Breast cancer survivors' attitudes about communication of breast cancer risk to their children
Abstract
Perceptions of breast cancer survivors regarding educating their children about hereditary breast cancer risk are underexplored. This study examined attitudes of early-onset breast cancer survivors concerning whether, at what age, and how their children should be educated about potential risk for hereditary breast cancer. Women with breast cancer diagnosed < age 50 years between 1994 and 1997 were recruited from 34 Virginia hospitals. Participants responded to two surveys. The Family History Questionnaire permitted participant classification into two groups based on risk for hereditary disease, as determined by personal and family history. The Knowledge, Attitudes and Beliefs Questionnaire addressed participants' concerns regarding their children's breast cancer risk, perceptions of their children's concerns about their own risk, and whether, at what age, and from what source their children should be informed about their own risk. Among 267 participants, the average age was 47.5 years; 90% were Caucasian, 7% were African American; 52% had suspected hereditary breast cancer. Of participants with children, 81% indicated concern about their children's breast cancer risk; 55% reported that their children had expressed concern about their own risk. Seventy-one percent of participants believed the age for informing children about hereditary breast cancer risk to be before 18 years. Eighty-four percent cited the child's parents and 65% cited health professionals as a preferred information source. No differences in responses between participants with suspected hereditary breast cancer versus presumed sporadic breast cancer were noted. These data establish the need for educational resources regarding hereditary breast cancer for children of early-onset breast cancer survivors. © 2003 Wiley-Liss, Inc.
INTRODUCTION
Scientific advances have resulted in the ability to predict genetically increased risk for a number of common adult-onset disorders, including breast cancer. Difficult issues may arise for the patient and family as they address their risk for hereditary breast cancer, as well as the ways to manage this risk. Some of the most sensitive of these issues relate to the impact of risk assessment on other family members, particularly children.
Despite a plethora of articles that have addressed the issue of genetic testing of children for adult-onset disorders [Harper and Clarke, 1990; Clarke et al., 1994; Wertz et al., 1994; American Society of Human Genetics Board of Directors and the American College of Medical Genetics Board of Directors, 1995; Michie and Marteau, 1996; Patenaude, 1996; Geller et al., 1997; McKinnon et al., 1997; Task Force on Genetic Testing, 1997; Cohen, 1998; Dickenson, 1999; Kodish, 1999; Eiger and Harding, 2000; American Academy of Pediatrics, 2001; Miesfeldt et al., 2001], little research has been conducted regarding the best ways to inform and counsel children and young adults about the possibility of inherited cancer risk, or about parental preferences in this regard [Strong and Marteau, 1995; Fanos, 1997; Cohen, 1998; Hamann et al., 2000].
Despite a plethora of articles that have addressed the issue of genetic testing of children for adult-onset disorders, little research has been conducted regarding the best ways to inform and counsel children and young adults about the possibility of inherited cancer risk, or about parental preferences in this regard.
This is striking, given that research has repeatedly shown that a major motivation for women to undergo BRCA1/2 testing is to help assess risks to their children [Lerman et al., 1994, 1995a, 1996; Struewing et al., 1995; Hughes et al., 1997; Jacobsen et al., 1997; Tessaro et al., 1997; Loader et al., 1998; Bluman et al., 1999; Pasacreta, 1999; Meiser et al., 2000]. The importance of education and counseling for young people about late-onset genetic disease risk and the need for further research in this area has been emphasized [Strong and Marteau, 1995; Fanos, 1997; Cohen, 1998; Lerman et al., 1998; Hamann et al., 2000; Tercyak et al., 2000]. Furthermore, an examination of these issues is important and timely, as communication about inherited breast cancer risk from parents to children is already occurring [Hughes et al., 1999; Tercyak et al., 2001].
We explored family issues regarding potential hereditary breast cancer risk among women from throughout Virginia who had been diagnosed with breast cancer prior to age 50 years between 1994 and 1997. Specifically, we examined these women's concerns regarding their children's risk for disease; perceptions of their children's concerns; and beliefs regarding whether, at what age, and how children should be informed about issues regarding potential hereditary breast cancer risk.
MATERIALS AND METHODS
Sample
This work was approved by the Institutional Review Board at the University of Virginia Health System. The study sample was recruited from all 71 Virginia medical facilities reporting breast cancer cases in women under age 50 years to the Virginia Cancer Registry between 1994 and 1997. Because younger breast cancer patients are at increased risk for hereditary breast cancer, the age limit of less than 50 years at diagnosis was chosen to ensure that a substantial proportion of participants would be at risk for hereditary disease.
To protect the confidentiality of potential study participants, women were contacted for participation by the individual hospitals where they were diagnosed. This resulted in a two-step sample recruitment process. Step one involved hospital recruitment. A detailed information packet describing the study and requesting the hospital's participation in recruiting participants was sent by our study coordinator to registry representatives at the 71 eligible medical facilities. Of these, nearly half (34) of the hospitals agreed to assist with recruitment. The most common reasons cited for hospital refusal were lack of time and lack of resources. Virginia Cancer Registry staff then sent each participating medical facility a list of the names of its breast cancer cases diagnosed at age less than 50 years and enrolled in the registry between 1994 and 1997.
Step two involved participant recruitment. Registry personnel at each participating medical facility sent an informed consent and the Family History Questionnaire to all 2,959 women with breast cancer that the facilities had enrolled in the Virginia Cancer Registry between 1994 and 1997. Women were asked to send the informed consent and completed Family History Questionnaire back to the medical facility. Each medical facility compiled the signed informed consent and completed Family History Questionnaires received from the 314 women who agreed to participate in our study and returned these to our study coordinator. Each consenting participant was assigned a study code that was placed on her informed consent and the completed Family History Questionnaire when received from the hospital. All 314 participants were subsequently mailed the Knowledge, Attitudes and Beliefs Questionnaire.
Measures
The Family History Questionnaire and the Knowledge, Attitudes and Beliefs Questionnaire were developed by the study team that included a medical oncologist, a cancer genetic counselor, and a survey methodologist (instruments available on request).
Family History Questionnaire
This was composed of questions to assess whether a woman was at risk for hereditary breast cancer. The questions included the number(s) and type(s) of previous cancers that the woman had been diagnosed with, the number(s) and type(s) of cancers in family members, and the ages at which these diagnoses were made.
Knowledge, Attitudes and Beliefs Questionnaire
The 56-item Knowledge, Attitudes and Beliefs Questionnaire asked questions related to the views of each woman regarding breast cancer and potential inherited breast cancer risk. Question areas included demographic information, information resources about inherited breast cancer, concerns about children's breast cancer risk, health history and experience, cancer screening behaviors, knowledge about breast cancer-associated risk factors, knowledge about inherited breast cancer, and beliefs about breast cancer causes. The majority of the questions were multiple-choice.
The content of the Knowledge, Attitudes and Beliefs Questionnaire was based on a literature review, examination of a previous instrument (Ethical, Legal, and Social Implications Program Psychometric Core Tools, National Human Genome Research Institute), and the clinical and research expertise of the investigators. This instrument was pilot-tested with women undergoing treatment for breast cancer at the University of Virginia who were not eligible for the study. Revisions were made based on feedback provided in one-on-one interviews.
Data Analysis
The completed Family History Questionnaires were reviewed by a medical oncologist and a cancer genetic counselor to separate the participants into two categories, women with suspected hereditary breast cancer and women with presumed sporadic breast cancer. Descriptive statistics for all questions included in the Knowledge, Attitudes and Beliefs Questionnaire were calculated using SPSS version 9.0. Selected study variables were compared between women with suspected hereditary breast and women with presumed sporadic breast cancer using the chi-square test.
RESULTS
Response Rate
A total of 314 women from throughout Virginia agreed to participate in the study. Of these, 273 returned both questionnaires for a completion rate of 87%. Data from six were not included in the analysis because the women indicated that their breast cancers were diagnosed in years other than between 1994 and 1997. Of the participants not returning the Knowledge, Attitudes and Beliefs Questionnaire, five were deceased, one refused, four had addresses deemed undeliverable by the post office, and the remaining 31 did not return the questionnaire for unknown reasons.
Demographic Information
Demographic characteristics of the 267 eligible participants are summarized in Table I. Four percent (10) of participants reported having received cancer-specific genetic counseling and 2% (4) of the participants reported having undergone DNA testing for mutations in breast cancer-associated genes. A total of 52% had suspected hereditary breast cancer.
| Characteristic | Category | % | n |
|---|---|---|---|
| Ethnicity | Caucasian | 90 | 237 |
| African American | 7 | 18 | |
| Other | 3 | 7 | |
| Education | Some high school | 7 | 18 |
| High school graduate | 23 | 60 | |
| Some college | 20 | 54 | |
| Technical graduate | 6 | 15 | |
| College graduate | 27 | 71 | |
| Graduate degree | 18 | 47 | |
| Income | <$8,000 | 3 | 7 |
| $8,000–$19,999 | 9 | 24 | |
| $20,000–$39,999 | 18 | 49 | |
| $40,000–$59,999 | 20 | 54 | |
| $60,000–$79,999 | 18 | 47 | |
| >$80,000 | 29 | 77 | |
| Don't know | 2 | 4 | |
| Marital status | Single | 6 | 15 |
| Married | 77 | 205 | |
| Separated | 14 | 38 | |
| Divorced | 3 | 7 | |
| Children | One or more | 87 | 231 |
| None | 13 | 36 |
Concern Among Women Regarding Their Children's Risk
Study participants with children (n = 231) were asked, “Have you been concerned about your children's breast cancer risk because of your diagnosis of breast cancer?” Eighty-one percent (188) responded yes. Of the remaining women, 16% (38) responded no, and 1% (3) responded “I don't know.”
Women's Perceptions of Children's Concern for Own Risk
When these same women were asked, “Have your children been concerned about their own breast cancer risk because of your diagnosis of breast cancer?” the majority, 55% (127), responded yes. Thirty-three percent (75) responded no and 13% (29) responded “I don't know.” Participants noted that both daughters and sons had expressed concern about their own risk for breast cancer. The mean age at which children were reported to have first expressed concern was 19.4 ± 6.7 years (range, 6–38 years) for daughters and 18.6 ± 7.9 years (range, 5–35 years) for sons.
Participants noted that both daughters and sons had expressed concern about their own risk for breast cancer. The mean age at which children were reported to have first expressed concern was 19.4 years for daughters and 18.6 years for sons.
Women's Beliefs Regarding Whether and at What Age Children Should Be Given Information Regarding Their Own Risk for Breast Cancer
Women were asked, “In a family where someone has had breast cancer, at what age should children first be given information about their own risk for getting breast cancer?” A total of 96% (257) women responded to this question; the remaining 4% (10) left the question blank. Only 1% (three) women responded that they felt that children should not be given information regarding their own risk for disease.
Eleven percent (27) felt that the information should be provided to the child before the age of 12 years while 60% (156) felt that it should be given to children ages 13–18 years. Twenty-three percent (59) believed that one should wait until the child is no longer a minor (18–21 years), and 5% (12) felt that individuals should be older than age 21 years before learning about their own risk for breast cancer.
Women's Beliefs Concerning Who Should Give Information About Risk to Children
Women could choose more than one response to the following question: “If you feel that children should be given information about their own risk for developing breast cancer, who should give it to them?” A total of 254 women responded to this question. Of these, 84% (214) felt that the parent would be the appropriate educator for children about their future risk; 65% (166) chose the child's primary care provider. A total of 33% (83) believed that this information should be delivered by a gynecologist, while 19% (48) felt that an oncologist should provide the information. Twelve percent (30) selected a genetic counselor as an individual who should provide this information, and 3% (7) believed that it should come from a geneticist. Four percent (9) of participants felt that friends should supply this information.
While the literature to date has focused primarily on the potential risks and negative impact of cancer genetics testing on minor children, the potential benefits and positive impact of counseling children about inherited cancer predisposition have been less emphasized.
Comparison of Attitudes and Beliefs Between Participants With Suspected Hereditary Breast Cancer and Participants With Presumed Sporadic Breast Cancer
Women with suspected hereditary breast cancer responded similarly to women with presumed sporadic disease when comparing answers to all questions related to the issues of breast cancer risk to their children, as well as issues related to educating their children about hereditary breast cancer.
DISCUSSION
The increasing capacity to predict genetically elevated risk for adult-onset, chronic, and life-impacting diseases has prompted discussion and research about genetic testing of children for a number of disorders, including cancer. However, there has been little research regarding whether and how to educate and counsel young people about potential inherited cancer risk, and parental attitudes and concerns about these issues. Our work explored early-onset breast cancer survivors' attitudes in this regard.
In this study, the vast majority of participants with children stated that, based on their own history of breast cancer, they were concerned about their children's breast cancer risk. Furthermore, the majority of participants stated that their children had expressed concern regarding their own risk for developing breast cancer. Notably, participants reported concern for disease not only among daughters, but among sons as well. Therefore, sons may represent a group of relatives of breast cancer patients whose concerns about cancer risk have been underrecognized. The psychological impact of these concerns and fears on children, including the effect on developmental and maturational processes, is unclear. While the literature to date has focused primarily on the potential risks and negative impact of cancer genetics testing on minor children, the potential benefits and positive impact of counseling children about inherited cancer predisposition have been less emphasized. It is possible that education and support may be comforting to a child with a family history of breast cancer because these may remove uncertainty and address misconceptions, as has been suggested for genetic testing for breast cancer risk [Michie and Marteau, 1996; Cohen, 1998; Eiger and Harding, 2000].
Despite a recognition of the need for education and counseling for young people as a preparation for genetic testing, limited research has addressed parental attitudes about the appropriate age at which to inform and counsel children about potential inherited breast cancer risk. When asked at what age children should be provided with information concerning their own breast cancer risk, the majority of participants gave a response of 18 years of age or younger.
When asked at what age children should be provided with information concerning their own breast cancer risk, the majority of participants gave a response of 18 years of age or younger.
When asked who should provide information to minors about hereditary breast cancer risk, the answer selected most frequently by participants was the child's parents. This raises concern, because research has identified parental misperceptions about and distortions of genetic information, sometimes even after the parents have obtained genetic counseling. Early research found that parents may discount genetic information as not applying to them specifically or may binarize probabilities (“it will happen or it won't happen”) [Lippman-Hand and Fraser, 1979]. In the area of breast cancer risk counseling, the emerging literature [Croyle and Lerman, 1999] suggests that women at all risk levels perceive their breast cancer risks as higher than predicted by objective assessment. One study also found evidence that such women binarize their risks [Lerman et al., 1995b]. Emotional distress about the family history may also play a role in parental risk perception and risk communication to children [Biesecker et al., 1993; Fanos, 1997]. Therefore, while parents will necessarily play a major role in supporting and educating their children about risk, others will need to play important and complementary roles in these same tasks.
Of participants choosing a health care provider to give information to children about breast cancer risk, most preferred the child's primary care practitioner. However, many primary care clinicians have not been trained in the area of inherited cancer predisposition [Jones et al., 1997], and studies regarding knowledge about genetics among primary care physicians have shown deficits [Hofman et al., 1993]. This raises questions about the adequacy of physicians in this role. The need for primary care (as well as specialty) physicians to become more knowledgeable about genetics has been recognized [American Society of Human Genetics, 1995; American Society of Clinical Oncology, 1997; Collins, 1997; Jones et al., 1997; American Academy of Family Physicians, 1999; National Coalition for Health Professional Education in Genetics, 2001].
Only a minority of participants felt that breast cancer risk information should come from a genetic counselor or geneticist. However, as almost all of our participants had not undergone breast cancer genetic counseling, they were likely not familiar with the role of genetic services for provision of inherited cancer risk information. Therefore, their declination may have been based on lack of familiarity with these services, rather than on an experience-based preference not to have their children attend these.
Concerns about parents' adequacy and physicians' limitations as information providers raise issues about how best to address family history-positive minors' concerns about their breast cancer risk. Clearly, one part of the development of breast cancer education and counseling resources for children is for primary care practitioners to develop more expertise in genetics [American Society of Human Genetics, 1995; American Society of Clinical Oncology, 1997; Collins, 1997; Jones et al., 1997; American Academy of Family Physicians, 1999; National Coalition for Health Professional Education in Genetics, 2001].
Clearly, one part of the development of breast cancer education and counseling resources for children is for primary care practitioners to develop more expertise in genetics.
In addition, genetic counselors may be called on to play a role in counseling children and adolescents, particularly when the family history is suggestive of an inherited disorder. However, this will require cancer genetic counselors to develop expertise and comfort in discussing breast cancer risk with minors in an age-appropriate way. The counseling of minors regarding breast cancer risk will represent a major challenge, given the complexities encountered in cancer risk counseling for adults [Croyle and Lerman, 1999], who are cognitively and psychologically more mature than children.
The lack of differences in responses regarding children, comparing women with suspected hereditary breast cancer and women with presumed sporadic breast cancer, suggests that breast cancer risk education will be important for breast cancer survivors with little evidence of inherited disease, as well as for those with suspected hereditary breast cancer.
There are several limitations to this work. First, the proportion of women from the target population who agreed to participate in the study was relatively small. This was likely a direct result of the two-step recruitment strategy used, in which potential participants were contacted by each of the 34 health care facilities involved in this work rather than by our study staff. While this was done to protect the women's confidentiality, it also made the recruitment process more complex and likely resulted in a lower response rate. However, the response rate was high (87%) among the women who agreed to participate in the study. Second, although the participating hospitals represented a cross-section of the health care facilities statewide, the recruitment strategy used in this study likely limited the generalizability of the data presented. Third, the opinions of these women are representative of a cohort of concerned breast cancer survivors diagnosed at a young age who were recruited from the community setting. Therefore, our results should be extrapolated to other breast cancer survivors with caution.
Future research in this area should focus on finding the best ways to inform and support children regarding their potential inherited cancer risk. Research should address the attitudes of both adolescents and younger children concerning whether, at what age, and how to inform and support them in coping with this issue. Attention should be given to sons as well as daughters. The impact of this information on children is unclear and needs further assessment, including its influence on a child's developmental processes.
In addition, the ability of parents to communicate information about inherited breast cancer risk clearly and accurately needs study. Potential attitudinal differences between mothers and fathers regarding this issue should be examined. Research in families with an identified mutation should explore the attitudes of both the transmitting and nontransmitting parent regarding counseling, as nontransmitting parents will also play a role in the decision about this issue. An important underexplored area involves the thoughts and beliefs of clinicians about how to inform and counsel children about the possibility of hereditary cancer risk. Lastly, this work needs to expand to include a broader spectrum of inherited cancer predisposition disorders.
Acknowledgements
The authors thank the staff of the Virginia Cancer Registry for their assistance in accomplishing this work as well as the hospital personnel who kindly dedicated their time to this project.
