth who ‘fall through the cracks in the system’ several years after discharge from the pediatric system. In summary, the current status of childhood disability transition literature is characterized by methodological limitations, lack of definitive results or focus on physical childhood-onset disability, and absence of mixed-method longitudinal studies that would capture both objective measures of functional status and patient/clinician experiences [
19].
The “LETS Study”
The “Longitudinal Evaluation of Transition Services” (“LETS Study”) study will attempt to address the many gaps identified in the literature by conducting a formal, prospective, longitudinal, mixed-method evaluation of the LIFEspan (“Living Independently and Fully Engaged”) model of transition care [
38]. LIFEspan is a recently funded, coordinated, client-centred model of linked health care across the lifespan, offered through a partnership between a pediatric rehabilitation centre and an adult rehabilitation centre. The study will compare adolescents with the diagnoses of CP and those with ABI, who receive LIFEspan, to a group of adolescents with SB who will not receive LIFEspan. A second cohort of young adults with CP and ABI who had been discharged prior to the formal launch of LIFEspan, will also be used as a comparison group. The findings of this work will inform the understanding of delivery of transitional care services for young people with childhood-onset disabilities. The objective of this paper is to serve as the first step in knowledge dissemination related to the LETS study. The current paper outlines the study protocol, explains the rationale for the study design and selection of outcome measures, and documents several methodological challenges encountered.
Hypotheses
The primary quantitative outcome of the study will be participants’ post-transition continuity of care, as defined in the “Measures” section of the protocol. We hypothesize that continuity within the health care system after transitioning from pediatric to adult care will be maintained by those participants who receive LIFEspan. Secondary outcomes of interest will include health and well-being, activities and social participation, transition readiness, and health care utilization (frequency, type and duration of emergency services and hospitalizations). It is further hypothesized that youth who will have experienced continuity of care will have enhanced access to health care professionals, reduced emergency health care use, better health status and well-being, more involvement in the community, and greater self-determination, self-efficacy, and self-management skills, as compared with youth not having received LIFEspan. In-depth qualitative analyses of interviews conducted with parents and youth will further inform our understanding of the transition experience. In addition, a detailed chart audit will provide a comprehensive description of the services received by participants, in order to ascertain the model’s treatment fidelity.
Discussion
The LETS study will be unique in conducting a prospective examination of a recently funded coordinated long-term approach to care. Demonstration of outcomes experienced by LIFEspan clients will quantify the value of a coordinated transfer approach and provide data for future studies examining the long-term implications of such a model on continuity of care, quality of life, inclusion, and participation. In a recent paper, McDonagh and Kelly [
19] attributed the current state of transition research in part to methodological challenges. Through a chronic illness lens, they identified key issues that have hindered development of a strong evidence base for the field of transition in solid organ transplantation, including a lack of consensus on basic terminology and adolescent age criteria, a lack of suitable measurement tools, the presence of heterogeneous financial and socio-cultural demographics, and in particular, the absence of guiding theoretical research frameworks to address “the complex multidimensional, multidisciplinary, and multiagency nature” (p. 690) of transition [
19]. McDonagh and Kelly’s careful consideration of such challenges is validating for transition researchers attempting to make headway. Based on our experiences as a multidisciplinary team of pediatric and adult rehabilitation clinicians and researchers, we argue that these issues are not unique to transplantation but are common to the field of health care transition for youth with complex chronic conditions and disabilities broadly [
27,
28,
59]. The current paper describes similar methodological challenges. We continue this discussion by providing concrete examples of such issues and the strategies used to address them as they were encountered in designing an active longitudinal study.
Despite these challenges, the LETS study has a number of methodological strengths. Unlike previous retrospective cross-sectional studies reported in the chronic condition and disability literature to-date [
9,
29‐
32,
34,
35], this prospective longitudinal study will examine the preparation for transition as well as their outcomes within one year post-transition. The outcome component of this study will determine whether LIFEspan establishes continuity of care within the first year post-transition, which is an important first step in maintaining long-term health. This window of time is previously unaccounted, as Young et al.’s study [
14] examined a longer time frame (5 – 15 years) and did not provide information on the transition itself or its immediate consequences. Second, while there are no available measures of transition readiness, we will use a combination of measures that assess skills central to successful transition and thus reflect the multidimensional approach to transition, as recommended [
19]. A wide range of measures with good psychometric properties and appropriate for individuals with disabilities will be implemented to assess outcomes such as quality of life, health, well-being, and emergency health care utilization. In addition, the qualitative component of the study will enrich this quantitative data and provide in-depth subjective perceptions of youth and their parents regarding their experiences of transition process, health care services, continuity of care, preparation interventions, etc. Finally, a comprehensive audit of implementation of LIFEspan (the process evaluation component) will allow for a detailed description of the intervention received by the participants. Previous evaluations described in the literature have not provided such detailed documentation of interventions.
Additionally, CP, ABI, and SB populations represent some of the most complex individuals who need support in transition and comprehensive, lifelong health and social services. Therefore, there is ample reason to believe that the findings from this study will generalize to a wide range of pediatric patients transitioning to adult systems. However, in general, heterogeneity is a huge issue in transition studies, due to the tremendous diversity across impairment groupings arising from variations in cognitive maturation, psychosocial development, skeletal and musculature growth, motor function and other co-morbidities [
59]. Such variations manifest themselves in very different clinical presentations with implications for self-management, multi-disciplinary treatment needs, and multi-agency service utilization [
59]. Conversely, exploring service needs and effective models of care within a single population is also extremely challenging given the vast number of potentially confounding factors associated with the potential spectrum of severity. While narrow inclusion criteria can be used to generate homogeneous samples and optimize data collection strategies, such efforts can diminish the representativeness of the sample by excluding participants who are more severely disabled, have worse health status, are least likely to achieve adequate societal participation, and who are, potentially, the most vulnerable with respect to lacking access to services that would respond to their complex needs. Thus, despite the demand for high level evidence, identification of an ethically sound and practical control group remains challenging within disability research and the field of transition [
58]. In the current study, the issue of potential differences among population groups will be addressed by using multiple baselines (i.e., participants serving as their own controls) and including a retrospective control group from the same populations as those of the intervention group. Through its inclusive design, this study will offer a unique insight in the experiences of non-verbal participants via proxy reporting and inclusion of augmentative and alternative communication users, whose transition experiences to-date have not been incorporated in formal evaluations [
60]. Studies supporting large samples and varied data collection techniques are still required as they will be critical to allow for natural variations in health and disability as well as socio-economic, geographical and cultural demographics among the youth.
Several limitations of this study should also be recognized. First, drop-out is always a significant risk in longitudinal designs. However, subject attrition for primary measures of continuity of care is expected to be minimal as permission for data release was sought at the outset of the study, and administrative database interrogation does not require continued participant contact. Unless a participant withdraws from the study, the primary outcome will be available on all participants, irrespective of loss to follow-up. In addition, key demographic data were also obtained at the time of consent; this information will allow for a detailed comparison of participants who completed the study with those that did not. However, subject attrition will still be concerning for secondary measures. Second, since this project is a longitudinal prospective cohort study of the LIFEspan model, the representativeness of the sample is also a potential risk. At present, the LIFEspan model is only available for clients of one pediatric centre. The centre is recognized as Canada’s largest rehabilitation centre and is located in a large multi-cultural urban area of Ontario. As such, LIFEspan does set the stage for an ideal model for which to strive in service delivery, where, for example, LIFEspan clients benefit from a publicly funded health care system and transportation services to and from the centre. However, generalizability of the LETS study is thereby somewhat limited, as these services and such multicultural populations might not be found in other jurisdictions. Third, the inclusion of three different disability groups also does raise the possibility of differences arising as a function of disability-related influences and not the intervention. While the standardized measures in this study have been selected based on their appropriateness for completion by people with disabilities, it is well recognized that individuals with physical and developmental disabilities may not follow the same developmental trajectory as their age-matched peers and as such, normative milestones may not be valid [
61].
While there is a wealth of evidence articulating challenges to implementing effective models of care, empirical evidence addressing processes and outcomes of transition remains limited. The importance of continuing research on adolescent health care transitions is indisputable for both practice and policy development, since due to a multitude of factors related to systemic deficiencies and inadequate preparation of clients for transition to adulthood, adolescents with childhood-onset disability are at an increased risk of health problems, secondary disabilities, and failure to achieve optimal adult societal roles and community participation. Generally, the population of young adults with disabilities and complex chronic conditions and their families will benefit greatly from stronger partnerships between the pediatric and adult sector, consistent financing, and dedicated human resources [
3]. However, there is a lack of written institutional guidance or policy regarding transition to influence such decisions, as existing research has not yet translated into policy [
5,
62]. Thus, systematic evaluations are essential to determine the success of transition programs and address this gap. The LETS study is unique in design, aims to address gaps in the literature, and will provide evidence to support the expansion of the LIFEspan model and its adaptability to other organizations, within different clinical settings, and for clients with other childhood disabilities. The current paper serves as an important step in the dissemination of the results by outlining the project background, explaining its key concepts, and providing a detailed description of methods adopted prior to the results of the analyses.
Competing interests
The authors declare that they have no competing interests.
Authors’ contributions
SK, JM, MB, CM, and HH conceptualized the study; SL, PM, AC, and YH contributed to the design of the study and were investigators/consultants on the grant application. IT participated in the coordination of the study. All authors participated in the drafting of this manuscript, and all authors read and approved the final manuscript.