Family Engagement at the Systems Level: A Framework for Action.

While family engagement at the individual level of health care, such as families partnering with providers in decision-making about health care for an individual child has been well studied, family engagement in systems-level activities (e.g., participation in advisory and other decision-making groups, or creation and revision of policies) that impact the health services families and children receive has not. This note from the field presents a framework that describes the information and supports that help families partner with professionals and contribute to systems-level activities. Without attention to these components of family engagement, family presence and participation may be only token. We engaged an expert Family/Professional Workgroup whose members represented key constituencies and diverse geography, race/ethnicity, and areas of expertise; conducted a review of peer-reviewed publications and grey literature; and conducted a series of key informant interviews to identify best practices for supporting meaningful family engagement at the systems level. Based on an analysis of the findings, the authors identified four action-oriented domains of family engagement and key criteria that support and strengthen meaningful family engagement in systems-level initiatives. Child- and family-serving serving organizations can use this Family Engagement in Systems framework to support meaningful family engagement in the design of policies, practices, services, supports, quality improvement projects, research, and other systems-level activities.

Child and Family Perspectives on Adjustment to and Coping With Pediatric Inflammatory Bowel Disease.

OBJECTIVES: Children with inflammatory bowel disease (IBD) are at increased risk for poor mental health. The etiology of this risk is not clear, though may be related to the disease, its treatment, and/or the experience of these. We sought to describe the challenges that children with IBD and their families face in living with a chronic condition and undergoing repeated intravenous infusions; and identify coping mechanisms to understand how medical systems may support resilience. METHODS: Semistructured qualitative interviews with 18 patient-guardian dyads at a tertiary outpatient infusion center, explored feelings related to IBD, the infusion process, and coping. Interviews were recorded, transcribed, and analyzed in ATLAS.ti. Two coders identified themes; developed a codebook and coded transcripts using the constant comparative method; and described themes/patterns. RESULTS: Participants identified challenges related to IBD (unpredictable nature, disrupted normalcy, treatment decisions, managing relationships, life transitions) and a subset of challenges related to the infusion procedure (anxiety of unknown, managing pain/anxiety during IV placement, logistics). Participants coped through social support, cognitive strategies (positive attitude) and/or behavioral strategies for managing emotions (preparation for intravenous [IV] placement), and confidence in the medical care. By employing these coping strategies, participants came to accept IBD, adapt to the "new norm," and learned life lessons and resilience. CONCLUSIONS: To support coping, clinical teams might provide anticipatory guidance to decrease anxiety of the unknown and identify cognitive-behavioral strategies for managing emotions. Delivery systems that build relationships, maintain normalcy, and consider needs of the family may further facilitate coping.

Child and Parent Perspectives on the Acceptability of Virtual Reality to Mitigate Medical Trauma in an Infusion Center.

INTRODUCTION: Children may experience medical trauma when undergoing medical procedures even when procedures are minor. While virtual reality (VR) is effective for managing procedural pain and anxiety, few studies address how families feel about using VR. We explore pediatric patient and guardian views regarding the acceptability of using VR during procedures to mitigate medical trauma. METHODS: Semi-structured qualitative interviews with 18 patient-guardian dyads at a tertiary outpatient infusion center for inflammatory bowel disease (IBD) treatment. Interviews explored how VR may change the infusion experience, including benefits, risks, and recommendations for clinical integration. Interviews were recorded, transcribed, and analyzed in ATLAS.ti. Two coders used a 3-step coding approach to: (1) identify themes; (2) develop a codebook and code transcripts using the constant comparative method; and (3) describe themes/patterns. RESULTS: Potential benefits of VR were distraction from infusion-related anxiety and pain and generating excitement for the appointment. Potential challenges were VR-side effects (dizziness, nausea), limited mobility during the procedure, disorientation/immersion leading to shock upon IV-placement, and a lost opportunity to build coping skills. Families queried when VR should first be introduced and when during the appointment use would be optimal. Parents expressed concerns about pushing VR when their child was already under stress. A limited number of families doubted the utility of VR. CONCLUSIONS: Patients and parents found VR to be an acceptable option for helping to manage medical trauma during infusions but highlighted that the VR experience must be carefully crafted to avoid unintended consequences, including lost opportunities to build resilience.

Psychometric Evaluation of a Consumer-Developed Family-Centered Care Assessment Tool.

The objective of this study was to create a psychometrically sound measure of family-centered care, the Family-Centered Care Assessment (FCCA), developed through a process led by families in collaboration with maternal and child health leaders. The items for the FCCA scale were initially developed by families of children and youth with special needs in partnership with pediatric providers and researchers. Using an Institutional Review Board-approved research protocol, the questions were revised based on input from focus groups of diverse parents in three states. Parental responses (N = 790) to the revised 59-item survey were collected online from families in 49 states. Item distributions uniformly showed excellent spread. A principal axes factor analysis confirmed the existence of a single factor. Rasch modeling item analyses identified a reduced subset of 24 items that demonstrated excellent psychometric properties. All items met the criteria for a linear Rasch scale. Empirical evidence in support of the construct validity of the 24-item measure was derived: all items had a positive and substantial item-total correlation; person alpha scale reliability was >0.80 and the item reliability was >0.90; both separation indices were >2.0; infit and outfit statistics were within 0.5-1.5; and item difficulties ranged between -2 and +2 logits. Strong rank-ordered associations and large effect sizes were observed for six indicators of quality of care. This study's family-led process produced a tool, the FCCA, to measure families' experience of care with excellent psychometric properties.

Psychometrics of the modified Family-Centered Care Assessment short version for childhood obesity.

Background: Incorporating principles of family-centered care into pediatric weight management interventions can improve the effectiveness and quality of treatment and reduce attrition rates. To assess the family-centeredness of interventions, reliable, valid, and easy-to-administer scales are needed. The purpose of the study was to develop a shortened version of the modifed Family Centered Care Assessment (mFCCA) and assess its psychometric properties. Methods: The mFCCA, a scale to assess the family-centeredness of interventions for childhood obesity, was administered to families following the Connect for Health randomized control trial evaluating the effectiveness of a primary care-based pediatric weight management intervention. We iteratively removed items from the mFCCA and used Rasch modeling to examine the reliability and validity of the shortened scale. Results: We included data from 318 parents and the exploratory factor analysis showed the presence of a single factor. The results of the Rasch modeling demonstrated acceptable internal consistency of the scale (0.7) and strong validity as evidenced by the overall model fit and range of item difficulty. Following the psychometric analyses, we reduced the number of items from 24 to 8 items. Conclusions: The mFCCA short version demonstrates good psychometrics and can be used to evaluate the family-centeredness of childhood obesity interventions with reduced participant burden, thereby improving outcomes for children with obesity. Trial registration: Clinicaltrials.gov NCT02124460 registered on April 24, 2014.

Engaging Families in Life Course Intervention Research: An Essential Step in Advancing Equity.

Life course intervention research requires a thorough understanding of complex factors that interact to affect health over time. Partnerships with families and communities are critical to understanding these interconnections and identifying effective interventions. Here, family and community engagement are presented, aligned with the 5 phases of the life course intervention research framework: planning, design, implementation, evaluation, and translation. During planning, the researcher considers their own starting position and what they need to learn from families and the community. The design phase produces a plan for family engagement that is layered, iterative, and includes qualitative methods that will inform life course modeling and the research process. The implementation phase includes administrative actions such as creating opportunities for contributions and providing compensation to family and community partners. The evaluation phase requires measurement of the quality of partnerships with families and community and includes making adjustments as indicated to improve these partnerships. This phase also calls for reflection on the impact these partnerships had on the intervention, including if they made a difference for those being served. During translation, the researcher works with all partners, including families and communities, about follow up steps toward project continuation, replication, or completion. The researcher also works collaboratively in determining how the study results are shared. A holistic approach to health over the life course that is designed and executed in partnership with families and their community can generate research findings with broad practical applicability and strong translational potential.

What Makes an Intervention a Life Course Intervention?

OBJECTIVES: To develop an initial list of characteristics of life course interventions to inform the emerging discipline of life course intervention research. METHODS: The Life Course Intervention Research Network, a collaborative national network of >75 researchers, service providers, community representatives, and thought leaders, considered the principles, characteristics, and utility of life course interventions. After an in-person launch meeting in 2019, the steering committee collaboratively and iteratively developed a list of life course intervention characteristics, incorporating a modified Delphi review process. RESULTS: The Life Course Intervention Research Network identified 12 characteristics of life course interventions. These interventions (1) are aimed at optimizing health trajectories; (2) are developmentally focused, (3) longitudinally focused, and (4) strategically timed; and are (5) designed to address multiple levels of the ecosystem where children are born, live, learn, and grow and (6) vertically, horizontally, and longitudinally integrated to produce a seamless, forward-leaning, health optimizing system. Interventions are designed to (7) support emerging health development capabilities; are (8) collaboratively codesigned by transdisciplinary research teams, including stakeholders; and incorporate (9) family-centered, (10) strengths-based, and (11) antiracist approaches with (12) a focus on health equity. CONCLUSIONS: The intention for this list of characteristics of life course interventions is to provide a starting point for wider discussion and to guide research development. Incorporation of these characteristics into intervention designs may improve emerging health trajectories and move critical developmental processes and pathways back on track, even optimizing them to prevent or reduce adverse outcomes.

Building a Life Course Intervention Research Framework.

OBJECTIVES: To report on first steps toward building a Life Course Intervention Research Framework (LCIRF) to guide researchers studying interventions to improve lifelong health. METHODS: The Life Course Intervention Research Network, a collaborative national network of >75 researchers, service providers, community representatives and thought leaders, participated in an iterative review process. Building on the revised Medical Research Council Guidance for Developing and Evaluating Complex Interventions, they identified 12 additional key models with features for inclusion in the LCIRF, then incorporated the 12 characteristics identified by the Life Course Intervention Research Network as actionable features of Life Course Interventions to produce the new LCIRF. RESULTS: The LCIRF sets out a detailed step-wise approach to intervention development: (1) conceptualization and planning, (2) design, (3) implementation, (4) evaluation, and (5) spreading and scaling of interventions. Each step is infused with life course intervention characteristics including a focus on (1) collaborative codesign (2) health optimization, (3) supporting emerging health development capabilities (4) strategic timing, (5) multilevel approaches, and (6) health equity. Key features include a detailed transdisciplinary knowledge synthesis to inform intervention development; formation of strong partnerships with family, community, and youth representatives in intervention codesign; a means of testing the impact of each intervention on biobehavioral processes underlying emerging health trajectories; and close attention to intervention context. CONCLUSIONS: This first iteration of the LCIRF has been largely expert driven. Next steps will involve widespread partner engagement in framework refinement and further development. Implementation will require changes to the way intervention studies are organized and funded.

Understanding Caregiving and Caregivers: Supporting Children and Youth With Special Health Care Needs at Home.

Caregiving encompasses the nurturing, tasks, resources, and services that meet the day-to-day needs of children and youth with special health care needs (CYSHCN) at home. Many gaps exist in the strategies currently offered by the health care system to meet the caregiving needs of CYSHCN. The work of family caregivers of CYSHCN is known to be extensive, but it is so poorly understood that it has been described as "invisible". This invisibility leads to poor communication and gaps in understanding between professional health care providers and family caregivers. To address these gaps, health care researchers must work with family caregivers to incorporate their expertise on caregiving and create meaningful and sustainable research partnerships. A growing body of research is attempting to remedy the problem of caregiving invisibility and lay better foundations for successful integration between health care settings, family caregiving, professional caregiving, and community supports for families of CYSHCN. We identify high-priority gaps in CYSHCN caregiving research and propose research questions that are designed to accelerate growth in evidence-based understanding of the work of family caregivers of CYSHCN and how best to support them.

Research on Family Health and Children and Youth With Special Health Care Needs.

Families of children and youth with special health care needs (CYSHCN) can face challenges with regard to health and well-being. Health systems are designed to support CYSHCN but do not often consider the health and well-being of their family. Despite a growing body of literature, substantial gaps remain in our understanding of the impact of caregiving on family health and well-being and mechanisms of supporting families. In order to better understand and address these gaps, a national CYSHCN network developed a national research agenda to prioritize key areas of insufficient understanding of health and well-being for families of CYSHCN. Questions identified by the research agenda include: 1) How can family resiliency and adaptability be measured and improved? 2) How can we better assess family mental health needs and implement appropriate interventions? 3) What is the impact of family health on CYSHCN health outcomes? This paper describes a review of what is currently known regarding health for families of CYSHCN, gaps in the literature focused on the research agenda questions, and recommendations for future research. Based on the research agenda and current state of research for family health of CYSHCN, the authors recommend focusing on resiliency and adaptability as outcomes, using implementation science to address mental health concerns of family members and to further assess the impact of family health on health outcomes of CYSHCN. In addition, research should have a special focus on diverse populations of families and consider these questions in the context of different family structures.

What Types of Hospitals Do Adolescents and Young Adults With Complex Chronic Conditions Use?

BACKGROUND AND OBJECTIVE: Hospitalizations for children with complex chronic conditions (CCC) at pediatric hospitals have risen over time. Little is known about what hospital types, pediatric or adult, adolescents, and young adults (AYA) with CCCs use. We assessed the types of hospitals used by AYAs with CCCs. METHODS: We performed a cross-sectional study of 856,120 hospitalizations for AYAs ages 15-to-30 years with ≥1 CCC in the 2017 National Inpatient Sample. We identified AYA with CCC by ICD-10-CM diagnosis codes using the pediatric CCC classification system version 2. Hospital types included pediatric hospitals (n = 70), adult hospitals with pediatric services (n = 277), and adult hospitals without pediatric services (n = 3975). We analyzed age trends by hospital type and CCC count in 1-year intervals and dichotomously (15-20 vs 21-30 years) with the Cochran-Armitage test. RESULTS: The largest change in pediatric hospitals used by AYA with CCCs occurred between 15 and 20 years with 39.7% versus 7.7% of discharges respectively (P< 0.001). For older AYA (21 to 30 years), 1.0% of discharges occurred at pediatric hospitals, compared with 65.6% at adult hospitals without pediatric services (P < 0.001). Older AYA at pediatric hospitals had more technology dependence (42.5%) versus younger AYA (27.6%, p < 0.001). CONCLUSIONS: Most discharges for AYAs ≥21 years with CCCs were from adult hospitals without pediatric services. Higher prevalence of technology dependence and neuromuscular CCCs, as well as multiple CCCs, for AYA 21-to-30 years discharged from pediatric hospitals may be related to specific care needs only found in pediatric settings and challenges transferring into adult hospital care.

Health System Research Priorities for Children and Youth With Special Health Care Needs.

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.

Hospital Readmission of Adolescents and Young Adults With Complex Chronic Disease.

Importance: Adolescents and young adults (AYA) who have complex chronic disease (CCD) are a growing population that requires hospitalization to treat severe, acute health problems. These patients may have increased risk of readmission as demands on their self-management increase and as they transfer care from pediatric to adult health care practitioners. Objective: To assess variation across CCDs in the likelihood of readmission for AYA with increasing age. Design, Setting, and Participants: Retrospective 1-year cross-sectional study of the 2014 Agency for Healthcare Research and Quality Nationwide Readmissions Database for all US hospitals. Participants were 215 580 hospitalized individuals aged 15 to 30 years with cystic fibrosis (n = 15 213), type 1 diabetes (n = 86 853), inflammatory bowel disease (n = 48 073), spina bifida (n = 7819), and sickle cell anemia (n = 57 622) from January 1, 2014, to December 1, 2014. Exposures: Increasing age at index admission. Main Outcomes and Measures: Unplanned 30-day hospital readmission. Readmission odds were compared by patients' ages in 2-year epochs (with age 15-16 years as the reference) using logistic regression, accounting for confounding patient characteristics and data clustering by hospital. Results: Of 215 580 participants, 115 982 (53.8%) were female; the median (interquartile range) age was 24 (20-27) years. Across CCDs, multimorbidity was common; the percentages of index hospitalizations with 4 or more coexisting conditions ranged from to 33.4% for inflammatory bowel disease to 74.2% for spina bifida. Thirty-day hospital readmission rates varied significantly across CCDs: 20.2% (cystic fibrosis), 19.8% (inflammatory bowel disease), 20.4% (spina bifida), 22.5% (type 1 diabetes), and 34.6% (sickle cell anemia). As age increased from 15 to 30 years, unadjusted, 30-day, unplanned hospital readmission rates increased significantly for all 5 CCD cohorts. In multivariable analysis, age trends in the adjusted odds of readmission varied across CCDs. For example, for AYA who had cystic fibrosis, the adjusted odds of readmission increased to 1.9 (95% CI, 1.5-2.3) by age 21 years and remained elevated through age 30 years. For AYA who had type 1 diabetes, the adjusted odds of readmission peaked at ages 23 to 24 years (odds ratio, 2.3; 95% CI, 2.1-2.6) and then declined through age 30 years. Conclusions and Relevance: These findings suggest that hospitalized AYA who have CCDs have high rates of multimorbidity and 30-day readmission. The adjusted odds of readmission for AYA varied significantly across CCDs with increasing age. Further attention is needed to hospital discharge care, self-management, and prevention of readmission in AYA with CCD.