Video Abstract

Video Abstract

Close modal
OBJECTIVES:

In this study, we sought to establish priorities for a national research agenda for children and youth with special health care needs (CYSHCN) through a structured, multistakeholder, mixed-methods approach.

METHODS:

Using surveys, we solicited responses from >800 members of expert-nominated stakeholder organizations, including CYSHCN families, health care providers, researchers, and policymakers, to identify what research with or about CYSHCN they would like to see in a national research agenda. From 2835 individual free-text responses, 96 research topics were synthesized and combined. Using an adapted RAND/UCLA Appropriateness Method (a modified Delphi approach), an expert panel rated research topics across 3 domains: need and urgency, research impact, and family centeredness. Domains were rated on 9-point Likert scales. Panelist ratings were used to sort research topics into 4 relative-priority ranks. Rank 1 (highest priority) research topics had a median of ≥7 in all domains.

RESULTS:

The RAND/UCLA Appropriateness Method was used to prioritize CYSHCN research topics and depict their varying levels of stakeholder-perceived need and urgency, research impact, and family centeredness. In the 15 topics that achieved rank 1, social determinants of health (disparities and rurality), caregiving (family resilience and care at home), clinical-model refinement (effective model elements, labor divisions, telemedicine, and system integration), value (stakeholder-centered value outcomes, return on investment, and alternative payment models), and youth-adult transitions (planning, insurance, and community supports) were emphasized.

CONCLUSIONS:

High-priority research topics identified by CYSHCN experts and family leaders underscore CYSHCN research trends and guide important directions. This study is the first step toward an efficient and cohesive research blueprint to achieve highly-effective CYSHCN health systems.

What’s Known on This Subject:

Achieving comprehensive, effective, and coordinated systems for children and youth with special health care needs (CYSHCN) has been elusive. Realizing ideal health systems for CYSHCN requires research to concentrate on the most pressing issues identified by diverse stakeholders.

What This Study Adds:

As a step toward a national CYSHCN research agenda, diverse experts developed a research topic taxonomy, depicting varying levels of need and urgency, research impact, and family centeredness. Highest-ranked research topics were centered on social determinants of health, family resilience, value, and youth-adult transitions.

Children and youth with special health care needs (CYSHCN) have, or are at an increased risk for, chronic physical, developmental, behavioral, or emotional conditions and also require health and related services of a type or amount beyond that required by children generally.1  A system of services for CYSHCN (ie, a “family-centered network of community-based services designed to promote the healthy development and well-being of CYSHCN and their families”2 ) includes their health care, mental health, education, public health, and social services. This health system is an important improvement target because CYSHCN make up nearly 20% of US children,3  and although by definition they require more health services than otherwise healthy children,1  they are the most vulnerable to problems with access.4  Health system design impacts cost and care quality for children with chronic illness,5  and effective systems provide mechanisms to improve CYSHCN outcomes.6,7 

Health system innovation has increased over the last 3 decades as CYSHCN definitions, data, and policies have increased in prominence.1,2,814  The US Maternal and Child Health Bureau (MCHB) 6 Core Outcomes in 200115  and Association of Maternal and Child Health Programs (AMCHP) Standards for Systems of Care in 201416  have advanced progress in developing comprehensive CYSHCN systems.2  The MCHB outcomes (eg, comprehensive care within a medical home and adequate insurance) provide means to measure progress toward achieving the health systems CYSHCN require. The AMCHP Standards for Systems of Care also represent structures (eg, capacity for timely access to services) and processes (eg, transportation assistance provided to families with difficulty) presumed to improve CYSHCN health outcomes.

Despite these efforts, a reliable, effective, comprehensive system of care for CYSHCN has been elusive.2  Only 16% of US CYSHCN receive care meeting all 6 MCHB core outcomes; only 43% receive care within a medical home.3  To create health systems that achieve the best possible CYSHCN outcomes, research needs to be concentrated on the most pressing issues identified by diverse stakeholders. However, CYSHCN research has myriad potential topics and stakeholders. If research priorities are not cohesive, researchers could conduct substantial work without driving demonstrable forward progress in population-level CYSHCN health and health care. A guiding framework thoughtfully developed by stakeholders might facilitate more efficient, cohesive, and, ultimately, transformative research.

In 2017, the Health Resources and Services Administration MCHB established a cooperative agreement to create a national research network, the Children and Youth with Special Health Care Needs National Research Network (CYSHCNet) (http://www.cyshcnet.org). CYSHCNet is an interdisciplinary, multisite, collaborative network to lead, promote, and coordinate national research activities to inform a high-quality system of care for CYSHCN. The network’s initial charge was to create a research agenda reflective of multistakeholder priorities. This agenda could help (1) prioritize the most important health system questions facing stakeholders, (2) define research goals for issues of greatest importance, and (3) provide a blueprint for research that CYSHCN investigators and other stakeholders can follow.

As an initial step in creating this agenda, we undertook the mixed-methods RAND/UCLA Appropriateness Method (RAM),17  a rigorous modified Delphi process, to integrate literature review and expert opinion. RAM has previously been used to develop quality measures and design clinical models for CYSHCN.1821  Our objective was to synthesize the opinions of a national multidisciplinary group of CYSHCN experts, including family caregivers, to prioritize research topics facing CYSHCN. With this study, we take a foundational step toward developing a national research agenda for CYSHCN systems of care.

We adapted RAM to set priorities for a CYSHCN research agenda (Fig 1). We used the following terms and processes. First, we developed a set of research topics from responses to an open-ended survey and published literature. Next, stakeholders evaluated every research topic according to 3 different domains, that is, the research topic’s perceived (1) need and urgency, (2) research impact, and (3) family centeredness. During a moderated expert panel, stakeholders gave every research topic 3 ratings (ie, 1 score in each domain). Finally, by using the median ratings of the 3 domains, each research topic was assigned into a rank order reflecting the research topic’s priority from 1 (high) to 4 (low). The Colorado Multiple Institutional Review Board considered this work exempt from human subjects review.

FIGURE 1

Overview of appropriateness method to establish national research agenda priorities for CYSHCN. Research topics were generated from synthesized and aggregated open-ended survey responses, following an ongoing, consensus-seeking approach. Conceptually similar research topics were sorted into themes. Themes emerged from a content analysis of survey responses. Each research topic was rated using 3 rating domains, corresponding to perceived (1) need and urgency, (2) research impact, and (3) family centeredness. Domains were rated by using 9-point Likert scales: 1 to 3 (low) was used when a research topic represented a domain to a low degree; 4 to 6 was used when a research topic represented an uncertain domain; and 7 to 9 (high) was used when a research topic represented a domain to a high degree. To convey relative prioritization, each research topic was assigned a rank order: rank 1: median of ≥7 in all 3 domains and >7 in at least 2 of 3 domains; rank 2: median of ≥7 in all 3 domains and >7 in 1 of 3 domains; rank 3: median < 7 in 1 or 2 of 3 domains, or median = 7 for all domains; and rank 4: median < 7 in all 3 domains or any disagreement.

FIGURE 1

Overview of appropriateness method to establish national research agenda priorities for CYSHCN. Research topics were generated from synthesized and aggregated open-ended survey responses, following an ongoing, consensus-seeking approach. Conceptually similar research topics were sorted into themes. Themes emerged from a content analysis of survey responses. Each research topic was rated using 3 rating domains, corresponding to perceived (1) need and urgency, (2) research impact, and (3) family centeredness. Domains were rated by using 9-point Likert scales: 1 to 3 (low) was used when a research topic represented a domain to a low degree; 4 to 6 was used when a research topic represented an uncertain domain; and 7 to 9 (high) was used when a research topic represented a domain to a high degree. To convey relative prioritization, each research topic was assigned a rank order: rank 1: median of ≥7 in all 3 domains and >7 in at least 2 of 3 domains; rank 2: median of ≥7 in all 3 domains and >7 in 1 of 3 domains; rank 3: median < 7 in 1 or 2 of 3 domains, or median = 7 for all domains; and rank 4: median < 7 in all 3 domains or any disagreement.

Close modal

Between February 2018 and March 2018, we sought input from professionally, demographically, and geographically diverse stakeholders. Members of stakeholder organizations identified by the CYSHCNet Executive Committee (Supplemental Information) were invited to complete an open-ended electronic questionnaire (Qualtrics, Provo, UT) developed and pilot tested with CYSHCNet family leaders. In the questionnaire, respondents identified “one or two things that they would most like to see improved in the approach to health for CYSHCN,” and “what research with or about CYSHCN they would like to see in a national research agenda.” Participants had 2 weeks to reply, after which nonresponders received up to 2 reminders at 1-week intervals. Although an individual could be a member of >1 stakeholder organization, they were asked to complete the survey only once.

Surveys from 269 respondents yielded 2835 responses, which 3 team members coded, edited, and combined into 86 unique research topics through an iterative, consensus-seeking approach. We used conventional inductive content analysis22  to identify key themes emerging during synthesis and aggregation of the CYSHCN research topics. Study team members reviewed the final list of research topics and themes to ensure representation of all submitted ideas.

Although a systematic literature review was not feasible because of the breadth of the topic of health systems for CYSHCN, stakeholder organizations were asked to submit articles perceived to guide future CYSHCN research directions. The research team updated the final list of research topics on the basis of a review of the submitted articles.

Members of the CYSHCNet Executive, Steering, and Advisory Committees (each of which included family members of CYSHCN), the network’s MCHB Division of Research project officers, and members of the MCHB services branch for CYSHCN were invited to participate in the RAM expert panel. The personal and professional characteristics of the 24 RAM panelists are summarized in Table 1. Three RAM participants were coauthors (D.Z.K., K.K., and C.G.H.) to ensure that the interpretation and presentation of the results was consistent and balanced from clinical, research, and family participant perspectives. Panelists received rating materials and a written summary of the research topic development that included the qualitative analysis of survey responses and the articles submitted by stakeholders.

TABLE 1

RAM Participant Characteristics

CharacteristicsRAM Research Topic Development Survey (N = 269)RAM Expert Panel (N = 24)
n (%)n (%)
Female sex 189 (70) 15 (63) 
Age, y   
 18–44 89 (33) 6 (25) 
 45–54 67 (25) 6 (25) 
 55+ 88 (32) 12 (50) 
 Not answered 25 (9) 
States represented   
 No. unique states represented by participants 41a 15 
Race and/or ethnicity   
 White 214 (80) 16 (67) 
 African American 7 (3) 
 American Indian or Alaskan native 2 (1) 
 Asian American 13 (5) 4 (17) 
 Native Hawaiian or Pacific Islander 1 (0) 
 Hispanic 6 (2) 2 (8) 
 Multiracial or other 13 (5) 2 (8) 
 Not answered 13 (5) 
Relationship to CYSHCNb   
 CYSHCN 3 (1) 
 Parent or family member 61 (23) 6 (25) 
 Advocate, child or family 45 (17) 5 (21) 
 Health care professional 197 (73) 22 (92) 
Health care professional roleb   
 Primary care 76 (28) 9 (38) 
 Specialty care 71 (26) 5 (21) 
 Behavioral health or therapist 25 (10) 2 (8) 
 Administrator 20 (7) 4 (16) 
 Policy 16 (6) 8 (33) 
 Researcher 40 (15) 17 (71) 
 Otherc 43 (16) 8 (33) 
CharacteristicsRAM Research Topic Development Survey (N = 269)RAM Expert Panel (N = 24)
n (%)n (%)
Female sex 189 (70) 15 (63) 
Age, y   
 18–44 89 (33) 6 (25) 
 45–54 67 (25) 6 (25) 
 55+ 88 (32) 12 (50) 
 Not answered 25 (9) 
States represented   
 No. unique states represented by participants 41a 15 
Race and/or ethnicity   
 White 214 (80) 16 (67) 
 African American 7 (3) 
 American Indian or Alaskan native 2 (1) 
 Asian American 13 (5) 4 (17) 
 Native Hawaiian or Pacific Islander 1 (0) 
 Hispanic 6 (2) 2 (8) 
 Multiracial or other 13 (5) 2 (8) 
 Not answered 13 (5) 
Relationship to CYSHCNb   
 CYSHCN 3 (1) 
 Parent or family member 61 (23) 6 (25) 
 Advocate, child or family 45 (17) 5 (21) 
 Health care professional 197 (73) 22 (92) 
Health care professional roleb   
 Primary care 76 (28) 9 (38) 
 Specialty care 71 (26) 5 (21) 
 Behavioral health or therapist 25 (10) 2 (8) 
 Administrator 20 (7) 4 (16) 
 Policy 16 (6) 8 (33) 
 Researcher 40 (15) 17 (71) 
 Otherc 43 (16) 8 (33) 
a

All but 9 states (Alaska, Arkansas, Connecticut, Hawaii, Idaho, Maine, Nebraska, Nevada, and West Virginia) had at least 1 respondent.

b

Self-identified; respondents were allowed to pick as many as applied.

c

Roles identified in “other” included funder, public health professional, community-based services, and advocates.

During 2 rounds, in March 2018 and April 2018, panelists rated the degree to which each research topic represented 3 domains: need and urgency, research impact, and family centeredness. The need and urgency domain was defined as the influence the research topic had on the health and well-being of CYSHCN, the severity of consequences (eg, number of CYSHCN affected by the issue, mortality risk, severity of symptoms, or functional loss), and the presence of critical gaps in knowledge to solve the problem. Research impact was defined as the ability of the knowledge gained from this research topic to translate to meaningful progress in 5 to 10 years and the potential to lead to improvements in health status, quality of care, public health, or policy over the longer-term. Family centeredness was defined as how well the research topic reflected the experiences and priorities of most CYSHCN and their families.

Using a 9-point Likert scale, panelists gave each research topic 3 ratings (1 rating in each of the 3 domains). A rating of 1 to 3 indicated low representation, a rating of 4 to 6 indicated uncertain representation, and a rating of 7 to 9 indicated high representation of a domain. Consistent with RAM convention, disagreement was defined if one-third or more of panelists rated an item in the low range (ie, 1–3), whereas one-third or more of panelists rated the same item in the high range (ie, 7–9). Panelists could include comments to improve clarity of each research topic and to suggest additional research topics.

During round 1, panelists completed the ratings electronically over 2 weeks, with 2 weekly reminders sent to nonresponders. During round 2, panelists participated in an in-person 2-day meeting to discuss the round 1 ratings. Panelists received a copy of their ratings and de-identified summary tables with group distributions for each research topic. Meeting moderators (R.J.C. and C.S.) did not rate topics. The facilitated group discussion goal was to ensure panelists had a consistent understanding of terms and concepts. The aim was to identify legitimate areas of disagreement and not to build rating consensus.17  After adjustments to rating materials (eg, wording changes for clarity) panelists rerated the research topics.

After round 2, we summarized rating distributions, areas of agreement and disagreement, and frequencies of each rating for research topics. Anticipating that the ratings would be skewed toward higher numbers, we ranked research topics into groups to convey their relative prioritization. The following criteria were used to define each rank group.

  • Rank 1: topics had a median in the high range (≥7) for all 3 domains (need and urgency, research impact, and family centeredness) and a median of >7 in at least 2 of 3 domains;

  • Rank 2: median of ≥7 in all 3 domains and >7 in 1 of 3 domains;

  • Rank 3: median < 7 in 1 or 2 of 3 domains, or median = 7 for all domains; and

  • Rank 4: median < 7 in all 3 domains or any disagreement.

Finally, to determine if family stakeholder priorities were distinct from those obtained through the RAM process, we reanalyzed the results from the 8 family caregivers separately. Differences in the highest-ranked research topics between family caregivers and the whole group were summarized.

Survey respondents (N = 269) reflected multidisciplinary stakeholders from across the United States, with at least 1 respondent from 41 of 50 states (Table 1). Nearly one-quarter (24%) were CYSHCN or family members. Approximately three-quarters (73%) self-identified as health professionals, including primary (28%) and specialty (26%) care providers, behavioral health providers and therapists (10%), administrators and policy experts (13%), and researchers (15%). Because reliable denominators were unavailable for surveyed groups and respondents could identify with multiple groups, response rates were not calculated. Moreover, our goal was to achieve thematic saturation and a broad list of candidate research topics from stakeholders rather than to achieve a high response rate.

As described previously, surveys generated 2835 individual free-text responses, which the research team synthesized and combined into 86 research topics. Nine themes emerged by consensus discussion: (1) access to services and supports (health system and community); (2) financing, payment models, and insurance; (3) caregiving at home and caregiving experience; (4) family health; (5) child health, vulnerability, and complexity; (6) care planning and coordination; (7) medical home and comprehensive care models; (8) technology and care; and (9) CYSHCN research methods innovations. Themes and representative text submissions are summarized in Table 2. Although research topics were sorted into a single theme, they did reveal overlap, with some research topics plausibly belonging to multiple themes.

TABLE 2

Summarized Results From Research Topic Development

Description of Research Topics Within Each ThemeRepresentative Respondent Perceptions
Caregiving at home and caregiving experience  
 Home environment as a health determinant; interventions to support caregiving at home; best practices for caregiver education and peer-to-peer support “Medical professionals need to be aware of the home environment, what interventions are realistic in a day-to-day environment, that will let the child live as much like a typically developing child as possible…” 
Technology and care  
 Technologies to complement caregiving and communication; dissemination or implementation of technology “Real-time symptom monitoring allowing just-in-time adjustments (like asthma meds in response to peak flows)…” 
Access to services and supports (health system and community)  
 Workforce capacity and expertise; CYSHCN, health care, and community links; durable equipment, education, and dental care “Having more access to behavioral services helps to keep children in the community rather than having to utilize inpatient [care].” 
Financing, payment models, and insurance  
 Payment model influence on quality, access, and outcomes; compensation for families, providers, and community services “…Funding can be used for what the family determines is most needed, even if it is for social determinants of health, housing, food, transportation, childcare…” 
Medical home and comprehensive care models  
 Key ingredients of effective models; family partnership, team efficiency, provider retention, and culturally sensitive care “In model practices, who provides care coordination, what do they do (especially what do they do that [is] successful), what are key qualities and qualifications for this position?” 
Care planning and coordination  
 Shared decision-making and goal setting; generalizable CYSHCN health screening and comanagement approaches “Improved family engagement through mutually defined goals (shared decision-making) that are family centered…” 
CYSHCN health, vulnerability, and complexity  
 Interventions to reduce consequences of social determinants, rural environments, mental health, and extreme complexity; health outcomes meaningful to families “…Need research in the area of CYSHCN living in rural locations; how to provide services and supports, what is available now, etc.” 
Family health  
 Interventions to promote family physical and mental health, resilience, and self-care; health system influence on family caregiver health “What are the caregiver benefits when they receive appropriate care support versus parents who do not receive same level of support—ability to work outside the home, caregiver stressors, and health and well-being?” 
Research methods innovations  
 Rare disease cohorts; CYSHCN as research team members; marketing findings to research consumers “…Increased participation in research by youth/young adults with chronic health conditions. What is important to them, what barriers do they experience, what are their suggestions to improve the system?” 
Description of Research Topics Within Each ThemeRepresentative Respondent Perceptions
Caregiving at home and caregiving experience  
 Home environment as a health determinant; interventions to support caregiving at home; best practices for caregiver education and peer-to-peer support “Medical professionals need to be aware of the home environment, what interventions are realistic in a day-to-day environment, that will let the child live as much like a typically developing child as possible…” 
Technology and care  
 Technologies to complement caregiving and communication; dissemination or implementation of technology “Real-time symptom monitoring allowing just-in-time adjustments (like asthma meds in response to peak flows)…” 
Access to services and supports (health system and community)  
 Workforce capacity and expertise; CYSHCN, health care, and community links; durable equipment, education, and dental care “Having more access to behavioral services helps to keep children in the community rather than having to utilize inpatient [care].” 
Financing, payment models, and insurance  
 Payment model influence on quality, access, and outcomes; compensation for families, providers, and community services “…Funding can be used for what the family determines is most needed, even if it is for social determinants of health, housing, food, transportation, childcare…” 
Medical home and comprehensive care models  
 Key ingredients of effective models; family partnership, team efficiency, provider retention, and culturally sensitive care “In model practices, who provides care coordination, what do they do (especially what do they do that [is] successful), what are key qualities and qualifications for this position?” 
Care planning and coordination  
 Shared decision-making and goal setting; generalizable CYSHCN health screening and comanagement approaches “Improved family engagement through mutually defined goals (shared decision-making) that are family centered…” 
CYSHCN health, vulnerability, and complexity  
 Interventions to reduce consequences of social determinants, rural environments, mental health, and extreme complexity; health outcomes meaningful to families “…Need research in the area of CYSHCN living in rural locations; how to provide services and supports, what is available now, etc.” 
Family health  
 Interventions to promote family physical and mental health, resilience, and self-care; health system influence on family caregiver health “What are the caregiver benefits when they receive appropriate care support versus parents who do not receive same level of support—ability to work outside the home, caregiver stressors, and health and well-being?” 
Research methods innovations  
 Rare disease cohorts; CYSHCN as research team members; marketing findings to research consumers “…Increased participation in research by youth/young adults with chronic health conditions. What is important to them, what barriers do they experience, what are their suggestions to improve the system?” 

After round 1, 24 of 86 research topics met rank 1 or rank 2 criteria. Experts proposed 10 additional research topics during round 1, leading to 96 total research topics. During round 2, as they reranked topics, panelists discussed tensions around breadth of research topic scope from 3 perspectives: (1) CYSHCN versus children and youth with medical complexity, (2) applicability to larger versus smaller populations, and (3) children versus youth versus adults. Guidance from MCHB team members was to focus on broadly applicable topics. Panelists were therefore encouraged to rate need and urgency lower if a research topic’s scope was felt to be narrow.

The results reveal the varying degrees of each research topic’s perceived urgency, impact, and family centeredness. Overall, median ratings for the family centeredness domain were >7 for the largest number of research topics (47 of 96 topics, Table 3). The domain research impact had the lowest number of topics with high ratings, with only 10 of the 96 having median ratings >7. Although research topics focused on financing were most often rated as having high impact, they were simultaneously the least family centered. No research topics were rated by experts as low (ie, no medians were ≤3) in either need and urgency, research impact, or family centeredness.

TABLE 3

RAM Round 2 Result Summary

ThemeNo. TopicsResearch Topics With Median Rating >7
Need and UrgencyResearch ImpactFamily Centered
Caregiving at home and caregiving experience 11 
Technology and care 11 
Access to services and supports (health system and community) 14 
Financing, payment models, and insurance 12 
Medical home and comprehensive care models 10 
Care planning and coordination 
Child health, vulnerability, and complexity 15 
Family caregiver health 
CYSHCN research methods innovations 
Total 96 14 10 47 
ThemeNo. TopicsResearch Topics With Median Rating >7
Need and UrgencyResearch ImpactFamily Centered
Caregiving at home and caregiving experience 11 
Technology and care 11 
Access to services and supports (health system and community) 14 
Financing, payment models, and insurance 12 
Medical home and comprehensive care models 10 
Care planning and coordination 
Child health, vulnerability, and complexity 15 
Family caregiver health 
CYSHCN research methods innovations 
Total 96 14 10 47 

. Research topics were generated from synthesized and aggregated open-ended survey responses, following an ongoing, consensus-seeking approach. Conceptually similar research topics were sorted into themes. Each research topic was rated using three rating domains, corresponding to perceived (1) need and urgency, (2) research impact, and (3) family centeredness. Each domain was rated by using a 9-point Likert scale: 1 to 3 (low) was used when a research topic represented a rating domain to a low degree; 4 to 6 was used when a research topic represented an uncertain rating domain; and 7 to 9 (high) was used when a research topic represented a domain to a high degree.

The rankings of research topics after round 2 are summarized in Table 4. The rank 1 group comprised 15 research topics emphasizing social determinants of health (SDH), family caregiving, refining clinical models, value, and youth-adult transitions. Although research topics related to these concepts were present across all ranks, the rank 1 topics highlight specific priorities within each. For example, SDH topics in rank 1 centered on research to effectively integrate SDH screening into care, understand the unique influence of rurality on CYSHCN, and develop innovative technology to reduce inequities.

TABLE 4

Descriptions of CYSHCN Research Topics in Each Rank After RAM

ThemeResearch Topic Descriptions by RAM Ranka
Rank 1 (Highest)Rank 2Rank 3 (Uncertainty in At Least 1 Rating Domain)
Caregiving at home and caregiving experience Optimizing care at home Home physical environment Peer support credentials and professional standards 
  Parent activation Home nurse core competencies 
  Family-identified education needs Independent living achievement 
  Behavioral health crises Peer mentor education 
  Training diverse parent populations Group care models 
Technology and care Telemedicine implementation Real-time symptom monitoring and treatment Parent contributing to health record documentation 
 Technology to reduce disparities Technology to train parents Data sharing across platforms and agencies 
Access to services and supports (health systems and communities) Insurance and community support through youth-adult transition Behavioral health access Medical and nonmedical service collaboration 
  Peer-peer coaching for self-management Documentation to minimize denials 
  Complex care and specialty care access Long-term supports and services across states 
   Durable medical equipment access 
   School integration 
   Oral health provision 
   Adult CYSHCN provider workforce size and competence 
Financing, payment models, and insurance Payment models that improve quality Paying families to deliver care Provider compensation 
 Care model return on investment Paid family medical leave Insurance delays and denials 
 Value outcomes meaningful to different stakeholders Payment models across life span Payment models supporting youth-adult transition 
  Medicaid-managed care impact Child health impact from health services to parents 
Medical home and comprehensive care models Effectiveness of integrated systems Parent advisory group best practices Staff/patient ratios 
 Key ingredients of effective models Conducting culturally sensitive care Family as professional team members 
 Palliative care incorporation into routine care   
Care planning and coordination Youth-adult transition planning models Shared decision-making and goal setting  
 Dividing labor and comanagement Family engagement in care planning  
 SDHb Professional care coordinators  
Child health, vulnerability, and complexity SDHb Health outcomes meaningful to families CYSHCN peer relationships 
 Rurality’s influence on health and care Identifying and reducing disparities Avoidable morbidity and mortality with youth-adult transition 
  Child and family mental health screening Youth-adult transition and mental health exacerbations 
   CYSHCN tiering systems 
   Family characteristics predicting acute care use 
   Unknown diagnoses 
   Developmental consequences that have an adult onset 
Family health Family resilience Family health influence on child health Parent mental health and addiction 
  Support for family self-care and emotional needs Parent stress during youth-adult transition 
  Family’s role in value-based decisions Shared care plans’ influence on family health 
   Family health as metric of CYSHCN system effectiveness 
ThemeResearch Topic Descriptions by RAM Ranka
Rank 1 (Highest)Rank 2Rank 3 (Uncertainty in At Least 1 Rating Domain)
Caregiving at home and caregiving experience Optimizing care at home Home physical environment Peer support credentials and professional standards 
  Parent activation Home nurse core competencies 
  Family-identified education needs Independent living achievement 
  Behavioral health crises Peer mentor education 
  Training diverse parent populations Group care models 
Technology and care Telemedicine implementation Real-time symptom monitoring and treatment Parent contributing to health record documentation 
 Technology to reduce disparities Technology to train parents Data sharing across platforms and agencies 
Access to services and supports (health systems and communities) Insurance and community support through youth-adult transition Behavioral health access Medical and nonmedical service collaboration 
  Peer-peer coaching for self-management Documentation to minimize denials 
  Complex care and specialty care access Long-term supports and services across states 
   Durable medical equipment access 
   School integration 
   Oral health provision 
   Adult CYSHCN provider workforce size and competence 
Financing, payment models, and insurance Payment models that improve quality Paying families to deliver care Provider compensation 
 Care model return on investment Paid family medical leave Insurance delays and denials 
 Value outcomes meaningful to different stakeholders Payment models across life span Payment models supporting youth-adult transition 
  Medicaid-managed care impact Child health impact from health services to parents 
Medical home and comprehensive care models Effectiveness of integrated systems Parent advisory group best practices Staff/patient ratios 
 Key ingredients of effective models Conducting culturally sensitive care Family as professional team members 
 Palliative care incorporation into routine care   
Care planning and coordination Youth-adult transition planning models Shared decision-making and goal setting  
 Dividing labor and comanagement Family engagement in care planning  
 SDHb Professional care coordinators  
Child health, vulnerability, and complexity SDHb Health outcomes meaningful to families CYSHCN peer relationships 
 Rurality’s influence on health and care Identifying and reducing disparities Avoidable morbidity and mortality with youth-adult transition 
  Child and family mental health screening Youth-adult transition and mental health exacerbations 
   CYSHCN tiering systems 
   Family characteristics predicting acute care use 
   Unknown diagnoses 
   Developmental consequences that have an adult onset 
Family health Family resilience Family health influence on child health Parent mental health and addiction 
  Support for family self-care and emotional needs Parent stress during youth-adult transition 
  Family’s role in value-based decisions Shared care plans’ influence on family health 
   Family health as metric of CYSHCN system effectiveness 

Rank 1 (highest): research topics had a median in the high range (≥7) for all 3 domains and a median of >7 in at least 2 of 3 domains; rank 2: research topics had a median of ≥7 for all 3 domains and >7 in 1 of 3 domains; rank 3: research topics had a median <7 for 1 or 2 of 3 domains, or median = 7 for all domains; rank 4 (not shown): research topics had a median <7 for all 3 domains or any disagreement. See the Supplemental Information for complete research topics and ratings.

a

Individual research topics were ranked on the basis of RAM results. Each topic was rated in 3 domains: need and urgency, research impact, and family centeredness.

b

Two SDH research topics were combined into a single topic.

Rank 1 caregiving topics were centered on families’ experience of caring for CYSHCN at home. These topics centered on research to optimize care delivered by families to CYSHCN at home, use a whole-family perspective, and examine caregiving by using a strength-based approach (eg, understanding and improving family resilience as a means to achieving CYSHCN and family health). Rank 1 clinical-model refinement topics emphasized research to integrate systems, uncover key elements of effective care models, and divide “labor” (routine care and coordination tasks) efficiently among team members, including families. Value-oriented rank 1 research topics centered on research used to determine what value means to different stakeholders and then to focus on return on investment in programs, personnel, and state-based systems. Finally, the rank 1 youth-adult transition topics emphasized interventions to effectively plan for transition and to ensure ongoing insurance and community supports into adulthood.

As illustrated in Table 5, rank 2 research topics had lower need and urgency ratings than rank 1 topics. Rank 3 research topics had lower research impact and family centeredness ratings than rank 2 topics. Rank 4 research topics (ie, those with medians <7 for all domains) were more technology focused (eg, family use of social media and application- or cloud-based care plans), were more provider centered (eg, workforce and compensation), or were more related to research methods (eg, standardizing CYSHCN subpopulations). Complete rating details are available in the Supplemental Information.

TABLE 5

Average Ratings of Research Impact, Need and Urgency, and Family Centeredness for Research Topics Within Each Rank

Summary of Research TopicsRating Domains
Need and UrgencyResearch ImpactFamily Centeredness
Rank 1 Social determinants, family caregiving, clinical-model refinement, youth-adult transitions, value 7.9 7.5 8.0 
Rank 2 Meaningful outcomes, behavioral health, home environment, peer-peer support, family engagement, family economics 7.0 7.1 7.9 
Rank 3 Linking family and child health, school and peers, dental health, youth-adult transitions, professional competencies, clinical staffing 6.8 6.4 7.2 
Rank 4 Technology, research methods, provider-centered concepts 5.9 5.7 5.7 
Summary of Research TopicsRating Domains
Need and UrgencyResearch ImpactFamily Centeredness
Rank 1 Social determinants, family caregiving, clinical-model refinement, youth-adult transitions, value 7.9 7.5 8.0 
Rank 2 Meaningful outcomes, behavioral health, home environment, peer-peer support, family engagement, family economics 7.0 7.1 7.9 
Rank 3 Linking family and child health, school and peers, dental health, youth-adult transitions, professional competencies, clinical staffing 6.8 6.4 7.2 
Rank 4 Technology, research methods, provider-centered concepts 5.9 5.7 5.7 

Each research topic was rated in 3 domains (need and urgency, research impact, and family centeredness) by using a 9-point Likert scale: 1 to 3 (low) was used when a research topic represented a domain to a low degree; 4 to 6 was used when a research topic represented an uncertain domain; and 7 to 9 (high) was used when a research topic represented a domain to a high degree. Shown are the average RAM round 2 ratings in each rank group. Rank 2 had lower need and urgency ratings than rank 1. Rank 3 had lower research impact and family centeredness ratings than rank 2. Average ratings of topics in ranks 3 and 4 were primarily in the uncertain range (4–6).

Notably, the RAM ratings obtained from the complete panel were consistent with the ratings generated by only the family caregivers on the panel. In particular, both analyses placed 10 of the same research topics in rank 1. Ranks 1 and 2, when combined, were nearly identical in both analyses. For example, none of the rank 1 topics from the complete analysis were ranked below rank 2 in the family caregiver analysis. Only 1 rank 1 research topic from the family caregiver analysis was below rank 2 in the complete analysis (“How do delays from insurance denials affect CYSHCN health, and how can they be minimized?”).

We adapted RAM with a professionally broad and nationally diverse group of CYSHCN experts, including family caregivers, to begin developing a national research agenda for CYSHCN systems of care. Our process resulted in a taxonomy of CYSHCN research topics depicting their varying levels of stakeholder-perceived need and urgency, research impact, and family centeredness. The highest-ranked research topics were focused on SDH, family resilience, value, and youth-adult transitions. These findings simultaneously reinforce current CYSHCN research trends and point toward important new directions that support achieving high-functioning systems of care.

Rank 1 topics reveal how our results could promote CYSHCN research advances. For example, directly supporting CYSHCN caregiving at home emerged as a top research priority. From topic development to discussion during RAM, our process framed caregiver challenges in terms of resilience and adaptability, providing a more positive orientation than studies that are focused on caregiving stresses and burdens.2330  This frameshift also aligns with recent research suggesting that strategies to support family caregivers may improve CYSHCN health. For example, emerging data support the theory that child and caregiver health are tightly interlinked3133  and that increasing resilience may mitigate negative effects of toxic stress.3436  Resilience in childhood disability appears to be dynamic,37  and panelists described resilience as a natural intervention target for future work. The latter echoes recent calls to apply real-time resilience measurement into routine care5,34,38  and is consistent with the American Academy of Pediatrics (AAP) clinical report on psychosocial factors faced by CYSHCN and families.38 

At present, effective interventions to support family caregiving are limited,39,40  and how best to achieve the rank 1 caregiving priorities across the vast array of CYSHCN conditions and family structures remains a key unknown. Promising approaches that emphasize family needs and strengths, build family capacity, and promote sustainable goal attainment have direct relevance to CYSHCN families.41  For example, the Family System-Illness Model42  is 1 framework for increasing family resilience in clinical settings for children with chronic illness. Strengths of the model are its linking of biological and psychosocial demands of chronic illness and its accounting for variation in patterns of illness onset, course, outcome, functional loss, and uncertainty about illness trajectory. As effective CYSHCN family resilience interventions materialize, adaptation to the different circumstances experienced by CYSHCN could lead to broadly generalizable and potentially transformative strategies.

Rank 1 research topics also extend current value-oriented CYSHCN research, such as return on investment in payments, staffing, and interventions.4347  Although value-oriented research could have major implications for financing and organization of systems of care for CYSHCN, the area remains poorly understood.43  Consistent with our findings, recent research reveals the central importance of defining value for CYSHCN care through incorporation of an array of stakeholder perspectives.45  Closing key value-oriented knowledge gaps could begin with mixed-methods studies to establish stakeholder-centered value measures and applying these with different stakeholders. Comparative effectiveness study designs could be used to evaluate the effects of various alternative payment models on CYSHCN health outcomes.

Rank 1 and rank 2 topics also highlighted integration across care sectors (home, school, community, and health care) as a CYSHCN research agenda priority. Particularly notable were numerous topics on behavioral health and the transition from childhood to adult systems. RAM participants rated more than half of all topics in these 2 areas as family centered, likely reflecting the central role of families in CYSHCN care and in building robust systems for their care. Conversely, relatively few topics in these areas were rated high in research impact. Although the significance of this finding is unclear, it may represent concerns about being able to translate research findings from these topics to meaningful impact in the short-term.

Our explicit motivation to develop research priorities and relative rankings distinguishes these results from related efforts. For example, the MCHB 6 Core Outcomes15  and the AMCHP Standards for Systems of Care16  provide health system metrics and were developed with multidisciplinary and family input; however, neither was designed to reflect specific research priorities. Although some aspects of AMCHP and MCHB frameworks may benefit from research, others may require different initiatives (eg, policy or advocacy). As an example of this distinction, the RAM research topics in the theme of access to services and supports have substantial overlap with AMCHP and MCHB frameworks (as well as high family-centered ratings); however, none rated high in research impact. One potential interpretation of this finding is that although these topics have substantial relevance to CYSHCN, panelists were skeptical that conducting research was the best way to address the underlying challenges.

Although panelists felt that lower-ranked research topics had some uncertainty in need and urgency, research impact, and/or family centeredness, this does not imply that those topics are unimportant. RAM does not readily facilitate interpreting why panelists gave specific ratings and is not intended to force consensus. Technology-centered research topics, despite being a focus area in the AMCHP Standards for Systems of Care, were consistently rated low or uncertain. On the basis of conversations during the RAM meeting, we speculate this was due to perceptions that technologies face important feasibility and generalizability barriers. Panelists also expressed that although technology might facilitate solutions, it might not inherently solve problems. Provider-centered research topics (eg, workforce, compensation, satisfaction, and efficiency) also tended to be lower ranked, presumably because they are less family centered, knowledge gaps are less clear, and the topics are seen as less urgent.

This work has limitations. The volume and content developed through this process precludes a comprehensive discussion of each research topic. Throughout RAM, it was challenging to be as inclusive as possible while simultaneously developing a well-defined, generalizable, and actionable set of high-priority research topics. Similarly, although we integrated solicited literature from stakeholder organizations into RAM, we did not conduct a formal systematic literature review. Whether a systematic literature review would have uncovered additional topics or influenced the panelist ratings is not known. Because we anticipated ratings would be skewed toward the high end of the scale, we established rank priority cutoffs, which might be overly sensitive. RAM also relies on experts having the necessary knowledge to make valid judgements, which we could not verify. Despite in-person discussion, experts may have had distinct CYSHCN contexts in mind, which could differentially influence some ratings. For example, thinking about children with autism versus asthma might affect ratings about caregiving research topics. Additionally, our RAM participants were less demographically diverse than we would have liked. We also recognize that priorities will change over time on the basis of new knowledge, practice patterns, payment models, and policies.

To translate RAM results into an actionable research agenda for CYSHCN health systems, additional steps are needed. First, comprehensive reviews should be used to synthesize current knowledge and distinguish key gaps for each research topic. Second, researchers should determine key barriers to progress. Do challenges result from limited knowledge about a topic or from implementation difficulties? Third, specific research initiatives that will close knowledge gaps and overcome barriers should be articulated. For some well-studied research topics, work may need to be focused on dissemination and implementation science,48  whereas for more emergent topics, basic discovery49  may be most needed.

Despite these limitations, this work uncovers research topics with high perceived need, research impact, and family centeredness and may offer a pragmatic guide for families, researchers, policymakers, and health system leaders interested in improving CYSHCN outcomes. The consistency of our results, whether evaluated across the complete stakeholder group or among family stakeholders alone, suggests that they accurately reflect priorities of CYSHCN families. The synthesis of opinions of a national multidisciplinary group of CYSHCN experts, including family caregivers, represents a fundamental first step toward the development of a national research agenda to improve CYSHCN health systems. The work prioritizes the most important systems-level questions, facilitates setting research goals related to these questions, and contributes to a blueprint to guide investigators to work cohesively toward achieving an idealized vision of comprehensive coordinated systems for CYSHCN.

We thank the expert stakeholders, youth, and families who contributed their invaluable knowledge during the various phases of this work. We also thank Jeffrey Sleeth, MD, for his invaluable review of an earlier draft of this article.

Dr Coller conceptualized and designed the study, conducted the primary data analysis and interpretation, and drafted the initial manuscript; Drs Berry, Chung, and Stille conceptualized the study, participated in data collection, analysis, and interpretation of data, and revised the manuscript; Drs Kuhlthau, Kuo, and Perrin and Ms Hoover critically interpreted data analyses and critically revised the manuscript; Dr Shelton and Ms Werner coordinated and supervised data collection and data management and critically reviewed the manuscript; Ms Thompson and Ms Garrity assisted with data collection and critically reviewed the manuscript; and all authors approved the final manuscript as submitted and agree to be accountable for all aspects of the work.

FUNDING: Supported by the Health Resources and Services Administration of the US Department of Health and Human Services under UA6MC31101 (Children with Special Health Care Needs Research Network). This information or content and conclusions are those of the authors and should not be construed as the official position or policy of, nor should any endorsements be inferred by, the Health Resources and Services Administration, the US Department of Health and Human Services, or the US Government.

     
  • AAP

    American Academy of Pediatrics

  •  
  • AMCHP

    Association of Maternal and Child Health Programs

  •  
  • CYSHCN

    children and youth with special health care needs

  •  
  • CYSHCNet

    Children and Youth with Special Health Care Needs National Research Network

  •  
  • MCHB

    Maternal and Child Health Bureau

  •  
  • RAM

    RAND/UCLA Appropriateness Method

  •  
  • SDH

    social determinants of health

1
McPherson
M
,
Arango
P
,
Fox
H
, et al
.
A new definition of children with special health care needs
.
Pediatrics
.
1998
;
102
(
1, pt 1
):
137
140
2
Perrin
JM
,
Romm
D
,
Bloom
SR
, et al
.
A family-centered, community-based system of services for children and youth with special health care needs
.
Arch Pediatr Adolesc Med
.
2007
;
161
(
10
):
933
936
3
Child and Adolescent Health Measurement Initiative
. 2016–2017 National Survey of Children’s Health (NSCH) data query. Available at: https://www.childhealthdata.org/browse/survey?s=2&y=24&r=1. Accessed December 15, 2018
4
Berman
S
.
Children with special health care needs: the canary in the health care reform coal mine
.
Pediatrics
.
2014
;
134
(
2
):
373
374
5
Institute of Medicine
.
Living Well with Chronic Illness: A Call for Public Health Action
.
Washington, DC
:
National Academies Press
;
2012
6
Kuhlthau
K
,
Hill
KS
,
Yucel
R
,
Perrin
JM
.
Financial burden for families of children with special health care needs
.
Matern Child Health J
.
2005
;
9
(
2
):
207
218
7
Smith
M
,
Saunders
R
,
Stuckhardt
L
,
McGinnis
JM
, eds.
Committee on the Learning Health Care System in America; Institute of Medicine. Best Care at Lower Cost: The Path to Continuously Learning Health Care in America
.
Washington, DC
:
National Academies Press
;
2013
8
Bureau of Health Care Delivery and Assistance, Division of Maternal and Child Health and United States, Public Health Service, Office of the Surgeon General
.
Children With Special Health Care Needs: Surgeon General’s Report: Campaign ’87
.
Washington, DC
:
Bureau of Health Care Delivery and Assistance, Division of Maternal and Child Health
;
1987
9
Newacheck
PW
,
Rising
JP
,
Kim
SE
.
Children at risk for special health care needs
.
Pediatrics
.
2006
;
118
(
1
):
334
342
10
Perrin
JM
.
Children with special health care needs and changing policy
.
Acad Pediatr
.
2011
;
11
(
2
):
103
104
11
Mooney
H
.
Children with special educational needs: what’s the agenda?
Community Pract
.
2012
;
85
(
12
):
18
19
12
Szilagyi
PG
,
Kuhlthau
KA
.
Children with special health care needs: a celebration of success!
.
Acad Pediatr
.
2011
;
11
(
2
):
94
95
13
Perrin
JM
.
Health services research for children with disabilities
.
Milbank Q
.
2002
;
80
(
2
):
303
324
14
Kuhlthau
KA
,
Bloom
S
,
Van Cleave
J
, et al
.
Evidence for family-centered care for children with special health care needs: a systematic review
.
Acad Pediatr
.
2011
;
11
(
2
):
136
143
15
McPherson
M
,
Weissman
G
,
Strickland
BB
,
van Dyck
PC
,
Blumberg
SJ
,
Newacheck
PW
.
Implementing community-based systems of services for children and youths with special health care needs: how well are we doing?
Pediatrics
.
2004
;
113
(
suppl 5
):
1538
1544
16
National Standards for Systems of Care for Children and Youth with Special Health Care Needs
.
Core domains for system standards.
Available at: http://cyshcnstandards.amchp.org/app-national-standards/#/coredomain. Accessed December 14, 2018
17
Fitch
K
,
Bernstein
SJ
,
Aguilar
MD
, et al
.
The RAND/UCLA Appropriateness Method User’s Manual
.
Santa Monica, CA
:
RAND Corporation
;
2001
18
Chen
AY
,
Schrager
SM
,
Mangione-Smith
R
.
Quality measures for primary care of complex pediatric patients
.
Pediatrics
.
2012
;
129
(
3
):
433
445
19
Coker
TR
,
Moreno
C
,
Shekelle
PG
,
Schuster
MA
,
Chung
PJ
.
Well-child care clinical practice redesign for serving low-income children
.
Pediatrics
.
2014
;
134
(
1
).
20
Coller
RJ
,
Nelson
BB
,
Klitzner
TS
, et al
.
Strategies to reduce hospitalizations of children with medical complexity through complex care: expert perspectives
.
Acad Pediatr
.
2017
;
17
(
4
):
381
388
21
Wang
CJ
,
McGlynn
EA
,
Brook
RH
, et al
.
Quality-of-care indicators for the neurodevelopmental follow-up of very low birth weight children: results of an expert panel process
.
Pediatrics
.
2006
;
117
(
6
):
2080
2092
22
Hsieh
HF
,
Shannon
SE
.
Three approaches to qualitative content analysis
.
Qual Health Res
.
2005
;
15
(
9
):
1277
1288
23
Carcone
AI
,
Ellis
DA
,
Naar-King
S
.
Linking caregiver strain to diabetes illness management and health outcomes in a sample of adolescents in chronically poor metabolic control
.
J Dev Behav Pediatr
.
2012
;
33
(
4
):
343
351
24
Martínez
KG
,
Pérez
EA
,
Ramírez
R
,
Canino
G
,
Rand
C
.
The role of caregivers’ depressive symptoms and asthma beliefs on asthma outcomes among low-income Puerto Rican children
.
J Asthma
.
2009
;
46
(
2
):
136
141
25
Raina
P
,
O’Donnell
M
,
Rosenbaum
P
, et al
.
The health and well-being of caregivers of children with cerebral palsy
.
Pediatrics
.
2005
;
115
(
6
).
26
Kuo
DZ
,
Cohen
E
,
Agrawal
R
,
Berry
JG
,
Casey
PH
.
A national profile of caregiver challenges among more medically complex children with special health care needs
.
Arch Pediatr Adolesc Med
.
2011
;
165
(
11
):
1020
1026
27
Romley
JA
,
Shah
AK
,
Chung
PJ
,
Elliott
MN
,
Vestal
KD
,
Schuster
MA
.
Family-provided health care for children with special health care needs
.
Pediatrics
.
2017
;
139
(
1
):
e20161287
28
Kuhlthau
KA
,
Perrin
JM
.
Child health status and parental employment
.
Arch Pediatr Adolesc Med
.
2001
;
155
(
12
):
1346
1350
29
Thyen
U
,
Terres
NM
,
Yazdgerdi
SR
,
Perrin
JM
.
Impact of long-term care of children assisted by technology on maternal health
.
J Dev Behav Pediatr
.
1998
;
19
(
4
):
273
282
30
Kuhlthau
K
,
Kahn
R
,
Hill
KS
,
Gnanasekaran
S
,
Ettner
SL
.
The well-being of parental caregivers of children with activity limitations
.
Matern Child Health J
.
2010
;
14
(
2
):
155
163
31
Brehaut
JC
,
Kohen
DE
,
Garner
RE
, et al
.
Health among caregivers of children with health problems: findings from a Canadian population-based study
.
Am J Public Health
.
2009
;
99
(
7
):
1254
1262
32
Cohen
E
,
Horváth-Puhó
E
,
Ray
JG
, et al
.
Association between the birth of an infant with major congenital anomalies and subsequent risk of mortality in their mothers
.
JAMA
.
2016
;
316
(
23
):
2515
2524
33
Epel
ES
,
Blackburn
EH
,
Lin
J
, et al
.
Accelerated telomere shortening in response to life stress
.
Proc Natl Acad Sci U S A
.
2004
;
101
(
49
):
17312
17315
34
Bethell
CD
,
Newacheck
P
,
Hawes
E
,
Halfon
N
.
Adverse childhood experiences: assessing the impact on health and school engagement and the mitigating role of resilience
.
Health Aff (Millwood)
.
2014
;
33
(
12
):
2106
2115
35
Garner
AS
,
Shonkoff
JP
;
Committee on Psychosocial Aspects of Child and Family Health
;
Committee on Early Childhood, Adoption, and Dependent Care
;
Section on Developmental and Behavioral Pediatrics
.
Early childhood adversity, toxic stress, and the role of the pediatrician: translating developmental science into lifelong health
.
Pediatrics
.
2012
;
129
(
1
).
36
Shonkoff
JP
,
Garner
AS
;
Committee on Psychosocial Aspects of Child and Family Health
;
Committee on Early Childhood, Adoption, and Dependent Care
;
Section on Developmental and Behavioral Pediatrics
.
The lifelong effects of early childhood adversity and toxic stress
.
Pediatrics
.
2012
;
129
(
1
).
37
Ten Hove
J
,
Rosenbaum
P
.
The concept of resilience in childhood disability: does the International Classification of Functioning, Disability and Health help us?
Child Care Health Dev
.
2018
;
44
(
5
):
730
735
38
Mattson
G
,
Kuo
DZ
;
Committee on Psychosocial Aspects of Child and Family Health
;
Council on Children with Disabilities
.
Psychosocial factors in children and youth with special health care needs and their families
.
Pediatrics
.
2019
;
143
(
1
):
e20183171
39
Dambi
JM
,
Jelsma
J
,
Mlambo
T
, et al
.
A critical evaluation of the effectiveness of interventions for improving the well-being of caregivers of children with cerebral palsy: a systematic review protocol
.
Syst Rev
.
2016
;
5
(
1
):
112
40
Edelstein
H
,
Schippke
J
,
Sheffe
S
,
Kingsnorth
S
.
Children with medical complexity: a scoping review of interventions to support caregiver stress
.
Child Care Health Dev
.
2017
;
43
(
3
):
323
333
41
Bethell
CD
,
Newacheck
PW
,
Fine
A
, et al
.
Optimizing health and health care systems for children with special health care needs using the life course perspective
.
Matern Child Health J
.
2014
;
18
(
2
):
467
477
42
Rolland
JS
,
Walsh
F
.
Facilitating family resilience with childhood illness and disability
.
Curr Opin Pediatr
.
2006
;
18
(
5
):
527
538
43
Bachman
SS
,
Comeau
M
,
Long
TF
.
Statement of the problem: health reform, value-based purchasing, alternative payment strategies, and children and youth with special health care needs
.
Pediatrics
.
2017
;
139
(
suppl 2
):
S89
S98
44
Ellis
RP
,
Bachman
SS
,
Tan
HR
.
Refining our understanding of value-based insurance design and high cost sharing on children
.
Pediatrics
.
2017
;
139
(
suppl 2
):
S136
S144
45
Anderson
B
,
Beckett
J
,
Wells
N
,
Comeau
M
.
The eye of the beholder: a discussion of value and quality from the perspective of families of children and youth with special health care needs
.
Pediatrics
.
2017
;
139
(
suppl 2
):
S99
S108
46
Perrin
JM
.
Innovative health care financing strategies for children and youth with special health care needs
.
Pediatrics
.
2017
;
139
(
suppl 2
):
S85
S88
47
Antonelli
RC
,
Turchi
RM
.
Care management for children with medical complexity: integration is essential
.
Pediatrics
.
2017
;
140
(
6
):
e20172860
48
Glasgow
RE
,
Vinson
C
,
Chambers
D
,
Khoury
MJ
,
Kaplan
RM
,
Hunter
C
.
National Institutes of Health approaches to dissemination and implementation science: current and future directions
.
Am J Public Health
.
2012
;
102
(
7
):
1274
1281
49
Khoury
MJ
,
Gwinn
M
,
Ioannidis
JP
.
The emergence of translational epidemiology: from scientific discovery to population health impact
.
Am J Epidemiol
.
2010
;
172
(
5
):
517
524

Competing Interests

POTENTIAL CONFLICT OF INTEREST: The authors have indicated they have no potential conflicts of interest to disclose.

FINANCIAL DISCLOSURE: The authors have indicated they have no financial relationships relevant to this article to disclose.

Supplementary data