Authentic and Sustained Family Partnership in Neonatal and Pediatric Research

Abstract

Improving the health and well-being of children requires sustained partnerships with parents, community members, advocates, and researchers to identify and answer high-impact research questions. Meaningful engagement with families and communities in the design, conduct, and dissemination of health research increases the likelihood of successful recruitment and retention, patient-centeredness, and end-user relevance.1 Guidance on establishing and sustaining patient, family, and community partnerships in health research is now widely available (Table 1). These detailed, educational resources share 2 main characteristics. First, they provide advice on how to bring together researchers committed to improving health care and outcomes for a specific population, to work in partnership with similarly committed, diverse community partners motivated to improve the lives of the same population. Second, the resources provide guidance for conducting the work within a cocreation framework. Effective cocreation involves establishing how the group will work together, including group agreements, agenda creation, and meeting facilitation processes that encourage all participants to share and listen to diverse perspectives and provide alternative ways for those less inclined to speak up during meetings to contribute. Cocreation also requires a collaborative, nonhierarchical organizing structure to foster respect, genuine interest, and psychological safety, enabling community members and researchers to share their lived experiences and work together to design and conduct studies. Despite the growth of guidance and consensus on common best practices in the literature, we found few published examples of sustained partnerships with families and communities in neonatal and pediatric research.2–4The Neonatal Seizure Registry (NSR) is a collaboration among 9 American pediatric hospitals with level IV neonatal intensive care units (NICUs) and National Association of Epilepsy Center level IV pediatric epilepsy centers. The NSR has conducted research on seizures in newborns since 2012 (https://neonatalseizureregistry.ucsf.edu). From its inception, NSR has partnered with parents with lived experience of having a child with neonatal seizures and parent-led advocacy organizations to design, conduct, and disseminate NSR research. Figure 1 illustrates the overarching conceptual framework that guides our collaboration. Over the past decade, more than a dozen parents of children with neonatal seizures, recruited from the study sites or referred by one of 3 participating parent support organizations, have met online monthly with study investigators and research coordinators to design, conduct, interpret, and disseminate original scientific research. When grant funding allowed, parent advisors received a small stipend to partially offset their time and effort, and conference expenses were covered for parent copresenters. New members receive an orientation to the group’s aims and processes, as well as the opportunity to contact current parent members individually if they feel more comfortable asking questions or sharing feedback with other parents. Members can leave the group and stay connected through group communications, with the option to rejoin later.As the NSR research portfolio has matured, so have the roles and contributions of the NSR Parent Advisory Panel. Our aim in this article is to share lessons learned from a decade of family and community partnership in conducting neonatal and pediatric research and to encourage replication and innovation by other research groups. The following perspectives from NSR Parent Advisory Panel members provide both specific examples of how bringing together researchers and parents, and the cocreation process, contribute to sustained research productivity and effective dissemination. Specific examples of the contributions and impacts of the NSR parent advisory group for each phase of the research process are shown in Table 2.When I joined NSR in 2019, Hope for HIE, a parent-led organization dedicated to awareness, advocacy, education, research, and support for neonatal and pediatric acquired hypoxic ischemic encephalopathy (HIE), was connecting 4500 families worldwide. I knew through my own experience of having a baby in the NICU at risk of neonatal seizures and from reading thousands of stories from the HIE community in my role as Executive Director of Hope for HIE that neonatal seizures and their management were topics of great concern. The use and duration of antiseizure medication were common worries for families—balancing the need for seizures to be controlled and the risks of medication.6 I joined because the published work, the quality of the investigators, and the integration of family perspectives were compelling.Serving as a conduit for families’ lived experiences in a way that is meaningful and effective for our community is personally and professionally rewarding. The information we have produced together for clinician and parent audiences is invaluable. We can now direct families, clinicians, and researchers to the NSR website, which features coproduced content with evidence- and research-based practical information promoting communication and collaboration in caring for children and families. The feedback from our community about the resources, output, and our collaboration is positive and validating.Several key features contribute to our panel’s longevity and success. First, there is continuity in the aims and processes. Our work continues uninterrupted, even if researchers or parents rotate on or off. We express gratitude for those rotating off, and we welcome and are excited to learn from new members. Second, everyone’s views are valued, respected, and integrated as collaborators, with thoughtful engagement leading to work that reflects what matters to the children and families in the studies. Over time, the research group has pursued the priority topics we proposed in the early days. For example, long-term functional developmental outcomes were an early research priority, and we have since contributed to the design of those studies and are now publishing exciting findings that fill significant knowledge gaps (Table 2).7–9 Finally, we have benefited from having parents with diverse experiences and whose children are at different ages and stages of diagnosis and treatment, as well as from having researchers from diverse disciplines and the clinical research coordinators who engage directly with parents in the day-to-day implementation of studies. Together, we have been effective at solving problems that arose during the research process, including addressing how to maintain recruitment during the COVID-19 pandemic and other disruptions to clinical care or funding. Whenever possible, our group has copresented the findings from NSR studies at local, national, and international professional meetings. Based on the feedback we received, including parents who have lived experience, were integrally involved in study planning and implementation, and are also involved in disseminating the findings increased clinician understanding, confidence, and motivation to implement the findings in practice.I have been involved with NSR since its inception and continue to proudly engage in advancing our shared commitment to improving the lives of families affected by neonatal seizures. My motivation for joining the NSR team was to offer the perspective of a parent of a now middle-aged adult who had neonatal seizures and is now living with epilepsy. Our transition from pediatric to adult care was particularly challenging, and I wanted to share that experience to help inform pediatric research. I have seen firsthand how incorporating the family perspective enables clinicians to better understand the lived experiences of patients and caregivers, providing them with insights that extend far beyond what can be captured in brief clinical encounters. As a parent of an older child, I bring a retrospective view on the developmental challenges we faced and how we partnered with our doctors to navigate them. This perspective has allowed me to contribute meaningfully to the qualitative research process (designing questions and giving feedback on the codebook and results interpretation), to help shape new research questions (particularly around parent well-being and long-term development), and to collaborate in the development of reports the study team provides to NSR study families to summarize their developmental assessment results.Parental input has been integrated throughout all stages of the research process, including study design, data analysis, article preparation, and professional conference presentations (Table 2). At the same time, we as parents benefit from gaining a deeper understanding of current research and best practices from leading researchers and clinicians in neurology. Like many parents of children living with epilepsy, I feel a strong sense of purpose in contributing to work that may help other families facing similar challenges. In addition, we, as parents, play an important role in shaping new research directions. For example, our observations highlighted the prevalence of sleep-related challenges in children with epilepsy.10 By highlighting the complexities of sleep hygiene, our team was able to explore an area that might otherwise have been overlooked, ultimately guiding the research into meaningful new directions.Like many of our members, I bring multiple perspectives to the NSR Parent Advisory Panel. I am a parent of a child who began experiencing seizures at 18 months, and I am also a parent and family advocate in my role as the virtual support program manager at Hand to Hold, an organization that provides early-intervention mental health support before, during, and after a NICU stay. When our organization’s executive director, Katrina Moline, had other responsibilities that prevented her from continuing to participate on the panel, I joined to offer a caregiver perspective grounded in lived experience and to ensure that the parent voice was represented in research that impacts families of infants with neonatal seizures. My primary contribution has been sharing what it feels like to navigate a child’s seizure diagnosis—the questions families have, the emotional weight of the process, and what meaningful communication from researchers looks like from a parent’s point of view. Our organization saw the partnership as an excellent way to contribute our perspective and advocate for meaningful family-centered research. Our expectation was that this collaboration would lead to more relevant, impactful research outcomes and foster stronger connections between researchers and the communities they serve, and indeed it has. Overall, the experience has been positive. Being part of the panel allowed us to share lived experiences and highlight the needs and gaps families face. One of the areas in which we had a significant impact was in innovations to how the longitudinal studies respectfully and effectively engaged with families in recruitment and retention. We carefully reviewed and refined participant-facing materials (eg, consent forms, surveys, educational resources) over multiple meetings to ensure clarity, cultural sensitivity, and alignment with the lived NICU family experience. We identified, discussed, and debated language use and assumptions that could be misinterpreted, stigmatizing, or emotionally triggering for families. We advised on the feasibility and acceptability of data collection methods, while being mindful of the cognitive, emotional, and logistical burdens on families (Table 2).The research collaboration met our expectations for engagement and mutual respect, and it has been rewarding to see how our work led to the codevelopment of several practical resources for families and clinicians to improve shared decision-making and caregiving (https://neonatalseizureregistry.ucsf.edu/resources). Our organization has been a key partner in sharing these resources widely through our extensive network so that more families can benefit from the new knowledge learned in the studies.We would like to see all neonatal and pediatric research conducted in this community-partnered way, yet we are aware that there are challenges to be overcome. For families and family advocacy organizations, these include balancing the time commitment required with other family or organizational priorities, and the effort and resources required to ensure that diverse parent voices are consistently and authentically represented. This will require research funders and collaboration among family advocacy organizations to identify and support opportunities to further increase parent engagement and address barriers to participation, thereby maximizing the benefits of family partnership across all neonatal and pediatric research.When we began this work more than a decade ago, partnering with parents and community organizations was new to us and was still emerging as a best practice. We quickly realized that the unique perspectives of parent advisors would strengthen our work. Integrating parents into every aspect of our research is now fundamental for NSR. We started by confirming that the questions we wanted to ask as clinicians and researchers were the right ones,6 and we engaged the NSR Parent Advisory Panel to help shape the vision and direction of our work. For example, it was the panel that suggested adding evaluation of parental mental health and family well-being to our studies.9 We reviewed domains of mental health, family stressors, and resilience with our parent partners, and they selected the validated instruments that most closely reflected the areas they felt would be most valuable. The panel also helped us select children’s outcome measures that would enable our studies to answer the questions that mattered most to families. They helped refine study protocols to make them as accessible as possible to diverse communities (eg, when and how a study coordinator should approach a family; prioritization of telephone or online outcome assessments). The panel members wrote compelling letters of support for our funding applications, demonstrating the feasibility and potential impact of the studies.A particularly valuable contribution of the panel has been to collaboratively draft and edit all participant-facing materials, such as consent forms, newsletters, flyers, and infographics to ensure that the language used is clear and meaningful to participants. This was especially important for genomic studies, which were complex to explain and had novel ethical implications for participants. The panel collaborates with us to analyze data, interpret results, and craft products that resonate with audiences beyond the purely academic; these products also speak to families and contribute to health care policy and advocacy. NSR parent advisors are true partners in our academic products and have consistently participated as coauthors of 14 papers to date, and often as copresenters of the research at local and national meetings. Moreover, they take the lead in disseminating our findings to community audiences, codeveloping infographics in several languages and materials to make our work relevant and accessible to the people for whom it matters most (Table 2).Developing trust in a partnership like ours takes time and intentional effort. For over a decade, parents and investigators have shared both professional and personal milestones, supporting each other through illness and loss and celebrating new jobs and promotions, births, and graduations. We have found that showing up authentically as investigators is meaningful to parent and community partners, who, in turn, are willing to share meaningful aspects of their lives that are reflected in our work. Disagreements are inevitable in group work, and our group has had occasional differences in perspectives on research priorities or the best approaches to engaging families of children with health conditions in our research. When these occur, we lean on our long-standing relationships and seek ways to slow down our usual decision-making pace so that we can more deeply listen to each other and gain a shared understanding of our varied perspectives. By doing so, our group has always been able to find a way forward to a consensus decision.The NSR Parent Advisory Panel demonstrates that maintaining a collaborative parent- and community-partnered research advisory team for over a decade is a feasible and effective strategy to advance neonatal and pediatric research. The NSR Parent Advisory Panel has achieved exceptional, sustained productivity across several large studies and has made significant global contributions to addressing high-priority research questions posed by families of affected infants and the health care professionals who care for them. The investment of time, effort, and expense in supporting the panel was modest, considering the achievements, and the virtual meetings enabled diverse participation.While the team’s success is evident in its sustained partnerships, impactful publications, practice-changing research, and ability to translate research findings into practical support for families affected by neonatal seizures, there remain opportunities to expand its reach even further. For example, as children mature and enter adolescence and young adulthood, they face new challenges that inspire future investigations to improve diagnosis, treatment, symptom management, and prevention. Addressing these challenges calls for sustained attention to and investment in longitudinal patient, family, and community research partnerships.We share the extensive available guidance (Table 1) and our more than a decade of experience in the hope that other research groups will be inspired and encouraged to begin, extend, or deepen their partnership with parents and family advocacy organizations to strengthen the quality and application of neonatal and pediatric research to address pressing clinical problems. While we have provided specific examples of how such a partnership has direct positive effects on the conduct of research across the entire life cycle of studies (Table 2), we caution against being overly prescriptive, as the main goal of these partnerships is the cocreation of both the partnership processes and the products. Embarking upon a family partnership in research with a genuine commitment to cocreation, developing common goals, and maintaining curiosity, respect for diverse perspectives, humility, and shared joy in discovery will provide an excellent foundation for authentic, sustained, and productive partnerships to advance health care for children and their families.Additional current members of the NSR Parent Advisory Panel (study site affiliation): Dana Annis (Children’s National, Washington, DC); Jennifer L. Guerriero, PhD (Boston Children’s Hospital, Boston, MA); Ashley Hamlett, MEd (Duke University Medical Center, Durham, NC); Justin Means (Cincinnati Children’s Hospital Medical Center, Cincinnati, OH); Katie Means (Cincinnati Children’s Hospital Medical Center, Cincinnati, OH); Adam Numis, MD (UCSF Benioff Children’s Hospital, San Francisco, CA); Stephanie Rau, BS (University of Michigan, Ann Arbor, MI); and Yasmeen Rezaishad, BS, (UCSF Benioff Children’s Hospital, San Francisco, CA). The NSR Parent Advisory Panel collaborators critically reviewed and revised the article.