Abstract
The transition from pediatric to adult care is the focus of growing research. It is important to identify how to direct future research efforts for maximum effect. Our goals were to perform a scoping review of the transition literature, highlight gaps in transition research, and offer stakeholder guidance on the importance and feasibility of research questions designed to fill identified gaps. The transition literature on rheumatic diseases and other common pediatric-onset chronic diseases was grouped and summarized. Based on the findings, a survey was developed and disseminated to pediatric rheumatologists and young adults with rheumatic diseases as well as their caregivers. The transitional care needs of patients, healthcare teams, and caregivers is well described in the literature. While various transition readiness scales exist, no longitudinal posttransfer study confirms their predictive validity. Multiple outcome measures are used alone or in combination to define a successful transition or intervention. Multimodal interventions are most effective at improving transition-related outcomes. How broader health policy affects transition is poorly studied. Research questions that ranked highest for importance and feasibility included those related to identifying and tracking persons with psychosocial vulnerabilities or other risk factors for poor outcomes. Interventions surrounding improving self-efficacy and health literacy were also ranked highly. In contrast to healthcare teams (n = 107), young adults/caregivers (n = 23) prioritized research surrounding improved work, school, or social function. The relevant transition literature is summarized and future research questions prioritized, including the creation of processes to identify and support young adults vulnerable to poor outcomes.
- pediatric transition to adult care
- transfer from pediatric to adult care
- transfer to adult care
- transferring to adult care
- transition to adult care
The transition from pediatric to adult medical care is a vulnerable period for adolescents (ages 10-17 yrs) and young adults (ages 18-25 yrs).1 Following transfer to adult care, young adults with chronic childhood-onset medical conditions are reported to have worse health outcomes, including increased rates of emergency room visits and hospitalizations2-4; nonadherence to care, loss to follow-up, and gaps in care4-9; poorer disease control and health-related complications including organ failure and death4,8,10,11; school/work dysfunction12; and increases in healthcare costs. These complications are well described among young adults with rheumatic diseases (RDs), especially among those with systemic lupus erythematosus (SLE).4,7,8,10,11 Research related to the transition of adolescents and young adults (AYAs) with pediatric RDs to adult health care has increased in recent years, but significant gaps in knowledge remain.
Goals for this project, adapted from the Agency for Healthcare Research and Quality Prioritization Criteria Methods13 (Supplement A, available with the online version of this article), were the following: (1) Perform a scoping review of existing pediatric to adult healthcare transition research as it relates to specific childhood-onset chronic illnesses (RDs, inflammatory bowel disease, type 1 diabetes, sickle cell disease, cystic fibrosis, as well as transplant recipients). These conditions were selected based on the overlap between the transition-related challenges encountered by persons with RDs and those with other complex chronic conditions. (2) Identify transition research gaps based on the scoping review and articulate questions for research in discussion with stakeholders—clinicians, young adults, and parents involved in the Childhood Arthritis and Rheumatology Research Alliance (CARRA) Transition Workgroup. (3) Survey stakeholders to prioritize transition research questions by importance and feasibility.
In order to organize existing literature surrounding transition, we identified the following 6 key domains: (1) transition outcome measures, (2) transitional care needs (of patients, the healthcare team, and caregivers), (3) transition readiness, (4) transition education and self-efficacy, (5) transition interventions or programs, and (6) transition-related health policy.
METHODS
Search strategy. We used the PRISMA (Preferred Reporting Items for Systematic Reviews and Meta-Analyses) scoping reviews methodology.14 Database searches were conducted in PubMed and EMBASE to identify articles from January 2000 through November 2020 (Supplement B, available with the online version of this article). Searches for each domain were conducted separately, and articles could be included in more than 1 domain.
Abstract inclusion criteria. For each domain, 2 research team members independently reviewed abstracts from the literature search for inclusion in the full manuscript review. A third reviewer broke ties in case of disagreement. Articles included for full review were English-language abstracts reflecting full manuscripts published in peer-reviewed journals as original research that addressed a domain topic. Reviews, opinion pieces, consensus statements, and conference articles or abstracts were excluded.
Data extraction, synthesis, and analysis. Full manuscripts were reviewed by 2 research team members and summarized in a domain-specific charting table. Elements extracted for the charting table included study design, study population, sample size, outcome measure(s), intervention, key findings, and limitations. The key findings for each domain were synthesized and summarized by subthemes. Finally, research gaps in the transition literature were underscored.
Survey development and dissemination. Two sessions were held among members of the CARRA Transition Workgroup, which discussed potential research questions based on the identified knowledge gaps. We then developed a 22-item survey to determine the importance and feasibility of transition-related research questions, which was reviewed by 2 young adults with RDs for clarity. We obtained institutional review board exemption from Boston Children’s Hospital (protocol number IRB-P00039656) for survey distribution.
The survey was disseminated to all CARRA members by email invitation, with up to 2 email requests welcoming participation. Young adult and parent stakeholders were recruited by social media through the Arthritis Foundation and the Lupus Foundation of America. Survey participants responded anonymously and were asked to self-identify as healthcare providers, young adults with RDs, caregivers, or other. Participants were asked to rank research questions based on the importance of each item on a 5-point Likert scale (1 = least important; 5 = most important). Mean and SD were calculated; scores based on self-identified group (healthcare team member vs young adult/caregiver) were also computed.
A separate 10-member task force from the CARRA Transition Workgroup ranked the feasibility of conducting a study to answer each research question (1 = least feasible; 5 = most feasible).
RESULTS
The electronic database search yielded 1491 abstracts across the 6 domains, of which 271 were included in the final scoping review (Supplement C, available with the online version of this article). For space considerations, references in this manuscript were significantly limited. Please reference Supplement D for this same manuscript with full references included.
Domain 1: Transition outcome measures. There are no standardized pediatric to adult healthcare transition outcome measures to determine the success or failure of a specific transition process or intervention. Multiple transition outcome measures across several categories have been used alone or in combination in the transition literature (Table 1; Supplement E). These include process outcomes, healthcare utilization, healthcare coordination, cost of care, insurance-related outcomes, and disease and clinical outcome measures.2-11,15-40 For juvenile idiopathic arthritis or SLE, clinical outcome measures included standardized and self-reported disease activity measures, self-reported end-organ manifestations during the previous 12 months, cumulative disease manifestations, progression to endstage renal disease in SLE, and death.8,10,11,18,37-39 Additional outcome measures included patient or family satisfaction and perceived quality of care, psychosocial and quality of life outcomes, parental outcomes, and self-management outcomes, including medication and appointment-related adherence.4-10,12,17,24,26-29,32,35,37,38,40-49 Four Delphi method studies on outcome measures suggested composite outcomes from the above categories.40,43
Transition outcome measures.
Domain 2: Transitional care needs. Patient, clinician, and caregiver transitional care needs are well described (Table 2; Supplement F, available with the online version of this article). From the patient perspective, during the transition to adult care, it is essential to address preparation for life-long disease self-management and the psychosocial and nonmedical logistical needs of AYAs.12,18,36,50-63 A structured transition plan with a clear point of contact is important, with early discussions about transition, recognition of adult care expectations, and positive yet realistic perceptions of adult care.51,52,57,59,60,64 Direction from the pediatric team in finding an adult clinician is valuable, ideally with a shared clinic appointment with all key stakeholders in a developmentally appropriate setting.18,51-53,55 A supportive family, with decreased parental involvement over time is also influential for long-term success.50,51,53,55
Transitional care needs.
Risk factors for poor transition outcomes include distance to an adult specialty center, transfer to adult care in a different city, public or no insurance, SLE diagnosis, Black race, social complexity, a high degree of stressful life circumstances, and uncontrolled disease prior to transfer.18,36
Clinicians favor transition programs that are formalized and accompanied by established protocols, a trained multidisciplinary team, institutional support, funding for transition services, and collaboration between pediatric and adult teams.52-54,59,60,62,65-69
Caregivers desire early discussions on how best to support a child’s development over time, as well as education about confidentiality, the transition and transfer processes, and the differences in pediatric and adult care cultures.59,60,64,70
Domain 3: Transition readiness. Transition readiness refers to indicators that AYAs can begin and successfully complete the transition process. There were several subthemes under the domain of transition readiness (Table 3; Supplement G, available with the online version of this article). With the exception of 1 longitudinal study,71 all studies describing transition readiness were cross-sectional. Predictors of transition readiness (often, though not exclusively, as determined by scores in validated transition readiness assessment tools such as the Transition Readiness Assessment Questionnaire [TRAQ] or TRxANSITION Scale) included self-efficacy, demographic variables, greater responsibility/lower parental involvement, family cohesion, provider communication, and higher adherence.71-82 However, these variables were less important after accounting for the individual psychosocial resilience and self-efficacy of AYAs.72-74 Developmental maturity and executive functioning are also important considerations for all AYAs when considering transition preparedness.83 Several transition readiness assessment tools were described, both disease-specific tools and general assessments.84,85 The only longitudinal study on transition readiness revealed that baseline TRAQ scores did not predict transfer or time to transfer over a 3-year period.71
Transition readiness.
Domain 4: Education and self-efficacy. Meaningful outcomes associated with higher educational attainment and self-efficacy included an improved transition experience and disease control.86 Thus, optimizing patient educational goals and building self-efficacy were important themes in the transition literature (Table 4; Supplement H, available with the online version of this article).
Education and self-efficacy.
Multiple educational interventions were described including group educational sessions,87 educational handouts,88 workshops,89 nurse-led efforts,90 and tool kits.91 These generally resulted in improved transition readiness91 and competency,89 medical knowledge,92 life-related skills,88 patient satisfaction,90 improved medication adherence and led to variable improvements in disease control.90 Technology-based educational interventions were also described including computer-based or online modules,17,93,94 telehealth sessions,15 and interactive tablet quiz games.95 Results ranged from no change in appointment adherence or self-management confidence ratings17,94 to increased transition readiness and self-management skills acquisition,15 and improved adherence, self-efficacy, and empowerment for those who actively contributed to a social media intervention.93
Interventions designed to improve self-efficacy or self-management included a tool kit promoting organization and goal setting, a transition coordinator focused on improving self-management,96 a randomized trial of a problem-solving skills training on medication adherence delivered by telephone,97 and an educational and case management-based group intervention. These interventions had variable outcomes, ranging from improvement in trust without change in self-management related outcomes, to modest improvements in adherence,97 to significant increases in self-management skills acquisition and disease remission.96,98-101
Domain 5: Transition interventions. We reviewed transition interventions beyond the education/self-efficacy interventions described above (Table 5; Supplement I, available with the online version of this article). Specialized or combined transition clinics were described, some with a multidisciplinary team19,30,34,42,62 and most including both a pediatric and adult clinician.25,42,102 These resulted in high patient/parent satisfaction,42,62,102,103 high rates of retention in adult care,62,102 posttransfer disease remission or disease control,25 high adherence, improved rates of hospitalizations,19 decreased death or allograft failure posttransfer,30 and greater cost-effectiveness.19,30 While most revealed favorable outcomes, some transition clinic interventions did not demonstrate an improvement in adherence, change in clinical outcomes,34,103 or consistent improvements in healthcare utilization outcomes.
Transition interventions.
Interventions focused on scheduling appointments in adult care had variable results, either not changing rates of disengagement posttransfer104 or improving dropout rates in adult care.105 Similarly, interventions centered on the role of a transition coordinator were inconsistent. Some demonstrated improved patient satisfaction,41,106 transfer rates,41 disease control,96,106 medication adherence, and hospitalizations.106 One transition coordinator-based intervention demonstrated improvement in patient satisfaction and clinic attendance during the study period, but not 1 year following the intervention.28,107 Other interventions resulted in no change in retention rates in adult care, disease control,29 or medication adherence. Only 1 study reported on transition-related interventions focused on a learning network for healthcare systems, which led to improvements in care. Educational interventions (Table 4) demonstrated positive results.15,17,48,88,89,90,98
The most effective interventions described several components implemented simultaneously. Multimodal interventions resulted in improved clinical and disease outcomes,26,27 posttransfer retention in care,21,44 shorter duration of transfer,108 improved medication adherence,108 reductions in stress and improvements in well-being,16,27,45,46,109 self-efficacy and self-advocacy,44 improved healthcare utilization variables,21,26 satisfaction with care,16,44,109 and improved process outcomes.110 All multimodal interventions resulted in improvement in some aspect of care, although 1 only resulted in improvements to documentation rather than transitional milestones110 and some resulted in no change to hospitalizations or other utilization variables, disease control,21 medication adherence,45,46 or disease knowledge. Some interventions experienced significant feasibility and sustainability challenges.16,109
Domain 6: Health policy. Very few studies addressed broader health policies and their effect on AYAs’ transition to adult care.31,111,112 One analysis of hospitalized patients did identify improvements in uninsured hospitalized young adults in the US following the implementation of the Affordable Care Act.31
Gaps in Transition Research. In summarizing the pediatric to adult healthcare transition literature, it became evident that gaps in research remain within each domain. While multiple outcome measures have been used, no study evaluated clinician satisfaction and few addressed how disease-related functioning and disability impact young adults longitudinally.10,12,40 The literature describes patients’, clinicians’, and caregivers’ needs during the transition process well, but the needs of healthcare organization leaders, administrators, and payors surrounding transition efforts were very rarely addressed.112 A rapid way to identify and track AYAs at risk for poor outcomes during the transition to adult care has not been established. For instance, a scoring system that accounts for social determinants of health has not been validated. Despite the myriad of studies on transition readiness, no transition assessment tool has been studied longitudinally posttransfer or has been shown to predict posttransfer outcomes.
There were no educational interventions described for parents, and very few studies focused on the importance of health literacy for transition.76 The role of community organizations in fostering education during the transition process is also poorly studied. While a few studies described self-efficacy as an outcome measure,96,98 it was unclear whether higher levels of self-efficacy are predictive of longitudinal successful posttransfer outcomes.49,99
Multimodal interventions were found to be most effective, but it was difficult to separate which components of an intervention were most important and relevant. Many of the interventions used proximal outcomes to determine success, and few interventions measured cost-effectiveness.19,30 While 1 study focused an intervention on AYAs with more complex disease, most did not specifically address the effect of interventions on people with complex medical and psychosocial needs. Only 1 study reported on a telehealth intervention.15 No study was designed to ensure equity in delivery of services or addressed implementation of complex interventions in settings with few resources.
Several opportunities for pediatric to adult healthcare transition research were recognized in the realm of health policy, including the need to investigate the effects of the Affordable Care Act on insurance and retention rates in adult care. It also remains to be seen whether the introduction of Current Procedural Terminology transition-related billing codes affects the provision of transitional care.113 There is no literature to date investigating the effect of insurance policies (eg, financial incentives for joint pediatric/adult transition clinics) or government mandates on transition-related care.
Survey of transition research priorities. A survey of transition research questions was formulated based on our identified gaps in transition research (Supplement J, available with the online version of this article). Between October and November 2021, all 509 CARRA members were invited to complete the survey, ranking the importance of each research question. There were 107 respondents recruited through the CARRA network: 103 healthcare team members, 1 researcher who did not self-identify as a healthcare provider, and 3 healthcare team members who also reported a history of childhood-onset RDs (whose responses were grouped with healthcare team members). There were 23 participants recruited through social media, of whom 21 were adults with RDs and 2 were caregivers.
Simultaneously, a 10-member volunteer task force that included clinicians and researchers as well as a young adult and a parent met to rank the feasibility of each research question. Feasibility was ranked by this expert task force only.
Survey results (Figure) comprise 4 ranked sets: more important and more feasible, more important but less feasible, less important but more feasible, and less important and less feasible. Accounting for all responses, the more important and relatively more feasible transition research questions address identifying, tracking, and improving outcomes for young people with psychosocial vulnerabilities and other risk factors for poor outcomes. In addition, interventions focused on improving self-efficacy and health literacy were rated highly in importance and feasibility.
Survey results.
We found differences in importance rankings based on self-reported group (healthcare team member vs young adults/caregivers; Figure). Given the very small number of caregivers who responded, young adults and caregiver responses were grouped together. For young adults/caregivers, the top research priorities were related to evaluating or improving work, school, and social functioning. In comparison, healthcare team members prioritized identification of young people at high risk for poor outcomes, understanding the contribution of psychosocial determinants of health to transition outcomes, and tracking at-risk patients.
DISCUSSION
In this scoping review of transition-related research relevant to AYAs with RDs, we summarized the transition literature across 6 key domains. We identified gaps in transition research and performed a survey of clinical team members as well as individuals with RDs and their caregivers. The research questions that were ranked highest in importance and feasibility were those related to identifying and tracking patients with psychosocial vulnerabilities and at high risk for poor outcomes. Young adults and their caregivers, in particular, ranked research questions assessing the determinants of work, school, or social functioning highest in importance; however, studies aimed at answering these questions may require more creative study designs to conduct based on lower feasibility scores. In the following paragraphs, we discuss several opportunity areas with high priority, including social functioning, predictors of adult outcomes beyond transition readiness, delivery of educational or self-efficacy interventions, effects of health policies, and tools to track high-risk individuals.
We found that multiple transition-related outcome measures have been used alone or in combination to define a successful transition or intervention. There are no standardized measures in this field of research, as has been noted by other reviews.114 A systematic review of transition research identified that only 3 studies used outcome measures across all domains of the triple aim of healthcare framework (patient experience of care, population health, and cost of care).115 Most commonly, population health measures are used to define outcomes.115 Inclusion of quality of life outcomes in this category is an important goal.116 Given differences in diseases, populations, and program resources, use of standardized measures across all transition-related studies may not be realistic in real-world situations. However, using a combination of outcome measures such as those defined by the quadruple aim of health care (which adds to the triple aim of health care by including the experience of providing care117) may be the ideal approach for future research studies in AYAs with RDs. Based on our survey results, future studies should also aim to include outcomes that address functioning of young adults in work, school, and social settings.
A review of factors associated with transition readiness118 identified several modifiable factors, including self-management and transition education, and nonmodifiable factors, including demographic, ecological, and disease factors. Our review further demonstrates the lack of longitudinal studies of outcomes after transfer to adult care confirming the predictive validity of published transition readiness scales. Future studies will need to explore whether tools that measure health literacy, self-efficacy, patient activation, self-management, or other psychosocial determinants of health are better predictors of transition-related outcomes in adulthood.
Transition education and self-efficacy are important themes in the literature that are potentially modifiable. Self-management skills, trust in adult care, sense of self-efficacy, social support, sociodemographic status, and interdisciplinary cooperation were identified as important factors influencing transition within an integrative framework in 1 review.119 Others have also concluded that knowledge and education are key facilitators to a successful transition for AYAs with childhood cancers,120 in agreement with our findings. Fortunately, effective interventions to improve education and self-efficacy have been described and can be implemented by individual institutions or on a broader scale to benefit AYAs with RDs. How community organizations can partner with health systems to effectively promote education of young adults during the transition period will require further study.
While education and other needs of patients, healthcare teams, and caregivers are well described in the literature, health policy as it relates to transition is a vastly underresearched topic, particularly in the US.113 There are rich opportunities for research in this area, especially utilizing large or administrative datasets.
Systematic reviews of transition interventions with statistically significant positive outcomes reveal that structured transition interventions improve outcomes.121 Successful studies describe transfer assistance and often include transition planning support and integration into adult care.122 Similarly, we found that multimodal interventions are the most effective in improving transition-related outcomes, although the current literature does not identify the critical component(s) of multimodal interventions. Further, the field is lacking evidence as to how healthcare teams can target interventions to those AYAs at highest-risk for poor outcomes. Therefore, next steps include developing mechanisms for identifying and tracking AYAs with RDs at risk for poor transition outcomes before, during, and after transition interventions. Tools to do so may consider incorporating known determinants of care gaps and poor outcomes123 to identify at-risk AYAs. As pediatric to adult care transition interventions are often complex, unique ways of evaluating them may be necessary.
There is overlap between the results of our survey and surveys of stakeholders involved in the care of Children and Youth with Special Health Care Needs (CYSHCN) program, who also prioritize research topics centered on social determinants of health and transition of care.124 A national research agenda on transition for CYSHCN recommended investigation of service models in partnership with youth and families, defining process and outcome measures, and evaluating fiscal policies to incentivize improved transitional care.125 While healthcare team members and young adults or their caregivers responding to our survey largely agreed regarding transition research priorities, there were also meaningful distinctions between the 2 groups. Research questions addressing psychosocial vulnerabilities, implementing interventions in settings with few resources, and measuring the effect of self-efficacy ranked as more important to healthcare team members compared to young adults and their caregivers. Meanwhile, young adults and their caregivers tended to rank research assessing the importance of healthcare policies and community organizations higher than healthcare team members. Top-ranked priorities for young adults and their caregivers pertained to work, school, and social function. Collectively, these rankings chart a course for future patient-oriented research in transition.
The results of our survey are limited in several ways. First, there were fewer young adults, and particularly caregivers, who responded compared to healthcare team members, which could affect generalizability. Given that most respondents outside of the CARRA network were recruited through social media, their RDs were self-reported rather than clinician-verified and no further demographic information was obtained from these individuals. Finally, the research prioritization efforts were offered primarily by pediatric rheumatology providers, with limited input from adult rheumatologists or other adult allied health members. Nevertheless, this is the first published survey, to our knowledge, that captures both clinician and patient/caregiver opinion regarding research prioritization. An additional strength is the dual categorization of research questions regarding both importance and feasibility.
We herein summarized the current state of the literature relevant to healthcare transition for AYAs with childhood-onset chronic illnesses. In addition to identifying remaining research questions in this field, we provide a useful construct for understanding the prioritization of these questions for rheumatology healthcare teams as well as for young adults living with pediatric RDs and their caregivers. This prioritized research agenda can inform and guide the design of future research aimed at improving transition from pediatric to adult rheumatology care.
ACKNOWLEDGMENT
The authors wish to acknowledge CARRA and the ongoing Arthritis Foundation financial support of CARRA.
Footnotes
The authors declare no conflicts of interest relevant to this article.
- Accepted for publication June 30, 2022.
- Copyright © 2022 by the Journal of Rheumatology
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ONLINE SUPPLEMENT
Supplementary material accompanies the online version of this article.
