Leading by example: How the Webster team's systematic review quietly rebuked the field's preoccupation with biomarker “disclosure,” and why it matters

Abstract

In this issue, Webster and colleagues report findings and provide direction following a systematic review and evidence synthesis designed to understand the decisional needs of research participants considering whether to receive biomarker or genetic risk information. The report serves as a foundation for a future of participant-centered approaches to the return of individual results in Alzheimer's disease research. This Ethics Response comments on the authors’ terminology choices highlighting the notable absence of references to disclosure. The Response concludes that alternative terms like return or sharing of results offer less stigmatizing and more patient-centered approaches to communicating biomarker and genetic test results.

The proliferation of imaging- and fluid-based biomarkers of Alzheimer's disease (AD) pathology has been accompanied by a corresponding set of concerns about whether, when, and how to return to the results of such testing to study participants ¹ and, increasingly, to patients. ² These concerns have spurred both theoretical inquiries and empirical investigations into the phenomenon of learning one's own AD biomarker status. Collectively, those endeavors have formed a new subfield in AD research, increasingly referred to as disclosure science. ³

Disclosure science has been defined as the systematic investigation of the psychological, behavioral, social, and legal implications of learning the results of one's own or a family member's biomarker testing for AD or related disorders (ADRD). ³ This growing body of literature has provided compelling evidence of the relative psychological safety of learning such information,^4,5 raised novel questions about how best to support those undergoing AD biomarker testing, ⁶ and pushed the field toward thoughtful anticipation of emerging risks and their implications for a future where AD biomarker testing is integrated into routine care of older adults and even those in mid-life.

In this issue of Journal of Alzheimer's Disease, Webster and colleagues detail findings from a systematic review and synthesis of qualitative evidence with the goal of understanding the decisional needs of research participants who are considering whether to receive individual AD biomarker or genetic risk information. ⁷ Findings provide clear direction for the field including the need for continued collective efforts to develop decision-support tools for participants, as well as the need for both general and project-specific work to identify participants’ information and support needs both prior to and following the return of results. In doing so the report serves as a foundation for a future of participant-centered approaches to the return of individual results in AD research.

Throughout the report, Webster and colleagues refer to the return, sharing, and communication of results to research participants. Notably absent are references to disclosure. While not explicitly declared, the authors apparently made an intentional decision to refrain from the use of the term. This commentary applauds the authors’ decision and urges leaders in the field to reconsider framing the work featured in Webster's review around the notion of disclosure, instead situating this line of research within the broader and extensive literature on health communication and health risk communication. This commentary asserts that use of the term disclosure may have unintended negative consequences for research, practice, and policy pertaining to ADRD. In full transparency, this commentary's author has been a contributor to the disclosure science literature including the development of a toolkit for “disclosure” of AD biomarker results.

As Webster and colleagues so clearly demonstrate, there are multiple ways of naming the process of providing a patient or research participant with the results of their AD biomarker or genetic testing. While various terms appear within the discourse on transparency in AD biomarker testing, disclosure has been used most consistently, appearing in titles of prior systematic and scoping reviews,^4,5 best practice guidelines, ⁸ and within the text of appropriate use criteria publications. ⁹ However, in contrast to the term's common usage in legal contexts, disclosure is not a term that is routinely used in medical settings. Indeed, when invoked in a medical context, disclosure seems to suggest that the information at hand is secretive and/or conveyed with reluctance. As protected health information with potentially life altering implications, there is no arguing the “high stakes” nature of one's AD biomarker status. However, there are innumerable medical tests that yield equally sensitive and potentially distressing information (i.e., cancer biopsies, psychiatric diagnoses, STI test results) but have not evoked the impulse to label their communication as a “disclosure.”

Two potential unintended consequences of using the term “disclosure” in context of AD biomarker testing warrant discussion. First, to the extent that the term connotes a sense of exceptional secrecy, its deployment at best succumbs to and, at worst, may indirectly perpetuate societal stigma around AD and dementia. Writings and research on the stigma associated with ADRD link the phenomenon to multiple negative consequences including rejection, discrimination, and exclusion from participation in socially rooted aspects of life. ¹⁰ Not surprisingly, fear of stigma has been implicated as a factor contributing to delays in detection of ADRD and hesitation to participate in research. ¹¹ Language used by experts should be sensitive to this and make every effort to avoid its perpetuation.

Second, disclosure is, by nature, a single, unidirectional event. While many would argue that use of the term is not intended to negate the importance of ongoing communication in the form of pre-disclosure counseling or post-disclosure support, use of the phrasing “disclose” nevertheless centers the communicative encounter around the action taken by the individual possessing rather than receiving the information. This is a subtle yet critical distinction as it is the information recipient whose AD biomarker status is in question, and to whom its life altering implications apply. By contrast, terms like communication and sharing imply a two-way interaction that includes both sending and receiving messages. ¹² These terms gives equal weight to both or all participants in the encounter and, to the point above, do so in a value neutral way that avoids implying that the information to be relayed is either good, bad, or exceptional in any way.

Each of the alternatives used by Webster and colleagues have advantages over the term disclose. As such, their review moves the field forward, not only in terms of its evidence synthesis, but as a model for honoring the agency of those learning of the AD biomarker status or genetic risk. If one accepts the assertion that participant-centered approaches to the return of research results begin with the adoption of participant-centered language, it follows to recognize Webster and colleagues for providing clear guidance to researchers striving to employ participant-centered approaches to human subjects research.