Self-Management Characterization for Families of Children With Medical Complexity and Their Social Networks: Protocol for a Qualitative Assessment.

BACKGROUND
Children with medical complexity (CMC) present rewarding but complex challenges for the health care system. Transforming high-quality care practices for this population requires multiple stakeholders and development of innovative models of care. Importantly, care coordination requires significant self-management by families in home- and community-based settings. Self-management often requires that families of CMC rely on vast and diverse social networks, encompassing both online and offline social relationships with individuals and groups. The result is a support network surrounding the family to help accomplish self-management of medical tasks and care coordination.


OBJECTIVE
The goal of this study is to use a theoretically driven perspective to systematically elucidate the range of self-management experiences across families of CMC embedded in diverse social networks and contextual environments. This approach will allow for characterization of the structure and process of self-management of CMC with respect to social networks, both in person and digitally. This research proposal aims to address the significant gaps in the self-management literature surrounding CMC, including the following: (1) how self-management responsibilities are distributed and negotiated among the social network and (2) how individual-, family-, and system-level factors influence self-management approaches for CMC from a theoretically driven perspective.


METHODS
This study will encompass a qualitative descriptive approach to understand self-management practices among CMC and their social networks. Data collection and analysis will be guided by a theoretical and methodological framework, which synthesizes perspectives from nursing, human factors engineering, public health, and family counseling. Data collection will consist of semistructured interviews with children, parents, and social network members, inclusive of individuals such as friends, neighbors, and community members, as well as online communities and individuals. Data analysis will consist of a combination of inductive and deductive methods of qualitative content analysis, which will be analyzed at both individual and multiadic levels, where interview data from two or more individuals, focused on the same experience, will be comparatively analyzed.


RESULTS
This study will take approximately 18 months to complete. Our long-term goals are to translate the qualitative analysis into (1) health IT design guidance for innovative approaches to self-management and (2) direct policy guidance for families of CMC enrolled in Medicaid and private insurance.


CONCLUSIONS
Multiple innovative components of this study will enable us to gain a comprehensive and nuanced understanding of the lived experience of self-management of CMC. In particular, by synthesizing and applying theoretical and methodological approaches from multiple disciplines, we plan to create novel informatics and policy solutions to support their care within home and community settings.


INTERNATIONAL REGISTERED REPORT IDENTIFIER (IRRID)
PRR1-10.2196/14810.

Overall Impact: This is a resubmission of a qualitative study that explores the self-management strategies of families of children with complex medical conditions in the context of social networks. The investigators make the point that despite the significant personal and financial burden of taking care of these children few studies have examined the process of self-management beyond the individual caretakers. The resubmission is responsive to prior critiques. The scientific premise is sound observing that secondary to advances in medical life saving strategies more and more children with complex medical needs are surviving. Understanding the families' self-management strategies, needs, problem solving and quality of life is an important first step in influencing policy and the provision of needed supportive services. The application has a number of strengths including the investigative team, an innovative theory driven design, and a rigorous and well described approach to analysis. Sex a biological variable is appropriately incorporated into the analysis. Weaknesses are minor and addressable. The overall impact of the results of the study are expected to support both families and policy makers.

Significance:
Strengths  The number of children with medical complexity is growing with advance in medicine  Prior research has primarily explored family self-management on an individual basis  The consideration of social networks strengthens and supports the scientific premise Weaknesses  This study is exploratory and the importance of social networks is hypothesized (minor weakness)

Investigator(s):
Strengths  The PI of this project has both clinical and research expertise with the population of children with complex medical needs and their families  Valdez brings unique and important research expertise to the project  Valdez has successfully managed a number of funded research projects  Multiple PI plan provided

Weaknesses
 A history of collaboration is not evident (minor weakness)

Innovation:
Strengths  Synthesis of theoretical approaches (nursing, public health, systems engineering will guide the research  Contextualization of self-management in the broader perspective of networks of care Weaknesses  None noted 1 R21 NR017991-01A1 4 ZRG1 NRCS-V (08) KEIM -MALPASS, J

Approach:
Strengths  Strategies to accomplish study activities are well thought out  Well designed and theory supported approach to analysis  Trustworthiness of data discussed and supported  Recruitment is designed to include a range of disabilities and races among the participants  De-identified qualitative data will be shared on a publicly accessible qualitative library Weaknesses  Consideration that in today's electronic world social networks may also be electronic. This is not discussed in the proposal or how it will be addressed. (minor weakness)  Although the family is proximal and can engage in face to face interviews It is not clear if this will be a limitation for the social network participants identified by the proband family (minor weakness) Overall Impact: This revised application focused on identifying constructs within the caregiver networks among children with chronic conditions. The processes by which these systems operate are not well understood. Understanding their complexities is important to provide a foundation to approach/alter care if there are appropriate targets for intervention that will improve distal outcomes. A systems engineering approach is proposed and is appropriate. The qualitative design is rigorous. The approach is sound and now includes children/networks outside the University of Virginia. Sex as a biological variable is included. The enrollment table does not reflect all persons who will be enrolled. If successful, these data will add to the literature and provide a foundation for intervention work to address the needs of the social networks that care for children with complex medical conditions.

Significance:
Strengths  This project will evaluate self-management strategies in the network of caregivers for children with complex medical conditions -to determine what works and how it works and what doesn't work and why.
 Children with complex medical needs have large networks of caregivers.

Weaknesses
 There is little focus on the distal outcome -the application focuses on process which is important but is only the beginning -a better understanding of how these data will be used to designed interventions/ameliorate what is broken would have strengthened the significance of the application.

Investigator(s):
Strengths  Strong appropriate team -extension of ongoing work -adequate engineering health policy and medical personnel on the team Weaknesses  None noted

Innovation:
Strengths 1 R21 NR017991-01A1 6 ZRG1 NRCS-V (08) KEIM -MALPASS, J  The application of the systems engineering approach and use of multiple theoretical perspectives to guide data collection and analysis is innovate and rich. Overall Impact: The proposed project will examine how self-management strategies for children with medical complexity (CMC) are negotiated across caregivers and members of a social network and how this impacts children's health outcomes. There is a strong scientific premise for the proposed project because CMC individuals account for ~1/3 of all pediatric healthcare expenditures. This is a descriptive, qualitative study with very rigorous design and analytical methods. There is a strong integrated theoretical framework underpinning the study. The MPI team is very strong and they have appropriate clinical and personnel resources to recruit the target sample. The PIs were very responsive to the previous critiques resulting in an exceptionally strong application. The proposed project has the potential to yield critical information and advance the science around self-management interventions for CMC.

Significance:
Strengths  The scientific premise of the proposed study is to examine the self-management experiences of children with medical complexities (CMC), their caregivers, and extended social network which to date, have not been well-studied.
 It is posited that effective, integrated family-centered approaches for self-management that predominantly occur in the home and community could reduce the frequency of acute hospitalization and inpatient care.
 Although only ~1% of children are classified as CMC, they account for over 1/3 of all pediatric health care expenditures.
 The proposed project extends the MPIs' previous research by including members of the caregivers' and child's social network in the process of self-management.
 The study is based on integration of theoretical models from nursing, public health, and human factors engineering research.
 The proposed project integrates several theoretical perspectives from different disciplines to examine barriers and facilitators of effective self-management  Little data exist on effective self-management interventions for CMC  A focus on the extended social network of the primary caregiver provides a more comprehensive picture of the roles of these individuals and how these roles are negotiated.

Weaknesses
 None noted.

Approach:
Strengths  Scientific rigor was demonstrated  Sex as biological variable (SABV) was addressed  Nicely detailed description of the qualitative approach and analytical strategy is provided and will be led by PI Valdez who has expertise in human factors engineering and cultural anthropology  Recruitment strategy is designed to enhance heterogeneity of participants  The focus on family management and extended social network who all play an active role in the child's healthcare management will identify how this network may improve the children's health outcomes.
 Consideration of caregivers' barriers (e.g., work, transportation) which impact their ability to optimally manage their child's healthcare needs

Weaknesses
 The enrollment table(s) should also include the distribution of caregivers and members of the social network.  The PIs were very responsive to the previous critiques resulting in an exceptionally strong application.

Resource Sharing Plans:
Acceptable  An acceptable data sharing plan is included

Budget and Period of Support:
Recommend as Requested

THE FOLLOWING SECTIONS WERE PREPARED BY THE SCIENTIFIC REVIEW OFFICER TO SUMMARIZE THE OUTCOME OF DISCUSSIONS OF THE REVIEW COMMITTEE, OR REVIEWERS' WRITTEN CRITIQUES, ON THE FOLLOWING ISSUES:
NIH has modified its policy regarding the receipt of resubmissions (amended applications). See Guide Notice NOT-OD-14-074 at http://grants.nih.gov/grants/guide/notice-files/NOT-OD-14-074.html. The impact/priority score is calculated after discussion of an application by averaging the overall scores (1-9) given by all voting reviewers on the committee and multiplying by 10. The criterion scores are submitted prior to the meeting by the individual reviewers assigned to an application, and are not discussed specifically at the review meeting or calculated into the overall impact score. Some applications also receive a percentile ranking. For details on the review process, see http://grants.nih.gov/grants/peer_review_process.htm#scoring. Notice of NIH Policy to All Applicants: Meeting rosters are provided for information purposes only. Applicant investigators and institutional officials must not communicate directly with study section members about an application before or after the review. Failure to observe this policy will create a serious breach of integrity in the peer review process, and may lead to actions outlined in NOT-OD-14-073 at https://grants.nih.gov/grants/guide/notice-files/NOT-OD-14-073.html and NOT-OD-15-106 at https://grants.nih.gov/grants/guide/notice-files/NOT-OD-15-106.html, including removal of the application from immediate review.