Navigating meaningful engagement: lessons from partnering with youth and families in brain-based disability research.

BACKGROUND: While patient and family engagement in research has become a widespread practice, meaningful and authentic engagement remains a challenge. In the READYorNot™ Brain-Based Disabilities Study, we developed the MyREADY Transition™ Brain-Based Disabilities App to promote education, empowerment, and navigation for the transition from pediatric to adult care among youth with brain-based disabilities, aged 15-17 years old. Our research team created a Patient and Family Advisory Council (PFAC) to engage adolescents, young adults, and parent caregivers as partners throughout our multi-year and multi-stage project. MAIN BODY: This commentary, initiated and co-authored by members of our PFAC, researchers, staff, and a trainee, describes how we corrected the course of our partnership in response to critical feedback from partners. We begin by highlighting an email testimonial from a young adult PFAC member, which constituted a "critical turning point," that unveiled feelings of unclear expectations, lack of appreciation, and imbalanced relationships among PFAC members. As a team, we reflected on our partnership experiences and reviewed documentation of PFAC activities. This process allowed us to set three intentions to create a collective goal of authentic and meaningful engagement and to chart the course to get us there: (1) offering clarity and flexibility around participation; (2) valuing and acknowledging partners and their contributions; and (3) providing choice and leveraging individual interests and strengths. Our key recommendations include: (1) charting the course with a plan to guide our work; (2) learning the ropes by developing capacity for patient-oriented research; (3) all hands on deck by building a community of engagement; and (4) making course corrections and being prepared to weather the storms by remaining open to reflection, re-evaluation, and adjustment as necessary. CONCLUSIONS: We share key recommendations and lessons learned from our experiences alongside examples from the literature to offer guidance for multi-stage research projects partnering with adolescents, young adults, and family partners. We hope that by sharing challenges and lessons learned, we can help advance patient and family engagement in research.

SUDEP: Living with the knowledge.

OBJECTIVE: To understand how knowledge of sudden unexpected death in epilepsy (SUDEP) impacted the lives of adult persons with epilepsy (PWE) and primary caregivers of both adults and children with epilepsy. METHODS: The principles of fundamental qualitative description guided this descriptive and exploratory qualitative study to document patients' and caregivers' perceptions and experiences. A purposeful sample of individuals (18 years or older) diagnosed with epilepsy or primary caregivers of PWE completed a single in-depth, semi-structured, one-to-one telephone interview. Categories of findings were developed using directed content analysis. RESULTS: A total of twenty-seven participants completed the study. This consisted of eight adult females and six adult males with epilepsy, ten female caregivers, and three male caregivers of PWE. All participants had become aware of SUDEP at least 12 months before their interview. Most were not informed about SUDEP by their treating neurologist and instead learned about SUDEP via alternative sources (e.g., the internet). All participants believed that knowledge of SUDEP outweighed the risks of being informed about it. Anxiety/fear related to SUDEP disclosure was generally not long-lasting. Caregivers of PWE were more directly impacted by SUDEP disclosure than adult PWE. Caregivers were more likely to make lifestyle/management changes due to learning about SUDEP (e.g., increased supervision and co-sleeping). Participants agreed that follow-up clinical support should be provided after SUDEP disclosure. CONCLUSIONS: Disclosure of SUDEP risk may have more significant impacts on caregivers of PWE than adult PWE in the form of lifestyle changes and epilepsy management. After SUDEP disclosure, follow-up support should be offered to PWE and their caregivers, which should be incorporated into future guidelines.

Valuing tacit nursing knowledge during the COVID-19 pandemic.

Public health nurses in Ontario, Canada, support the healthy growth and development of children across the province through a variety of programs including home visits for pregnant individuals and families with young children. During the COVID-19 global pandemic the needs of families increased while access to health and social services decreased. During this time, home visiting teams closely involved in supporting families also experienced staff redeployment to support pandemic efforts (e.g., case and contact management, vaccinations) and changes to the nature of home visiting work, including shifts to remote or virtual service delivery. To support nursing practice in this new and evolving context, a framework for capturing and sharing the tacit or how-to knowledge of public health nurses was developed. A valuing of this type of knowledge for informing future public health nursing practice - well beyond the pandemic response - was recognized as a pandemic silver lining when reflecting on two years of supporting home visiting teams in our province.

A commentary on the healthcare transition policy landscape for youth with disabilities or chronic health conditions, the need for an inclusive and equitable approach, and recommendations for change in Canada.

There is a growing number of youth with healthcare needs such as disabilities or chronic health conditions who require lifelong care. In Canada, transfer to the adult healthcare system typically occurs at age 18 and is set by policy regardless of whether youth and their families are ready. When the transition to adult services is suboptimal, youth may experience detrimental gaps in healthcare resulting in increased visits to the emergency department and poor healthcare outcomes. Despite the critical need to support youth with disabilities and their families to transition to the adult healthcare system, there is limited legislation to ensure a successful transfer or to mandate transition preparation in Canada. This advocacy and policy planning work was conducted in partnership with the Patient and Family Advisory Council (PFAC) within the CHILD-BRIGHT READYorNot™ Brain-Based Disabilities (BBD) Project and the CHILD-BRIGHT Policy Hub. Together, we identified the need to synthesize and better understand existing policies about transition from pediatric to adult healthcare, and to recommend solutions to improve healthcare access and equity as Canadian youth with disabilities become adults. In this perspective paper, we will report on a dialogue with key informants and make recommendations for change in healthcare transition policies at the healthcare/community, provincial and/or territorial, and/or national levels.

Building a culture of engagement at a research centre for childhood disability.

BACKGROUND: Engaging patients and family members as partners in research studies has become a widespread practice in healthcare. However, relatively little has been documented about what happens after the research study ends. For example, is patient and family engagement embedded in the wider infrastructure of organizations, and if so how? What are the long-term effects of engaging parents on research teams on the culture of how research is conducted? This study seeks to address these two gaps by examining how a culture of family engagement has been built over time at CanChild Centre for Childhood Disability Research at McMaster University in Ontario, Canada. METHODS: This study is based on ethnographic research methodology and combines elements of organizational ethnography, interviews, and collaborative auto-ethnography with parent partners, researchers, staff, and trainees. RESULTS: Since the inception of CanChild Centre for Childhood Disability Research at McMaster University in 1989, parents have been involved in research studies. Over time, this involvement evolved from being consulted on research studies to undertaking decision-making roles as partners and most recently as co-principal investigators. A growing infrastructure fosters a community of engagement that goes beyond the individual research study, and often beyond CanChild. This infrastructure consists of training, knowledge mobilization and social networking. In addition, the "softer" building blocks of CanChild's culture of engagement are an openness to learning from others, a commitment to relationship building, and a drive to grow and improve. These values are espoused by the leadership and are instilled in the next generation of researchers to inform both research and clinical work. While some challenges should be acknowledged when researchers and family partners work together on research studies, we identify a number of strategies that we have used in our studies to foster authentic and meaningful family-researcher partnerships. CONCLUSION: Engaging patients and families as partners in research constitutes a culture shift in health research, whereby studies about patients and families are carried out with them. Developing a community of engagement that transcends an individual research study is a step towards creating a culture of research that is truly shaped by the people about whom the research is being done.

More and more patients and family members are getting involved in health research studies as partners. However we do not know much about what happens after the research study ends. This article looks at how parents have been involved in research studies at CanChild Centre for Childhood Disability Research at McMaster University in Ontario, Canada. CanChild researchers, staff, students and parents were asked about their experiences of working together on research studies. One of the researchers then pooled together all of these stories, shared them with everyone to get their feedback, and wrote the initial draft of this article. All the people interviewed were invited to read the article and to add their thoughts and opinions until everyone was satisfied with the final product. Our shared stories show that a lot has changed since CanChild was established in 1989. At first, researchers consulted with parents when they were doing a study. Now, many parents are partners and co-principal investigators on research studies. CanChild has also developed opportunities for parents and researchers to get training in patient-family engagement and to network with each other outside of research studies. Researchers, staff, students and parents talked about what makes research partnerships successful, including: being open to learning from each other; taking the time to get to know each other as people; and always trying to do better. They also shared some of the challenges that come up on research studies and suggested strategies for working through them.

CHILD-BRIGHT READYorNot Brain-Based Disabilities Trial: protocol of a randomised controlled trial (RCT) investigating the effectiveness of a patient-facing e-health intervention designed to enhance healthcare transition readiness in youth.

INTRODUCTION: Youth with brain-based disabilities (BBDs), as well as their parents/caregivers, often feel ill-prepared for the transfer from paediatric to adult healthcare services. To address this pressing issue, we developed the MyREADY TransitionTM BBD App, a patient-facing e-health intervention. The primary aim of this randomised controlled trial (RCT) was to determine whether the App will result in greater transition readiness compared with usual care for youth with BBD. Secondary aims included exploring the contextual experiences of youth using the App, as well as the interactive processes of youth, their parents/caregivers and healthcare providers around use of the intervention. METHODS AND ANALYSIS: We aimed to randomise 264 youth with BBD between 15 and 17 years of age, to receive existing services/usual care (control group) or to receive usual care along with the App (intervention group). Our recruitment strategy includes remote and virtual options in response to the current requirements for physical distancing due to the COVID-19 pandemic. We will use an embedded experimental model design which involves embedding a qualitative study within a RCT. The Transition Readiness Assessment Questionnaire will be administered as the primary outcome measure. Analysis of covariance will be used to compare change in the two groups on the primary outcome measure; analysis will be intention-to-treat. Interviews will be conducted with subsets of youth in the intervention group, as well as parents/caregivers and healthcare providers. ETHICS AND DISSEMINATION: The study has been approved by the research ethics board of each participating site in four different regions in Canada. We will leverage our patient and family partnerships to find novel dissemination strategies. Study findings will be shared with the academic and stakeholder community, including dissemination of teaching and training tools through patient associations, and patient and family advocacy groups. TRIAL REGISTRATION NUMBER: NCT03852550.

SUDEP: To discuss or not? Recommendations from bereaved relatives.

PURPOSE: The overarching purpose of this descriptive and exploratory qualitative study was to understand the experiences of relatives of individuals whose deaths were identified as SUDEP and to explore their preferences regarding SUDEP counseling. METHODS: The principles of fundamental qualitative description informed all design decisions. Stratified purposeful sampling included 27 bereaved relatives (parent, sibling, spouse or child), aged at least 18 years, of 21 persons who passed away because of SUDEP. In-depth one-to-one interviews were conducted. Directed content analysis was used to code, categorize, and synthesize the interview data. RESULTS: There was consensus among all participants that the risk of SUDEP should be discussed with patients by their healthcare providers. Relatives opted for information on SUDEP at the time of, or shortly following, the diagnosis of epilepsy. Neurologists were identified as the healthcare providers who should discuss SUDEP with patients during a face-to-face encounter, subsequently supplemented with written information. It was identified that, when discussing SUDEP, emphasis should be on the risk factors, possible preventive strategies, and the rarity of incidence. CONCLUSION: The results of this study indicated that bereaved relatives wanted neurologists to inform patients about the risk of SUDEP, with optimal timing and setting of SUDEP counseling determined on a case-by-case basis.

Quality of life questionnaires for children with cancer and childhood cancer survivors: a review of the development of available measures.

OBJECTIVE: To identify and appraise all published quality of life (QOL) measures developed for use with children with cancer and childhood cancer survivors. METHODS: MEDLINE, CINAHL, EMBASE, PsycINFO, CancerLit, and Sociological Abstracts were searched from the inception of each database to 15 June 2009. Included articles were ones that described the development and/or psychometric evaluation of a QOL measure developed for use with children with cancer or childhood cancer survivors. Articles were appraised for adherence to internationally recommended guidelines for item generation, item reduction, and psychometric evaluation. RESULTS: Thirteen QOL questionnaires were identified by our search. Eleven measures are applicable to measuring QOL in children with any type of cancer, and two are specific to children with brain cancer. Four measures can be used to measure QOL in children undergoing cancer treatment, six can be used with children on or off treatment, and three are specific to childhood cancer survivors. While all measures underwent some degree of formal development and validation, item generation often did not involve children with cancer or their parents, and a number of measures did not describe or utilize recommended methods for item reduction and psychometric evaluation. CONCLUSIONS: Most of the measures identified in this review were designed to measure QOL concerns of children with any type of cancer and at any time, during treatment or survivorship. Our findings can help researchers and clinicians identify scientifically sound measures.