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.