Introduction
Information on possible heightened risks for developing hereditary cancer, after completion of cancer genetic counseling, has implications for both the counselees and their at-risk relatives. Based on the results of a possible DNA test and counselees’ family cancer history, counselees receive individualized medical recommendations. These recommendations may include risk reduction surgery, regular screening, and DNA testing of relatives (Stichting Opsporing Erfelijke Tumoren and Vereniging Klinische Genetica Nederland Werkgroep Klinische Oncogenetica
2005; Menko et al.
2013). However, at-risk relatives for whom this information might also be of medical benefit do not always receive this information (Peterson et al.
2003; Claes et al.
2003; MacDonald et al.
2007). Thus, they lack the opportunity to make a well-informed decision regarding whether to attend genetic counseling, DNA testing and/or surveillance activities. The possible barriers playing a role in the family communication process are manifold and complex, and have been reviewed by Chivers Seymour et al. (
2010) and Wiseman et al. (
2010).
Current Dutch guidelines (
2012) state that counselees and clinical geneticists are responsible for optimal dissemination of information to the family, in the case of hereditary and familial cancer syndromes for which effective preventive interventions are available, in line with international recommendations (Godard et al.
2006). It is recommended that, during cancer genetic counseling, counselees be informed about the possible implications of genetic testing for their relatives. As part of the genetic counseling, counselees receive a letter which summarizes and reaffirms the information provided during the counseling sessions. In hereditary cancer syndromes, in which a pathogenic mutation has been detected, a separate family letter is provided to be distributed by the index patient to all at-risk family members. This letter provides information on the diagnosis within the family, as well as the possibilities regarding DNA testing and preventive options.
Although counselees report feeling responsible about disclosing information to at-risk relatives (Hughes et al.
2002; McGivern et al.
2004; Wiseman et al.
2010), they may face several barriers in communicating hereditary cancer risk information to their relatives; these barriers can be categorized as: 1) lack of knowledge, i.e., a limited understanding of which family members ought to be informed about what, 2) motivation due to the desire to protect the relative or oneself e.g., from negative emotions, and 3) self-efficacy, i.e., feeling unable to inform relatives because one may not be able to reach them or does not feel confident about informing them correctly (Claes et al.
2003; MacDonald et al.
2007; Wilson et al.
2004; van den Nieuwenhoff et al.
2007). The counselees report a need for information and support in communicating genetic risk information to others (Hayat et al.
2012; Ratnayake et al.
2011). Counselees also express that their lack of confidence in disseminating information may be improved by more professional backup (Suthers et al.
2006).
Recent interventions aimed at improving disclosure of hereditary risk include the provision of an educational tool (Kardashian et al.
2012), enhanced cancer genetic information and an extra consultation with a specialist nurse who specifically addresses family communication (Roshanai et al.
2009), communication skills training (Montgomery et al.
2013), or additive counseling focusing specifically on discussing at-risk information with relatives (Forrest et al.
2008). In the two controlled studies, the interventions showed positive results on satisfaction, but not on family communication (Montgomery et al.
2013; Roshanai et al.
2009). A cohort study found an increase in the proportion of relatives attending genetic counseling services. Although it was assumed that this increase was due to improved family communication, this was not specifically investigated (Forrest et al.
2008).
We designed a counseling intervention targeted at assisting counselees in informing their at risk relatives as an addition to the genetic counseling services already provided in the Netherlands by genetic counselors. The intervention is based on the most important principles of Motivational Interviewing (MI) (Rollnick et al.
1999) and addresses the above-mentioned barriers to accurate dissemination of hereditary risk within families, with the aim to increase the efficacy of the intervention. We describe the design of the intervention, its fidelity (i.e., the degree of confidence that it can be delivered as intended) and its acceptability to counselees.
Discussion
We developed a brief intervention as an adjunct to usual care, to assist counselees in making an informed decision about disclosing hereditary risk information to relatives and to act on this. The intervention focuses on i) enhancing counselees’ knowledge of which relatives to disclose important genetic-related information to, ii) exactly what information should be disclosed, and ii) increasing their motivation and self-efficacy in informing their at-risk relatives. The intervention appeared to be widely accepted by participating counselees; both the content and practicality were highly rated.
The intervention is grounded in MI, a well-known clinical method with an encouraging evidence base for its efficacy (Miller and Rollnick
2009). The choice for MI as the guiding model for the intervention is based on the MI tenet that it is important to elicit the person’s own inherent arguments for relevant health behavior, rather than imposing someone else’s (Miller and Rollnick
2009). In line with this view, the psychosocial workers adopted a collaborative role and promoted a well-informed decision about which relative to inform about what, rather than to determine whom the counselee ‘should’ inform. The psychosocial worker has to facilitate the counselee’s ability to make a conscious decision about the communication of hereditary risk, with an awareness of what affects this decision and how relatives are subsequently informed. Thus, the goal of the intervention is to address the process of informing relatives. Although this process may result in more relatives being informed, this was not the primary goal of our intervention. Our approach to the dissemination of hereditary risk information within families via the counselee might be considered as ‘indirect’ or ‘passive’, as opposed to direct contact between the genetic counselor and relatives. Although the latter may result in more relatives being informed and seeking genetic counseling, a direct approach raises issues about counselees’ confidentiality, the relative’s right to privacy and the right not to know.
Counselees evaluated the additive counseling as not being confronting, long, or superfluous. On the other hand, over half of the counselees did not experience the counseling as clarifying and/or supportive. These might be the counselees who had already informed their relatives and who experienced no problems in this respect. However, although the intervention did not provide them with additional support, they seemed to appreciate the offer to discuss family communication, as indicated by the high rates for usefulness, pleasantness and for the way in which the psychosocial workers delivered the intervention. In sum, although not all counselees may have benefitted, the intervention did not appear to cause them any harm.
It is noteworthy that less than half of the eligible counselees accepted the invitation to participate in this study. Other Dutch psychosocial studies in the field of Clinical Genetics reported comparable response rates (Albada et al.
2012; Vos et al.
2011) suggesting that these generally healthy users of this medical service are less likely to contribute to research than populations of (chronically) ill patients. Nevertheless, the limited uptake suggests that, for the majority of counselees, additional counseling in discussing hereditary risk with relatives does not serve a high need for support.
Analysis of the audio-recorded consultations leads us to conclude that many elements of the intervention were delivered as intended. Thus, the MI approach seems feasible for experienced psychosocial workers, after appropriate training. Agenda setting and exploring which relatives were informed was carried out very systematically. Not all elements of the second phase were prevalent in the audio-taped consultations; however, this is in line with the manual which states that one or more of these elements should be used only if a counselee did not correctly inform their relatives. It is nevertheless noteworthy that, of the counselees who had not yet informed all relatives at-risk, 41 % reported that the psychosocial worker had done little to motivate them to inform their relatives. This supports the finding that psychosocial workers infrequently used the motivation-enhancing strategy to have the clients themselves list what is good about informing relatives, suggesting room for improvement in this respect. Also, the psychosocial workers rarely checked whether counselees’ had understood the information they had provided.
The training for psychosocial workers comprised two sessions, with ample opportunity for practice in between sessions and for obtaining individualized feedback based on audio-recorded consultations; this appears to have been beneficial. Initially, psychosocial workers were inclined to present information themselves concerning which relatives needed to be informed and what information should be provided, rather than to explore counselees’ knowledge on whom to inform about what. Increased exploration of counselees’ knowledge as a result of the second training session appears to have led to an increase in the detection of counselees’ who had not yet informed all at-risk relatives and, consequently, in more frequent additional or corrective information. This finding underscores the importance of a thorough exploration to obtain proper understanding of the counselees’ knowledge level.
Only in a minority of consultations was a second phase needed to correct/add information and build counselee’s motivation and self-efficacy to further inform relatives. Although there are many examples of problematic risk communication within families, our results tentatively suggest that many counselees succeed in disseminating risk information.
Practice Implications
Results of this feasibility study show that psychosocial workers who are required to assist in the process of communication of genetic information within families can acquire and apply the skills necessary for a theory-based approach such as MI. We also demonstrated that counselees who consent to a conversation about family communication, appreciate the psychosocial workers’ approach. Therefore, this intervention appears to be feasible for clinical practice. We emphasize, however, that in families with hereditary or familial cancer, we recommend that the subject of informing relatives be considered an integral part of the genetic counseling process and be addressed in the first counseling session (Menko et al.
2013). We do not yet know whether the intervention is effective, i.e., improves counselees’ ability to make a well-informed decision about whether or not to inform relatives about their possible heightened risk and preventive options, and to what extent they feel better equipped to do so. If found to be effective, a telephone intervention is more likely to be implemented than a face-to-face type of intervention.
Research Recommendations
Currently, we are conducting a randomized controlled trial, based on a new study sample, to test the efficacy of this additional telephone intervention in clinical practice (Geus de et al.
2014). We hypothesize that the intervention will improve counselees’ knowledge, motivation and self-efficacy to inform their at-risk relatives. In addition, we will investigate whether the intervention leads to more relatives being informed by the counselee. If efficacious, implementation of this relatively brief intervention may be warranted.
Acknowledgments
This study was financially supported by the Dutch Cancer Society (grant number UVA 2010-4658). The authors thank the research assistants Jane van der Vloodt, Anne Sijmons, and Laurine Knol for their work during the study, and also thank the psychosocial workers Alma Schiphorst, Annemiek Blom, Anja van Nunen, Tine Tijmstra, and Jo Jacobs for participating in the study.
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