Returning Individual Research Results to Participants Guidance for a New Research Paradigm (2018) / Chapter Skim
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5 Advancing Practices for Returning Individual Research Results
5 Advancing Practices for Returning Individual Research Results
Pages 189-240
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From page 189... ...
The committee acknowledges, however, that expanding the return of research results places new demands on the research enterprise, including the development of needed expertise on study teams and assembling the resources needed to offer and return individual research results appropriately. Inconsistency in practices will need to be addressed in order to minimize the risk of harm from the return of results, an evidence base will be needed for the development of best practices for returning results, best practices will need to be developed and disseminated, and these best practices will need to be broadly implemented in order to prevent inequities.
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Returning research results can be done (and it can be done well) , up-front investments can be scalable, and the development of best practices over time will improve the consistency and quality of the process of returning individual research results.
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From page 191... ...
Challenges communicating clinical test results and other medical information effectively may stem, in part, from gaps in health literacy1 and other forms of literacy, such as graph literacy and health numeracy.2 In 2006 the National Center for Education Statistics released a National Assessment of Adult Literacy and found that "the majority of adults (53 percent) had intermediate health literacy while about 22 percent had basic and 14 percent had below basic health literacy" (National Center for Education Statistics, 2006, p.
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Learning from Current Practices in Return of Individual Research Results Research results differ substantially from clinical test results in a number of ways, which limits the degree to which clinical experience can offer guidance on the return of research results. Most notably, research results are often associated with a greater degree of uncertainty as a result of incomplete scientific knowledge, and the uncertainties present at the level of individual results are even larger than the uncertainties present in aggregate results.
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Returning Individual Genetic Research Results In the field of genetics, some research investigators and direct-to-consumer (DTC) companies have been using and exploring methods for returning individual 3 "A reference range is a set of values that includes upper and lower limits of a lab test based on a group of otherwise healthy people.
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From page 194... ...
, a group at the University of Michigan developed a process for returning results to family members involved in a linkage study.4 The process involved pre-counseling education and assessment, during which the risks and benefits of receiving results were explained and informed consent was obtained, and also a post-testing disclosure of results with clinical counseling by a multidisciplinary team (Biesecker et al., 1993)
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Wendy Chung, the Kennedy Family Professor of Pediatrics and Medicine at the Columbia University College of Physicians and Surgeons, has discussed the variety of methods used by her team to return research results in their studies of the genetic basis of human diseases (Wynn et al., 2017) .7 The communication methods employed included giving participants the option of receiving results with a genetic counselor present to enable in-depth interpretation and contextualization of the genetic results or providing participants their nucleotide sequence data in a BAM file,8 leaving interpretation up to the participant (perhaps through the use of outside interpretive services the participant could pay for)
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. The return of results in this field is done because the research participants generally have a significant interest in learning their individual research results for their 9 Testimony of Wendy Chung of Columbia University at the public meeting of the Committee on the Return of Individual-Specific Research Results Generated in Research Laboratories on September 6, 2017.
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. Therefore, determining the meaning and clinical interpretation of such test results can be a challenge, and "reference ranges do not provide conclusions on safety or risk.
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Haynes et al. concluded that "scientists should include community partners from the target population in the development of research and data disclosure strategies in order to enhance the quality of research, to support the rights of the study participants to know their individual results, and to increase environmental health literacy" (Haynes et al., 2016, p.
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. Researchers reviewed existing risk communication strategies, networked with more experienced investigators, sought the community's input, and assessed local health literacy and numeracy.
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. GROWING UP FEMALE During the consent process, the child participants were informed that elevated blood sugar, insulin, blood pressure, and cholesterol would be reported back to their parents and that "the investigators will tell you about significant new findings developed during the course of the research and new information that may affect your health, welfare, or willingness to stay in this study" (Hernick et al., 2011, p.
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For example, the discovery of a genetic variant in a participant provides information about that individual participant's future disease risk but, if the variant is heritable, the discovery may also offer information about family members' risks and lead to generalizations about a group's risk. Similarly, an environmental exposure result may be relevant not only to the participant, but potentially to others who share that environment (e.g., family members, neighbors, coworkers)
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SETTING PARTICIPANT EXPECTATIONS IN THE CONSENT PROCESS AND BEYOND In returning research results to participants, investigators should set participants' expectations up front (Tarrant et al., 2015)
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Second, due to the variability in research results and the frequent lack of clear reference information, participants may need help in determining whether they want the results and, if so, what the results might look like. To further shape participant expectations and guide decision making during the consent process regarding which results, if any, they would like to receive, it may be helpful to provide participants with examples of what results may look like (NHGRI, 2018)
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. Third, receiving either clinical or research test results can result in both benefits and harms, and it is critical to address these during the consent process.
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For example, technology can be a particularly helpful way to incorporate the principles of health literacy (as discussed previously)
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TRAINING AND STAFFING • Train staff on the informed consent process for the return of individual research results, including how to address possible challenges that may occur, how to encourage participant questions, and how to prioritize information shared with the participant. MATERIALS FOR CONSENT • Write with little to no technical jargon.
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• Rely on verbal exchange for supporting and reminding participants about the most important aspects of returning research results, including risks and benefits as well as confidentiality. • Use the "common language" used by participants as well as plain language.
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CONCLUSION: The heterogeneity of research study designs and populations means that different consent processes will be appropriate in different situations, but regardless of the type of consent process, clear communication appropriate to varying levels of health literacy is essential. Recommendation 9: Ensure Transparency Regarding Return of Individual Research Results in the Consent Process.
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From page 209... ...
Staged Consent • Reduces time spent discussing secondary findings • Following-up and recontacting participants for during initial consent; more detailed information consent could be costly and burdensome provided later if secondary findings occur • Participant's decision to enroll in study made • Participant makes decisions on secondary findings without full information about potential return of closer to the time of receipt, can consider current secondary findings circumstances • Depending on procedure, recontacting participant • More detailed and specific information for participant may reveal unwanted information about a secondary finding, with negative impact on participant • Participant maintains choice about types of secondary findings to receive, or about opting out altogether continued 209
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Outsourcing • Researchers do not have to spend time explaining • Though participant receives all data, may not implications of secondary findings -- would be become aware of medically significant data outsourced to entities that specialize in interpretation/ • Services for genomic interpretation and counseling communication of, for example, genomic information not widely available at present -- could be mitigated if • Costs associated with return of results avoided, demand increases including recontacting participants, hiring additional • May exacerbate health disparities, since further staff to communicate results, etc. interpretive services may be costly and hence limited • Participant spared immediate task of deciding which to wealthy participants secondary findings to receive; can pursue this question later with entity of their choice • Researchers' obligations simplified to the return of each participants raw data SOURCE: Adapted from Appelbaum et al., 2014.
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. Consequently, the processes for returning individual research results must either (1)
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Facilitating Understanding of the Meaning and Limitations of Results Through Reference Information Having access to information is not the same as being able to understand and use that information. In particular, studies in both the consumer product marketing and medical decision-making fields have shown that people find it difficult to interpret unfamiliar data in the absence of relevant reference standards (Hsee, 1996; Zikmund-Fisher et al., 2004)
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To be clear, providing reference information for a result is not the same as providing personalized interpretation, such as clinical guidance. Clinical guidance requires integrating a research test result into the participant's individual circumstances (e.g., known medical conditions, family history)
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When reference standards for a result are not known, investigators should weigh the benefits and risks of return and consider whether a return of aggregate results only would be more appropriate than a return of individual results. Regardless of whether aggregate or individual results are returned, the fact that reference information does not exist should be explicitly communicated to participants.
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Note that prevalence is of high value in determining the likelihood that a test result represents the true value 5. Prevalence observed for the study
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Because relative standards provide only limited and potentially misleading meaning, it is generally preferable to provide absolute reference information (just as absolute risk communication is generally preferred over relative risk communications) , though the committee acknowledges that this will not always be possible (Dunagan et al., 2013; Trevena et al., 2013)
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. The use of plain language can help research participants D better understand the limitations related to the validity of the test result and the implications in terms of whether the data should be relied on for decision making.
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. For example, it may be difficult to communicate to research participants how much or how little effect a particular genetic marker may have on the incidence or severity of a condition -- and, accordingly, whether an intervention or other action is appropriate.
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CONCLUSION: The state of scientific knowledge about a particular test guides the types of reference information that are available and can be provided to research participants when returning individual research results. When the context for a test result is well established and standardized, then a strong presumption is that this reference information will be provided.
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Changes in community, federal, or industrial practices may be needed to develop better guidance for how the research committee needs to approach these situations. To deal with the fact that research participants often struggle to make sense of consent documents, the 2018 proposed revisions to the Common Rule mandate that consent documents provide a "key information" section at the beginning of the consent document that contains a "concise and focused" description of the research and summarizes the project information most important to potential subjects in making their decision whether to participate (Federal Register, 2017)
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Most research participants, however, are unlikely to think about these threats to validity and interpretability. Hence, research results are prone to misinterpretation (e.g., confusing a research result with an established clinical test result)
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. Because people tend to assume that any test results they receive are both precise and accurate, providing information that conveys the uncertainty of the result is critical, particularly since the potential for error increases in research contexts.
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, and • mailing of printed materials. Other reports have described a number of different factors that go into the selection of an appropriate communication method for returning individual research results (Fitzpatrick-Lewis et al., 2010; MRCT Center, 2017a)
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In some instances, the research results could be tethered to an existing patient portal or EHR, such as in cases where a research participant may also be a patient receiving clinical care within the institution. Although such portals typically feature a secure two-way e-mail communication option, there are a number of potential disadvantages, including a lack of opportunity for the synchronous communication of a phone or in-person return and the fact that the portal is less likely to be used by racial and ethnic minority and rural populations and those with limited health literacy or technology proficiency (Sarkar et al., 2010, 2011; Sharit et al., 2014)
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( is open-source software that allows a user to create tailored messages and visualization outputs that are responsive to overlapping participant characteristics such as language, age, and level of health literacy (Arcia et al., 2015; Unertl et al., 2016)
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. The below example of a number-line display includes both color gradients and an additional harm anchor to help users identify the values at which a particular test result becomes clinically concerning (Zikmund-Fisher et al., 2018)
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From page 227... ...
and also includes a brief visual summary of the key take-home messages to display the risks associated with the test results. The design was based on best practices for visual displays as well as the research participants' preferences for communication that provided important context about the result including reference range and associated risks.
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From page 228... ...
. In many situations, a multimodal approach to returning individual results will be beneficial (e.g., delivering results via mail or electronic portal and then following up with a phone discussion or in-person meeting to offer participants a chance to ask questions and seek clarification)
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From page 229... ...
among various electronic clinical information systems. For the return of results through EHR or a patient portal, this would include core content such as participant name, name of laboratory test, and laboratory test result (Fast Healthcare Interoperability Resources, 2017)
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The committee notes that research to study the impact of returning individual research results is already under way, but more work will be required to generate best practices (Genomes 2 People, 2018; Miller
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As discussed in Chapter 3, institutions may be able to assist research teams by developing the required infrastructure for the return of results, and this could include infrastructure that enables investigators access to core communication expertise. As the return of individual research results becomes more widely practiced, including research communication cores into institutional development grants may be considered and would provide investigators access to experts and a standardized mechanism for communication and avoid potential costs associated with study-by-study assessments.
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In Informed consent and health literacy: Workshop summary. Washington, DC: The National Academies Press.
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2013. Method for the development of data visualizations for community members with varying levels of health literacy.
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in electronic health records: A systematic review. Journal of the American Medical Informatics Association 24(2)
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2017b. Evaluation of automatic class III designation for the 23andme personal genome service c arrier screening test for Bloom syndrome: Decision summary correction.
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2015. The patient portal and abnormal test results: An exploratory study of patient experiences.
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2014. Returning individual research results for genome sequences of pancreatic cancer.
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2016. Getting it right with the Precision Medicine Initiative: The role of health literacy.
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2010. The literacy divide: Health literacy and the use of an Internet-based patient portal in an integrated health system-results from the Diabetes Study of Northern California (DISTANCE)
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2017. Graphics help patients distinguish between urgent and non-urgent deviations in laboratory test results.
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