IRB: Ethics & Human Research
The Research and Family Liaison: Enhancing Informed Consent
The advancement of medical diagnosis and treatment requires scientifically sound and ethically appropriate research involving human participants. The inclusion of children in research necessitates assuring adequate protection for a population that generally relies on others to safeguard its interests. Although regulations have created a framework to promote ethical research with children, they represent a minimal requirement, and simple adherence to these regulations does not guarantee that a given research project is ethically optimal or meets the highest ethical standards. Family confusion regarding the purpose of research and the therapeutic misconception,1 failure to make child assent a meaningful process,2 cross-cultural challenges,3 and the dual role of the clinician/re-searcher4 can all compromise the ethical integrity of informed consent.
Some limited studies describe demonstrated solutions or interventions to meet these challenges, but no consensus exists regarding how best to present research information to families, to educate families about the research process, to ensure that families comprehend information presented to them, or to facilitate the voluntary participation of families throughout a research study. There is a growing consensus, however, that more effort should be focused on the informed consent process.5 Our institution addressed these challenges by creating the Research and Family Liaison (RFL) position within the pediatric research setting. This article describes how our institution wrestled with improving the informed consent process through the creation of this position, the methods employed to develop the role of the liaison, and the evolution of the liaison’s role over time. Future plans include a formal evaluation of the liaison role.
The Research and Family Liaison
The Research and Family Liaison (RFL) is a new staff position whose role was designed to assist families and to collaborate with members of the pediatric research team to facilitate the informed consent and assent processes. The RFL also creates informational materials relevant to the needs of child participants and their families. While the RFL’s role is similar to that of a patient advocate, distinct differences exist.
The concept of advocacy discussed in the research ethics literature ranges from advocates who serve in compliance and monitoring roles6 to persons focused solely on the research participant, with many iterations in between.7 The research advocacy role most often referred to in the literature is the Research Subject Advocate (RSA). This advocacy role falls within the compliance/monitoring model and is meant to support investigators who conduct studies at an institutions’ General Clinical Research Center (GCRC) that is funded by the National Center for Research Resources (NCRR).8 Our primary concern was that the RSA works mainly with a specified population (participants in GCRC studies) and does not usually oversee participation in non-GCRC studies. At our institution—a large urban tertiary care pediatric hospital located in the western United States—many research studies fall outside of the GCRC’s purview, thus limiting the RSA’s access to research participants. Another concern is the RSA’s strong focus on compliance and less direct interaction with families or involvement in research staff education. Thus, the RFL role resembles the RSA position with respect to the element of advocacy yet is distinct in its scope and focus.
When selecting a title for the position, we were concerned that the term “advocate” created the potential to generate a perception that families need to be protected from the research team and that team members do not themselves advocate for or seek to safeguard the interests of child participants and their families. This perception might set an adversarial tone in the relationship between an advocate and research team members, making it more difficult for the new staff position to be seen as valuable not just by families, but also by the research team. Thus, we chose the title “Research and Family Liaison.”
Due to the innovative nature of the project, two individuals were hired to shape the position, support each other in day-to-day role activities, effectively manage the workload, provide different skill sets, and allow for creative interaction. The RFL role requires individuals with excellent communication skills to facilitate interactions between the investigator and family. Bicultural and bilingual communication skills are valuable in this position if an institution is committed to improving the informed consent and assent process for children and families with limited English proficiency. Persons with and without healthcare backgrounds were considered for the RFL positions, but individuals without healthcare backgrounds were ultimately selected. The lack of formal clinical training offers a significant advantage: if the RFL does not understand the consent form or study information presented by the researcher, the child and the child’s family will likely also not understand.
While an RFL can provide insight into communication, he or she must also be able to navigate medical and research cultures in order to be successful. With that in mind, core competencies (Table 1) were developed to guide RFL orientation and skill training.
The two RFLs performed an initial literature review to identify problem areas related to informed consent that might benefit from further attention. The RFLs sought out stakeholders’ thoughts and opinions through family focus groups, multiple interviews with researchers, and informal discussions with research staff. Additionally, the RFLs directly observed informed consent conferences between investigators and families. These observations further elucidated needs in the research arena and increased the RFLs’ understanding of the established framework within which they would interact with staff and families.
We conducted nine semistructured interviews using a sample of pediatric investigators from a variety of departments selected to represent the types of research conducted at the hospital, as well as research presenting varying levels of complexity to participants. We employed a qualitative, descriptive approach to characterize researchers’ perceptions of factors that influence informed consent, challenges faced by investigators, and strategies for improving the informed consent process. These transcripts were reviewed along with associated field notes, and a coding scheme was developed. QSR N6 Qualitative Data Analysis Software was used to examine the themes. To assure trustworthiness or reliability of data analysis, a primary researcher read the transcripts and clustered the major themes. A second investigator then did the same. Reviewers identified, clarified, and adjudicated discrepancies.
Four themes emerged from these interviews. The first involved challenges the RFL would face when interacting with individuals who were physician-researchers. Many investigators who exercised this dual role noted that they were often more knowledgeable about the study than anyone else, and therefore believed they could be an invaluable resource to families who were considering enrolling their child in a study. However, the dual role carries the potential for conflict of interest that could undermine both the clinical and research relationship with the family.
The second theme revolved around ambivalence towards the concept of child assent. Many researchers seemed to have trouble distinguishing child assent from informed consent or parental permission. They believed that child assent was merely a version of informed consent for children rather than a separate, more nuanced process. The third theme involved investigators feeling inadequately prepared to appreciate and address cross-cultural issues in the context of research. Researchers anecdotally noted that children and families with limited English proficiency seemed more likely to be confused regarding the informed consent process. Finally, investigators sensed a need for training in ethical issues related to research, recognizing that presenting study information to families in a meaningful way requires skill and training.
Family Focus Group
A family focus group was conducted with nine parents from seven families whose children had been diagnosed with some form of cancer in the past 12 years and who were in remission. RFLs utilized a grounded theory approach for the initial focus group. Grounded theory looks to discover theory by examining concepts grounded in the data; it is a good method to use when inquiring about how processes—in this case, the informed consent discussion—influence the way in which things are accomplished through a given set of social interactions.9 The focus group was semistructured, and the discussion was recorded and transcribed verbatim. RFLs coded and analyzed the data to arrive at the core categories or themes discussed below.
The focus group provided the RFLs with further insight into family experiences during oncology trials, parents’ emotional state at the time of the informed consent process, the impact of hearing about research-related side effects, the reasons for enrolling their child in a study, and how laypersons understand clinical trials. Four main themes emerged from the focus group. First, parents agreed that the combination of their emotional state at the time of their child’s diagnosis and medical complexities related to the disease made understanding details of their child’s condition very difficult. The presentation of enrolling their child in research as an option for treatment increased the degree of complexity and required parents to make decisions for which they felt ill prepared. Second, most parents felt a sense of urgency regarding decision-making about their child’s treatment options—a result of their own emotional state, the language and behavior used by physicians, and time constraints dictated by the disease. Third, the parents’ sense of trust in the physicians and institution played a significant role in their decision-making. Finally, parents mentioned several resources they believed would have made their situation easier to handle by reducing some of the burdens they experienced. These resources included: 1) a person assigned to each family who could act as their main point of contact and guide them through their experience and 2) more information, along with different types and formats for receiving that information. Families wanted additional information about general research participation, specific information related to the study in which they had the option of enrolling their child, and a resource sheet to help them formulate questions to ask when considering research participation for their child. Parents also wanted access to this information in both written forms and via the Internet.
From February to December 2006, the RFLs observed more than 65 informed consent conferences to learn about the informed consent process, identify communication needs, and ascertain ways they might be helpful. Utilizing a qualitative descriptive design and an observational approach, they collected data from May to September 2006. This convenience sample consisted of research staff from two departments. A quality improvement instrument developed and pilot-tested by the project was used that focused on the way information was presented, the family’s ability to comprehend that information, and the effect of explanations, descriptions, and examples used during the conference discussions. The RFLs took field notes and also met with the child and parent(s) whenever possible to solicit their impressions of the conference. Data were entered and analyzed using the Statistical Package for the Social Sciences 12.0.1 for Windows. RFL field notes were triangulated with tool results to amplify findings.
Based on observations of informed consent conferences recorded via the quality improvement tool, the RFLs noted that families appeared to better understand information when medical jargon was avoided, the informed consent conference was limited to an hour, and visual aids were used to assist in the presentation of research information. This was corroborated by discussions with families conducted after the informed consent conference and confirmed during the family focus group. In addition, many families were left with the impression that they had to make a decision rather urgently, but did not know if that sense of urgency translated into hours, days, or weeks. Thus, the RFLs concluded that it would be helpful to families if investigators gave them a specific time frame for making a decision about whether to enroll their children in a study.
Other factors that appeared to aid families in the research process included: the investigator asking if the family had any questions before beginning the informed consent conference, especially when the family was dealing with a new diagnosis; acknowledging that the family was not expected to remember all the information presented; identifying the consent form as a reference document for information they may not remember; explaining the purpose of research in general; asking families’ preferences surrounding presenting specific information, such as percentages or general ideas; and explaining randomization and why it was used in studies. Families also seemed to have a better understanding of the research study when key issues were highlighted for them (e.g, “The most important things to remember are . . .”). Moreover, it appeared that families participating in informed consent conferences where the presenter was accompanied by another research team member (e.g., a fellow or research nurse) found the presence of that additional person and the ensuing dialogue that occurred between them helpful. Often if one person was not communicating an aspect of the research in a way that was understandable to the family, the other member was able to clarify the matter.
Finally, the RFLs identified problems in presenting study information to families with limited English proficiency. The presence of an interpreter at an informed consent conference changed its dynamics. At times, research staff would summarize study information, providing less detail than usual, so that even if interpretation was necessary, the conference would not increase in length. The experience research staff had in working with interpreters and the interpreter’s competency and familiarity with research concepts affected the quality of the conference. Cultural views regarding research in general and child assent specifically posed challenges to the research team working with these families.
RFL Activities and Lessons Learned
The RFL position was designed to improve the quality of informed consent, parental permission, and child assent in research at our institution. Based on observations of the informed consent process and discussions with investigators, staff, and families, we identified three RFL activities: direct engagement; education; and consultation. Our experiences with these activities have yielded important lessons for those seeking to improve the informed consent process for pediatric research. These lessons include 1) identifying potential barriers to incorporating the RFL position into the pediatric research setting, 2) clarifying the scope of the RFL’s role, and 3) determining how the RFL will interact with other institutional members.
We encountered several challenges when introducing a new staff position in the pediatric research setting that are likely to be common at other institutions. These challenges included finding the resources to support the position, concerns by the current research staff about the RFL’s role and operational barriers.
With regard to financial resources, the RFL position was originally funded for two years through a grant from the Health Resources and Service Administration Maternal and Child Health Bureau of the U.S. Department of Health and Human Services. Since the grant ended, our research institute has supported the position so that the RFL can continue to evolve to best meet the needs at our institution. Several research staff questioned the value of the RFL and expressed fear that incorporating such a position into the pediatric research setting might have negative results, such as fewer families agreeing to enroll their child in research. These fears were not realized. This and other concerns have been resolved over time as the research staff continually worked with the RFLs. The majority of staff appreciate and look for ways to further incorporate the RFL in their research activities.
Perhaps the most significant barrier to incorporating the RFL into the pediatric research setting had to do with operational issues. Ensuring that the RFLs were notified in advance about scheduled informed consent conferences was difficult initially. New activities take time to incorporate into existing routines. The RFLs found ways to alleviate the burden on research staff by familiarizing themselves with research processes in various departments and contacting key individuals who notify them of upcoming consent conferences.
Scope of the RFL’s Role
A “one-size fits all” approach to informed consent discussions is not optimal since it fails to address the seemingly endless variations that exist among research studies and those presenting the details of a research project to parents and children. The three activities of the RFL—direct engagement, education, and consultation—are an attempt to further clarify and meet the multiple needs of all involved in the research process. Direct engagement, education, and consultation define the scope of the role. These RFL activities will vary with the study, the investigator, and the family.
Direct engagement involves interactions with families, potential child participants, and research team members related to research and the informed consent process. The RFL’s involvement in a particular study may be due to a recommendation from the Institutional Review Board (IRB) or the request of a principal investigator based on particular features of the protocol (e.g., the study involves multiple study arms). Direct engagement may take a variety of forms depending on the level of risk involved and complexity of the study process.
Our focus group and RFL observations suggested that difficulty in understanding study information seemed to increase when studies presented complex research concepts (e.g., randomization), when stress levels were higher (e.g., a recent diagnosis), or when the study posed greater than minimal risk (as defined by the federal regulations and determined by the IRB). The RFL may act as a facilitator between families and research team members at such times. While establishing a relationship with the family and the research team, the RFL becomes knowledgeable about the communication styles and preferences of both the family and the investigator. This allows the RFL to act as a liaison and to mediate style and communication differences. It also helps to create an optimal environment within which a meaningful discussion of the research project can occur. Additionally, the RFL may follow the family throughout the entire study, acting as a sounding board for decisions regarding continued participation. An RFL can be a resource to families who may be uncomfortable raising certain issues with the research team. With less complex studies, the RFL is available to work with the presenters to improve their explanation of the study or to engage directly with a specific family if an investigator is concerned about comprehension.
Direct engagement also includes ongoing focus groups and discussions with families and child participants designed to identify new or recurring issues that need to be addressed. One example of such an activity is a family focus group with Spanish-speaking families led by an RFL. (Spanish-speaking families comprise 70% of our institution’s limited English proficiency patient population.) The purpose of the focus group was to better understand how families with limited English proficiency view and experience the research process and to discover ways to meet these families’ specific needs.
Several factors determine whether direct engagement by an RFL in a particular study will be helpful or necessary. Since an RFL cannot attend every informed consent conference, identifying those aspects related to the study, child participant, family, and research personnel will help clarify when an RFL’s involvement would be most valuable. Some studies may be more difficult to understand because of their complexity, the nature of the risks and benefits, or the setting (e.g., research being introduced within the context of a child’s treatment plan).
Because each family is unique, we discovered that the RFL has been helpful in assisting families with different needs and preferences by providing different services. For example, one family may need additional sources of information; the RFL would then gather that information for them with the help of the research team. Another family may require assistance formulating questions that will best help them receive the information they are seeking. It is difficult to say which families will benefit since the needs of each are so unique. However, we have found that, overall, families are assisted by the RFL’s presence.
Researchers who believe families are struggling with research information can refer them to RFLs. RFL involvement can be valuable when the researcher is also the clinician, when she has little consent process experience, or if she wants to improve her delivery of study information to families.
Another activity of the RFL is to educate. The RFLs created the Consent, Assent, and Parental Permission Mentoring Program (CAPP) (Table 2). The program aims to educate new research associates, nurses, fellows, and investigators. RFLs provide educational lectures and discussion sessions on topics such as ethics, research regulations, contextual issues of the informed consent conference, and aspects specific to presenting study information to families with limited English proficiency. The mentoring portion of the program includes two components. In the first, CAPP participants observe other research staff presenting study information to families “in the field,” followed by a discussion with RFLs about what worked well and what could have been improved. The second component involves the RFLs observing each CAPP participant as they present information to families. RFLs then provide these research staff members with feedback on their presentation skills. The CAPP program offers staff the opportunity to discuss issues about the informed consent process and serves as an environment where they feel supported and have access to resources to improve their interactions with families.
The RFL is in a unique position to identify issues and needs during the informed consent process. Providing education, training, and mentoring to research staff is a critical aspect of the role since it provides a large number of staff with the opportunity to gain information at one time, creates a forum to discuss challenges in the consent process, and defines methods to meet those challenges. Most institutions will not have the resources to utilize an RFL for each research visit, so training staff will likely become a primary focus of the RFL’s role.
In addition, RFLs are currently available at the request of individuals within the institution to consult about ethical issues related to the informed consent process in research. For example, RFLs have helped research teams think about optimal ways to present multiple studies to families during a single research visit and have aided in the construction of parental permission and child assent forms.
Consultation meets the needs of research teams prior to any interaction with potential participants and families. Consultation can be done at the individual study level, with an RFL assisting the research team in clarifying consent materials—for example, by creating diagrams and flow charts that can be used as visual aids when presenting study information to families. The RFL can also assist at an institutional level by constructing global parental permission and assent form templates and by providing consultation for informed consent and child assent policy.
These three activities—direct engagement, education, and consultation—have helped focus the role of the RFL and continue to give it shape as we seek to meet the needs of those involved in research and encourage a truly informed consent process.
RFL Interaction with Other Institutional Members
Hospital administrators may question why an RFL is needed if an RSA or other similar position already exists at an institution. We will focus on the RSA role since it is currently a position that may be similar to the RFL role. At our institution differences exist between these two roles. The RSA model includes many functions, including unbiased observation, provision of information in higher risk research study scenarios, and monitoring data and safety plans.10 Certainly the role of individual RSAs differ by institution, and certain tasks of a particular RSA may overlap with the activities of the RFL. However, the RFL role was meant to move beyond compliance and observation to further support the informed consent process through direct engagement, education, and consultation. The usual system of IRB review has not been sufficient to genuinely promote informed consent. It produces a consent document but provides little or nothing to help carry out the process of informed consent. The RFL role seeks to support the process of consent, not only its documentation, after IRB approval has been granted.
Many challenges exist when conducting research with children. The Research and Family Liaison position was conceived after examining current dilemmas in research, investigating advocacy-related literature, and observing and conversing with families whose children were research participants. The lessons that were learned were then applied in a specific research setting. Rather than advocating for an individual or group, the RFL advocates for the informed consent process. Broadening the traditional scope of informed consent (which focuses on recall and understanding), the RFL brings attention to the many factors that affect the presentation of study information such as the setting, the presenter, and the informational and emotional needs and preferences of each family. The RFL has been well received by families and research staff at our institution. The next step is to formally evaluate the impact of the RFL position as it is currently outlined and continue to encourage the evolution of the RFL’s role as a resource for research teams and families.
This project was supported by the Department of Health and Human Services Administration grant number H1SMCO6346. Many thanks to James Hendricks, President of Seattle Children’s Hospital Research Institute, for his continued support of the Research and Family Liaison position. Our gratitude to Mary Beth Foglia, who provided consultation and qualitative analysis. Finally, thanks to Dr. Benjamin Wilfond, Dr. Douglas J. Opel, and Carolyn Newcom for assistance in reviewing and editing drafts of this paper.
The University of Washington School of Medicine’s IRB approved the researcher interviews, the observation process, the information gathering process, and the family focus groups.
Halle Showalter Salas, MPhil, is a Research and Family Liaison, Zuraya Aziz, MS, is a Research and Family Liaison, and Nanci Villareale, RN, MSN, is the Administrative Director, Treuman Katz Center for Pediatric Bioethics, Seattle, WA; and Douglas S. Diekema, MD, MPH, is the Director of Education, Treuman Katz Center for Pediatric Bioethics, Seattle, WA, and Professor, Department of Pediatrics, University of Washington School of Medicine, Seattle, WA.
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Halle Showalter Salas, Zuraya Aziz, Nanci Villareale, and Douglas S. Diekema, “The Research and Family Liaison: Enhancing Informed Consent,” IRB: Ethics & Human Research 30, no. 4 (2008): 1-8.