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Transitions of Care From Pediatric to Adult Services for Children With Special Healthcare Needs

Research Protocol

I. Background and Objectives for the Systematic Review

In the United States, nearly 20 percent of children under age 18 have special healthcare needs,1 defined as having or being at increased risk for chronic physical, developmental, behavioral, or emotional conditions.2 This population, which is growing in number, often require lifelong health-related services. Between 2011 and 2017, approximately 4.5 million youth ages 12–18 with special healthcare needs transitioned from pediatric to adult providers and settings of healthcare.3 Such transitions are often difficult and can lead to gaps in care, adverse health outcomes, and frustration for patients and families.3-5

One reason transitions from pediatric to adult healthcare services are so challenging is lack of clarity around how to best manage them. In 2011, the American Academy of Pediatrics, the American Academy of Family Physicians, and the American College of Physicians sought to address this issue by publishing a framework for implementing care transitions for youth beginning in early adolescence.6 Subsequently, Got Transition (a federally funded national resource center on healthcare transitions) developed a structured clinical approach for transitioning patients from pediatric to adult healthcare services. This approach, called "Six Core Elements of Health Care Transition," includes: transition policy, transition tracking and monitoring, transition readiness, transition planning, transfer of care and transfer completion.7

Healthcare providers have adopted various approaches to integrating the Six Core Elements, including direct interventions for healthcare transitions with children and their caregivers, implementation strategies such as provider training, and tools to facilitate communication between pediatric and adult providers. However, the broad spectrum of the Six Core Elements has led to questions regarding the best transition intervention designs and the implementation tools and strategies. Further, questions persist regarding how certain variables might affect the benefits and harms of specific approaches, and how those approaches could potentially be modified accordingly. Variables that that might affect outcomes are patient characteristics (e.g., age, ethnicity, disease type), provider/hospital characteristics (e.g., access to specialty services, specialty training) and care setting (e.g., specialty center, telemedicine). Identifying and understanding intervention characteristics that lead to more successful transitions will help patients, caregivers, and providers make more informed decisions about which interventions (or components) might work for whom and under what circumstances.

Intervention components are not consistently packaged. Instead, interventions vary widely in their components, structure, and processes. For this reason, we are not rigidly defining or describing the potential array of interventions we might encounter in the literature, and nor are we excluding any care transition intervention. Readers interested in more detail about potential interventions are encouraged to explore the Got Transitions website.

Key Decisional Dilemmas

The National Cancer Institute (NCI) requested this review as part of a series of projects for The Childhood Cancer Survivorship, Treatment, Access, and Research (STAR) Act, which is devoted to advancing the state of science and improving the healthcare and quality of life for children and adolescent cancer survivors. As NCI anticipated limited research specifically addressing healthcare transitions for children and youth with pediatric cancer, the review will examine transitioning to adult health care for all children with special health care needs. The key decisional dilemmas to be addressed by the review include the effectiveness and harms of (1) health care interventions targeted toward children with special healthcare needs and their families/caregivers, (2) strategies to implement interventions for healthcare transition including provider-related training, and (3) tools to facilitate communication between pediatric and adult providers.

II. The Research Questions

Key questions (KQ) address the effects of interventions for transition from pediatric to adult services for children with special healthcare needs. Healthcare transition interventions can be complex or multicomponent, while involving behavior changes on the part of patients and care providers. Therefore, we include a subquestion to help understand barriers and facilitators for such interventions. Contextual questions will support and inform the review by providing information on the context within which care transitions happen.

We have made no changes to the key questions in response to public comments (n=2). The majority of the comments will be addressed as considerations in contextual pieces and will be dependent on the landscape of the literature.

  • KQ1: What are the effectiveness, comparative effectiveness, harms, and costs of care interventions for transition from pediatric to adult medical care services, including primary care, for children with special healthcare needs and their families/caregivers?
    • KQ1a: How do outcomes vary by intervention characteristics or components?
    • KQ1b: How do outcomes vary by patient/caregiver or provider characteristics or setting?
    • KQ1c: What are the barriers and facilitators to effective transitions?
    • KQ1d: What are the gaps in evidence for the effectiveness of the interventions?
  • KQ2: What are the effectiveness, comparative effectiveness, harms, and costs of implementation strategies for care interventions for transition, including provider-related training?
    • KQ2a: How do outcomes vary by intervention characteristics or components?
    • KQ2b: How do outcomes vary by patient/caregiver or provider characteristics or setting?
    • KQ2c: What are the barriers and facilitators to effective implementation?
    • KQ2d: What are the gaps in evidence for the effectiveness of the interventions?
  • KQ3: What is the effectiveness, comparative effectiveness, harms, and costs of tools to facilitate communication between pediatric and adult providers for care transitions from pediatric to adult medical care for children with special healthcare needs and their families/caregivers?
    • KQ3a: How do outcomes vary by intervention characteristics or components?
    • KQ3b: How do outcomes vary by patient/caregiver or provider characteristics or setting?
    • KQ3c: What are the barriers and facilitators to effective tools to facilitate communication?
    • KQ3d: What are the gaps in evidence for the effectiveness of the interventions?

In addition to the identified key questions, NCI expressed interest in a formal, mixed-methods evaluation of the contexts in which interventions for transitioning children with special healthcare needs from pediatric to adult services are developed and used. Questions to be evaluated include:

  1. How is effectiveness defined and measured for transitions of care from pediatric to adult services for children with special healthcare needs?
  2. What transition care training and other implementation strategies are available to prepare pediatric medical providers (e.g., pediatricians and other specialists) and adult medical providers (e.g., primary care providers, nurse practitioners, physician assistants) for transitioning children with special healthcare needs to adult care?
  3. What training is available for linguistic- and culturally competent care?
  4. What transition care training and other implementation strategies are available to prepare pediatric patients and their families for transitioning children with special healthcare needs to adult care?
  5. What care interventions including primary care have been used for transition from pediatric to adult medical care for children with special healthcare needs?
  6. What strategies have been proposed to increase availability of adult care providers for people transitioning from pediatric to adult care?

We will prioritize synthesis of literature for populations that best approximate the healthcare experiences of pediatric and adolescent cancer survivors. Even so, the literature might encompass a wide range of care providers, including primary care/ family medicine physicians, specialty care physicians, nurse practitioners, physician assistants, and social workers. Note that in Table 1, outcomes are loosely listed to correspond with key questions. Importantly, the final outcomes collected will depend on the outcomes reported in individual studies and the interventions and measures used by study authors.

Table 1. PICOTs for transition interventions

PICOT KQ1: Benefits and Harms of Care Intervention KQ2: Implementation Strategies KQ3: Communication Tools
Population

Adolescents and young adults (diagnosed with cancer or other special healthcare condition before 21 years old) with a chronic physical or mental illness or physical, intellectual, or developmental disability, also including parents and/or care givers.

Patient subgroups: disease condition (including cancer), age of diagnosis, sex/sexual orientation, race/ethnicity, religion, socioeconomic status, adverse childhood events.

Provider subgroups: age, sex, race/ethnicity, education, socioeconomic status, specialty, care setting.

Multi-disciplinary care providers (e.g. primary care/ family medicine physicians, specialty care physicians, nurse practitioners, physician assistant, etc.) caring for adolescents and young adults with a special healthcare need.

Patient subgroups: disease condition (including cancer), age of diagnosis, sex/sexual orientation, race/ethnicity, religion, socioeconomic status, adverse childhood events.

Provider subgroups: age, sex, race/ethnicity, education, socioeconomic status, specialty, care setting.

Multi-disciplinary care providers (e.g. primary care/family medicine physicians, specialty care physicians, nurse practitioners, physician assistant, etc.) providers caring for adolescents and young adults with a special need.

Patient subgroups: disease condition (including cancer), age of diagnosis, sex/sexual orientation, race/ethnicity, religion, socioeconomic status, adverse childhood events.

Provider subgroups: age, sex, race/ethnicity, education, socioeconomic status, specialty, care setting.

Intervention Intervention related to the care transition from pediatric to adult medical care (e.g., any single or multi-component intervention that addresses the Six Core Elements of healthcare transition such as educational materials, patient care documents, processes, etc. There are not widely established neat packages of intervention components; interventions vary widely in their components, structure, and processes.) No healthcare transition intervention is explicitly excluded. However, transition interventions that address the full spectrum of transition to adult life, such as transition to independent living from foster care or among people with developmental disabilities, will be excluded. Implementation strategies, including training (e.g., any single or multi-component intervention that addresses implementing the Six Core Elements of healthcare transition such as trainings). Tools for provider communication (e.g., any single or multi-component intervention that addresses communication that supports the Six Core Elements of healthcare transition such as patient care documents).
Comparators Comparator required, but no exclusion based on comparator type. Comparator required, but no exclusion based on comparator type. Comparator required, but no exclusion based on comparator type.
Outcomes
  • Transition readiness (e.g., patient, family, provider, and system level)
  • Quality of life
  • Mortality
  • Morbidity
  • Disease-specific clinical outcomes
  • Wellness visits/screenings (e.g., depression, anxiety, STIs, other risk and resiliency factors such as alcohol use, substance abuse, violence)
  • Treatment or care adherence
  • Engagement in care (e.g., no shows, time between providers, satisfaction, loss to follow-up, time between leaving pediatric setting to going to adult)
  • Satisfaction (patient and family)
  • Family caregiver outcomes
  • Harms
  • Unintended consequences (e.g., ethics of transition)
  • Psychosocial (e.g., social-emotional, mental health, etc.)
  • Insurance
  • Cost
  • Resource utilization (ER visit, hospitalization, length of stay)
  • Intervention
    • Adoption
    • Fidelity
    • Sustainability
    • Feasibility
    • Acceptability
  • Satisfaction (physician and other formal caregiver)
  • Quality of life
  • Mortality
  • Morbidity
  • Disease-specific clinical outcomes
  • Family Caregiver outcomes
  • Harms
  • Unintended consequences (e.g., ethics of transition)
  • Cost of implementation
  • Insurance
  • Transition readiness
  • Quality of life
  • Mortality
  • Morbidity
  • Disease-specific clinical outcomes
  • Treatment or care adherence
  • Engagement in care (e.g., no shows, time between providers, satisfaction, loss to follow-up, time between leaving pediatric setting to going to adult)
  • Satisfaction (patient and family)
  • Family Caregiver outcomes
  • Harms
  • Unintended consequences (e.g., ethics of transition)
  • Insurance
  • Cost
  • Resource utilization (ER visit, hospitalization, length of stay)
Timing At least 6 months post transition for tests of interventions. No exclusions for qualitative or mixed studies for barriers and facilitators subquestion. At least 6 months for tests of interventions. No exclusions for qualitative or mixed studies for barriers and facilitators subquestion. At least 6 months for tests of interventions. No exclusions for qualitative or mixed studies for barriers and facilitators subquestion.
Setting All settings (e.g., primary care, specialty care, schools, rural, resource limited settings, and telehealth) All settings (e.g., primary care, specialty care, schools, rural, resource limited settings, and telehealth) All settings (e.g., primary care, specialty care, schools, rural, resource limited settings, and telehealth)

Abbreviations: ER=Emergency room visit; PICOT=population, intervention, comparator, outcome, timing, setting

 

III. Analytic Framework

Figure 1 shows a visual representation of the analytic framework for the KQ's, illustrating the relationship of interventions and outcomes.

Figure 1. Analytic Framework

Figure 1 is the analytical framework describing the flow of patients diagnosed with cancer or other special healthcare need and care providers for individuals diagnosed with cancer or other special healthcare need. Patients receive healthcare transition interventions from pediatric to adult medical care. Care providers receive training and or other implementation strategies and tools to facilitate communication related interventions. Interventions may be associated with barriers and facilitators to care transition or potential gaps in evidence. Final outcomes include effectiveness, comparative effectiveness, and or harms of care interventions, provider-related training and other implementation strategies, and tools to facilitate communication between pediatric and adult providers. Outcomes may differ by patients baseline characteristics (such as age of diagnosis, sex/sexual orientation, race/ethnicity, religion, education, SES, adverse childhood events, or disease condition) or care providers (such as age, sex, race/ethnicity, education, SES, specialty, or care setting).

IV. Methods

Criteria for Inclusion/Exclusion of studies in the review:

Studies will be included in the review based on the PICOTS framework outlined above in Table 1 and the study-specific inclusion criteria described in Table 2.

Table 2. Study inclusion criteria (key questions & contextual questions)

Category Criteria for Inclusion
Study Enrollment

KQ1: Individuals diagnosed with cancer or other special healthcare need (diagnosed before 21 years of age) and their parents and/or caregivers.

KQ2-3: Multidisciplinary care providers for individuals diagnosed with cancer or other special healthcare need (diagnosed before 21 years of age).

CQ1 & CQ5: Individuals diagnosed with cancer or other special healthcare need (diagnosed before 21 years of age) and their parents and/or caregivers.

CQ2–4 & CQ6: Multidisciplinary care providers for individuals diagnosed with cancer or other special healthcare need (diagnosed before 21 years of age).

Study Objective

KQ1 and CQ1 & CQ5: Evaluate the effectiveness, comparative effectiveness, harms, and/or costs of care interventions for transition from pediatric to adult medical care services, explore relationships between PICOTS characteristics and outcomes, or experiences of participants and stakeholders

KQ2, CQ2–4: Evaluate the effectiveness, comparative effectiveness, harms, and/or costs of provider-related training and other implementation strategies for care transitions.

KQ3: Evaluate the effectiveness, comparative effectiveness, harms, and/or costs of tools to facilitate communication between pediatric and adult providers for care transitions from pediatric to adult medical care for children with special healthcare needs and their families/caregivers.

KQ sub-questions a and b: Evaluate outcomes of interventions identified for KQ1–3 by intervention characteristics or components; by patient/caregiver or provider characteristics or setting; 

KQ sub-questions c: Identify barriers and facilitators to effective transition interventions, implementation, and tools to facilitate communication.

Study Design Randomized and non-randomized controlled trials, cohort studies with comparator arms, single arm pre/post design. Supplemented by qualitative studies (e.g. ethnography, phenomenology, narrative research, grounded theory, case study, qualitative descriptive); quantitative non-randomized studies (e.g. non-randomized controlled trials, cohort study, case-control study, cross-sectional analytic study); quantitative descriptive studies (e.g. incidence or prevalence study without comparison group, survey, case series with >9 participants, case report of transition tool, component, or program); mixed methods studies (e.g. convergent design, sequential explanatory design, sequential exploratory design)
Outcomes Outcomes listed in Table 1. Actual outcome measures will be defined by authors of published studies.
Publication Type Published in peer-reviewed journals and grey literature with full text available (if sufficient information to assess eligibility and risk of bias are provided). Letters and abstracts are excluded due to the inability of such short publications to provide the information needed to fully describe the interventions. Consensus statements from professional organizations and agencies from grey literature for contextual questions. Individual or small group opinion or perspective pieces will be excluded.
Language of Publication English only, due to resource limitations

Abbreviations: KQ=key question; CQ=contextual question

 

Searching for Evidence: Literature Search Strategies for Identification of Relevant Studies to Answer the Key Questions and Contextual Questions

We will search Ovid Medline, CINAHL, Ovid Embase, and the Cochrane register of Controlled Trials (CENTRAL) to identify studies published and indexed in bibliographic databases. The search algorithm will include relevant controlled vocabulary and natural language terms for the concepts of transitions of care (pediatric to adult services), and special healthcare needs (Appendix A). We will supplement our search strategies with backward and forward citation searches of recent relevant systematic reviews.

We will review bibliographic database search results for studies relevant to our PICOTS framework and study-specific criteria. Search results will be downloaded to PICO Portal, an on-line systematic review platform, for screening. Titles and abstracts will be reviewed by two independent investigators to identify studies meeting PICOTS framework and inclusion/exclusion criteria. All citations deemed appropriate for inclusion by at least one of the reviewers will be retrieved for full-text review. Two reviewers will independently perform full-text screening to determine if inclusion criteria are met. Differences in screening decisions will be resolved by consultation between reviewers, and, if necessary, consultation with a third investigator. We will document the inclusion and exclusion status of citations undergoing full-text screening. Throughout the screening process, members of the review team will meet regularly to discuss training material and issues as they arise to ensure that inclusion criteria are applied consistently.

We will conduct additional grey literature searching to identify relevant completed and ongoing studies that meet the study design inclusion criteria noted in Table 2. Grey literature search results will be used to identify studies, outcomes, and analyses not reported in the published literature to assess publication and reporting bias and inform future research needs. We will also search for grey literature sources for additional material to address the contextual questions. Potential sources of grey literature will include relevant governmental agencies, professional associations, and consensus statements. We will also track, using ClinicalTrials.gov, ongoing trials that have yet to publish results, emphasizing their contributions to a potential research agenda.

For the draft report, the search will be conducted from database inception to January, 2021. We will update searches while the draft report is under public/peer review.

Data Abstraction and Data Management

Studies meeting inclusion criteria will be distributed among investigators for data extraction. For all study designs, these data fields will include author, year of publication, study design, population (including patient and/or provider/clinician characteristics of interest noted in Table 1), intervention, study followup, and setting. Intervention duration, comparison, outcomes cited, and risk of bias elements will be abstracted for studies used to address KQ1–3.

For the key questions, additional information pertaining to effectiveness, harms, costs, barriers, facilitators, and cited gaps will be abstracted. These fields include subject inclusion and exclusion criteria, intervention and comparison characteristics, descriptions and results of included outcomes and harms, and study funding source. Intervention characteristics will include components and activities, timing, frequency, duration, use of technology, training, delivery approach (prescriptive or manualized vs tailored), other delivery modalities, and use of cultural adaptations or modifications. We will note the age, developmental stage, or cognitive ability for which the intervention is intended. For the contextual questions additional information pertaining to the definition and measurement of effectiveness, implementation strategies (including training available to providers, patients, and families), care intervention, and strategies for increasing provider availability will be abstracted from studies that pass risk of bias. For studies using qualitative data collection and analysis, we will also abstract if theoretical or conceptual frameworks were used. We will use the PICOTS framework for quantitative studies and the PerSPEcTiF framework for qualitative studies.8

Relevant data will be extracted into Microsoft Excel by a primary reviewer. Evidence tables will be reviewed and verified for accuracy by a second reviewer. Data will be extracted to evidence and outcomes tables by one reviewer and reviewed and verified for accuracy by a second reviewer.

Assessment of Methodological Risk of Bias of Individual Studies

Based on AHRQ guidance,9 two independent reviewers will assess risk of bias of eligible studies. Reviewers will consult to reconcile discrepancies in overall risk of bias. Overall risk of bias assessments for each study will be classified as low, moderate, or high based on the collective risk of bias inherent in each domain and confidence that the results are believable given the study's limitations. We will focus on studies with the least potential for bias and the fewest limitations.

Because of the wide array of study designs that will contribute to the mixed studies review methodology, we will use the Mixed Methods Appraisal Tool (MMAT) for all included studies.10-13 For randomized and nonrandomized controlled trials studies that contribute to a synthesis that requires grading strength of evidence, we will supplement the MMAT with the ROBINS-I.14 While the MMAT is likely sufficient for descriptive qualitative syntheses, the ROBINS-I provides a deeper, systematic decision tool for risk of bias assessment which in turn will support more confidence in the study limitations domain (discussed below in the Grading Strength of Evidence section).

Data Synthesis

We will summarize results in evidence tables and synthesize evidence for each unique population, comparison, and outcome or harm. The evidence tables will be organized by intervention targets, interventions, comparators, and patients with special health care needs, care provider, or other system-level outcomes. Considering the complexity and variety of adolescents and young adults with special health care needs identified during a scoping of the literature, we are proposing this broad categorization of patients by condition/disease type (subject to change according to available literature) in Appendix B, Figure 2. This will ensure pediatric oncology populations are presented as specific groupings. Further, to maximize the potential to learn from the wide array of non-oncology patient populations represented in the health care transition literature, we anticipate grouping patients into categories based on the general patterns of patient needs or experience and which may suggest different care transition approaches. For example, adapting the patient groupings from the PiCASO study,15 disease/conditions present in the literature for pediatric to adult care transitions could be described by 4 groups: low daily management or monitoring only, frequent hospitalizations or emergency department visits, high daily management with intermittent flares, or survivor of life-threatening condition requiring ongoing treatment/symptom management.

Because we have not identified an agreed-upon taxonomy of interventions by general purpose, we will categorize interventions empirically by intervention and comparator pairs using the Six Core Elements as a framework. We will use the Implementation Research Logic Model as a framework for studies on implementation strategies.16

Additionally, a potential way of sorting core outcome sets stratified by conditions/diseases identified during the scoping review are shown in Appendix B (Figure 3 & Figure 4). Additional core outcome sets identified can be seen in Appendix C, which is subject to change according to available literature.

For the key questions, we will assess the efficacy and comparative effectiveness of outcomes using minimal important differences when they are well established, but for many outcomes this will not be the case. Although we are not anticipating this to be the case, if certain comparisons can be pooled using typical forest plots and meta-analysis, we will analyze data using appropriate methods. We will calculate risk ratios and absolute risk differences with the corresponding 95 percent confidence intervals for binary primary outcomes. Weighted mean differences and/or standardized mean differences with the corresponding 95 percent confidence intervals will be calculated for continuous outcomes. We will assess the clinical and methodological heterogeneity and variation in effect size to determine appropriateness of pooling data.17 We will assess statistical heterogeneity with Cochran's Q test and measure magnitude with I2 statistic.18

If we determine there are sufficient randomized controlled trials, nonrandomized controlled trials, and single arm pre/post studies testing transition care interventions for a given intervention/comparison/outcome set to allow aggregation, we will conduct a qualitative comparative analysis (QCA) to explore patterns of intervention components for transition programs or models.19 QCA is a method for synthesizing comparatively rich information, including more complex interventions and intervention contexts and can be helpful in revealing the relative importance of intervention components and contextual features. QCA uses set-theoretic logic to suggest areas of overlap among studies, such as study outcomes and similarities in interventions and similarities within intervention context. Any results from a QCA will be viewed and presented as hypothesis-generating analyses, not tests of hypotheses. We will file a protocol amendment with specific actions if a qualitative comparative analysis is to be conducted. At minimum, a qualitative narrative synthesis using matrix table approaches will be used for all research questions.

For studies that address barriers and facilitators, we will create an evidence map of all contributing studies, organized by disease/condition. Because variation in concepts is the main goal, we will sample the literature for themes, using thematic analysis, and end sampling when we reach saturation. We will develop a sampling frame based on disease/condition, data richness, and closeness of the study data to the review questions. We will include all studies that specifically enrolled subpopulations of interest, including pediatric oncology diagnosis, race/ethnicity, or other patient subgroups noted in Table 1. We will then cross-check the qualitative findings against findings from the quantitative pre/post single arm studies for intervention fit to patient-reported issues.

Grading the Strength of Evidence for Major Comparisons and Outcomes

We will evaluate overall strength of evidence for select quantitative outcomes for KQs 1 through 3 within each comparison based on five required domains: (1) study strengths and limitations (risk of bias); (2) directness (single, direct link between intervention and outcome); (3) consistency (similarity of effect direction and size); (4) precision (degree of certainty around an estimate); and (5) reporting bias.20 Based on study design and risk of bias, we will rate study limitations as low, medium, or high. Consistency will be rated as consistent, inconsistent, or unknown/not applicable (e.g., single study) based on whether intervention effects are similar in direction and magnitude, and statistical significance of all studies. Directness will be rated as either direct or indirect based on the need for indirect comparisons when inference requires observations across studies. That is, reaching the conclusion requires more than one step. Precision will be rated as precise or imprecise based on the degree of certainty surrounding each effect estimate or qualitative finding. An imprecise estimate is one for which the confidence interval is wide enough to include clinically distinct conclusions. For outcomes found to have at least moderate or high strength of evidence, we will evaluate reporting bias by the potential for publication bias, selective outcome reporting bias, and selective analysis reporting bias by comparing reported results with those mentioned in the methods section and an assessment of the grey literature to assess potentially unpublished studies. Other factors we may consider in assessing strength of evidence include dose-response relationship, the presence of confounders, and strength of association.

For mixed studies qualitative results, we will use CERQual for short statements of qualitative findings. CERQual follows a similar process for assessing the degree of confidence in the finding statement.21 The domains include methodological limitations, relevance, coherence, and adequacy, and follow the same general philosophical approach as the domains discussed above.

Based on these factors, the overall strength of evidence or CERQual confidence for each outcome will be rated as:

  • High: Very confident that estimate of effect lies close to true effect. Few or no deficiencies in body of evidence, findings believed to be stable.
  • Moderate: Moderately confident that estimate of effect lies close to true effect. Some deficiencies in body of evidence; findings likely to be stable, but some doubt.
  • Low: Limited confidence that estimate of effect lies close to true effect; major or numerous deficiencies in body of evidence. Additional evidence necessary before concluding that findings are stable or that estimate of effect is close to true effect.
  • Insufficient: No evidence, unable to estimate an effect, or no confidence in estimate of effect. No evidence is available, or the body of evidence precludes judgment.

An overall rating of high strength of evidence would imply that the included studies were randomized controlled trials with a low risk of bias and with consistent, direct, and precise domains. We will assess strength of evidence for key final health outcomes measured with validated scales.

V. References

  1. Children with Special Health Care Needs. 2020. (Accessed April 21, 2020).
  2. McPherson M, Arango P, Fox H, et al. A new definition of children with special health care needs. Pediatrics 1998;102:137-40.
  3. McManus MA, Pollack LR, Cooley WC, et al. Current Status of Transition Preparation Among Youth With Special Needs in the United States. Pediatrics 2013;131:1090-7.
  4. Sadak KT, Dinofia A, Reaman G. Patient-perceived facilitators in the transition of care for young adult survivors of childhood cancer. Pediatric blood & cancer 2013;60:1365-8.
  5. Bloom SR, Kuhlthau K, Van Cleave J, Knapp AA, Newacheck P, Perrin JM. Health care transition for youth with special health care needs. The Journal of adolescent health: official publication of the Society for Adolescent Medicine 2012;51:213-9.
  6. Supporting the Health Care Transition From Adolescence to Adulthood in the Medical Home. Pediatrics 2011;128:182-200.
  7. Got Transitions. 2020 (Accessed July 29, 2020.
  8. Booth A, Noyes J, Flemming K, Moore G, Tunçalp Ö, Shakibazadeh E. Formulating questions to explore complex interventions within qualitative evidence synthesis. BMJ global health. 2019 Jan 1;4(Suppl 1).
  9. Viswanathan M, Ansari M, Berkman N, et al. Assessing the Risk of Bias of Individual Studies in Systematic Reviews of Health Care Interventions AHRQ. 2012.
  10. Pace, R., Pluye, P., Bartlett, G., Macaulay, A. C., Salsberg, J., Jagosh, J., et al. (2012). Testing the reliability and efficiency of the pilot Mixed Methods Appraisal Tool (MMAT) for systematic mixed studies review. International Journal of Nursing Studies, 49(1), 47-53.
  11. Pluye, P. (2013). Critical appraisal tools for assessing the methodological quality of qualitative, quantitative and mixed methods studies included in systematic mixed studies reviews. Journal of Evaluation in Clinical Practice, 19(4), 722.
  12. Pluye, P., Gagnon, M. P., Griffiths, F., & Johnson-Lafleur, J. (2009). A scoring system for appraising mixed methods research, and concomitantly appraising qualitative, quantitative and mixed methods primary studies in mixed studies reviews. International Journal of Nursing Studies, 46(4), 529-546.
  13. Pluye, P., Hong, Q. N., Granikov, V., & Vedel, I. (2018). The wiki toolkit for planning, conducting and reporting mixed studies reviews. Education for Information (Special Issue).
  14. Sterne JAC, Hernán MA, Reeves BC, Savović J, Berkman ND, Viswanathan M, Henry D, Altman DG, Ansari MT, Boutron I, Carpenter JR, Chan AW, Churchill R, Deeks JJ, Hróbjartsson A, Kirkham J, Jüni P, Loke YK, Pigott TD, Ramsay CR, Regidor D, Rothstein HR, Sandhu L, Santaguida PL, Schünemann HJ, Shea B, Shrier I, Tugwell P, Turner L, Valentine JC, Waddington H, Waters E, Wells GA, Whiting PF, Higgins JPT. ROBINS-I: a tool for assessing risk of bias in non-randomized studies of interventions. BMJ 2016; 355; i4919; doi: 10.1136/bmj.i4919.
  15. Anderson, R. A., Bailey Jr, D. E., Wu, B., Corazzini, K., McConnell, E. S., Thygeson, N. M., & Docherty, S. L. (2015). Adaptive leadership framework for chronic illness: framing a research agenda for transforming care delivery. ANS. Advances in nursing science, 38(2), 83. 
  16. Smith JD, Li DH, Rafferty MR. The Implementation Research Logic Model: a method for planning, executing, reporting, and synthesizing implementation projects. Implementation Science (2020) 15:84. DOI: 10.1186/s13012-020-01041-8.
  17. Morton SC, Murad MH, O'Connor E, Lee CS, et. al. Quantitative Synthesis—An Update. Methods Guide for Comparative Effectiveness Reviews. (Prepared by the Scientific Resource Center under Contract No. 290-2012-0004-C). AHRQ Publication No. 18-EHC007-EF. Rockville, MD: Agency for Healthcare Research and Quality; February 2018.
  18. Fu R, Gartlehner G, Grant M, et al. Conducting quantitative synthesis when comparing medical interventions: AHRQ and the Effective Health Care Program. Journal of Clinical Epidemiology. 2011 Nov;64(11):1187-97. PMID 21477993.
  19. Kahwati L., Jacobs S., Kane H., Lewis M., et al. Using qualitative comparative analysis in a systematic review of a complex intervention. Systematic Reviews (2016) 5:82 DOI 10.1186/s13643-016-0256-y
  20. Berkman ND, Lohr KN, Ansari M, et al. Grading the strength of a body of evidence when assessing health care interventions for the effective health care program of the Agency for Healthcare Research and Quality: an update. 2013.
  21. GRADE Working Group. CERQual Tool. 2018. Accessed 20 June 2019. 29. Lewin S, Booth A, Glenton C, Munthe-Kaas H, Rashidian A, Wainwright M, et al. Applying GRADE-CERQual to qualitative evidence synthesis findings: introduction to the series. Implement Sci. 2018;13(Suppl 1):2.

VI. Definition of Terms

N/A.

VII. Summary of Protocol Amendments

If we need to amend this protocol, we will give the date of each amendment, describe the change and give the rationale in this section. Changes will not be incorporated into the protocol. Example table below:

Date Section Original Protocol Revised Protocol Rationale
State the effective date of the change in protocol Specify where the change would be found in the protocol Describe the language of the original protocol. Describe the change in protocol. Justify why the change will improve the report. If necessary, describe why the change does not introduce bias. Explain what the change aims to accomplish.

 

VIII. Review of Key Questions

The Agency for Healthcare Research and Quality (AHRQ) posted the Key Questions on the AHRQ Effective Health Care Website for public comment. The Evidence-based Practice Center (EPC) refined and finalized them after reviewing of the public comments and seeking input from Key Informants and the Technical Expert Panel (TEP). This input is intended to ensure that the Key Questions are specific and relevant.

IX. Key Informants

Key Informants are the end-users of research; they can include patients and caregivers, practicing clinicians, relevant professional and consumer organizations, purchasers of health care, and others with experience in making health care decisions. Within the EPC program, the Key Informant role is to provide input into the decisional dilemmas and help keep the focus on Key Questions that will inform health care decisions. The EPC solicits input from Key Informants when developing questions for the systematic review or when identifying high-priority research gaps and needed new research. Key Informants are not involved in analyzing the evidence or writing the report. They do not review the report, except as given the opportunity to do so through the peer or public review mechanism.

Key Informants must disclose any financial conflicts of interest greater than $5,000 and any other relevant business or professional conflicts of interest. Because of their role as end-users, individuals are invited to serve as Key Informants and those who present with potential conflicts may be retained. The AHRQ Task Order Officer (TOO) and the EPC work to balance, manage, or mitigate any potential conflicts of interest identified.

X. Technical Experts

Technical Experts constitute a multi-disciplinary group of clinical, content, and methodological experts who provide input in defining populations, interventions, comparisons, or outcomes and identify particular studies or databases to search. The Technical Expert Panel is selected to provide broad expertise and perspectives specific to the topic under development. Divergent and conflicting opinions are common and perceived as healthy scientific discourse that fosters a thoughtful, relevant systematic review. Therefore, study questions, design, and methodological approaches do not necessarily represent the views of individual technical and content experts. Technical Experts provide information to the EPC to identify literature search strategies and suggest approaches to specific issues as requested by the EPC. Technical Experts do not do analysis of any kind; neither do they contribute to the writing of the report. They do not review the report, except as given the opportunity to do so through the peer or public review mechanism.

Members of the TEP must disclose any financial conflicts of interest greater than $5,000 and any other relevant business or professional conflicts of interest. Because of their unique clinical or content expertise, individuals are invited to serve as Technical Experts and those who present with potential conflicts may be retained. The AHRQ TOO and the EPC work to balance, manage, or mitigate any potential conflicts of interest identified.

XI. Peer Reviewers

Peer reviewers are invited to provide written comments on the draft report based on their clinical, content, or methodological expertise. The EPC considers all peer review comments on the draft report in preparing the final report. Peer reviewers do not participate in writing or editing of the final report or other products. The final report does not necessarily represent the views of individual reviewers. 

The EPC will complete a disposition of all peer review comments. The disposition of comments for systematic reviews and technical briefs will be published 3 months after publication of the evidence report.

Potential peer reviewers must disclose any financial conflicts of interest greater than $5,000 and any other relevant business or professional conflicts of interest. Invited peer reviewers with any financial conflict of interest greater than $5,000 will be disqualified from peer review. Peer reviewers who disclose potential business or professional conflicts of interest can submit comments on draft reports through the public comment mechanism.

XII. EPC Team Disclosures

EPC core team members must disclose any financial conflicts of interest greater than $1,000 and any other relevant business or professional conflicts of interest. Direct financial conflicts of interest that cumulatively total more than $1,000 will usually disqualify an EPC core team investigator.

XIII. Role of the Funder

This project was funded under Contract No. 75Q80120D00008 from the Agency for Healthcare Research and Quality, U.S. Department of Health and Human Services. The AHRQ Task Order Officer reviewed the EPC response to contract deliverables for adherence to contract requirements and quality. The authors of this report are responsible for its content. Statements in the report should not be construed as endorsement by either the Agency for Healthcare Research and Quality or the U.S. Department of Health and Human Services.

XIV. Registration

This protocol will be registered in the international prospective register of systematic reviews (PROSPERO).

Appendix A. Search Strategies: Transitions of Care From Pediatric to Adult Services for Children With Special Healthcare Needs

Date Searches Ran: September 4, 2020

Ovid MEDLINE(R) and Epub Ahead of Print, In-Process & Other Non-Indexed Citations, Daily and Versions(R) 1946 to September 03, 2020

  1. (adolescen* or child* or emerg* adult* or p?ediatr* or teen* or young adult* or youth).mp.
  2. (transition* adj5 (care or plan* or process*or program* or service*)).mp.
  3. limit  to english language

Total: 4297

Database—CINAHL Plus with Full Text

EBSCOhost Research Databases

( ((adolescen* or child* or emerg* adult* or p?ediatr* or teen* or young adult* or youth)) AND (transition* N5 care OR transition* N5 plan* OR transition* N5 process* OR transition* N5 program* OR service* N5 )

Limit to English, exclude MEDLINE

Total: 3071

Embase Classic+Embase 1947 to 2020 September 03 results exported 2020-09-04

  1. (transition* adj5 (care or plan* or process* or program* or service*)).ti,ab. 
  2. (adolescen* or emerg* adult* or p?ediatr* or teen* or young adult* or youth).ti,ab.
  3. Limit  to English language
  4. Limit 4 to Conference Abstract Status records
  5. 3 not 4 (excludes conference abstracts from results)

Total: 3191

Cochrane Library

Cochrane Library

Date Run: 2020/09/04

#1    ((adolescen* or child* or emerg* next adult* or p?ediatric? or teen* or young next adult* or youth))    
#2    (transition* near/5 (care or plan* or process* or  program* or service*))    
#3    #1 and #2

Trials = 395

Appendix B. Figures

Appendix B. Figure 2. Adolescents or Young Adults with Special Healthcare Need. Populations Identified in Evidence Mapping.

Figure 2 is a suggested broad categorization of patients by condition/disease type that was found during evidence mapping of the literature (subject to change according to available literature).

Appendix B. Figure 3. Outcomes of Interest Stratified by Condition Group. Example.

Figure 3 is a potential way of sorting core outcome sets stratified by conditions/diseases.

Appendix B. Figure 4. Outcomes of Interest Stratified Condition Group. Example.

Figure 4 is a potential way of sorting core outcome sets stratified by conditions/diseases.

Appendix C. Identified Core Outcome Sets. Additional.

Populations and Outcomes

Disease (Dx) Category

Dx Name (No included)

Core Outcome Sets Reference(s)

AUTOIMMUNE DISEASES

Juvenile idiopathic arthritis

Pain, fatigue, activity limitations, overall physical and mental health impact, work/school/housework ability and productivity, severity of uveitis (measured as anterior chamber cell grade) and duration of active inflammation

OMERACT 2018 JIA (Delphi Survey)

Lupus

Immunological tests for dx activity and severity, the kidney function parameters, Health status, QoL, cost effectiveness, mortality (as part of adverse effect)

EULAR Task Force

PRINTO core outcome sets

HIV

All-cause mortality, cost/cost effectiveness for patients, QoL, CD4 count, viral load, hospitalization, antiretroviral treatment, mental health/social interaction, knowledge/attitudes, health services access/uptake

SLR

BLOOD DISORDERS
Sickle cell Pain, depression/negative emotional impact, fatigue, physical, cognitive domains, occupational status assessment, self-efficacy

ASH and USFDA

CONGENITAL CONDITIONS
Congenital heart defects QoL, HRQoL/perceived health status, growth and development, Physical activity level, arrhythmias, heart failure, survival, pregnancy, financial burden, productivity, anxiety, depression

ICHOM announced the release of a Congenital Heart Disease Standard Set (CHDSS)

Spina bifida

Obesity, health-related quality of life, levels of physical fitness and physical activity

Childhood condition Review

GENETIC DISORDERS
Cystic fibrosis Reduction in muscle mass, decrease in lung function, decrease in habitual physical activity levels, malnutrition, and/or an use of oxygen as energy substrate (reduced exercise capacity) Childhood condition review
GI DISEASES
Inflammatory bowel disease Survival, disease activity/ remission, colorectal cancer, anemia, treatment-related complications, healthcare utilization IBD-related admissions, emergency room visits and patient-reported outcomes such as QoL, nutritional status and impact of fistulae SLR (Delphi)
KIDNEY DISEASES

Chronic kidney disease

Life participation, survival, kidney function, infection

Standardized Outcomes in Nephrology—Children and Adolescents (SONG-Kids) (Delphi)

METABOLIC CONDITIONS

Type 1 Diabetes

Hypoglycemia, hyperglycemia, time in range, diabetic ketoacidosis (DKA), and patient-reported outcomes (PROs),

Standardizing Clinically Meaningful Outcome Measures Beyond HbA1c for Type 1 Diabetes

Diabetes

Glycemic control, global QoL, ADL, BW, Kidney function, hyperglycemia, hypoglycemia, blindness, neuropathy, gangrene, non-fatal MI, HF, CVD, hospitalization, Hyperglycemic emergencies, medicine related side effects, overall survival, mortality

SCORE-IT study (Delphi)

NEUROCOGNITIVE AND BEHAVIORAL DISORDERS

Autism

Anxiety, distress, hypersensitivity, sleep problems, happiness, relationships with siblings and parental stress. Faddiness about food, aggression to others, toileting difficulties

SLR1

SLR2

ADHD

DSM-criteria-based rating scales, Clinical Global Impression scales, direct observation and others

SLR

NEUROLOGICAL CONDITIONS

Cerebral palsy Impaired selective voluntary muscle control, muscle paresis, and spasticity, limited performance in exercise tests and training exercises, aerobic and anaerobic capacity, agility and muscle strength, HRQoL

Childhood condition review

Epilepsy Complete seizure freedom, seizure frequency, ability to perform normal day-to-day activities, cognitive function and QoL

Delphi Survey

Mulitple Sclerosis MRI, cumulative new active lesions, Cognitive function through neuropsychological tests, fatigue, depression, and quality of life International Pediatric MS Study Group

REPRODUCTIVE CONDITIONS

Polycystic ovary syndrome (PCOS)

Final core outcome set included 3 generic outcomes (BMI, quality of life, treatment satisfaction).

Harmonizing research outcomes for polycystic ovary syndrome (Delphi)