Wendy Ungar, Co-Lead of CHILD-BRIGHT's Health Economics team, Senior Scientist at SickKids and a Professor at the University of Toronto, presented a Researcher Lightning Round talk following the TEDxYorkUSalon AutismInnovations on April 22, 2017 on the science of Health Economics. Watch 'Where does the money go? Shifting the curve in early autism intervention'
Youth engagement in the research enterprise has been growing in prominence over the last number of years. Multiple regional Young Person’s Advisory Groups (YPAGs) have become established not only to raise awareness of research studies that target youth, but to be actively engaged in the research process by providing input into study protocols and influencing research priorities aimed at the pediatric population.
Due to the unique perspective that these groups possess, CHILD-BRIGHT has partnered with KidsCan, the YPAG based out of the British Columbia Children’s Hospital Research Institute. The unique perspective of KidsCan advisors is expected to strengthen the pediatric focus of our Network’s research projects as well provide invaluable feedback on how to more meaningfully engage with youth and build research capacity.
The Training Core has been fortunate to engage with KidsCan over the last few months. In December of 2016, CHILD-BRIGHT collaborated with Can-SOLVE CKD and piloted a module of CIHR’s Patient-Oriented Research workshop (“Introduction to Health Research in Canada”) to a current group of Advisors. The session was intended to solicit feedback on practical strategies that can be employed to make the material more accessible to youth aged 13-18. After piloting the material, an intense hour-long focus group delineated a set of suggested practices that would lead to designing a more interactive and engaging workshop targeted at youth.
In February of 2017, the Training Core had the opportunity to employ the suggested recommendations and design an interactive workshop to orient newly recruited KidsCan advisors to the basic tenets of healthcare research in Canada. Participants were oriented as to how research projects unfold, how basic biomedical research differs from clinical research, and the role that ethics and peer review play in the process. Through this workshop, participants were further challenged with how the principles of patient-oriented research will play a role in Network-wide research projects to engage patients as partners.
Throughout the training session, it was clear to the facilitator that these youth are not only highly engaged, but strongly committed to having their voices represented at the table to influence research agendas that impact the pediatric population. Together, researchers and representatives from KidsCan will be able to roll out research projects that more meaningfully engage with youth through various stages of the research process.
“My daughter Ella is all rainbows and lollipops! She is always giggling and makes me so happy. But life as a single mom of three children, including one with special needs, is not always easy. I often feel quite lonely.”
Alyssa Keel lives in the Toronto area and is mom to three young children. Big brother Braeden is four years old, and twin sisters Raegan and Ella are three.
“Raegan and Ella are rare mono mono twins; monochorionic (meaning they shared the same placenta) and monoamniotic (meaning they shared the same amniotic sac). They were born premature at 29 weeks of gestation, and weighed only two pounds, 4 ounces and two pounds, 10 ounces at birth. They spent 80 days in the NICU at Sunnybrook,” says Alyssa.
“I gave birth via emergency c-section. Raegan was born first but it took longer to get Ella out. She suffered a grade 4 (severe) brain bleed. They warned us that it was likely she wouldn’t survive, and a few days later, she almost died. But she pulled through and now, three years later, she is a sassy, strong-willed preschooler!”
But Ella’s birth experience left her with special needs.
“Approximately one week after birth, Ella was diagnosed with hydrocephalus (a condition where there is an abnormal accumulation of cerebrospinal fluid in the brain). And shortly after her first birthday, she was diagnosed with cerebral palsy. She cannot walk, sit unassisted, or stand. She needs lots of support and equipment.”
“It’s a lot of work but my kids make me happy. I’m also very lucky to live in a big city where Ella can attend a special needs preschool, and where we are close to hospitals like Sunnybrook and SickKids. But my experience has changed my dream of what it would be like to have three kids. I’ve had to adapt, and I often feel very lonely. I don’t fit in. Most single moms don’t have as many children as I do, and many don’t understand the challenges I face with a special needs child. It’s hard to join their activities. For example, in the winter, they like to meet at indoor play centers, but the slides and games aren’t accessible to Ella. I want all three of my children to play and have fun.”
“It’s not easy to find accessible places, and finances in general are also a challenge, but I want Ella to have fun, to enjoy life, and to play in the park too. I do everything I can to help all my children live life to the fullest.”
Why CHILD-BRIGHT Matters to Me
“CHILD-BRIGHT matters to us because our story is only one of many; each family has a unique voice and story to be told, and in doing so, we open the possibilities of helping other families and children with different abilities. I always hope that our story will connect us with those families with similar stories, so that we can all feel understood and valued. It is my hope that the difficulties my children may face or that our experience navigating the system may help other families in the future, so that one day brain injuries, and cerebral palsy, will no longer feel so isolating.”
By Frank Gavin, National Liaison, Canadian Family Advisory Network & CHILD-BRIGHT Citizen Engagement Council Chair
For a long time (at least a few decades) families, mainly parents, have contributed to research focused on children’s health in two main ways: by consenting to and facilitating the participation of their own children as research subjects and by responding to surveys, questionnaires and interview questions, thus making themselves research subjects, an odd term since in both situations those who are called “subjects” often feel themselves regarded as “objects.”
Increasingly in the last decade or so—and accelerating very quickly in the last few years—patients and families have been invited and encouraged to participate in quite different capacities in health research, particularly as advisors and as co-investigators. This has been an international movement. (Check out, for instance, INVOLVE (http://www.invo.org.uk/ ) in the U.K. and the Patient-Centered Outcomes Research Institute (http://www.pcori.org/) in the U.S. In Canada the Canadian Institutes for Health Research’s (CIHR’s) Strategy for Patient-Oriented Research (http://www.cihr-irsc.gc.ca/e/41204.html) is the largest and best-known (but far from the only) driver of patient—a term that in this context includes the family members and other informal caregivers of patients—participation in all aspects of the research process.
The extensive and often deep involvement of patients in research is intended to ensure that the research directly addresses the needs and priorities of patients and has an important impact on their health and on the health of the system they depend upon. Patient-oriented research makes extensive use not only of the lived experience of patients but of the range of skills, interests, and areas of expertise that patients possess. It requires true partnership and collaboration.
The New Roles
Before identifying what can get in the way of such partnership and collaboration and what families and researchers can do to overcome the barriers, I’ll list some of the new roles family members have begun to play. (Note that in all these roles they are not working alone but with other research team members.)
- Identifying potential topics
- Ranking potential topics in order of importance, urgency, or relevance
- Refining research questions
- Asking questions, especially “why” and “how” ones
- Ensuring the language used is understandable—and not alienating—to patients and families
- Helping to recruit other patients and families as team members
- Mentoring new patient and family team members
- Mentoring researchers inexperienced in partnering with patients in this new way
- Suggesting ways of recruiting patients and families as research subjects
- Identifying ways to ease the anxiety and confusion of patients and families who are completing questionnaires and surveys (leading to more considered, less guarded responses), e.g. by indicating why particular questions are being asked, who will be using the information provided, who will and will not have access to it, and what use will be made of it in the short and long term.
- Participating in some interviews as interviewers
- Selecting the tools or measures (perhaps especially related to quality of life) that will be used
- Analyzing the information / data gathered
- Identifying the most effective ways of communicating information / findings to the public, especially patients and families
- Participating in knowledge translation (KT) activities
- Participating in advocacy activities, especially for policy- and decision-makers, arising out of the research findings / recommendations
Problems and Solutions
Here is a partial list of problems or barriers both families and researchers face in making their partnership effective and some solutions:
Being Representative of Families: Family members engaged in patient-oriented research and sometimes in other kinds of research are often explicitly or implicitly regarded as representative of other families. Sometimes they regard themselves in this way, in some cases slipping into such a role. Not being very careful about being a “representative” can be dangerous. If asked to represent other families, ask yourself and your team members how you can have the kind of regular access to a range of families that will help you be more truly representative. Even then, think carefully of what families you cannot—probably for a variety of reasons—contact or represent. It also helps to make sure there are at least two family members on each team. Among other benefits, this reminds everyone that there are many family experiences, perspectives, and voices and not a single patient or family voice or story.
Time and the Pace of Research: Many if not most research projects can take several years to complete. And there are often long periods (e.g. during the time ethics approval is being sought or while data are being collected) when family members of teams may have little if anything to do. These “time-related” realities should prompt everyone to recognize that some family members may be able to play their roles for only a portion of the research project or may have to change their roles. There should also be an explicit plan and a designated “go-to person” to insure family members aren’t left out of the loop during periods when they have little to do.
The Language of Research: The language of research might be more specialized, i.e. more unfamiliar to most people, than the language of a particular scientific discipline. Ordinary words, e.g. power, bias, control, etc. don’t have their usual meanings. Some researchers seem to love speaking in code; some have a hard time using plain language, understandably worried, perhaps, that something important will be lost or distorted. Obviously, highly specialized language can be a barrier to people new to this world. Glossaries can help, but more helpful is an explicit commitment to respond readily and un-condescendingly to requests for “translations” or plain-language terms. Families should also be willing to learn some of the more important and frequently used specialized terms since doing so may help them better understand the work they are part of.
Research Methodology: Researchers are often focused on methodology (e.g. sample sizes, inclusion and exclusion criteria, degrees of uncertainty, the sequences of tests, etc.). Researchers can make methodology less opaque by preparing some simple explanations of the most important elements of research methodology, by referring family members to existing accessible resources, by participating in training sessions where they can explain to patients and families why the “how” is so important, and by answering questions clearly and collegially. It’s important that such sessions not be “front-loaded” but rather spaced out over the course of the project. Too much information at the start can be more than daunting.
The Place of Stories / Lived Experience: Families sometimes report getting contradictory signals about the place or relevance of their own—or their child’s—experience. They are sometimes encouraged to join because of their “rich lived experience” and then surprised, disappointed, or hurt when they are not invited to contribute or even refer to that experience or when their recounting of part of that experience is--or seems to be--ignored or discounted. Researchers should discuss with family members at the outset whether and to what degree their experience, often conveyed through stories, should be explicitly shared. And families should also consider at the start whether they would prefer not to disclose particular parts of their experiences. In short, the place of stories should be determined by all, not assumed.
Disappointing Results: Sometimes—often, in fact—research yields answers different from the answers everyone hoped for. A new treatment or a new way of providing care may be no better and may even be worse than the tried and true, however limited the tried and true is. It’s important to know about this potential for disappointment at the outset and to remind oneself of it during the course of the project. Projects with “negative outcomes” also make long-term contributions.
The “Feel Good” Factor: This is still frequently present in research teams that include patients and families as partners. Patient-oriented research is a new phenomenon and researchers often know that engaging patients and families and having a team that has the attractive look of such engagement are traits funders are looking for and rewarding. All the more reason, then, to make sure the engagement and the work also feel good—and worthwhile—to patients and families. The team should periodically ask what difference this partnership is making to the quality and the applicability of the research. Evaluation tools are needed and are very much in the works.
Role Definition: Family members have sometimes been told their role was “to keep the researchers honest” or to “remind the researchers why they are doing what they are doing” or to be “inspiring.” There are obvious problems with such roles. It’s much more helpful for family members and researchers to discuss the roles family members will play as explicitly and realistically as possible, to review those roles during the course of the project, and to negotiate changes.
Money: Most everyone working within national or provincial contexts now agrees that patients and families who provide advice or act as co-investigators for research projects should not only have their expenses covered (travel, meals, childcare, parking, etc.) but also be given some remuneration for their work. On the other hand, some people, including some family members, would like to see the option to volunteer remain.
As for amounts, there is wide variation and sometimes strong disagreement about what constitutes appropriate remuneration. Should distinctions be made based on how much expertise or experience is required? Will such work become a career for some people? If so, is that a good or a bad thing? Issues related to remuneration are best dealt with early in the process and candidly. They should also be revisited over time. Any project budget should detail remuneration arrangements and indicate clearly which expenses will be covered.
The partnerships (among patients, families, researchers, clinicians, and policy-makers) required to make patient-oriented research succeed require much more than good intentions and well-developed social skills. All the participants need some training, some of it undergone together, other parts of it best undergone by patients alone, families alone, researchers alone, etc. CIHR is now developing a foundational curriculum in patient-oriented research and INVOLVE has many training resources on its website. There are two points to emphasize: 1) training should be viewed as an ongoing activity, not just as something that happens at the start of a person’s or a team’s work, and2) while everyone needs some training, everyone can, with experience, also deliver some part of the training, often to individuals playing quite different roles in the research project.
Many, probably most, family members who have found themselves in these new roles in health research report experiencing various frustrations and difficulties. In an often highly credentialed world it isn’t easy to feel confident and find one’s voice, and being asked “how do you know that?” when the answer is intimately linked to a painful experience can cause more than discomfort. Still, families can offer—and have offered—what no one else can; they bring a commitment that can fortify others and the whole enterprise; and they, knowing how the elements of their children’s care and of their children’s lives are connected or disconnected, can illuminate both the dark corners and the big picture.
“Nowhere is the importance of investing in health more apparent than in our care of the developing child: the earlier the investment the greater the return on investment.”
Read more about Three To Be's generous commitment to our work here.
From October 25 to 27, members of our CHILD-BRIGHT team attended CIHR's Strategy for Patient-Oriented Research (SPOR) Summit in Ottawa, an event that brought together 300 SPOR stakeholders, including patients, federal/provincial/territorial ministries, partners, researchers and clinicians.
A marketplace opened the three-day event, during which time our team hosted a widely visited booth and exchanged with other attendees on the vision and mission of our new network. We were joined by some of our network funding partners and also got to visit the booths and meet other SPOR network members, SPOR support unit staff, and affiliated organization from across Canada and most importantly, families and patients.
Minister of Health Jane Philpott then opened the three-day series of workshops, panel discussions and informal gatherings during which time all attendees learned and exchanged on various issues relating to patient-oriented research.
Attendees on behalf of CHILD-BRIGHT were:
Dr. Annette Majnemer, CHILD-BRIGHT Network Director
Dr. Steven Miller, CHILD-BRIGHT Network Co-Director
To Nhu Nguyen, CHILD-BRIGHT Network Manager
Frank Gavin, patient representative and CHILD-BRIGHT Citizen Engagement Council (CEC) Chair
Donna Thomson, patient representative and CHILD-BRIGHT committee member
Kate Robson, patient representative and CHILD-BRIGHT committee member
Christine Marcotte, patient representative and CHILD-BRIGHT Communications Manager
Denise Lo, SickKids Foundation
Dr. Keiko Shikako-Thomas, CHILD-BRIGHT Principal Investigator, Jooay Project
Read about our partnership with the Women and Children's Health Research Institute