ORIGINAL RESEARCH

Experiences and Decisions that Motivate Womenat Increased Risk of Breast Cancer to Participatein an Experimental Screening Program

Michelle Proulx & Marie-Dominique Beaulieu &

Christine Loignon & Marie-Hélène Mayrand &

Christine Maugard & Nathalie Bellavance &

Diane Provencher

Received: 6 March 2008 /Accepted: 29 October 2008 /Published online: 14 February 2009# National Society of Genetic Counselors, Inc. 2009

Abstract Although the discovery of mutations on BRCA1and BRCA2 genes associated with high breast cancer riskhas given rise to screening and surveillance initiatives, thereis little documentation on why high-risk women choose toenter screening programs. The objective of this qualitativestudy was to develop a detailed understanding of theexperiences and decisions that motivate women with in-creased risk of hereditary breast cancer to participate in themulticentered Quebec experimental breast screening program.Our study involved 21 participants who were either BRCAcarriers or at risk and untested. These women were inter-

viewed while participating in the screening program. Ourstudy demonstrates that intensive screening programs mayprovide valuable reassurance for women with increasedfamilial risk of hereditary breast cancer, who count on earlydetection and rapid response from professionals if and when aproblem arises. Health professionals must take these andothers concerns into account to ensure their interventions aremost consistent with women’s needs.

Keywords Hereditary breast cancer . BRCA1 or BRCA2diagnostic testing . Surveillance monitoring

J Genet Counsel (2009) 18:160–172DOI 10.1007/s10897-008-9202-z

M. Proulx (*)Faculty of Pharmacy,Université de Montréal,7426 St-André Street, # 2020,Montreal, Quebec, Canada H2R 2P9e-mail: proulxmichelle@videotron.ca

M.-D. BeaulieuDepartment of Family Medicine,CHUM Research Centre,Université de Montreal,Hôpital Notre-Dame,L.-C. Simard Pavilion,1560 Sherbrooke Street East, 8th floor,Montreal, Quebec, Canada H2L 4M1e-mail: marie-dominique.beaulieu@umontreal.ca

C. LoignonDivision of Oral Health and Society,Faculty of Dentistry,McGill University,3640 University Street (Strathcona building),Montreal, Quebec, Canada H3A 2B2e-mail: christine.loignon@mcgill.ca

M.-H. MayrandDepartment of Social and Preventive Medicine,Faculty of Medicine, Université de Montréal,1420 Mont-Royal Blvd.,Montreal, Quebec, Canada H2V 4P3e-mail: mhmayrand@videotron.ca

C. Maugard :N. BellavanceDepartment of Medicine, CHUM Research Centre,Université de Montréal,Hôtel-Dieu CHUM, Room Y-4621, J.A. de Sève Pavilion,2099 Alexandre de Sève Street,Montreal, Quebec, Canada H2L 2W5

C. Maugarde-mail: christinemaugard@sympatico.ca

N. Bellavancee-mail: nathalie.bellavance.chum@ssss.gouv.qc.ca

D. ProvencherDivision of Gynecology-Oncology, Faculty of Medicine,Université de Montréal,CHUM - Hôpital Notre-Dame, 1560 Sherbrooke Street East,Montreal, Québec, Canada H2L 4M1e-mail: diane.provencher.chum@ssss.gouv.qc.ca

Introduction

The discovery of mutations on the BRCA1 and BRCA2genes associated with high risk of breast and ovariancancers, and the proven capacity of certain surgicalinterventions (prophylactic mastectomy or salpingo-oophorectomy) to reduce the incidence of these cancers,have led to the development of genetic diagnostic programsfor women at high familial risk. Nevertheless, studiessuggest that women may decline to undergo preventivesurgery for a variety of reasons, such as the psychosocialsignificance of the breast in Western cultures (aesthetics,personal identity, intimacy), or issues related to accessto care and physician preferences (Metcalfe et al. 2007,Sauven et al. 2004). Current studies on screening docu-ment the set of women who undergo modalities, particu-larly mammography, and then neglect follow-up; thesestudies fail, however, to explain convincingly the reasonsbehind these women’s decisions (Lerman et al. 2000,Wainberg and Husted 2004).

Very little conclusive data are in fact available onwhat motivates women at high risk of breast and ovariancancer to decide to participate in ongoing screeningprograms. In cases of high risk, we might expect a highrate of participation. Taking this premise for granted, fewresearchers have looked at how such programs have beenreceived. Yet some studies have suggested that up to30% of women confirmed as having a high risk offamilial cancer abandon the screening programs to whichthey have been referred (Isaacs et al. 2002). Even moreworrisome, these women generally have not availedthemselves of other options (Isaacs et al. 2002, Lermanet al. 2000, Metcalfe et al. 2000, Wainberg and Husted2004).

This paper presents the results of a qualitative, exploratorystudy carried out within a large experimental breast screeningprogram (Provencher et al. 2004). The objective of thequalitative component was to develop a detailed understand-ing of the subjective experiences and decisions that motivatewomen to participate in the program.

Background

At a theoretical level, this exploratory study is based mainlyon the work of Rees et al. (2001), who developed aframework that is useful in linking three dimensions centralto the problem under study: the subjective experience ofillness, the response to risk information, and the perceptionof risk. Rees et al. (2001) conclude—based on studiesshowing positive associations between perceived suscepti-bility to breast and ovarian cancers and adherence to

screening strategies—that perception of risk is an interme-diate but strong component of decisions for action. Theyconsider that, among women at high risk, perceptionsand beliefs influence thoughts, emotions and decisions.While they recognize that these perceptions and beliefsnot only vary according to culture, but evolve and aretransformed over time (Gurmankin et al. 2005, Payne1990), the researchers suggest that the subjective experi-ence of illness is a key determinant. Thus it is mainlythrough their direct experience of breast or ovarian cancerin family members who have survived or succumbed toillness that high-risk women represent the illness forthemselves and behave accordingly (Richards et al. 1995,Wellisch et al. 1992, 1996). This experience influencestheir responses to risk information that can includeemotional or affective reactions (feelings of powerlessnessand fear) and engaging in screening and surveillanceactivities. Rees et al. (2001) suggest it is not so much theobjective details of the information that spur action, butrather the meaning given to this information (Senior et al.2000).

While cognitive bias may intervene (tendency to over—or underestimate one’s own risk), they conclude thatperception of risk is also a product of other factors, suchas number of persons in the family who have had cancer,physical resemblance to a stricken family member, proximityin age to another family member at the time of her diagnosis,exposure to media reports, and interactions with the health-care system, friends, and work colleagues (Rees et al. 2001,Vernon et al. 1993). In addition, the researchers note thathigh-risk women must make difficult decisions about theirpersonal risk in relation to a whole range of screening andpreventive measures (e.g. breast self-examination, mam-mography, preventive surgeries) without clear indicationsof the best course to follow. This aspect is also mentionedby Eaden et al. (2001). Rees et al. (2001) conclude, as doWroe et al. (1998), that women’s decisions are likely to bestrongly determined by the subjective experience of illnessand their understanding of the pros and cons of the proposedpreventive and screening measures.

The framework proposed by Rees et al. (2001) presentscore dimensions that have been causally linked withdecision-making in women with increased familial riskof hereditary breast cancer. The objective of the presentstudy was to explore these and other emergent dimensionsusing an interpretative approach, to see how they fit intowomen’s experiences and decisions. This approachallowed us to uncover the meanings ascribed to experienceand to explore the dynamic and complex nature of thisexperience and its expression in the decisions of theprogram participants (Denzin and Lincoln 1994, Pourtoisand Desmet 1989).

Experiences and Decisions that Motivate Women 161

Methods

The Multicentered Experimental Screening Program

The multicentered Quebec experimental breast screeningprogram operates at the Hôtel-Dieu Clinic for HereditaryCancers, in the Genic Medicine service of the CHUM(Centre hospitalier de l’Université de Montréal). Theprogram is the subject of a broad evaluative study whichis designed to measure the sensitivity of different imagingmethods among women at high risk of hereditary breastcancer (Provencher et al. 2004). Women admitted to theprogram must either be carriers of BRCA1 or BRCA2mutations or have a higher than 30% risk of carrying amutation without having agreed to be tested or to knowthe results of their tests. The risk is established usingBRCAPRO software (Berry et al. 2002, Provencher et al.2004). Participants are followed by the clinic’s care teamusing five screening methods: clinical examination of thebreasts, mammography, ultrasound, scintimammographyand magnetic resonance imaging (MRI). Women are taughtbreast-self examination at their first visit.

The first round begins with the complete series of testsbeing carried out within four weeks. Six months later,participants undergo a clinical exam and an ultrasound. Inaccordance with standard clinic follow-up practice, womenwith positive (abnormal) test results are referred for furtherinvestigation. Over the course of their participation, certainpreventive surgical options (prophylactic bilateral salpingo-oophorectomy or mastectomy) may be proposed to them bythe team.

Sampling Strategy, Process and Characteristics

We wanted the sample to represent the experimental screeningprogram’s population of women. To this end, we usedpurposive sampling because it provided for continuousselection of information-rich cases judged to be representativeof all the program participants, i.e., women who were eitherBRCA carriers or at risk and untested (Deslauriers 1991).

The sample was constructed progressively by means of aconcurrent process of data collection using semi-directedinterviews and analysis, up to the point of conceptualsaturation, where the addition of new data no longercontributed to a better understanding of the phenomenonunder study (Deslauriers 1991). Recruitment was done bythe coordinator of the multicentered screening program,who identified eligible participants based on the abovecharacteristics and according to the chronological order inwhich their names appeared on a computerized list from theevaluative study. Of the 37 women on the list (from the 187participants in the experimental program at the time of

recruitment), 18 agreed to an in-depth interview—seven whohad completed a first round of screening procedures andeleven who had completed more than one round. This allowedus to capture the evolution of the experience. Among thewomen who declined to participate (19 women), some told usthey did not want to be interviewed again. Others were toobusy; some explicitly or implicitly refused to meet with us forfear of reviving negative emotions or painful memories. Stillothers were unavailable because of vacations or business trips.

Upon observing that some women (a total of 25 at thetime of recruitment), regardless of health status, had chosento abandon the program, we decided to recruit some ofthese to explore their decision. Three women were theninterviewed by telephone, to facilitate their recruitment.These women had moved or had quit the program becauseof access difficulties or schedule conflicts; at the time oftheir interviews, they were being followed by a familydoctor. While these telephone interviews were brief (about15 min), they covered the same content as the face-to-faceinterviews with women in the program and were retained inthe final sample, bringing the total number of participantsto 21. We decided to stay at this number because of thelimitations (e.g. on expression of affect) imposed bytelephone interviews (Kirsch and Brandt 2002).

The participants were relatively equally distributedamong three health statuses: seven (of whom only onewas a carrier) had previously had breast cancer and decidedto be tested; eight others were unaffected with cancer, haddecided to be tested and were carriers; and six othersunaffected with cancer declined to be tested. Among thethree participants who withdrew from the study, two had apersonal history of cancer and were not carriers, and theother had no prior history of cancer but was a carrier.

Of the 21 participants, seven women had also receivedfalse positive results from at least one of the screening tests;three had already received a diagnosis of breast cancer (one ofthese was a carrier); three were unaffected and were carriers;and one woman, unaffected, had declined testing. Eight of theparticipants also underwent prophylactic bilateral salpingo-oophorectomy before or during their participation in theprogram, and of these, three (unaffected and carriers) alsounderwent prophylactic bilateral mastectomy.

The participants ranged in age from 24 to 67 years; themajority were married. They held a variety of types ofemployment and enjoyed generally high household income.We had hoped, but were unable, to recruit participants frommore disadvantaged socioeconomic situations.

The Interviews

Participants underwent an in-depth interview of approxi-mately 90 min. The interview guide (Table 1) was based on

162 Proulx et al.

the literature and emerging data; it was reworked after pre-testing with three participants (included in the final sample).Interviews were tape-recorded and transcribed. They wereconducted at home or in the workplace—at the convenienceof the participants—or even by telephone. The study wasapproved by the Ethics Committee of the CHUM ResearchCentre. In direct encounters, consent forms were signed by theparticipant at the time of the interview, and for the telephoneinterviews, forms were mailed, signed and returned.

Analyses

The analyses were carried out in three phases. First, the datawere reduced and organized through a process of coding.Two members of the team (MP and CL) read through thetranscripts of six interviews, each undertaking their own

coding. Then two meetings were held to compare and agreeon a coding system (summarized in Table 2). The data werethen categorized and indexed by category and sub-categoryusing QSR NUDIST software. The next phase involvedintra-individual synthesis, as proposed by Ayres et al.(2003), to capture the unique dimensions of each partic-ipant’s experience and to explore trends, consistency anddifferences among the 21 interviews. Statements from eachinterview were synthesized in relation to the indexedcategories and whatever was core to the participant wasextracted. The above-mentioned two researchers eachanalyzed independently one-half of the final corpus andthen discussed them together. The final phase consisted ofinter-individual syntheses (Ayres et al. 2003) in which thecontent of the various discourses was compared to identifycommon and specific themes by health status. Our aim was

Table 1 Summary of the Interview Guide

1. Course of treatment prior to genetic screening•Progression up to genetic screening•Information known before counselling on BRCA1/2 testing and genetic counselling•Information retained and understanding of this information and of the communication; how was it done?•Evolution of understanding after genetic counselling2. Genetic counseling and testing•Understanding of implications associated with BRCA1 or BRCA2 diagnostic testing•Evolution of understanding about the test; perceived advantages vs disadvantages•What was acceptable and what was not•Reasons for having the test or for not wanting to have it•Personal reaction after the decision; evolution•Discussion and impact both individually and in terms of family; evolution of understanding3. Unfolding of the process between the experience of genetic counselling and participation in the surveillance monitoring tests•Exploration of the proposed options (preventive surgery, medications, others)•Reaction to the various options, reasons for undergoing one option or another•What is acceptable and not acceptable about each option•Impact individually and in terms of family4. Surveillance monitoring•Motivation for participation in the monitoring•Manner in which the monitoring was presented•Understanding of the monitoring•Process and description of the monitoring received•Understanding of each of the tests (self-examination, mammography, ultrasound, scintimammography, magnetic resonance):- Inconvenience, procedures, number of tests- Advantages and disadvantages- What is satisfactory and not satisfactory- What is acceptable and not acceptable- How the tests compare to each other- Impact individually and in terms of family- In the case of false positive results: reactions, understanding, evolution

5. Improvements that might be made to the entire program (counselling, options, surveillance monitoring) in terms of follow-up, treatment, andsupport

6. Other comments

Experiences and Decisions that Motivate Women 163

to determine which themes were shared by certainparticipants according to their health status and which wereapplicable to all participants regardless of their status. Therigour of the analyses was assured throughout by a processof verification and validation of results using feedbackloops for reviewing the transcripts and the intra- and inter-individual syntheses. The initial categorization providedbenchmarks to ensure all essential and nuanced dimensionswere effectively covered by the analyses. The resultstherefore represent the most faithful and balanced renderingof the statements of all the participants.

Results

Women’s Subjective Experience of Breast Cancerand of Being at High Risk

For all women, the decision to participate in the screeningprogram was clearly the culmination of a difficult process.All the participants, regardless of their health status, invariably

and systematically asserted that breast cancer was anemotionally highly-charged term. At the time of the interview,some who had already had the illness said they were stillliving the painful experience of cancer and the ever-presentfear of recurrence. These women, and even those who had nothad the disease, conceptualized breast cancer as an insidiousand implacable evil—and more so, when they knew them-selves to be at high risk of hereditary breast cancer.

There is no question that the genetic experience is afamily affair, not only because of the technical aspects ofconstructing the family genealogy, but also because ofdimensions related to identity. Clearly, the family experi-ence of cancer influenced the way each woman conceivedof her own risk of developing the disease. Some womensaid they tended to evaluate their own risk by basingthemselves on resemblance to a close family member.

…I resemble my mother, like two drops of water; Ihave her hands, her legs…It was like, at a givenmoment, genetics takes over…You have the same toesas your father, because that’s how it works (womanwithout history, carrier: interview 19).

Table 2 Data Codification System

1. Perception of the case management1.1 Healthcare and treatment experiences, on the part of both the interviewee and close family, related to breast cancer and other cancers

(previous experiences, other experiences related to health or illness)1.2. Genetic testing: BRCA1/BRCA2 (absence or presence of a mutation) (relevance, motivation, understanding, information received, results,

satisfaction, evolution of perceptions)1.3. Screening program: mammography, magnetic resonance, scintimammography, ultrasound, clinical exam (relevance, motivation,

understanding, information received, results, satisfaction, evolution of perceptions)1.4 Treatment options at the time of genetic counselling (excluding all options preceding the counselling; these are included in category 2.1)1.4.1. Cancer-preventive drugs (Raloxifene, Tamoxifen)1.4.2. Surgeries1.4.3. Others1.5. Results of screening tests (biopsy, normal, abnormal)1.6. Improvements2. Risk (genetic testing, screening tests): a family story!2.1. Reactions and consequences among close family members (everything that explicitly mentions reactions of those in the family circle in

relation to risk, participation, motivation, discussion, etc.)2.2. Cancer in the family (comments by the interviewee on family history, gene transmission, related to risk, etc.)2.3. The patient’s role and responsibility (strategy regarding members of the family and her close circle, personal attitude assumed).2.4. Formulation of the risk (understanding, acceptability, beliefs, family values, images/signs)2.5. Affects surrounding the risk (fear, confidence, emotions, etc.)3. Perception of breast cancer and comparison with another illness (definition, causes, consequences, extent, subjective representation of risk

related to the cancer)4. Course of treatment (all factual data related to the patient’s course of treatment, plus expectations and needs)4.1. Before genetic counselling (expectations, needs)4.2. During genetic counselling (expectations, needs)4.3. Screening program (expectations, needs)

164 Proulx et al.

Several others seemed to have assessed their ownchances of survival or their own risk of developing theillness on the basis of family experience, and particularly onthe number of cancer deaths in the family and theirperception of the pain and suffering endured by the familymembers in these illnesses.

Moreover, almost all the women appeared to have agood understanding of the implications of being a mutated-gene carrier and were able to speak at length about thenumbers and the probability statistics when discussing theirown risk. Some of them stressed the fact that they knewbeing the carrier of a mutation is not the same as having acancer diagnosis, nor does it predict with certainty that theillness will develop. Others expressed concern that knowl-edge in this field, particularly of the uncertain nature of thegenetic tests (for example, in terms of prognosis), iscurrently limited and that a lot remains unknown.

I understand that this is not a cancer diagnosis. Theway I think it should be interpreted is, if you’rediagnosed, you maybe have to pay more attention andtake it a bit more seriously. This is not a guarantee thatthe cancer will move. It’s also not a guarantee, ifyou’re not diagnosed, that you won’t have it (womanwith history, not a carrier: interview 3).

While the vast majority of the participants seemed tohave very good information on risk, this informationnevertheless provoked a great deal of anxiety and worryin women who had already had breast cancer and otherswho had not, but had lived through the recent death of aclose family member. One young woman described herdifficulty in absorbing the information because of her intenseemotions after having just lived through her mother’s deathfrom breast cancer.

You think about the person you’ve lost…. It’s hard tomake the connection between the two things when it’svery scientific, while what you’re hearing is allemotional…. It’s connected to someone… these are notjust impersonal data…. Everything they are explaining,it’s like… my mother. It’s very difficult to reconcile thetwo…. The scientific data is very cold, very numerical,and what you experience in relation to this, and whatbrought me to this study, it’s very emotional… (womanwithout history, not tested: interview 13).

This seemed to be particularly significant for three womenwho opted for bilateral mastectomy. These women felt thescreening procedures alone—while indispensable—did notprovide enough reassurance. They concluded their only viableoption was to undergo the preventive surgery to bring theirpersonal risk, in terms of percentages, down to a more

acceptable level, i.e., equivalent to that of the generalpopulation.

I don’t know… I feel much less at risk, I feel it, Ilive it, it’s in my gut, that I’m much less at risk thanthose who decide not to do these operations or whodon’t even know about the genetics. I feel much,much less at risk… (woman without history, carrier:interview 20).

What Motivates a Decision to Undergo or DeclineDiagnostic Testing

All the women, upon entering the screening program, hadbeen invited by the team to consider undergoing BRCA1 orBRCA2 diagnostic testing. In the interviews, they all spokeat length about what led them to opt for the test or todecline it. For all of them, the decision was closely linkedto the availability of the screening monitoring. Somewomen even said that the testing, on its own, would beunacceptable without the availability of close follow-up.Several also spoke of their desire to contribute to theadvancement of knowledge in the field. However, as weexplored the key motivations put forward by the partic-ipants, we saw that these were expressed according towhether the women had elected to be tested or not andwhether or not they had already had the illness.

Motivations in Women, Affected and Non Affected,Who Accepted the Testing

Women Affected with Cancer Felt they had Nothing to Loseand Everything to Gain By Being Tested

The seven participants who were affected with cancer andaccepted the testing seemed ultimately to feel they hadnothing to lose and even everything to gain by being tested.Some, having come through the illness relatively well, saidthat the decision to undergo testing was much easier forthem emotionally and psychologically compared with otherparticipants who had never had cancer. For all theseparticipants, knowing their status seemed to be one moretool giving them control over their lives and thus providinga certain measure of security.

She [her sister] wanted me to go. She wanted to know,but for me, I was worried about her knowing. I said tomyself, how can you live knowing that you mighthave, or that you have an 80% chance of having,cancer? I had a hard time telling myself that washealthy. It was mostly that. For myself, I had nothingto lose, and it was even a good thing. But I was a little

Experiences and Decisions that Motivate Women 165

bit worried about her (woman with history, not acarrier: interview 3).

However, several of them did say that they were hesitantat first, because it seemed to them they were extending theexperience of their illness. Most, in fact, were comforted tolearn they were not carriers; only one in this group knewshe was a carrier. Although she did not regret havingundergone the testing, this woman described feelingsometimes very alone and anxious.

Women Unaffected with Cancer Emphasizedthat they Preferred Knowing Over not Knowing

The other eight participants who never had cancer andunderwent the testing said that ultimately they preferredknowing their health status, rather than not knowing.Knowing removed any doubt. In fact, some explained thatthey would not be able to live with the uncertainty and feltmuch more reassured knowing. Even more importantly, thediagnostic testing offered them the possibility of receiving anegative result, which had the potential to be liberating andreassuring.

My family history. I wanted to know, yes, but in myown mind I always knew I was very, very, very muchat risk. I always told myself that. Since, and evenbefore, my sister became ill. And to know the resultof that test — that, it was just the possibility ofhaving a positive result. Just the possibility of beingable to say, no, you’re worried about nothing. Youdon’t have it. But the fact that it was confirmed, thatdidn’t surprise me (woman without history, carrier:interview 7).

While these women did not regret having undergone thetesting, some described the destabilizing effects of thenews, in terms of emotion and of identity.

I think of it as a house, your own interior is like ahouse and you’re used to functioning in it, andsuddenly, from one day to the next, it’s as if a sectionof wall has fallen and there are a whole lot of peoplearound you, and who are part of your reality, but it’snot so easy… being prepared for that is not so easy…(woman without history, carrier: interview 9).

It appears that these women gradually came to create anew identity and to deal better with this new status in theirdaily lives.

We must add that women who accepted the testing,whether affected or unaffected, also expressed other

common themes that had to do with their decision toundergo testing. Almost all showed concern that knowingwhether they were carriers might make them much moresensitive to physical symptoms, and they were hopefulabout being more carefully followed by the team. Somealso stated that they would be better prepared to decide onthe best preventive measures to take (e.g. preventivesurgery). Finally and above all, many of these participantsstressed the fact that they felt obligated to do everything intheir power to deal with their situation, particularly for theirchildren and grandchildren. Some women who werehesitant at first eventually accepted the testing, reasoningthat they had a responsibility to know their status for thesake of their children, so as to be better prepared.

As for the rest, knowing what type it is, that wasn’timportant. The important thing was to get rid of itand to find out if there is any chance my childrenwill inherit it (woman with history, not a carrier:interview 3).

Women who Declined Testing Based their Decisionon a Desire to Protect themselves Emotionallyand on the Uncertain Nature of the Prognosis

The six other participants, who had never had cancerand had declined the testing, had, like the precedingparticipants, a very painful family history (deaths ofseveral close family members, sudden death of someoneclose who had the disease at a very young age) andtherefore considered themselves also to be at high risk.However, they reacted differently to the idea of beingtested. These participants stressed that they wanted toprotect themselves and those close to them. They seemedto have asked themselves how they would react if theylearned they were carriers. These women concluded thatknowing their status could have negative repercussions,emotionally and psychologically, and might even acceler-ate the development of the illness. In addition to beingconcerned about the confidentiality of their medicalrecords, some women stated that they did not want tolive in dread of the illness, and they felt that knowingthey were carriers might create worries or arouse a lot ofanxiety in their daily lives. Some chose instead to liveday by day, relying on the program for early screening ofany problem.

So I was not tested, on the other hand, because theyexplained to me that in this situation I could have thesame follow-up as a woman or a person who wouldhave a positive result. So I said, listen, somehow, now,today, to live with this, I’m not sure, psychologically

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speaking, that I could do it… (woman without history,not tested: interview 2).

Some of these women who had children (three out ofsix) also indicated that they decided to refuse the testingbased on the recommendations of their daughters. Thesedaughters seemed to have considered there was a risk thatdiagnostic testing would unsettle their mothers too muchemotionally, and so they suggested that they refuse to betested. They also seemed to be sufficiently reassured thattheir mothers would be closely monitored.

Finally, some of these six participants expressed anotherconcern. They felt that, given the uncertain nature of thetest and of the prognosis, the testing would not lead toanything more definitive. These women explained that evenif they were carriers, they might very well never developthe disease, and that the only obvious choices were to beclosely followed or to undergo preventive surgery. Feelingthey were sufficiently covered by the screening procedures,these women did not think they would be better offknowing their genetic status.

Benefits and Challenges of the Experimental ScreeningProgram

Screening and Monitoring Offer Reassurance

Among all the participants, the decision to undergo ordecline diagnostic testing seemed to represent a majorturning point—much more difficult to live through,emotionally and psychologically, than the screening tests.Indeed, a central theme among all women was that thescreening program provided a sense of reassurance andsecurity because it offered the possibility of rapid responseand therefore hope of better recovery. Many women alsoseemed to consider the screening program to be an efficientpreventive strategy for identifying problems and treatingthem as soon as they appear.

It’s like having all the luck on our side to detect it intime and have the appropriate treatments withouthaving to go under the scalpel. I have the impressionthat if they know it, and I know it, they’ll put muchmore effort into early detection, and the monitoringwill be stricter. This gives me a better chance to fight it(woman without history, carrier: interview 11).

These women said that being carefully examined wasalso reassuring, and they seemed especially to appreciatethe number and complementary of the tests. They consid-ered that it would be hard for the team to overlook aproblem, given the number and frequency of tests.

I have the impression that if they make us go throughthese four tests, it’s for a reason, they are complemen-

tary to each other. They probably can’t find everythingthey would want to with just one test, so they haveto use several to really look at everything as muchas possible (woman without history, not tested:interview 6).

They seemed to feel the professionals on the team wereexperts in what they did, and they were reassured that theteam was conscientious and meticulous, leaving nothing tochance, always on the lookout.

Because I know that if there were something, theywould jump right away into high gear and I would knowthe same day or the next (woman without history, carrier:interview 11).

They also appreciated being reminded by the team aboutroutine exams, and they were relieved not to have to worryabout planning these, which offered some respite fromdwelling on their situation. Some felt they were in aprivileged group, being well looked-after by the technicalstaff and the care team in everything having to do with thetests. They stressed the fact that they were very aware thatthe reality was different for others in the current healthcaresystem.

Just being followed by the clinic reassures me. It’s asif I told myself, I don’t have to worry about this,they’ll look after it for me. Or if they call me for areturn visit, I go for the tests. It’s like I’m leaving thisin someone else’s hands, and I think for me it’shealthier psychologically (woman with history, not acarrier: interview 3).

This concern was raised by several participants, eventhose who left the experimental program. One of them, wholeft the program because she moved away and was unableto travel the long distance required to undergo the screeningtests, expressed the wish to have this type of program in herregion.

Emotions, Worries or Stress Felt by Women, Particularlyin Case of False Positive Results

Many participants, especially those who had already hadcancer, talked about the emotions, worries or stress they felteven just at the thought of participating in the screeningprogram. Because of the painful experiences they livedthrough in their illness, some of them seemed to have felt alot of anxiety when undergoing different screening tests,especially in the beginning.

Since then, I don’t like going into hospitals… I stopbreathing and I only start breathing again when I getout at the end of the day, and now I carry along my

Experiences and Decisions that Motivate Women 167

little bottle of Purel. I definitely don’t like being in ahospital, I don’t feel well, and every time I have anappointment coming up, for about a month ahead, Ilose some of my control over my anxiety. Sometimesthere are really days where it’s harder to fight the fear, areal feat (woman with history, a carrier: interview 10).

Yes, because each time we undergo a test, we wonderif they will find something else? I time, I think of that.But they tell me that if there’s something, they will callme back. After a certain time, I let go. And I say tomyself, if we had to do it, we would (woman withhistory, not a carrier: interview 3).

Women who never had the illness, whether they hadbeen tested or not, also talked about their emotions and thestress they felt when undergoing the tests, especially thefirst round of tests.

There were a lot in my family and so I want to live…for sure, it’s not always obvious, but emotionally,when I go to be tested, whether I like it or not, that’sthe only thing on my mind and it worries me…especially the first time I went to the hospital and I puton the blue smock, it was difficult, and I told myself itwas for my own good… It’s not obvious… Things gomore easily now, but the first times were not easy(woman without history, not tested: interview 14).

Whatever their health status, the participants were notfully satisfied with the way in which results were reported.Some preferred to get a follow-up phone call immediatelyafter the tests, no matter what the results were. However, allagreed the procedure should be formalized and clearly setout from the first round of tests. Some also would haveliked the processes and procedures, and what they shouldexpect, to be explained clearly from the start, so that theycould be better prepared psychologically. Unfortunately, thispreparation was uneven, as several participants lamented.

The way things happen… how this is done… themachine, the noise… I like to know in advance whatto expect … have all the information on the tests suchas they are, because I was not used to them and it wasunfamiliar to me… (woman with history, not a carrier:interview 17).

It would be nice to be called to be told everything is fine… rather than to say that no news is good news (womanwithout history, not tested: interview 14).

All the participants seemed also to be very concernedabout how long they had to wait for results.

Yes, because you have a mammogram and you don’tget the results right then, you get them three weekslater… When you’re worried, three weeks is a very

long time. I think it shouldn’t be like that. You shouldnever have to wait weeks and weeks for any result,because you imagine the worst… It depends on thekind of person you are, but when you have livedthrough cancer, you imagine the worst (woman withpersonal history, not a carrier: interview 12).

For sure, it can seem like a long time… You think thatif you have something in six months, you have time todie… Then I have to tell myself they’re the expertsand they know what they’re doing and what they’retalking about, so I have to stop worrying and get onwith other things (woman without history, not tested:interview 15).

This wait was even more difficult for women who had togo through other investigations with positive (abnormal)test results. Whatever their health status, these women toldabout their stress and worries when given news of a falsepositive result and while waiting for the results ofinvestigations. Some found it particularly difficult emotion-ally to go through the waiting, worried they might havesomething serious and that it would not be treated rapidlyenough. Generally, these women seemed to find it goteasier over time, and all the more so if the results werenormal.

The time I had to wait for the biopsy results, it lastedtwo weeks and couldn’t go on much longer, because itwould have been unreasonable, because whether youlike it or not, you think of all kinds of scenarios whileyou’re waiting like that, especially if you’re high risk.What is unacceptable is to wait for treatments… Iknow today everyone is doing their best, but when youknow you’re ill and you might have to wait to betreated, I think that is absolutely unacceptable …(woman without history, not tested: interview 14).

… but I was stressed out for a month until I got thecall from the generalist … and then I really stoppedbeing stressed because I knew that if there weresomething, they would call. Basically, what wasstressing me was not knowing (woman withouthistory, a carrier: interview 9).

Some mutation carriers and some women who refusedthe testing, and who felt isolated because they lived aloneor received little support from their families, said theywould have liked to receive psychological support or beable to talk with other women in the same situation.

I don’t know if there should be a psychologistinvolved in it or … I don’t know, but I know thatthe people you meet are very professional, and veryrespectful of the fact that it’s emotional and that you

168 Proulx et al.

can get overwhelmed like that, but I just don’t know if apsychologist could help with that. At a certain point, it’shard to face the test results, the analyses of tissue andblood, and also the questions. So at a certain point, it’sall going too fast … For me, at that moment, the datameant nothing… For me it was people, my grandmother,my mother, an aunt, my family, my heritage… (womanwithout history, not tested: interview 13).

Traditional Screening Methods Called into Question

Several women mentioned the discomfort of the tests, suchas scintimammography, where they had to stay in the sameposition for several minutes. Some complained that MRIwas very noisy. Some others who said they were claustro-phobic were apparently very afraid of entering the machine.One woman even had to take medication to calm herselfthe first time she had the test.

Magnetic resonance. They had explained this to me alittle, but when you don’t see the machine. When Iwas in front of the machine, I saw the little opening. Itworried me. At the time, I wondered if I would be ableto stay in it for a long time. Ten long minutes? Thetechnician explained it to me. You didn’t go in head-first, but feet-first. So you can see the exterior, you feelless stuck. Maybe they could have explained this tome a bit more. Maybe they could have told me it’simportant not to be claustrophobic (woman withhistory, not a carrier: interview 3).

For sure, when you’re in a big machine and you hearthe noises and it’s impressive … you have theimpression that they can see everywhere in your body,so if there is something, they will find it for sure.Ultrasounds and mammograms, but I’ve always hadlumps in my breasts and I told myself that it wassomething they wouldn’t see (woman without history,a carrier: interview 21).

Most, however, appreciated ultrasound, which involvesno significant discomfort and offers the possibility ofgetting immediate feedback on the screen.

Actually, it’s only with ultrasound that you getimmediate feedback because you’re right there on thescreen, so ultrasound is great. You can say it’sreassuring because you see the results on the spot (6).

This is less the case for breast self-examination andmammography. Some participants no longer did breast self-examination because this made them too anxious. Someseemed also to have very little confidence in mammogra-phy, particularly women who had breast cancer and had abad experience with the technology. In addition to the pain,

they wondered about the potentially carcinogenic effects ofmammography due to exposure to radiation.

My level of confidence is lower in terms of what theycan find with mammography… I think that if one daythey could stop doing mammograms and concentratejust on the two others, I would like that (woman withhistory, a carrier; interview 10).

Actually, the great majority of participants put a lot ofhope in MRI for cancer detection, much more than inmammography, which is considered a less sensitive test.

If we compare the tests against each other… for sure,magnetic resonance and nuclear are more, comparedwith a mammogram. I don’t know… I can’t tell you, Ican’t come to conclusions because it’s not finished…As mammogram doesn’t find all cancers… It would begreat if they could find a test that could really findbreast cancer… but it’s not accessible to everyone,either. There aren’t machines everywhere (womanwith history, not a carrier, interview 17).

Tests. Mammograms, they see what they see, I’vealways been told that resonance was clearer and moreadvanced but I don’t believe in it anymore (womanwithout history, carrier: interview 19).

Discussion

The results of this exploratory study broaden our under-standing of the experiences and decisions that motivatewomen to participate in a screening program and suggestthat issues of security are central concerns. We see thatintensive screening procedures offer women at high risk ofhereditary breast cancer some reassurance and a sense ofcontrol over their lives. However, participation in suchprograms can exacerbate emotional suffering by makingwomen relive difficult experiences or by keeping them inan illness mode, preventing them from putting the cancerbehind them. Some participants also spoke of a loss ofconfidence, and even of fears (such as fear of radiation) atthe thought of certain screening methods, even to the pointof abandoning breast self-examination because of the fear itgenerates (Wainberg and Husted 2004, Nelson et al. 2005).

There is currently very little information available on theparticipation of women at high risk of hereditary breastcancer in intensive screening programs (Warner 2004).Most studies are quantitative and not very informative. Forinstance, the sense of security offered by screeningprograms is rarely mentioned and hardly developed in theliterature (Bonadona et al. 2002, Lodder et al. 2003),whereas it was a core dimension of the experience among

Experiences and Decisions that Motivate Women 169

the women we encountered. As demonstrated by otherswith regard to genetic diagnostic testing, we also observeddifficulties related to waiting for screening results. Thisaspect has been reported in the literature (Lerman et al.2000, Nelson et al. 2005, Sauven et al. 2004). We notedthat participants felt strongly that communication of resultsshould be formalized, an observation also made by Wardleet al. (1993) in a cohort of women at risk of ovarian cancer.This aspect appears particularly important in the case offalse positive results which could lead to undue anxiety(Spratt and Spratt 1992). Some women felt isolated andwould have liked to see individual or group counsellingoffered over the course of their participation in the screeningprogram. In fact, it seems the importance of psychologicalsupport is very often underestimated (Metcalfe et al.2000).

Our study also demonstrates that decisions relating todiagnostic testing form part of a very emotionally andpsychologically difficult process. A review of the partic-ipants’ decisions revealed some pivotal motivations thatentered concurrently into their decisions, and which wereclosely linked to the availability of screening monitoring.The screening was reassuring to some women who hadalready had cancer and who ultimately felt they had nothingto lose by this experience, and even much to gain, asobserved by Biesecker et al. (2000). We see that thedecision to undergo genetic testing can also reflect a desireto know one’s health status, and that some women foundthe idea of living in uncertainty unbearable. Others wouldrather protect themselves emotionally and therefore de-clined testing (Lodder et al. 2003). However, this protectivereaction is only possible because of the availability ofscreening monitoring, which allows some women to placethemselves in the care of health professionals on the team,providing some emotional respite.

The qualitative nature of our study allows us tocorroborate the framework proposed by Rees et al. (2001).We see that the direct experience of breast cancer—either inthe woman herself or in someone close—has an impact onthe subjective experience of illness, on the response to riskinformation, and even on the perception of risk, thatbecomes an important determinant of decision-making,particularly with regard to diagnostic testing. Here weshould highlight one dimension that is particularly signif-icant to the subjective experience of illness, which is familysolidarity. Indeed, even though the genetic experienceappears to be intimate and personal to the woman, clearlyit involves many people—her sister, her mother, andespecially her children. Many participants expressed amoral responsibility to those close to them to undergogenetic diagnostic testing or even surgery, for the sake ofprevention. Like other researchers, we consider this a coredimension of subjective experience and a key determinant

for action (Bonadona et al. 2002, Cappelli et al. 1999,Lodder et al. 1999, Metcalfe et al. 2000).

Our observations also suggest that the prior experienceof illness and the response to risk information have animportant influence on how participants within the programdeal with their risks on the basis of deeply subjectiveperceptions. These lay perceptions introduce not onlynormative (values, attitudes toward diagnostic testing), butalso symbolic (figures or images associated with awarenessof status) and affective (fears, emotions) dimensions. Forexample, we see that some women—such as the three whounderwent bilateral mastectomy—have a fundamental need,in the course of the program, to reduce their personal risk tothe lowest possible level, and thus they elect to undergopreventive surgery. As some researchers have observed, weclearly see here that information on survival rates associ-ated with different screening methods does not sufficientlyreassure women at high risk, who would rather opt forpreventive surgery (Schwartz et al. 2000, Lim et al. 2004).All in all, these results demonstrate the importance ofclearly presenting high-risk women with the pros and cons(e.g. potential physical and emotional sequelae) of theproposed preventive and screening measures and ofexplaining clearly all the evidence and uncertaintiessurrounding the available data (Giordano et al. 2005,Hallowell 2000, Lerman et al. 2000). Furthermore, we seethat perceptions of risk evolve and are transformed andtranslated into action over the course of the program. Thisis reminiscent of the notion of chronic risk developed byKenen et al. (2003), whose perspective is useful in lookingat the chronicity of the risk experience among high-riskwomen and the numerous adjustments it entails. Our resultsclearly show that after learning of their status as carriers,some participants felt their self-identity to be profoundlyshattered and had to learn to deal with this new reality not onlyin relation to their sense of identity, but also in their relation-ships with others, and in terms of their genetics and their healthstatus. This was also observed by Lim et al. (2004).

Study Strengths and Limitations

The strength of this study is in describing the experiencesand decisions that motivate women’s participation in ascreening program in terms of a sample that is diversifiedaccording to health status. This made it possible to contrastand compare different aspects of the experience as lived byparticipants in the experimental program (Popay et al.1998). However, the study is limited, in that it does notreally explain why some women withdrew from theexperimental program, even knowing they were at highrisk of hereditary breast cancer. The telephone interviewspresented significant limitations, since the interviewer didnot have access to non-verbal cues that are valuable

170 Proulx et al.

expressions of affect and that are helpful in bettercomprehending ideas (Kirsch and Brandt 2002). Likewise,it would have been useful to interview certain women who,although participating in the program, refused to take partin our exploratory study—especially those who citedemotional reasons for this refusal. Such interviews mighthave provided additional, enlightening information. Lastly,our study did not enable us to document why some womenrefused to participate in the experimental program at all. Itmay be that these women have very different attributesfrom our participants in terms of sociodemographiccharacteristics and sensitivity to risk, as suggested byGeller et al. (1999). Indeed, those researchers observedthat women who are likely to participate in genetic testingprotocols are a self-selected group: they generally exhibita heightened sensitivity to personal risk, are relatively well-educated, generally have a high level of income and oftenexpress a desire to participate in the advancement ofknowledge in this field (Geller et al. 1999). Our studytherefore offers a basis for generalizing the conclusions, on atheoretical level, primarily to women with the same socio-demographic backgrounds as our participants (Kvale 1995).

Genetic Counseling Practice Implications

The participants raised a number of dimensions relatedto genetic counseling, including information on risk andBRCA1/BRCA2 testing (nature of the information andhow it is transmitted), as well as on the screening programprocess. From our results, we can see that althoughinformation needs vary from one participant to another,this information is gradually assimilated by all theparticipants. The results also indicate that participants donot all receive the same information at intake on how theprogram operates. Among other things, participants whojoined a few months into the program, after the team hadbecome more experienced with the procedures, appeared tobe better prepared than the others. These two aspects—information (e.g. risk, diagnostic testing) and clarity aboutthe programs’s processes (e.g. results reporting, wait times)—must be front and centre in counseling activities. Counselingshould be geared toward ensuring each participant has the bestpsychosocial support for her specific situation. The counselormust also communicate information on risks and proposedoptions (with pros and cons) at a rhythm that respects eachwoman’s situation and, especially, must ensure that informa-tion is clearly understood.

Conclusion

This study demonstrates that intensive screening programsmay provide valuable reassurance for women with increased

familial risk of hereditary breast cancer. We observed thatparticipants count on early detection and rapid response fromthe professionals, if and when a problem arises. From thisstudy, we see the extent to which security is a core concernamong women who choose to participate in a screeningprogram, along with important concerns about personaland family risk. Health professionals must take all theseconcerns into account to ensure their interventions aremost consistent with women’s needs.

This study was funded by the Quebec Breast CancerFoundation (March 2004) in the context of a study entitledMultimodality Breast Cancer Screening Program for High-Risk Women (Presence or High Probability of BRCAMutation) directed by Diane Provencher, Lucie Lalonde,Christine Maugard, Williams Foulkes, Ann Aldis, Marie-Dominique Beaulieu and Michelle Proulx.

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