GENETIC TESTINGVolume 11, Number 1, 2007© Mary Ann Liebert, Inc.DOI: 10.1089/gte.2006.9998

Factors Associated with an Individual’s Decision to Withdrawfrom Genetic Testing for Breast and Ovarian Cancer

Susceptibility: Implications for Counseling

BÉATRICE GODARD,1 ANNABELLE PRATTE, MARTINE DUMONT,3

ADÈLE SIMARD-LEBRUN,3 and JACQUES SIMARD3

ABSTRACT

Our study aimed to examine why individuals withdraw from genetic testing for breast and ovarian cancersusceptibility. We explored the characteristics of 334 individuals from high-risk breast and ovarian cancerfamilies who declined genetic testing for BRCA1/2 mutations, when, and why they did so. Individuals who de-clined genetic testing were older, and a greater proportion had never developed breast or ovarian cancer.Fifty one per cent (51.1%) of individuals withdrew after the first genetic counseling session. Most of thosewho declined were afraid of the psychological effects of genetic testing (36.3%). The next most-cited expla-nations concerned logistic problems such as a limited ability to travel, lack of time, personal issues, advancedage, or health problems (21.7%). The third category included individuals who did not see any advantage inbeing tested (14.5%). Insurability was a concern (5.9%), mainly for men. Surprisingly, confidentiality was nota frequently reported issue (1.3%). Sixty eight per cent (68%) of individuals belonging to a family in whichat least one individual has been tested withdrew after the presence of a deleterious BRCA1/2 mutation in arelative was disclosed, compared to 42% after the disclosure of a nonconclusive test result in at least one rel-ative. Concern about the psychological effects of the result was still one of the major reasons. Several factorsmay influence an individual’s decision to decline genetic testing; a greater understanding of these issues mayhelp health professionals to better meet the needs and concerns of individuals from high-risk families, thuspossibly improving their health outcomes.

45

INTRODUCTION

ADVANCES IN HUMAN GENETICS will play a crucial role inmedicine and public health by providing genetic informa-

tion for the prediction and prevention of disease (Khoury et al.2004). It has been estimated that by the age 60, 6 in 10 peoplewill develop a disease with a genetic component (Our Inheri-tance, Our Future 2003). Family history may be especially im-portant in helping to identify individuals at increased risk forcommon complex diseases, such as cancer, in whom effectiveinterventions might produce the greatest health benefit and inassessing the clinical relevance of testing for a genetic suscep-

tibility, thus refining risk prediction (Khoury et al. 2004). Testsfor BRCA1/2 mutations represent an early example of this par-adigm. Indeed, clinically significant or deleterious mutations inBRCA1 and BRCA2 are the most important known causes ofinherited susceptibility to breast and ovarian cancer (Ford et al.1998). A combined analysis using data from 22 studies indi-cated that the cumulative risks in BRCA1 carriers by the age of70 were 65% for breast cancer and 39% for ovarian cancer,whereas the corresponding estimates for BRCA2 carriers were45% and 11% (Antoniou et al. 2003). The identification of dele-terious mutations in these genes would encourage optimal sur-veillance and provide a rationale for personalized risk-reduc-

1Department of Preventive and Social Medicine, Bioethics Programs, University of Montreal, Montreal, Quebec, Canada.2Department of Human Genetics, McGill University, Montreal, Quebec, Canada.3Cancer Genomics Laboratory, Oncology and Molecular Endocrinology Research Center, CHUQ Research Center and Laval University, Que-

bec, Canada.

tion strategies and therapeutic approaches, which have been es-timated to add years of life for carriers and free noncarriers toconsider drastic surgical interventions (Armstrong et al. 2004;Eisinger et al. 2004; McIntosh et al. 2004; Nelson et al. 2005;U.S. Preventive Services Task Force 2005).

However, choosing whether or not to undergo testing forBRCA1/2 mutations is a complex and personal decision for anindividual and his/her family. (Armstrong et al. 2000; Keoghet al. 2004; McInerney-Leo et al. 2005; Schwartz et al. 2005).BRCA1/2 testing presents the potential for benefits and disad-vantages. It delivers crucial information that facilitates informeddecisions about individual and familial cancer optimal surveil-lance and risk-reduction interventions. If a carrier is identified,her survival could be improved with tailored early detection,risk-management, and prevention strategies. Genetic testing canreassure noncarriers and alleviate their anxiety (Schwartz et al.2005). On the other hand, genetic testing for breast and ovar-ian cancers can lead to adverse psychological consequences,such as anxiety and worrying about relatives (Armstrong et al.2000; Meiser et al. 2002; Thompson et al. 2002; Lodder et al.2003; McInerney-Leo et al. 2005). The perception is that itcould lead to insurance or employment discrimination. In non-carriers, it could create a false sense of security. In brief, asearch for mutations and the disclosure of results generate im-portant ethical issues related to the autonomy of individuals,nonmaleficence, and responsibility of health-care professionals(Julian-Reynier et al. 2000; Daly et al. 2001; Tercyak et al.2001, 2002; Hughes et al. 2002; Claes et al. 2003; Forrest etal. 2003; Hallowell et al. 2003).

Uptake for breast and ovarian cancer genetic testing has beenlargely documented (Ropka et al. 2006), whereas little is knownabout declining genetic testing for BRCA1/2 mutations. Indi-viduals are entitled to know or not know their genetic status.However, little is known about what motivates high-risk indi-viduals to withdraw from genetic testing for breast and ovariancancer predisposition. And while withdrawals are not limitedto the field of genetics, we can assume they are more frequentin predictive medicine, which can involve severe treatments andpsychosocial and familial implications. To date, most studieshave focused on patients’ refusal to participate in clinical can-cer trials at large (Primo et al. 2001) and breast cancer clinicaltrials (Kotwall et al. 1992). Very few studies have focused onfactors associated with an individual’s refusal to undergo or de-cision to withdraw from genetic testing for breast and ovariancancer. A recent British study examined the attributes, the lev-els of cancer worry, and the barriers to testing for a group ofrelatives of known BRCA1/2 carriers (315 participants; 34 testdecliners). The results from the group revealed that barriers totesting included apprehension about results, concerns abouttravel to the genetics clinic, and the need to take time off workor away from family and other obligations. Women were morelikely than men to worry about subsequently receiving lessscreening if found to be noncarriers. This study also found thatwomen who decline tests worry less about cancer than thewomen who choose to be tested (Foster et al. 2004).

A Dutch study explored self-reported motives for not un-dergoing genetic testing and factors related to this decision inwomen at risk to be a BRCA1/BRCA2 mutation carrier; theywere participants in a surveillance program for breast and ovar-ian cancers (85 tested women and 13 nontested). These women

learned about the possibility of genetic testing more than 1 yearbefore the study and did not volunteer for it (Lodder et al. 2003).Participants in a U.S. study of potential barriers to the wide-spread use of cancer genetic counselling services (37 partici-pants) revealed the following reasons for choosing not to pro-ceed with counseling: concern over health insurability for selfor family, cost; emotional effects, the inability to perceive ben-efits, and the time commitment (Geer et al. 2001). Another U.S.study reported that 8 years after establishing a familial cancergenetic program, only 18% of the 162 recommended familieshad proceeded with BRCA1/2 testing (Hartenbach et al. 2002).Heightened anxiety, potential discrimination, and the cost oftesting were cited as barriers. Another study sought to deter-mine the proportion of women undergoing BRCA1/BRCA2 test-ing and the factors associated with this decision among womenundergoing genetic counseling in the context of a clinical breastcancer risk assessment program in the United States (Armstronget al. 2000). The results reveal the 86 women who declined ge-netic testing were more likely to have important concerns aboutlife insurance and job discrimination than the 125 women whounderwent testing. Finally, a sixth study consisted of a prospec-tive investigation of psychosocial predictors of genetic coun-seling and testing among 76 African-American women whowere offered free BRCA-related services. The participants whodeclined genetic counseling “had significantly less knowledgeof breast cancer genetics than those who accepted both coun-seling and testing.” They also “reported greater anticipation ofnegative emotional responses to testing and more concern aboutstigmatization” (Thompson et al. 2002). Finally, undergoing ge-netic testing and learning one’s BRCA1/2 status may affect fam-ily relationships (McInerney-Leo et al. 2005, D’Agincourt-Can-ning 2006). In other respects, previous work has shown thatdifferent populations have different beliefs, different valuesand, for this reason, make different decisions regarding genetictesting. In countries without a public health-care system, thecost of BRCA1/2 testing may be a significant barrier to testing(Cho et al. 1999; Hartenbach et al. 2002).

Our study aimed to determine the context and the reasonswhy French-Canadian individuals coming from high-risk fam-ilies declined genetic testing for breast and ovarian cancer sus-ceptibility in the context of an integrated clinical research pro-gram (the Interdisciplinary Health Research International Teamon Breast Cancer Susceptibility, or BRCAs). The Interdiscipli-nary Health Research International Team on Breast Cancer sus-ceptibility (INHERIT BRCAs) is an international and interdis-ciplinary team working in the field of oncogenetics. This teamwas created in January, 2001, and is composed of 17 investi-gators: clinicians and scientists from seven hospitals and fiveCanadian universities as well as from the University of Cam-bridge, U.K., and from the International Agency for Researchon Cancer (IARC) in Lyon, France, which is part of the WorldHealth Organization. This team also includes some 15 re-searchers and clinicians (general practitioners, clinical geneti-cists, onco-surgeons, onco-gynecologists, hemato-oncologists)who cooperate closely with INHERIT BRCAs, as well as about40 professionals, genetic counselors, research assistants and nurses, biocomputer scientists, and psychologists. Thus, INHERIT BRCAs consists of a forum of experts in molecularand clinical genetics, genetic epidemiology, population health,genetic demography, psychosocial evaluation, health care and

GODARD ET AL.46

health services evaluation, ethics, and public law. Other mem-bers of INHERIT BRCAs involved in the clinical aspects ofprogram are listed in Appendix. More specifically, we exploredwhich individuals declined genetic testing, along with their cir-cumstances and motivations. We also aimed to identify betterthe barriers keeping at-risk individuals from genetic testing. Ourtranslational research started in 1996, giving individuals suffi-cient time to weigh the pros and cons and to reconsider theirrefusal.

MATERIALS AND METHODS

Study setting

The recruitment of high-risk French-Canadian breast and/orovarian families started in 1996 through a translational researchproject, which thereafter evolved into a large ongoing interdis-ciplinary research program designated INHERIT BRCAs (Godard and Simard 2003; Avard et al. 2006). A major com-ponent was to identify and characterize BRCA1 and BRCA2 mu-tations in the French-Canadian population and to determine therole of the demographic history in their origin and diffusion.This integrated clinical research program was composed of anetwork of referring clinicians across the province of Quebec.Approval was obtained from eight ethics committees corre-sponding to the different institutions participating in the re-search program. Potential index cases were referred by theirclinicians and were invited to sign a consent form authorizinga member of the research team to make contact. An initial phonecall was made to explain briefly the objectives and proceduresof the research program, to evaluate the likelihood of eligibil-ity of the individual, and to schedule an appointment for a firstgenetic counseling session when appropriate. Relatives were in-vited to participate by index cases and were contacted after ex-pressing their potential interest in the study. The genetic coun-seling session explained research protocol and hereditary breastand ovarian cancers and discussed the importance of familycommunication about these issues. The session also covereddifferent types of genetic test results, their potential psychoso-cial repercussions, and other key ethical, legal, and societal is-sues. Thereafter, the information concerning the family historywas collected and analyzed to evaluate the eligibility of familymembers for further steps in the study.

Individuals were recruited if their family met one of the fol-lowing strict criteria: (1) families with at least 4 individualswith breast and/or ovarian cancer diagnosed in first- or second-degree relatives; (2) families presenting 3 individuals diagnosedwith breast and/or ovarian cancer in first-degree relatives; and(3) families bearing a mutation already identified in the BRCA1or BRCA2 genes. A few additional families that did not meetthose strict criteria were recruited when the analysis of pedi-grees was suggestive of a genetic component (e.g., monozy-gotic twins diagnosed with breast cancer at an early age; 4 re-lated individuals with early-onset breast cancer, one case ofmale breast cancer plus a women diagnosed with early breastcancer). All participants had to be at least 18 years of age andmentally capable. In most instances, a pathology report con-firmed the diagnoses of breast and/or ovarian cancer. After ob-taining a signed informed consent from each participant, a blood

sample was drawn for mutation screening as previously de-scribed (Moisan et al. 2006; Simard et al. 2006).

Finally, participants consenting to be informed of their re-sults were seen by a clinician from one of the seven hospitalsdirectly involved in this research program and responsible forthe disclosure of genetic test results. The result disclosure ses-sion included a review of medical recommendations pertainingto each participant’s test result and a discussion of how the re-sult could affect the individual and his or her family.

The data used in these analyses came from 1,220 individu-als from 385 high-risk families; 886 participants consented tobe informed of their genetic test results and had received theirresults at the time the analyses of this study were performed(i.e., April, 2005). A total of 364 individuals decided to with-draw from the research program at different steps during theprotocol, either before or after the first genetic counseling ses-sion or after a blood sample was taken. The current analysesare based on those 334 individuals from whom 234 individu-als voluntarily explained their withdrawal.

Procedures

Our study is based on a pedigree analysis and a content anal-ysis of notes and comments received from the research subjectsor taken by grenetic counselors and genetic nurses. We exploredwhich individuals withdrew from genetic testing for breast andovarian cancer, when and why. Analysis was conducted usingstandard inductive methods: all notes and comments were readand categorized into codes representing emergent themes, ratherthan following predetermined codes. New codes were createduntil a point of redundancy was reached. The research team wasalso consulted to obtain more information when necessary.

Statistical analyses

The �2 test was used to test for significant association be-tween withdrawn status and some demographic variables. Thisstatistics was performed using SAS (SAS Institute Inc., Cary,NC).

RESULTS

Characteristics of individuals

Characteristics of the individuals who withdrew fromBRCA1/2 genetic testing were compared to participants whohave already received their test result (Table 1). Data were not available for several subjects, especially for those who withdrew before the first genetic counseling session. (The INHERIT-BRCAs project has never been designed for collect-ing data on individuals who declined genetic testing. Conse-quently, among individuals who voluntarily provided reasonsfor declining genetic testing (n � 234), demographic data weremissing for several subjects.) In regard to the subjects for whomdata were available, there was a significant difference in theage distribution of individuals in the two groups (p � 0.0001),with a greater proportion who declined being over 60 years ofage. There is also a greater proportion of individuals with nohistory of breast or ovarian cancer in the subset of individualswho withdrew (p � 0.0001). Although there was a significant

WITHDRAWAL FROM GENETIC TESTING 47

difference in the number of children between the two groups(p � 0.0154), there was no statistical difference between beingchildless and having at least one child (p � 0.7914). The sig-nificant difference observed in marital status between the twogroups was mainly due to separated individuals versus otherstatus (that is, versus married persons (p � 0.0005), versus sin-gle persons (p � 0.001), versus widowed persons (p � 0.003)).There was no statistical difference between the two groups inthe distribution of single individuals and widowed individualscompared to married individuals (p � 0.8367 and p � 0.9837,respectively).

Reasons for withdrawal from genetic testing for breastand ovarian cancer susceptibility

One hundred of the 334 individuals (30%) who declinedBRCA1/2 genetic testing did not say why. It is important to noteexplanations were provided voluntarily. In fact, according toethical guidelines, “asking for a justification would be perceived

as an infringement of the right of the individuals to withdrawor refuse to participate in a research project with complete free-dom” (Tri-Council 1998). It would go against the principle ofprivacy and would therefore be considered unethical. For theother 234 individuals (70%), one or more reasons were men-tioned. However, only the primary reason given by the indi-vidual was retained for this study to compare individuals’ re-sponses (Table 2).

Anxiety of living with the result was the most frequent rea-son for declining genetic testing (85 individuals or 36.3%).Some individuals were afraid a positive test result would cre-ate anxiety, whereas others stated that waiting for their resultwould make them too anxious and would have a significant psy-chological impact on them.

Fifty two respondents (22.2%) withdrew from genetic test-ing for a contextual reason, such as limited ability to travel,lack of time, particularly because of their work and their im-possibility to get off from work, their advanced age, health prob-lems, or other personal problems. Thirthy four individuals

GODARD ET AL.48

TABLE 1. CHARACTERISTICS OF THE INDIVIDUALS WHO WITHDREW FROM BRCA1/2 GENETIC TESTING

COMPARED TO PARTICIPANTS WHO HAVE ALREADY RECEIVED THEIR TEST RESULT

Individuals who Patients who receivedwithdrew their test result

Socio-demographic characteristics n % n % p

GenderMan 47 14.1 116 13.1 0.654Woman 287 85.9 770 86.9Total individuals 334 100.0 886 100.0

Agea

�40 44 16.9 164 18.540–59 113 43.3 510 57.6 �0.0001�60 104 39.8 212 23.9Total individuals 261 100.0 886 100.0

Marital statusb

Spouse 161 76.7 579 70.5Separated 7 3.3 106 12.9Single 23 11.0 71 8.6 0.0011Widowed 19 9.0 65 7.9Total individuals 210 100.0 821 100.0

Childrenc

0 39 16.7 160 18.71 49 21.0 122 14.3 0.01542 64 27.5 308 36.0�3 81 34.8 266 31.1Total individuals 233 100.0 856 100.0

Breast/ovariancancer statusDiagnosed 69 20.7 295 33.3Not diagnosed 265 79.3 591 66.7 �0.0001Total individuals 334 100.0 886 100.0

Mutation statusd

Family with mutation 103 54.2 421 47.5Family without mutation 87 45.8 465 52.5 0.094Total individuals 190 100.0 886 100.0

aNo data were available for 73 individuals who withdrew.bNo data were available for 124 individuals who withdrew and 65 individuals who have received their results.cNo data were available for 101 individuals who withdrew and 30 individuals who have received their results.dNo data were available for 144 individuals who withdrew.

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(14.5%) did not perceive any benefit from genetic testing or didnot feel concerned by the study’s topic; i.e., they did not con-sider their familial cancer history important. The explanationgiven was that individuals considered their medical follow upwas appropriate and they were childless and therefore expressedthat knowing their result would not change anything.

As indicated in Table 2, 25 individuals (10.6%) withdrewfor various reasons: either they did not want to continue dis-cussing cancer because they or a family member had had a can-cer case and family suffering was deemed too high, or they pre-ferred to be clinically tested or were either participating or hadalready participated in a similar research program. In other re-spects, 20 respondents (8.5%) were at first interested in partic-ipating in the BRCAs program but later declined because theirfamily refused or they alluded to difficulty contacting familymembers because of poor relationships, geographical distance,or difficulty in talking to them about cancer. Fourteen individ-uals (5.9%) feared the possible effects of genetic testing forbreast cancer on their own or their family members’ insurabil-ity, but surprisingly, confidentiality was not an issue. Only 3individuals (1.3%) expressed a fear of a confidentiality breachor worry that unauthorized persons could gain access to theirpersonal data.

Relation between the time and the reason of withdrawal

Almost half of the individuals (163/334 or 48.8%) withdrewfrom BRCA1/2 genetic testing before the first genetic counsel-ing session. A total of 125 individuals (37.4%) withdrew afterthe first genetic counseling session and 46 (13.8%) after a firstblood sample was drawn. As reported in Table 1, approximately80% individuals who decided to withdraw had never been di-agnosed with breast and/or ovarian cancer.

As for the relation between the time and the reason of with-drawal, Table 2 shows that among individuals who withdrewbefore the first genetic counseling session (98/234), 38 put for-ward logistic constraints. Nineteen individuals did not want toknow their genetic test result because of the expected psycho-logical impact. An equivalent number did not want to continuediscussing cancer or preferred to be tested in a clinical setting.Finally, 11 did not see any advantage to genetic testing and only3 evoked concerns about insurance.

Among individuals who withdrew from genetic testing afterthe first genetic counseling session (136/234), 66 feared the psy-chological impact of the genetic test result, whereas 23 indi-viduals did not see any personal advantages to genetic testing.Logistic constraints were reported in 14 cases. Among the 20individuals who declined due to relative’s refusal to participateor because of difficult contacts with other family members, 14withdrew after the first genetic counseling session. Eleven in-dividuals evoked concerns about the possible effects ofBRCA1/2 genetic testing for breast and ovarian cancer suscep-tibility on insurability.

Relation among the testing status of the family, the cancer status of the individuals, and the reason of withdrawal

More than 85% of withdrawals from families where nobodyhad been tested had never been diagnosed with cancer (82/95)

(Table 2). In this subset, the main reason was again the fear ofthe psychological impact of genetic test results, followed by lo-gistic problems and by the family refusal to undergo genetictesting. In a foreseeable way, the other important reason wasthe perception of any advantage of genetic testing.

When considering the subset of individuals belonging to fam-ilies in which at least one individual has been tested (n � 139),68% (55/80) withdrew after the disclosure of the presence of adeleterious mutation in a relative, compared to 42% (25/59)who withdrew after the disclosure of a nonconclusive test re-sult in at least one relative. Among the 55 decliners from aBRCA1/2-positive family forgoing genetic testing, 47 had neverbeen diagnosed with breast or ovarian cancer and 7 individu-als did not see any advantage of being tested. It is also inter-esting to observe that worries about insurability were the mainreason men declined genetic testing, especially after disclosureof the presence of a mutation in their family (6/14), whereasonly 3 women stated this reason and they all belonged to a non-conclusive BRCA1/2 family.

DISCUSSION

What stands out? What is noteworthy?

The current study is the largest reported study having fo-cused on factors associated with individuals’ refusal to undergoor decision to withdraw from genetic testing for breast and ovar-ian cancer (334 decliners and 886 participants). Although somelimitations, such as missing demographic data and reasonsgiven voluntarily, we examined reasons for withdrawal fromgenetic testing for breast and ovarian cancer, the relation be-tween the time and the reason of withdrawal, as well as the re-lation between the testing status of the family, the cancer sta-tus of the individuals, and the reason of withdrawal. Ourfindings indicate that several factors may influence the decisionto decline genetic testing, such as age, marital status, or neverhaving been diagnosed with breast or ovarian cancer. Anotherstudy showed just less than half of the participants who wereapproached for BRCA1/2 mutation testing chose to receive theirtest results and 13% were undecided (Keogh et al. 2004). Theyfound no evidence that the decision to learn of mutation statusdepended on age, gender, family history, or having been diag-nosed with breast cancer (Keogh et al. 2004). These results goagainst another study showing that people with children oftenfeel guilty about transmitting the mutation and want to give off-spring the chance to benefit from tailored prevention and riskmanagement strategies (Lodder et al. 2003). In fact, there is un-explained variation in uptake across studies and no strong, con-sistent predictors of uptake within studies (Ropka et al. 2006).

In our study, among decliners, a greater number of individ-uals decided not to undergo genetic testing for BRCA1/2 mu-tations after the first genetic counseling session. These indi-viduals might have exercised their right not to know, becausethey probably received sufficient information to make an in-formed decision. Genetic counseling has been shown to enhanceknowledge and to promote a more realistic appraisal of the ben-efits and risks of testing (Lerman et al. 1998; Briss et al. 2004).However, this right not to know may be associated with the fearof the psychological impact of genetic testing. In fact, anxiety

GODARD ET AL.50

was the most frequent reason for withdrawing from genetic test-ing, mainly after the first genetic counseling session in ourstudy. It is to be noted that participants in the INHERIT-BR-CAs project were not charged any fees; consequently, theirwithdrawal was not a question of money, but rather a psy-chosocial issue. These findings are consistent with anotherstudy according to which “rather than adhering to the notionthat genetic information is power or that it is better to knowthan to not know, those who declined perceived genetic infor-mation as something that would affect their lives adversely.Learning their carrier status did not offer the promise of control but posed a potential risk” (D’Agincourt-Canning2006:110).

Embedded in the fear of the psychological impact of genetictesting is the notion of genetic determinism—that those whohave the mutation are destined to get cancer (D’Agincourt-Can-ning 2006:110). Some previous research addressing the psy-chosocial aspects of genetic testing for inherited cancer predis-position has also found that many individuals fear negativepsychological consequences if they test positive (Lerman et al.1998; Geer et al. 2001). Lerman et al. (1998) reported that ad-verse psychological effects were seen primarily in individualswho declined genetic counseling and testing. Thus, accordingto this latter study, the decision to decline or defer genetic test-ing may promote, rather than alleviate, adverse psychologicaleffects.

Our findings indicate that advanced age, health problems,limited ability to travel, lack of time, or other personal prob-lems were the second category reasons for withdrawing fromgenetic testing and the first ones for declining testing before thefirst genetic counseling visit. These reasons might be an ex-pression of a lack of interest for being tested or a form of de-nial, since the researchers and clinicians of the team offered theindividuals a wide range of availabilities in various location.The right of an individual to choose whether or not to receiveinformation about their personal risk status is a fundamentalright. However, it must be an informed decision and the resultof personal soul-searching rather than a lack of education orsupport. Other studies showed that participants who did not per-ceive a benefit to the cancer genetic counseling service in factmisunderstood it (Cho et al. 1999; Geer et al. 2001; Harten-bach et al. 2002).

“No perceived benefit” was the third most frequently citedreason for declining genetic testing. Participants consideredthey had adequate medical follow-up or they were childless and,for these reasons, genetic testing would not change anything forthem. More that half of them came to this conclusion after afirst genetic counseling session. This reason is troubling be-cause it concerns high-risk individuals: did they understand lit-tle or did they deny their risk and so did not see advantages ofgenetic testing? In D’Agincourt-Canning’s study (2006),women who declined testing did not reject a genetic mutationas the underlying etiology of breast or ovarian cancer; rather,they did not think genetic testing offered them any furtherchoice. They felt constrained by the narrow range of medicaloptions available to them. It is true that a positive result canhave important implications for the individual and can lead toother difficult decisions concerning, for example, prophylacticmastectomy or oophorectomy. This could explain the attitudeof this subgroup in the present study.

The debate surrounding the use of genetic information by in-surers generates a lot of concern and unanswered questions. Thefear of health and life insurance discrimination is usually oneof the most important factors in the decision to not undergoBRCA1/2 testing (Geer et al. 2001; Armstrong et al. 2003; Pe-terson et al. 2002). Nevertheless, one of these studies found noevidence of life insurance discrimination among women whoparticipated in risk assessment and genetic testing through aclinical program (Armstrong et al. 2003). In the INHERIT-BRCAs program, the impact on insurability was part of the rea-son individuals expressed for declining genetic testing for breastand ovarian cancer, although it was not the most important one.In the INHERIT-BRCAs project, the age of the participants andthe fact that the majority was already insured made the needfor life insurance less relevant. However, it was the main rea-son men declined genetic testing.

Given the lack of documented instances of life and healthinsurers using genetic test results in underwriting decisions(Low et al. 1998), this issue needs to be taken into account. InCanada, no rule prevents insurers from asking for a genetic testresult. They may have access to the medical records in whicha genetic test result could be recorded under certain circum-stances (Godard and Simard 2003). The position of the Cana-dian Life and Health Insurance Associate is that “ . . . insurerswould not require an applicant for insurance to undergo genetictesting. However, if genetic testing has been done and the in-formation is available to the applicant for insurance and/or theapplicant’s physician, the insurer would request access to thatinformation just as it would for other aspects of the applicant’shealth history” (Knoppers et al. 2004). Finally, confidentialityis closely related to insurability. A breach of confidentiality re-lates to the possibility of discrimination. Nevertheless, very fewindividuals were afraid genetic information could be sharedwith unauthorized persons. Do these results mean that for themajority of these decliners genetic information doesn’t differfrom other medical information and therefore warrants the sameprotection?

What can be done?

Our study shows that the fear of the psychological impact ofknowing one’s result was the most frequent reason given by in-dividuals for declining genetic testing. Although a greater num-ber of withdrawals occurred after the first genetic counselingsession, this result emphasizes the importance of heighteningsome elements of the genetic counseling interaction, namelyhelping at-risk individuals cope with vulnerability, optimizefamily interaction, and improve health behaviors (Patenaude etal. 2002; McInerney-Leo et al. 2005). Counselors can play acrucial role in suggesting a psychological as well as a medicalfollow up. It has been shown that assessing and possibly mod-ifying people’s appraisal of their condition and of its impact ontheir family (that is, the threat of the genetic test) may help toreduce subsequent anxiety (Michie et al. 2002; McInerney-Leoet al. 2005).

Our results acknowledge the need to understand the personalexperience of individuals and how genetic information is as-similated into their lives. Jointly eliciting a patient’s preferencesmay be useful to determine the level of counseling servicesneeded (Audrain et al. 1998). Support groups may also have an

WITHDRAWAL FROM GENETIC TESTING 51

effect on psychosocial functioning and health-related quality oflife (Di Prospero et al. 2001; Till, 2003). Our findings are con-sistent with those of D’Agincourt-Canning (2006:112) that “de-cision making about genetic testing involves an integration ofdifferent elements of self. These aspects of self refer to the man-ner in which participants view genetic testing when thinkingabout their own physical health and their family’s well-being.( . . . ) These aspects of self are not distinct entities but tightlyintegrated. Furthermore, they are not static. Expressions of selfand, accordingly, the choices people make might shift andchange as circumstances within the family, within relationshipswith others, or within one’s life change”.

In other respects, we may question individuals’ denial. De-nial can be viewed as a useful defense mechanism, but also aspotentially capable of interfering with treatment (Ross et al.1992). Dialogue between health-care professionals and at-riskindividuals should begin early to foster preventive steps, andcommunication and education must continue throughout the in-dividual’s life. It may be recommended that the research teamor the genetic counselor should communicate with the referringphysician about such a patient’s perception and that they re-view with the latter the reasons why the evaluation might pro-vide useful information and support. A free and informed de-cision is crucial in the context of genetic testing, but cannot beachieved if there is a lack of information and education.

More broadly, health professionals (clinicians, counselors,nurses), as a whole, can play a crucial role in responding to ed-ucational needs and in raising public awareness. Informationcampaigns will always remain beneficial for at-risk individu-als, in particular for those who put forward a lack of time andavailability or for those who do not perceive benefits in genetictesting. A lot of educational print materials have been devel-oped to provide effective preliminary education about BRCA1/2mutation testing and they may serve as a model (Schwartz etal. 2001). These information tools could be distributed byhealth-care professionals to patients and by the latter to theirrelatives. These kits would be useful for translating complexscientific concepts inherent to genetics and inheritance into amore readily understandable format, as well as for explainingthe multistep process of genetic testing. Information kits couldalso be useful for family physicians who could address theirpatient’s reluctance and fears, although it may be time con-suming. Furthermore, some physicians may have limitedknowledge about genetic testing for BRCA1/2 mutations tocounsel patients fully about the possible outcomes. In that case,education programs developed for health professionals in re-sponse to the information and counseling needs of family mem-bers of breast cancer patients may serve as reference (Halver-son et al. 2000; Watson et al. 2001).

In conclusion, our findings suggest some key elements playa role in the decision-making process for declining genetic test-ing for breast and ovarian cancer. Considering that differentmanagement options exist for women at increased genetic risk,factors associated with withdrawal from genetic testing forbreast and ovarian cancer are important to know. A better un-derstanding of the reasons preventing at-risk individuals fromknowing their genetic test result may allow health profession-als to better account for their needs and concerns, and to bet-ter identify information that is relevant to individuals. Forinstance, knowing that many women decline genetic testing

because it would cause them increased anxiety suggests thathealth professionals should play attention to whether psycho-logical counseling can help the person to live with their deci-sion. The patient’s beliefs, knowledge, attitudes, and expecta-tions should be taken into account during this process (Miesfeldtet al. 2000). This also implies that health professionals shouldstrongly encourage these high-risk individuals to opt for fre-quent surveillance and personalized risk-reduction strategies,thus possibly improving their health outcomes.

ACKNOWLEDGMENTS

The authors are indebted to the participants for their gen-erosity. We would also like to thanks Nathalie Bolduc, ClaireBrousseau, Dr Sylvie Délos, Marie-Andrée Lajoie, PascaleLéger, Hélène Malouin, Josée Rhéaume, Andrée McMillan, andTina Babineau for genetic counseling and clinical data man-agement and Valérie Gaborieau for statistical analysis. Wewould like to thank clinicians for participant referral, especiallyDrs. Paul Bessette, Peter Bridge, Jocelyne Chiquette, RachelLaframboise, Jean Lépine, Bernard Lespérance, Marie Plante,and Patricia Voyer, who referred more than five high-risk fam-ilies to this reseach study. We also appreciated advice receivedfrom ethics committees. This work was supported by the Cana-dian Institutes of Health Research (CIHR) for the INHERITBRCAs research program. Jacques Simard is Chairholder of theCanada Research Chair in Oncogenetics. Béatrice Godard is re-search scholar from the Fonds de la Recherche en Santé duQuébec (FRSQ).

REFERENCES

Antoniou A, Pharoah PD, Narod S, Risch HA, Eyfjord JE, Hopper JL,Loman N, Olsson H, Johannsson O, Borg A, Pasini B, Radice P,Manoukian S, Eccles DM, Tang N, Olah E, Anton-Culver H, WarnerE, Lubinski J, Gronwald J, Gorski B, Tulinius H, Thorlacius, S,Eerola H, Nevanlinna H, Syrjakoski K, Kallioniemi OP, ThompsonD, Evans C, Peto J, Lalloo F, Evans DG, Easton DF (2003) Aver-age risks of breast and ovarian cancer associated with BRCA1 orBRCA2 mutations in case series unselected for families history: acombined analysis of 22 studies, Am J Hum Genet 72:1117–1130.

Armstrong K, Calzone K, Stopper J, Fitzgerald G, Coyne J, Weber B(2000) Factors associated with decisions about clinical BRCA1/2testing. Cancer Epidemiol. Biomarkers Prev 9:1251–1254.

Armstrong K, Weber B, Fitzgerald G, Hershey JC, Pauly MV, LemaireJ, Subramanian K, Asch DA (2003) Life insurance and breast can-cer risk assessment: adverse selection, genetic testing decisions, anddiscrimination. Am J Med Genet 120A:359–364.

Armstrong K, Schwartz S, Randall T, Rubin SC, Weber B (2004) Hor-mone replacement therapy and life expectancy after prophylacticoophorectomy in women with BRCA1/2 Mutations: a decision anal-ysis. J Clin Oncol 22:1045–1054.

Audrain J, Rimer B, Cella D, Garber J, Peshkin BN, Ellis J, Schild-kraut J, Stefanek M, Vogel V, Lerman C (1998) Genetic counselingand testing for breast-ovarian cancer susceptibility: what do womenwant? J Clin Oncol 16:133–138.

Avard D, Bridge P, Bucci LM, Chiquette J, Dorval M, Durocher F,Easton D, Godard B, Goldgar D, Knoppers BM, Laframboise R, Les-perance B, Plante M, Tavtigian SV, Vezina H, Wilson B, INHERITBRCAs, Simard J (2006) Partnering in oncogenetic research—the

GODARD ET AL.52

INHERIT BRCAs experience: opportunities and challenges. FamCancer 5:3–13.

Briss P, Rimer B, Reilley B, Coates RC, Lee NC, Mullen P, Corso P,Hutchinson AB, Hiatt R, Kerner J, George P, White C, Gandhi N,Saraiya M, Breslow R, Isham G, Teutsch SM, Hinman AR, LawrenceR, Task Force on Community Preventive Services (2004) Promotinginformed decisions about cancer screening in communities andhealthcare systems. Am J Prev Med 26:67–80.

Cho MK, Sankar P, Wolpe PR, et al (1999) Commercialization ofBRCA 1&2 testing: practitioner awareness and use of a new genetictest. Am J Med Genet 83:157–163.

Claes E, Evers-Kiebooms G, Boogaerts A, Decruyenaere M, DenayerL, Legius E (2003) Communication with close and distant relativesin the context of genetic testing for hereditary breast and ovarian can-cer in cancer patients. Am J Med Genet 116A:11–19.

D’Agincourt-Canning L (2006) Genetic Testing for Hereditary Breastand Ovarian Cancer: Responsibility and Choice. Qualitat Health Res16:97–118.

Daly MB, Barsevick A, Miller SM, Buckman R, Costalas J, Mont-gomery S, Bingler R (2001) Communicating genetic test results tothe family: a six-step, skills-building strategy. Fam Commun Health24:13–26.

Di Prospero LS, Seminsky M, Honeyford J, Doan B, Franssen E,Meschino W, Chart P, Warner E (2001) Psychosocial issues follow-ing a positive result of genetic testing for BRCA1 and BRCA2 mu-tations: findings from a focus group and a needs-assessment survey.CMAJ 164:1005–1009.

Eisinger F, Bressac B, Castaigne D, Cottu PH, Lansac J, LeFranc JP,Lesur A, Nogues C, Pierret J, Puy-Pernias S, Sobol H, Tardivon A,Tristant H, Villet R (2004) Identification et prise en charge desprédispositions héréditaires aux cancers du sein et de l’ovaire. BullCancer 91:219–237.

Ford D, Easton DF, Stratton M, Narod S, Goldgar D, Devilee P, BishopDT, et al (1998) Genetic heterogeneity and penetrance analysis ofthe BRCA1 and BRCA2 genes in breast cancer families. Am J HumGenet 62:676–689.

Forrest K, Simpson SA, Wilson BJ, Van Teijlinger ER, McKee L,Haites N, Matthews E (2003) To tell or not to tell: barriers and fa-cilitators in family communication about genetic risk. Clin Genet64:317–326.

Foster C, Evans DGR, Eeles R, Eccles D, Ashley S, Brooks L, Cole T,Cook J, Davidson R, Gregory H, Mackay J, Morrison PJ, Watson M(2004) Non-uptake of predictive genetic testing for BRCA1/2 amongrelatives of known carriers: Attributes, cancer worry, and barriers totesting in a multicenter clinical cohort. Genet Test 8:23–29.

Geer KP, Ropka ME, Cohn WF, Jones SM, Miesfeldt S (2001) Fac-tors influencing patients’ decisions to decline cancer genetic coun-seling services. J Genet Couns 10:25–40.

Godard B, Simard J (2003) Les enjeux éthiques de l’identification d’unesusceptibilité génétique au cancer de sein dans un contexte derecherche clinique intégrée. In: Hervé C, Knoppers BM, MolinariPA. Les pratiques de recherche biomédicale visitées par labioéthique. Éditions Dalloz, Paris.

Hallowell N, Foster C, Eeles R, Ardens-Jones A, Murday V, Wat-son M (2003) Balancing autonomy and responsibility: the ethicsof generating and disclosing genetic information. J Med Ethics29:74–83.

Halverson PK, Mays GP, Rimer BK, Lerman C, Audrain J, Kaluzny A(2000) Adoption of a health education intervention for family mem-bers of breast cancer patients. Am J Prev Med 18:189–198.

Hartenbach EM, Becker JM, Grosen EA, Bailey HH, Petereit DG, Lax-ova R, Schink JC (2002) Progress of a comprehensive familial can-cer genetic counseling program in the era of BRCA1 and BRCA2.Genet Test 6:75–78.

Hughes C, Lerman C, Schwartz M, Peshkin BN, Wenzel L, Narod S,Corio C, Tercyak KP, Hanna D, Isaacs C, Main D (2002) All in the

family: evaluation of the process and content of sisters’ communi-cation about BRCA1 and BRCA2 genetic test results. Am J MedGenet 107:143–150.

Julian-Reynier C, Eisinger F, Chabal F, Lasset C, Noguès C, Stoppa-Lyonnet D, Vennin P, Sobol H (2000) Disclosure to the family ofbreast/ovarian cancer genetic results: Patient’s willingness and asso-ciated factors. Am J Med Genet 94:13–18.

Keogh LA, Southey MC, Maskiell J, Young MA, Gaff CL, Kirk J,Tucker KM, Rosenthal D, McCredie MR, Giles GG, Hopper JL(2004) Uptake of offer to receive genetic information about BRCA1and BRCA2 mutations in an Australian population-based study. Can-cer Epidemiol Biomarkers Prev 13:2258–2263.

Khoury M, Little J, Burke W (eds) (2004) Human genome epidemiol-ogy: A scientific foundation for using genetic information to improvehealth and prevent disease. Oxford University Press, London.

Knoppers BM, Lemmens T, Godard B, Joly Y, Avard D, Clark T,Hamet P, Hoy M, Lanctôt S, Lowden S, Martin H, Maugard C, Mil-lette Y, Simard J, Vachon M-H, Zinatelli F (2004) Genetics and lifeinsurance in Canada: Points to consider. CMAJ 170:1–3.

Kotwall CA, Mahoney LJ, Myers RE, Decoste L (1992) Reasons fornon-entry in randomized clinical trials for breast cancer: a single in-stitutional study. J Surg Oncol 50:125–129.

Lerman C, Hughes C, Lemon SJ, Main D, Snyder C, Durham C, NarodS, Lynch HT (1998) What you don’t know can hurt you: Adversepsychologic effects in members of BRCA1-linked and BRCA2-linked families who decline genetic testing. J Clin Oncol16:1650–1654.

Lodder L, Frets PG, Trijsburg RW, Klijn JGM, Seynaeve C, TilanusMMA, Bartels CCM, Meijers-Heijboer EJ, Verhoog LC, NiermeijerMF (2003) Attitudes and distress levels in women at risk to carry aBRCA1/BRCA2 gene mutation who decline genetic testing. Am JMed Genet 119A:266–272.

Low L, King S, Wilkie T (1998) Genetic discrimination in life in-surance: empirical evidence from a cross sectional survey of ge-netic support groups in the United Kingdom. Br Med J 317:1632–1635.

McInerney-Leo A, Biesecker BB, Hadley DW, Kase RG, GiambarresiTR, Johnson E, Lerman C, Struewing JP (2005) BRCA1/2 testing inhereditary breast and ovarian cancer families II: Impact on relation-ships. Am J Med Genet 133A:165–169.

McIntosh A, Shaw C, Evans G, et al (2004) Clinical guidelines andevidence review for the classification and care for primary womenat risk of familial breast cancer. National Collaborating Centre forPrimary Care/University of Sheffield, London, http://www.nice.org.uk.

Meiser B, Butow P, Friedlander M, Barratt A, Schnieden V, WatsonM, Brown J, Tucker K (2002) Psychological impact of genetic test-ing in women from high-risk breast cancer families. Eur J Cancer38:2025–2031.

Michie S, French DP, Marteau TM (2002) Predictive genetic testing:mediators and moderators of anxiety. Int J Behav Med 9:309–321.

Miesfeldt S, Jones SM, Cohn WF (2000) Informed consent for BRCA1and BRCA2 testing: What clinicians should know about the processand content. J Am Med Womens Assoc 55:275–279.

Moisan AM, Fortin J, Moisan MD, Samson C, Bessette P, ChiquetteJ, Laframboise R, Lepine J, Lesperance B, Pichette R, Plante M,Provencher L, Voyer P, Goldgar D, Bridge P, Simard J (2006) No evidence of BRCA1/2 genomic rearrangements in high-riskFrench-Canadian breast/ovarian cancer families. Genet Test 10:104–115.

Nelson HD, Hoyt Huffman L, Rongwei F, Harris EL (2005) Geneticrisk assessment and BRCA mutation testing for breast and ovariancancer susceptibility: systematic evidence review for the U.S. pre-ventive services task force. Ann Int Med 143:362–379.

Our Inheritance our Future: releasing the potential of genetics in theNHS (2003). White paper. Department of Health, United Kingdom.

WITHDRAWAL FROM GENETIC TESTING 53

Patenaude AF, Guttmacher AE, Collins FS (2002) Genetic testing and psychology. New roles, new responsibilities. Am Psychol 57:271–282.

Peterson EA, Milliron KJ, Lewis KE, Goold SD, Merajver SD (2002)Health insurance and discrimination concerns and BRCA1/2 testingin a clinic population. Cancer Epidemiol Biomarkers Prev 11:79–87.

Primo LN, Higdon R, Lim N, Kwan K, Tanaka M, Lau DHM, Wun T,Welborn J, Meyers FJ, Christensen S, O’Donnell R, Richman C,Scudder SA, Tuscano J, Gandara DR, Lam KS (2001) Prospectiveevaluation of cancer clinical trial accrual patterns: identifying po-tential barriers to enrolment. J Clin Oncol 19:1728–1733.

Ropka ME, Wenzel J, Phillips EK, Siadaty M, Philbrick JT (2006) Up-take rates for breast cancer genetic testing: a systematic review. Can-cer Epidemiol Biomarkers 15:840–855.

Ross DM, Peteet JR, Medeiros C, Waksh-Burke K, Rieker P (1992)Difference between nurses’ and physicians’ approach to denial in on-cology. Cancer Nurs 15:422–428.

Schwartz MD, Benkendorf J, Lerman C, Isaacs C, Ryan-Robertson A,Johnson L (2001) Impact of educational print materials on knowl-edge, attitudes, and interest in BRCA1/BRCA2. Cancer 92:932–940.

Schwartz MD, Lerman C, Brogan B, Peshkin BN, Isaacs C, DemarcoT, Halbert CH, Pennanen M, Finch C (2005) Utilization ofBRCA1/BRCA2 mutation testing in newly diagnosed breast cancerpatients. Cancer Epidemiol Biomarkers Prev 14:1003–1007.

Simard J, Dumont M, Moisan AM, Gaborieau V, Vézina H, DurocherF, Chiquette J, Plante M, et al (2006) Evaluation of BRCA1 andBRCA2 mutation prevalence, risk prediction models and a multi-steptesting approach in French-Canadian high-risk breast and/or ovariancancer families. J Med Genet 11:1–33.

Tercyak KP, Hughes C, Main D, Snyder C, Lynch JF, Lynch HT, Ler-man C (2001) Parental communication of BRCA1/2 genetic test re-sults to children. Patient Educ Couns 42:213–224.

Tercyak KP, Peshkin BN, Demarco TA, Brogan BM, Lerman C (2002)Parent-child factors and their effect on communicating BRCA 1/2test results to children. Patient Educ Couns 47:145–153.

Thompson HS, Valdimarsdottir HB, Duteau-Buck C, Guevarra J, Bovb-jerg DH, Richmond-Avellaneda C, Amarel D, Godfrey D, Brown K,Offit K (2002) Psychosocial predictors of BRCA counseling and test-ing decisions among urban African-American women. Cancer Epi-demiol Biomakers Prev 11:1579–1585.

Till, JE (2003) Evaluation of support groups for women with breastcancer: importance of the navigator role. Health Qual Life Outcomes1:16.

Tri-Council (1998) Policy statement: Ethical conduct for research in-volving humans. Ottawa, Canada.

U.S. Preventive Services Task Force (2005) Genetic risk assessmentand BRCA mutation testing for breast and ovarian cancer suscepti-bility: recommendation statement. Ann Int Med 143:355–361.

Watson E, Clements A, Yudkin P, Rose P, Bukach C, Mackay J (2001)Evaluation of the impact of two educational interventions on GP man-agement of familial breast/ovarian cancer cases: a cluster randomisedcontrolled trial. Br J Gen Pract 51:817–821.

Address reprint requests to:Dr. Béatrice Godard

Department of Preventive and Social MedicineBioethics Programs

University of MontrealC.P. 6128, succ. Centre-ville,

Montreal, Quebec, Canada H3C 3J7

E-mail: beatrice.godard@umontreal.ca

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