Disclosure of individual research results

Most existing ethical guidelines justify disclosure based on the ethical principle of beneficence, and set a high threshold for disclosing individual research results. Research results that are unreliable (ie, have not been replicated in subsequent studies) or meaningless (ie, lack utility) are of little value to research participants, may cause unnecessary anxiety, and may lead participants to make ill-advised medical decisions.8–10 Because the goal of research is to generate knowledge for the benefit of society, disclosing individual results contradicts this aim if it compromises scientific validity. This goal may be over-ridden, however, when the information to be disclosed has sufficient potential benefit to the participant. Accordingly, most guidelines stipulate that research results must be clinically useful if they are to be disclosed to participants. For example, the US National Bioethics Advisory Commission states that disclosure should occur only when ‘(a) findings are scientifically valid and confirmed, (b) findings have significant implications for the subject's health concerns, and (c) a course of action to treat or ameliorate these concerns is readily available’.8

Critics of these guidelines reject the notion that disclosure should be limited to results that have clinical utility or that relate to conditions for which no treatment exists and argue that respect for persons generates an obligation to make individual research results available to participants.3 Accordingly, much of the debate in this domain has centred on extending the threshold for disclosure, to include results that have personal utility (eg, those that might influence life-planning or reproductive decision-making) or that might otherwise be meaningful to participants.

We argue that researchers have an obligation to disclose individual results to participants when doing so is consistent with both the ethical principles of beneficence and respect for persons. Beneficence requires that researchers minimise the potential harms and maximise the potential benefits of their research, and ensure that the potential benefits of the research (both to the participant directly and to society) are reasonably balanced in relation to the potential harms of research.

Respect for persons requires that researchers respect the autonomy of decisionally capable individuals, and protect those with developing, impaired, or diminished autonomy. A researcher satisfies this obligation by ensuring that participants are informed about the research, and allowing them to make decisions regarding participation based on their own values and preferences. In cases in which research participants are incapable of exercising autonomy, researchers show respect for them by appealing to a proxy decision-maker. The proxy decision-maker is charged with making an informed decision on behalf of the participant, based on either his or her expressed wishes or best interests.

Given these principles, we present four criteria for the disclosure of individual research results. Satisfaction of these criteria is sufficient to justify disclosure.

1. Disclosure does not undermine the scientific validity of the study.

2. The results are informative and reliable.

3. The potential benefits of disclosure to the participant outweigh the potential harms.

4. The participant consents to be informed of the results.

Before applying these criteria to the question posed at the outset, we provide some relevant background information on the vegetative state, the mental imagery task used to assess covert awareness, and the individuals participating in this research.

Vegetative state

In the context of clinical neurology, consciousness is typically understood as consisting of two components: ‘wakefulness (also known as arousal, referring to the level of consciousness) and awareness (the contents of consciousness)’.11 Wakefulness is a state in which the eyes are open and may be accompanied by a degree of motor arousal; awareness, meanwhile, entails having certain experiences. While patients in the vegetative state may demonstrate wakefulness, they are unable to voluntarily (ie, non-reflexively) respond to visual, auditory, tactile or noxious stimuli and fail to provide evidence of language comprehension or meaningful expression.12 It is on this basis that awareness is assumed to be absent. Accordingly, the vegetative state is often referred to as ‘wakefulness without awareness’. The vegetative state can be caused by ‘acute traumatic and non-traumatic brain injuries, degenerative and metabolic brain disorders, as well as severe congenital malformations in the nervous system’.12 Recovery of consciousness is unlikely after 12 months if the vegetative state is caused by brain trauma, and rare after 3 months if it is caused by anoxia or other brain disorders. The life expectancy of patients in the vegetative state is between 2 and 5 years, although survival beyond 15 years is not unheard of.13

The prevalence of the vegetative state in adults is between 40 and 168 per million in the USA, although precise epidemiological data are lacking.14 This is due in part to the fact that the clinical assessment of vegetative patients is based on subjective interpretation of inconsistent behaviours in patients with severely limited motor skills.15 It is possible that a patient who is aware, but behaviourally non-responsive, might be diagnosed as vegetative. In fact, several studies have shown that as many as 40% of patients diagnosed as vegetative actually possess at least minimal responsivity and awareness.16–18 Moreover, of the 60% who are correctly diagnosed as vegetative on the basis of behavioural diagnostic criteria, an estimated 17% retain covert awareness undetectable through routine clinical assessment.19

Mental imagery task

As neuroimaging has become increasingly sophisticated, new techniques have emerged to supplement traditional, bedside assessment methods. One technique used to detect covert awareness has been successfully developed.19 Study participants are placed in a functional MRI scanner and repeatedly instructed to imagine playing tennis or walking from room to room in their house for 30 s intervals. These tasks have been consistently and reliably shown to activate distinct areas of the brain in 100% of healthy volunteers.20 Successful activation of these areas in response to the researcher's commands reveals that these participants retain the capacity for sustained attention (required to maintain focus), language comprehension (required to understand instructions), response selection (required to switch between alternative tasks or conditions) and working memory (required to remember which task to perform when instructed).

The ingenuity of this technique stems partly from the fact that it relies on an established model of awareness assessment and translates it to a new modality.21 In the absence of specific neural markers, the only way in which an individual can demonstrate awareness is by responding to commands through some form of behaviour; this is true of both healthy individuals and those with disorders of consciousness. Given that patients in the vegetative state are behaviourally non-responsive, the mental imagery task provides an alternative mode by which they can demonstrate command-following abilities and thereby indicate awareness.

Researchers have also employed a modified version of the mental imagery task to successfully communicate with participants diagnosed as vegetative. In two cases, participants imagined playing tennis to indicate ‘yes’ and imagined walking from room to room in their house to indicate ‘no’ in response to externally verifiable questions, including their name, the current date, and the name of their personal support worker.19 ,22

Both the mental imagery task and its modified versions yield robust results. Successful completion allows researchers to be highly confident that the participant possesses awareness. However, if the participant is unable to complete the task, it is virtually impossible for researchers to determine whether he or she lacks awareness or if there was a failure in the test conditions. This may occur if the participant fails to understand the instructions, falls asleep during the test, or moves frequently, thereby preventing researchers from obtaining images suitable for analysis.23

Participant population and families

Since 2012, 24 individuals with an average age of 35.2 years (range 18–60 years) have participated in some form of mental imagery task. Of this participant group, 14 were diagnosed as vegetative, 9 as minimally conscious, and 1 as having locked-in syndrome. The median time since injury was 5.0 years (range 1–19 years); 11 of the participants had suffered traumatic brain injury; 8 were living at home; the remaining 16 were being cared for in hospital. Most participants had already undergone MRI or CT imaging and repeated behavioural tests to assess awareness. In all cases, the severity of the patient's neurological condition requires that the decision to participate in research and to be informed of results is made by the patient's family.

The families have discussed the participant's diagnosis and prognosis with a neurologist. Yet, few studies have examined the needs and attitudes of these families. In one study, Tresch and colleagues interviewed 33 family members of vegetative patients (ranging from 12 to 204 months after the injury with a mean duration of 54 months) and found that 90% of them believed the patient had some awareness of pain, environment, taste, conversation, or the presence of others. However, 32 of the 33 family members did not expect the patient to improve.24 Families also express a strong desire to be informed by physicians about what is done to their family member and to be involved in decision-making. The experiences in this study are consistent with Tresch's results; while family members commonly believe that the participant remains aware on some level, they have come to accept his or her condition and do not anticipate recovery.

Disclosure of individual research results in a disorder of consciousness context

What action guidance do our proposed criteria provide for researchers regarding the disclosure of individual research results in this study context? In the case of positive results, the four criteria we have identified above clearly support disclosure.

Disclosure of negative results

The obligation to disclose inconclusive or negative individual research results is a more complex issue. Failure to detect awareness can occur for a number of reasons, as mentioned above, and the benefits associated with disclosure of negative results are not easy to discern; for instance, families may misinterpret the fact that no awareness was detected to mean that no awareness is present. There is also the potential that the family may suffer emotional harm (eg, disappointment, anxiety, confusion) if negative results are disclosed.

Nevertheless, there are over-riding moral reasons why researchers should provide these results to families. First and foremost, if the researcher had previously agreed to disclose positive individual results during the informed consent process, failure to do so upon the conclusion of the research will likely be interpreted by families as a negative result. The potential harm may be greater if negative results are not disclosed and the family is left to interpret a lack of results on their own rather than having the matter explained to them by a competent professional. Leaving families to draw their own conclusions suggests a lack of respect and may cause greater anxiety than disclosure of negative results. Available data indicate that research participants want individual research results made available to them even if they are inconclusive or negative.32 ,33 While the desires of participants should not be the sole determinant of policy regarding disclosure (especially with respect to uncertain results), these data suggest that participants find the disclosure of individual research results potentially beneficial. It therefore seems plausible that family members acting on behalf of such participants might derive similar benefit from disclosure.

Future directions

Having shown that researchers ought to disclose individual research results to participant's families, how should the actual process of disclosure unfold? Although the disclosure of positive results may seem unproblematic, a skilled clinical communicator who is familiar with the research protocol and is able to convey the nuances of the results to each family will be invaluable. Clearly articulating to families the possible outcomes of research before participation will be instrumental in ensuring that families are able to give informed consent not only to participation but also to disclosure. We believe that engaging the families of participants in discussing these issues is critical to maximising the potential benefits of disclosure and minimising the potential harms. Further work is required to develop educational materials for families, and the impact of disclosure of research results on families should be studied systematically.

Of course, family members are not the only ones to whom the research results are relevant. Many of the practical benefits to the participant mentioned above must be implemented by the physician and other staff in charge of care. This means that, in addition to making individual results available to the participant's family, researchers ought to make positive results available to the participant's physician provided that the family agrees. If further research demonstrates that the potential benefits of disclosure to the patient are substantial, it may even be permissible for researchers to disclose positive results to the patient's physician or caregiver when the family declines to be informed.