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Models of Care That Include Primary Care for Adult Survivors of Childhood Cancer

Research Protocol

I. Background

Statement of the Problem and Decisional Dilemmas

The American Cancer Society estimates that there were over 110,000 cancer survivors age 0–19 as of January 1, 2019; when added to adult survivors of childhood (age 0–14) and adolescent (age 15–19) cancer, the total number of survivors approaches 400,000.1 The growth in the number of survivors reflects new and more effective therapies, better risk stratification, and progress in supportive care that have improved treatment outcomes over the past decades, with 5-year relative survival increasing from 58 percent (1975–1977) to 84 percent (2008–2014) for children and from 68 percent (1975–1977) to 85 percent (2008–2014) for adolescents.1 Unfortunately, due to their cancer and its treatment, the majority of adult survivors experience life-long, chronic morbidities such as cardiomyopathy, metabolic syndrome, and subsequent malignant neoplasms.2-6 However, the impacts of cancer and its treatment are not uniform, with risks related to the previous cancer, its therapy, genetic predispositions, lifestyle behaviors, and comorbid health conditions.7 Thus, the National Academy of Medicine (formerly the Institute of Medicine) recommends life-long follow-up based on these factors.8

There is a lack of clarity regarding the appropriate models of survivorship care. Models of care range from specialized survivorship follow-up to general oncology follow-up to primary care follow-up. Multiple barriers exist that preclude many childhood cancer survivors from receiving specialized long-term follow-up care, but other barriers exist to receipt of quality survivorship care in primary care.9,10

Given the growing number of childhood cancer survivors, the extensive morbidity and mortality experienced by these survivors, the need for risk-based survivorship care, and the lack of clarity regarding the appropriate models of childhood survivorship care, the key decisional dilemmas include (1) what models of childhood cancer survivorship care improve short- and long-term outcomes, for which survivors, and under what circumstances and (2) what tools, trainings, resources, and processes can promote quality survivorship care across the various models? The National Cancer Institute (NCI) requested a realist review as part of a series of projects for The Childhood Cancer Survivorship, Treatment, Access, and Research (STAR) Act to better understand the state of the science and ultimately improve the care and quality of life for childhood and adolescent cancer survivors.

Models of Care

There is no consistent taxonomy for survivorship models of care used in the literature. The American Society of Clinical Oncology describes eight different models, classified primarily based on provider(s) and setting: oncology specialist care, multi-disciplinary survivorship clinics, disease/treatment-specific survivorship clinics, general survivorship clinics, consultative survivorship clinics, integrated survivorship clinics, community generalist model, and shared-care (see Appendix A for a more detailed description).11

However, studies generally have limited data on patterns of care receipt to use to classify models and tend to use broader categories. Using data from the Childhood Cancer Survivor Study, Nathan et al. organized health care receipt as follows: no health care, general medical care, general survivor-focused care, and risk-based survivor-focused care.7 Oeffinger et al. focused on health care system interactions: general contact, general physical examination, cancer-related medical visit, cancer center visit.12 Mueller et al. organized care by provider type seen: primary care provider, specialty care physician, nurse practitioner/physician assistant, survivorship clinic team.13 Surveys of providers have asked about categories such as continued care in pediatric oncology, referral to primary care, shared-care with primary care, and follow-up through a specialized long-term follow-up program.14-16 One systematic review simply categorized models as general practitioner (GP) led vs. shared care between a GP and pediatric oncology or a late-effects clinic.17

Resources

In contrast to the taxonomy for the models of survivorship care, the taxonomy for the tools/training/resources/processes (hereinafter "resources") is more straightforward. Surveys conducted by Nathan et al. and Suh et al. have asked about the value of options such as patient-specific standardized letters; long-term follow-up guidelines; expedited routes of contact for consultation, re-referral, support services; and websites, medical education, pamphlets.15,16 A systematic review by Singer et al. identified a well-organized transition, treatment summary, survivorship care plan, education of generalist providers, and guidelines as components of successful follow-up.17

Project Aims

Our project has two aims: (1) to identify and analyze models of survivorship care that include primary care using realist synthesis and (2) to identify available tools, training, and resources for childhood cancer survivorship and use realist synthesis to understand how and why they produce effects on health outcomes. To accomplish these aims we will: (1) identify models of survivorship care and analyze the program theories (underlying ideas and assumptions) about how they are intended to work, (2) identify available tools, training, and resources for childhood cancer survivorship and analyze the program theories underlying how they are intended to be implemented, and (3) test and refine the identified program theories through reviewing empirical evidence evaluating the use of models and resources in practice to explain how different contexts shape the mechanisms through which they work.

II. Methods/Approach

A realist synthesis or review answers the broad question "What works, how, why, for whom, to what extent and in what circumstances, in what respect and over what duration?" It is underpinned by a generative model of causation and posits that the underlying mechanisms (M) through which an intervention works and its outcomes (O) are shaped by contextual features (C) such as the way the intervention is designed, how it is implemented, in what settings, and the different characteristics of recipients. It seeks to build explanations about how contextual features shape the mechanisms through which interventions work and their outcomes that can be expressed as CMO configurations. The review process aims to first identify the underlying ideas and assumptions about how an intervention works (known as program theories) and then to synthesize empirical evidence to identify how different contexts trigger different mechanisms that result in different intended and unintended outcomes. The product of a realist review is a 'transferable' theory that describes what works for O, for what individuals, in what kinds of situations (C). We will develop and refine theory (around the CMO configurations) for models of and resources for care of adult survivors of childhood cancer.

Key Questions

Questions were initially identified by NCI and AHRQ and focused through a topic refinement process. We have developed contextual questions to guide our preliminary discussions with the stakeholders, as well as the specific review questions (or key questions) to be addressed.

Contextual Questions (CQ)

CQ1. How is effectiveness defined and measured for survivorship care models for adult survivors of childhood cancer?

CQ2. What are the models of survivorship care for adult survivors of childhood cancer?

  1. Which of these models include primary care?
    1. What is the evidence of effectiveness of the different models that include primary care?

CQ3. What survivorship care resources are available for adult survivors of childhood cancer and their families?

  1. What are the intended outcomes of the different resources available for adult survivors of childhood cancer and their families?
  2. What is the evidence of effectiveness of the different resources available for adult survivors of childhood cancer and their families?
  3. What are the monetary costs to access these resources?

CQ4. What survivorship care resources are available to providers who care for adult survivors of childhood cancer?

  1. What are the intended outcomes of the different resources available to care providers?
  2. What is the evidence of effectiveness of the different resources available to care providers?
  3. What are the monetary costs to access these resources?

Key Questions (KQ) for the Realist Review

KQ1. For whom and under what circumstances could different survivorship care models for adult survivors of childhood cancer that include primary care be effective?

  1. What are the key mechanisms by which these models could be effective?
  2. What are important contexts that determine whether different mechanisms could be effective?

KQ2. For whom and under what circumstances could different survivorship care resources for adult survivors of childhood cancer be effective in achieving their intended outcomes?

  1. For survivors and their families
    1. What are the key mechanisms by which these resources could lead to their intended outcome?
    2. What are important contexts that determine whether different mechanisms could lead to outcomes?
  2. For care providers
    1. What are the key mechanisms by which these resources could lead to their intended outcome?
    2. What are important contexts that determine whether different mechanisms could lead to outcomes?

For the purposes of this project, the term "resources" includes:

  • long-term follow-up guidelines
  • educational materials directed at either patient/family or care providers regardless of media (i.e., electronic, hard copy),
  • in-person or virtual trainings (i.e., workshops, conferences, CME courses) directed at either patient/family or care providers,
  • patient care documents (i.e., patient-specific standardized letters, treatment summaries, survivorship care plans), and
  • survivorship care management processes (i.e., expedited routes of contact for consultation, re-referral, support services; methods for digitizing and securely distributing health records; and other provider-to-provider and provider-to-patient communications)

PICOTS

We will adapt the PICOTS framework (populations, interventions, comparators, outcomes, timing, and setting) to inform our realist review. The PICOTS include but are not limited to the following.

Population(s)

  • Adult survivors of childhood cancer (cancer diagnosed prior to age 21years old) with no evidence of clinical disease; and their families
  • Care providers of adult survivors of childhood cancer

Interventions

  • Models of childhood cancer survivorship care for use in adult survivors
    • Models of childhood cancer survivorship care for use in adult survivors that include primary care
  • Survivorship resources available to adult survivors of childhood cancer and their families
  • Survivorship resources available to care providers of adult survivors of childhood cancer

Comparators

  • Optional (will not require a comparison)

Outcomes

List of outcomes will be informed by contextual questions but may include:

  • Intermediate patient health outcomes
  • Morbidity
  • Mortality
  • Relapse
  • Quality of life
  • Psychosocial outcomes
  • Mental health outcomes
  • Caregiver burden
  • Satisfaction with care
  • Educational attainment
  • Adherence with care
  • Cost and resource utilization
  • Unintended consequences
  • Additional burdens
  • Late effects—new cancers, cardiac or respiratory issues, etc. from original treatment

Timing

  • After the transition from childhood cancer care

Settings

  • All care settings

Work Completed to Date

We have completed initial discussion with our stakeholders. Stakeholders are individuals with a diverse range of perspectives including clinicians with expertise in cancer survivorship and survivorship care; clinicians with expertise in cancer survivorship research; and childhood cancer survivorship patient advocates and caregivers.

We convened three small group meetings of the stakeholders to help clarify the relevant concepts related to models of care and resources. This was the first meeting with stakeholders where information was gathered to assist in development of the initial program theory.

Overview of Iterative Approach

As with all realist reviews, our methods will be iterative, including multiple searches of the literature and consultations with stakeholders. The objective is to develop an initial program theory and then refine it as our understanding grows based on the literature and our stakeholders' input. For clarity, the term "model(s)" will only be used to refer to "models of care," and "theory(ies)" will only be used to refer to program theory(ies) and mid-range theory(ies).

  • We will follow an iterative processWe will develop an initial program theory by engaging stakeholders (completed) and reviewing mid-range theories and literature that describes expectations for models of care and resources.18,19 This process aims to identify evidence from a range of sources, including qualitative, quantitative, and mixed-methods studies. In addition to searching the published literature in a non-systematic manner to identify ideas, we will search for other policy and guidance documents.20
  • Following the development of the initial program theory, we will conduct targeted literature searches and consult with our stakeholders to refine this theory.

Below, we describe the approaches will use for consulting with the stakeholders, searching, screening, abstracting, and synthesizing the literature throughout our process of theory refinement.

Stakeholders

We will engage our stakeholders in meetings through the progression of the realist review. As noted above, we have engaged with them regarding the development of the initial program theory and they will be engaged at least two additional times. Meetings will take place via webinar. In order to allow for open and candid discussions, multiple webinars will be held at each time point—with at least one for clinicians and researchers and at least one for caregivers and advocates. In the meetings with both groups, our approach will be the same: we will present our initial (and then refined) program theories and elicit their feedback regarding how the proposed theory reflects (or fails to reflect) their expert understanding of the issues and their experiences with care for adult survivors of childhood cancer. A realist synthesis expert will be engaged separately to advise on realist methods, but may be asked to attend other webinars as well.

Searches

Our search process will be iterative and will evolve over time depending on the findings of each level of searching. As described earlier, we will search first for information on mid-range theories and other ideas about how the models of care and resources are intended to work to inform development of our initial program theory. The initial exploratory searching to identify relevant data on childhood cancer survivorship will include:

  • Collecting information from the team regarding their knowledge of mid-range theories related to access to care, knowledge specialization, coordination across the continuum of care, and uptake and use of resources.
  • Searching for opinion pieces, editorials, and commentaries about how models of care and resources are intended to work.
  • Convening a second meeting with the stakeholders to present the initial program theory that we will have developed, obtain their feedback on how it does (or does not) reflect their experience and understanding of the issues, and obtain their recommendation for additional theories that may be useful for integration.21

We will next conduct additional searches to refine our theory by identifying empirical evidence that supports or refutes the context, mechanism, outcome configurations outlined in our initial program theory. Searching for this stage will include:

  • Searching three databases (PubMed, CINAHL, PsycInfo). See Appendix B for a preliminary search strategy.
  • Searching other sources, such as websites suggested by stakeholders.
  • Conducting a general search of Google Scholar to identify documents relevant to ‘childhood cancer’ (or related terms) and survivorship.

We will use snowballing and berry picking22 with key studies. As relevant sources are identified we will conduct 'cluster searching'.21-23 This is part of the purposive sampling approach to identify 'clusters' of data from connected documents. We will use methods similar to those described above (i.e., searching databases, websites, Google Scholar) to identify clusters of information that support the refinement. Search terms will be informed by the initial program theory. We will not limit our searches by study design, or location of study; however, we will limit our search to English-language studies only.

The above second stage of searching to refine the program theory may occur more than once. As the program theory evolves, we will re-conduct searches to support or refine it further.

Screening

Studies will be included based on their potential to inform development or testing of program theories. Screening will occur iteratively along the lines described above for searching. For theory development, we will include resources that inform theory development; for theory refinement we will include studies based on their capacity to support or refute the initial program theory.

The organizing elements are outlined in the PICOTS table. To screen abstracts, we will follow methods described by Carrieri.24 Each abstract will be screened by one of the core team members. A random sample of 10 percent of the screened abstracts will be checked by another senior team member for consistency. Disagreements that cannot be resolved by the two screeners will be discuss and resolved by team discussion.24 The same process will be applied to full text articles.

Data Extraction and Organization

Information will be extracted according to the two phases described in the iterative approach. For theory development, we will focus on extracting key ideas; for theory refinement, we will focus on extracting evidence to support or refine fragments of the initial program theory.

From identified studies for theory refinement only, we will extract characteristics (e.g., study design, study period, and follow-up), context (e.g., setting, number of model components, policy and historical context), mechanism (e.g., author-identified mechanisms), and outcomes (e.g., satisfaction with care, morbidity). We will use MAXQDA (VERBI Software, Germany), a qualitative data analysis tool, for the collection, coding, and analysis of information from identified studies. Data extraction will be completed by one study team expert or methodologist and a 10 percent random sample will be checked by a second reviewer.

For theory refinement, included studies will be divided into those that can make major or minor contributions in informing and testing the program theory.24 This classification will be completed by one team member and a random sample of 10 percent of the studies will be checked by a second senior reviewer. Our criteria for classifying studies are adapted from the categorization criteria from Carrieri et al., 2018:24

Major:

  • Studies which contribute to the research questions and are conducted in a childhood cancer survivorship context.
  • Studies which contribute to the research questions and are conducted in contexts with similarities to childhood cancer survivorship such as adult survivors of adult cancer; and adult survivors of serious childhood illness (e.g., meningitis).

Minor:

  • Studies conducted in models of care that are markedly different from childhood cancer survivorship but where the mechanisms could plausibly operate in the context of childhood cancer survivorship models.

Data Synthesis

For theory development, data synthesis will focus on identifying and incorporating the key ideas in the initial program theory.

For theory refinement, data synthesis will involve the following processes. Included studies will be assessed as to whether (i) the data are relevant to the theories being tested (relevance) and (ii) if the research methods support the conclusions (rigor). Determinations of rigor will be informed by a set of questions derived from the Critical Appraisal Skills Programme (CASP) Checklist for Qualitative Research.25 While not a formal quality assessment, these questions will help determine whether the method used is credible and trustworthy. Examples include: Is the methodology appropriate for addressing the research goal? Has the researcher justified the research design? Were the data collected in a way that addressed the research issue? Assessment will be completed by one team member and a 10 percent sample will be checked by a second reviewer.

If a study is determined to be relevant and has sufficient rigor based on the CASP questions above, we will review it to determine if its contents provide data that may be interpreted as functioning as context, mechanism, or outcome, or associations between any two (C-M, C-O, M-O).26 We will judge the context-mechanism- outcome configurations (CMOCs) per Abrams, 201818: What is the CMOC (partial or complete) for the data that may be interpreted as functioning as context, mechanism or outcome?26 Is there further data to inform this particular CMOC contained within this document or other documents? If so, which other documents? How does this particular CMOC relate to other CMOCs that have already been developed?

We will review included studies to determine how a particular (full or partial) CMOC relates to the program theory. We will also search for further data to inform how the CMOC relates to the program theory. If this information is not available in the document under review, we will look for information in other related documents. Once we complete the extraction of information of the CMOC and any supporting data we will discuss whether the program theory needs to be changed.18

Additionally, we will use interpretive cross-case comparison to explain how and why outcomes occurred. This will be accomplished by comparing interventions that have worked to those that have not to help us understand how context has impacted findings. We will continue to interpret that data to determine if the relationships between CMO change.24

Throughout the course of this realist review we will move between examples in the literature, and iterative searching to test the program theory. We will consider the following analytic thinking process: look for evidence on one outcome in one source enabling insights about other outcomes in other sources (juxtaposition); when results differ in similar circumstances investigate further to find explanations (reconciling); look at methodological strengths or weaknesses (adjudication of sources); explain where and why outcomes differ in particular contexts (consolidation).24

Refine Program Theory

Moving through the iterative process above, we will refine the initial program theory.19,24 We will look at the range of generalities to move from a lower-level to a higher-level of describing which models of care or resources work in what contexts, how, and why. As we approach the conclusion of the project, we will conduct a final meeting with our stakeholders. During this final round of webinars, we will present our refined program theory and CMOCs and elicit feedback regarding whether they reflect our stakeholders’ understanding and experience of the issues. Based on this feedback, we will make final revisions to the refined program theory.

III. References

  1. Cancer Treatment & Survivorship Facts & Figures 2019-2021 American Cancer Society. Atlanta, GA: 2019.
  2. Hudson MM, Ness KK, Gurney JG, et al. Clinical ascertainment of health outcomes among adults treated for childhood cancer. Jama. 2013 Jun 12;309(22):2371-81. doi: 10.1001/jama.2013.6296. PMID: 23757085.
  3. Bhakta N, Liu Q, Ness KK, et al. The cumulative burden of surviving childhood cancer: an initial report from the St Jude Lifetime Cohort Study (SJLIFE). Lancet (London, England). 2017 Dec 9;390(10112):2569-82. doi: 10.1016/s0140-6736(17)31610-0. PMID: 28890157.
  4. Armstrong GT, Kawashima T, Leisenring W, et al. Aging and risk of severe, disabling, life-threatening, and fatal events in the childhood cancer survivor study. Journal of clinical oncology : official journal of the American Society of Clinical Oncology. 2014 Apr 20;32(12):1218-27. doi: 10.1200/jco.2013.51.1055. PMID: 24638000.
  5. Meacham LR, Chow EJ, Ness KK, et al. Cardiovascular risk factors in adult survivors of pediatric cancer--a report from the childhood cancer survivor study. Cancer epidemiology, biomarkers & prevention : a publication of the American Association for Cancer Research, cosponsored by the American Society of Preventive Oncology. 2010 Jan;19(1):170-81. doi: 10.1158/1055-9965.epi-09-0555. PMID: 20056636.
  6. Geenen MM, Cardous-Ubbink MC, Kremer LC, et al. Medical assessment of adverse health outcomes in long-term survivors of childhood cancer. Jama. 2007 Jun 27;297(24):2705-15. doi: 10.1001/jama.297.24.2705. PMID: 17595271.
  7. Nathan PC, Greenberg ML, Ness KK, et al. Medical care in long-term survivors of childhood cancer: a report from the childhood cancer survivor study. Journal of clinical oncology: official journal of the American Society of Clinical Oncology. 2008 Sep 20;26(27):4401-9. doi: 10.1200/jco.2008.16.9607. PMID: 18802152.
  8. Institute of M, National Research Council National Cancer Policy B. In: Hewitt M, Weiner SL, Simone JV, eds. Childhood Cancer Survivorship: Improving Care and Quality of Life. Washington (DC): National Academies Press (US) Copyright 2003 by the National Academy of Sciences. All rights reserved.; 2003.
  9. Hudson MM. A model for care across the cancer continuum. Cancer. 2005 Dec 1;104(11 Suppl):2638-42. doi: 10.1002/cncr.21250. PMID: 16258932.
  10. Nathan PC, Ford JS, Henderson TO, et al. Health behaviors, medical care, and interventions to promote healthy living in the Childhood Cancer Survivor Study cohort. Journal of clinical oncology : official journal of the American Society of Clinical Oncology. 2009 May 10;27(14):2363-73. doi: 10.1200/jco.2008.21.1441. PMID: 19255308.
  11. American Society of Clinical Oncology. Models of long-term follow-up care. 2020. Accessed on June 10, 2020.
  12. Oeffinger KC, Mertens AC, Hudson MM, et al. Health care of young adult survivors of childhood cancer: a report from the Childhood Cancer Survivor Study. Annals of family medicine. 2004 Jan-Feb;2(1):61-70. doi: 10.1370/afm.26. PMID: 15053285.
  13. Mueller EL, Park ER, Kirchhoff AC, et al. Insurance, chronic health conditions, and utilization of primary and specialty outpatient services: a Childhood Cancer Survivor Study report. Journal of cancer survivorship : research and practice. 2018 Oct;12(5):639-46. doi: 10.1007/s11764-018-0700-1. PMID: 29943170.
  14. Henderson TO, Hlubocky FJ, Wroblewski KE, et al. Physician preferences and knowledge gaps regarding the care of childhood cancer survivors: a mailed survey of pediatric oncologists. Journal of clinical oncology : official journal of the American Society of Clinical Oncology. 2010 Feb 10;28(5):878-83. doi: 10.1200/jco.2009.25.6107. PMID: 20038717.
  15. Nathan PC, Daugherty CK, Wroblewski KE, et al. Family physician preferences and knowledge gaps regarding the care of adolescent and young adult survivors of childhood cancer. Journal of cancer survivorship : research and practice. 2013 Sep;7(3):275-82. doi: 10.1007/s11764-013-0271-0. PMID: 23471729.
  16. Suh E, Daugherty CK, Wroblewski K, et al. General internists' preferences and knowledge about the care of adult survivors of childhood cancer: a cross-sectional survey. Annals of internal medicine. 2014 Jan 7;160(1):11-7. doi: 10.7326/m13-1941. PMID: 24573662.
  17. Singer S, Gianinazzi ME, Hohn A, et al. General practitioner involvement in follow-up of childhood cancer survivors: a systematic review. Pediatric blood & cancer. 2013 Oct;60(10):1565-73. doi: 10.1002/pbc.24586. PMID: 23813795.
  18. Abrams R, Wong G, Mahtani KR, et al. Understanding the impact of delegated home visiting services accessed via general practice by community-dwelling patients: a realist review protocol. BMJ open. 2018 Nov 13;8(11):e024876. doi: 10.1136/bmjopen-2018-024876. PMID: 30429150.
  19. Greenhalgh J, Pawson R, Wright J, et al. Functionality and feedback: a protocol for a realist synthesis of the collation, interpretation and utilisation of PROMs data to improve patient care. BMJ open. 2014 Jul 22;4(7):e005601. doi: 10.1136/bmjopen-2014-005601. PMID: 25052175.
  20. Morton T, Atkinson T, Brooker D, et al. Sustainability of community-based interventions for people affected by dementia: a protocol for the SCI-Dem realist review. BMJ open. 2019 Jul 26;9(7):e032109. doi: 10.1136/bmjopen-2019-032109. PMID: 31350255.
  21. Booth A, Briscoe S, Wright JM. The "realist search": A systematic scoping review of current practice and reporting. Research synthesis methods. 2020 Jan;11(1):14-35. doi: 10.1002/jrsm.1386. PMID: 31714016.
  22. Booth A, Harris J, Croot E, et al. Towards a methodology for cluster searching to provide conceptual and contextual "richness" for systematic reviews of complex interventions: case study (CLUSTER). BMC medical research methodology. 2013 Sep 28;13:118. doi: 10.1186/1471-2288-13-118. PMID: 24073615.
  23. Doing Realist Research. 2020/06/17. 55 City Road, London: SAGE Publications Ltd. 2018. DOI: 10.4135/9781526451729.
  24. Carrieri D, Briscoe S, Jackson M, et al. 'Care Under Pressure': a realist review of interventions to tackle doctors' mental ill-health and its impacts on the clinical workforce and patient care. BMJ open. 2018 Feb 2;8(2):e021273. doi: 10.1136/bmjopen-2017-021273. PMID: 29420234.
  25. CASP. Qualitative Research Checklist. Secondary Qualitative Research Checklist. 2013.
  26. The RAMESES Projects. 2020. Accessed on June 10, 2020.
  27. Davidson P, Halcomb E, Hickman L, et al. Beyond the rhetoric: what do we mean by a 'model of care'? The Australian journal of advanced nursing: a quarterly publication of the Royal Australian Nursing Federation. 2006 Mar-May;23(3):47-55. PMID: 16568879.
  28. R P. Evidence based policy: a realist perspective: SAGE Publications; 2006.
  29. An Introduction to Program Theory. Social Solutions. Accessed on September 23 2020.

IV. Definition of Terms

List of Acronyms

Acronym Definition
AHRQ Agency for Healthcare Research and Quality
AYA Adolescent/young adult
CASP Critical Appraisal Skills Programme
CME Continuing medical education
CMO Context, Mechanism, Outcome
CMOC Context, Mechanism, Outcome Configurations
CQ Contextual Questions
EPC Evidence-based Practice Center
GP general practitioner
IOM Institute of Medicine
KI Key Informants
KQ Key Questions
MD Medical Doctor
NAM National Academy of Medicine (Health and Medicine Division (HMD) of the National Academies)
NCI The National Cancer Institute
NP Nurse Practitioner
PA Physician Assistant
PCP primary care provider
PICOTS Populations, Interventions, Comparators, Outcomes, Timing, and Setting
STAR The Childhood Cancer Survivorship, Treatment, Access, and Research
TEP Technical Expert Panel
TOO Task Order Officer

 

List of Terms

Term Definition (as used in this report)
Resources The term "resources" includes guidelines, educational materials, in-person or virtual trainings, patient care documents, and survivorship care management processes.
Models Refers to models of survivorship care.
Models of care Approaches to organizing and delivering care, in this case to adult survivors of childhood cancer.27
Realist review A realist review answers the broad question "What works, how, why, for whom, to what extent and in what circumstances, in what respect and over what duration? It is used for complex interventions to explore systematically how contextual factors influence the link between intervention and outcome.28
Rigor Whether the research methods support the conclusions, are credible, and trustworthy.18,20,28
Relevance Whether the data are relevant to the theories being tested; whether a document can contribute to developing or testing a theory.20,24,27
Theories Refers to initial program theory, program theory, and mid-range theory
Theory An explanation of how an intervention may or may not work.
Program Theory

The assumption that an intervention's design, activities, and execution will lead to the achievement of the outcomes you intend for your clients.29

 

V. Summary of Protocol Amendments

If needed, protocol amendments will be added in the future.

VI. 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. There were no public comments.

VII. Stakeholders

Stakeholders 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; they are also the end users of research, including patients and caregivers, practicing clinicians, relevant professional and consumer organizations, purchasers of health care, and others with experience in making health care decisions. They are 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 results in a thoughtful, relevant systematic review. Therefore, study questions, design, and methodological approaches do not necessarily represent the views of individual Stakeholders.

Within this realist review, the Stakeholder role is to provide input into identifying the Key Questions, developing the initial theory and providing feedback on the refined theory. Stakeholders are not involved in analyzing the evidence or writing the report and have not reviewed the report, except as given the opportunity to do so through the peer or public review mechanism.

Stakeholders 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 role as clinical/content experts as well as end-users, individuals are invited to serve as Stakeholders 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.

VIII. 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 preparation of 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 three months after the 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 may not have any financial conflict of interest greater than $5,000. Peer reviewers who disclose potential business or professional conflicts of interest may submit comments on draft reports through the public comment mechanism.

IX. 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. Related financial conflicts of interest that cumulatively total greater than $1,000 will usually disqualify EPC core team investigators.

X. Role of the Funder

This project was funded under Contract No. 75Q80120D00003 from the Agency for Healthcare Research and Quality, U.S. Department of Health and Human Services. The AHRQ Task Order Officer reviewed 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 the Agency for Healthcare Research and Quality or the U.S. Department of Health and Human Services.

XI. Registration

We will not be registering this realist review protocol.

Appendix A. Eight Models of Cancer Survivorship Care as Described by American Society of Clinical Oncology.11

Model Description
Oncology Specialist Care
  • Follow-up care occurs in the oncology setting with treating oncologist
  • Can be implemented in private practice, community hospitals or cancer centers
  • Can be implemented in disease-specific clinics or modality-specific practices
  • Communication and coordination with PCP
Multi-disciplinary Survivorship Clinics
  • Care provided by a specialized team (i.e. oncologist, psychologist, cardiologist, etc.) in a separate clinical area
  • Can be implemented at cancer centers and other facilities with extensive resources
  • Ideal patient populations include: pediatric cancer survivors, adolescent/young adult (AYA) survivors and adult survivors of pediatric cancer
  • Communication and coordination with PCP
Disease/Treatment-specific Survivorship Clinics
  • Care can be provided by a physician, NP, PA, or multispecialty team
  • Care provided in oncology setting
  • Can be developed for a common diagnosis, such as breast cancer, or treatment modality, such as stem cell transplant
  • Can be developed in private practice, community hospital, or cancer center
  • Communication and coordination with Primary Care
General Survivorship Clinics
  • Care can be provided by MD, NP, or PA (not multispecialty)
  • Can be implemented at a cancer center, community hospital, or private practice
  • Communication and coordination with Primary Care
Consultative Survivorship Clinics
  • Care can be provided by MD, NP, or PA
  • One-time visit with no follow-up, but has potential for another visit
  • Ideal for providing treatment summary and care plan
  • Can be implemented at a cancer center, community hospital, or private practice
  • Communication and coordination with Primary Care
Integrated Survivorship Clinics
  • Embedded in the treatment focused oncology setting
  • Care can be provided by an MD, NP, or PA
  • Care is ongoing
  • Can be implemented at a cancer center, community hospital, or private practice
  • Provider communicates and coordinates care with primary care provider and specialists as needed
Community Generalist Model
  • Survivorship care is provided by primary care physician, NP, or PA
  • Care setting can be at a health care system or private practice
  • Communication and coordination with PCP
Shared-care
  • Care for all survivors is coordinated between oncology specialist, and PCP generalists

 

Appendix B: Preliminary Literature Search Strategy (PubMed)

# Term Results Notes
1 "Cancer Survivors"[Mesh]   cancer survivor/survivorship string
2 Cancer[tiab]    
3 survivorship[Mesh]    
4 Survivors[mh]    
5 survivorship[tiab]    
6 survivors[tiab]    
7 survivor[tiab]    
8 1 OR 2 AND (3 OR 4 OR 5 OR 6 OR 7)) 29,662  
9 "health planning"[tiab]    
10 "health care planning"[tiab]    
11 "patient care planning"[tiab]    
12 "patient care bundles"[tiab]    
13 "patient care bundle"[tiab]    
14 "patient care documents"[tiab]    
15 "patient care document"[tiab]    
16 "patient care guidelines"[tiab]    
17 "educational materials"[tiab]    
18 "education materials"[tiab]    
19 Workshop[tiab]    
20 Workshops[tiab]    
21 "training program"[tiab]    
22 "training programs"[tiab]    
23 "management processes"[tiab]    
24 "management process"[tiab]    
25 "management plans"[tiab]    
26 "management plan"[tiab]    
27 "care plan"[tiab]    
28 "care plans"[tiab]    
29 "care model"[tiab]    
30 "Care models"[tiab]    
31 "Health Planning Guidelines"[Mesh] 4086 Did not include Education[mh] or “models, theoretic*”[mh]
32 "Patient Care Planning"[Mesh]  63,386  
33 "Patient Care Bundles"[Mesh] 823  
34 9 OR 10 OR 11 OR 12 OR 13 OR 14 OR 15 OR 16 OR 17 OR 18 OR 19 OR 20 OR 21 OR 22 OR 23 OR 24 OR 24 OR 25 OR 26 OR 27 OR 28 OR 29 OR 30 OR 31 OR 32 OR 33 170,701
 
Resources and models string
 
35 8 AND 34 1091 Add term "narrative": 1
Add term "commentary": 12
Add term "editorial": 12
Add term Letter": 14
      136 reviews
Limit to: (child OR childhood OR pediatric OR paediatrics OR adolescent), 53