Attitudes Toward the Genetic Testing of ChildrenAmong Adults in a Utah-Based Kindred Tested for aBRCA1 Mutation

Heidi A. Hamann,1* Robert T. Croyle,2 Vickie L. Venne,3 Bonnie J. Baty,4 Ken R. Smith,3,5 andJeffrey R. Botkin3,4

1Department of Psychology, University of Utah, Salt Lake City, Utah2National Cancer Institute, National Institutes of Health, Bethesda, Maryland3Huntsman Cancer Institute, University of Utah, Salt Lake City, Utah4Department of Pediatrics, University of Utah, Salt Lake City, Utah5Department of Family and Consumer Studies, University of Utah, Salt Lake City, Utah

Advances in molecular biology and geneticshave led to the identification of the breast/ovarian cancer susceptibility genes BRCA1and BRCA2, along with tests to detect muta-tions in these genes. Although the appropri-ateness of BRCA1/2 genetic testing for chil-dren has been debated in the literature,little is known about the attitudes of indi-viduals who have undergone cancer suscep-tibility testing. The present study focusedon attitudes toward BRCA1 testing for chil-dren among 218 adults from a Utah-basedkindred who had received BRCA1 test re-sults. Results indicated that approximatelyone-fourth of the participants would permitBRCA1 testing for children under the age of18. General attitudes about genetic testingwere predictive of attitudes toward the test-ing of children. In addition, men and indi-viduals without a BRCA1 mutation weremore likely to agree that minors should beallowed BRCA1 testing. Individuals whosemother had been affected with breast can-cer were less likely to permit testing for mi-nors. Among parents of minor children, lessthan one-fifth indicated that they wouldwant BRCA1 testing for their own children;carrier status was not predictive of atti-tudes toward testing their own children. Asbreast/ovarian cancer susceptibility testingcontinues to be disseminated into clinical

settings, there may be an increase in thenumber of test requests for minors. Thefindings of the present study represent animportant step in exploring attitudes aboutgenetic testing of children among individu-als who have received cancer susceptibilitytest results. Am. J. Med. Genet. 92:25–32,2000. © 2000 Wiley-Liss, Inc.

KEY WORDS: ethics; patients’ attitudes;BRCA1 testing; children

INTRODUCTION

Advances in molecular biology and human geneticshave led to the identification of genes related to a va-riety of diseases and the development of tests to detectmutations in these genes. Recently, a number of mu-tations in two genes, BRCA1 and BRCA2, have beenshown to predispose women to breast and ovarian can-cer [Miki et al., 1994; Wooster et al., 1995]. With thedevelopment of high-risk cancer clinics and the com-mercial availability of BRCA1/2 tests, testing for mu-tations in these genes has recently moved from the re-search domain to the clinical realm. The rapiddissemination of this new technology increases the im-portance of addressing ethical and psychosocial consid-erations concerning breast/ovarian cancer susceptibil-ity testing.

One of the most controversial issues surrounding ge-netic testing for BRCA1/2 is whether such tests areappropriate for children. Unlike testing for cancersthat may have clinical manifestations and/or the op-portunity for medical management in childhood (i.e., LiFraumeni syndrome, retinoblastoma, familial adeno-matous polyposis, and Wilms tumor), BRCA1 andBRCA2 are generally thought to contribute only toadult cancers [Li et al., 1992; Offit, 1998]. Based ondata from the Breast Cancer Linkage Consortium

Grant sponsors: National Cancer Institute, NIH; Grant num-ber: CA 63681; Grant sponsor: the National Center for ResearchResources; Grant number: MO1-RR00064; Grant sponsor: NIH;Grant number 5-T32-NG00042-05.

*Correspondence to: Heidi A. Hamann, M.S., Department ofPsychology, University of Utah, 1530 East 390 South, Rm 502,Salt Lake City, UT 84112. E-mail: heidi.hamann@m.cc.utah.edu

Received 25 November 1999; Accepted 20 January 2000

American Journal of Medical Genetics 92:25–32 (2000)

© 2000 Wiley-Liss, Inc.

(BCLC), it has been estimated that the cumulativerisks for women with BRCA1 mutations vary with agefrom approximately 16% by age 40, to 59% by age 50,and 82% by age 70 [Easton et al., 1993, 1995; Ford etal., 1994]. Recent studies have documented lower pen-etrance for carriers with differing ascertainment and/or subset of mutations [e.g., Ford et al., 1998; Levy-Lahad et al., 1997; Struewing et al., 1997]. Sur-veillance techniques such as clinical breast examina-tion and mammography are advised to begin no earlierthan age 25 for individuals found to have BRCA1/2mutations [Burke et al., 1997]. Thus, having informa-tion about one’s BRCA1/2 mutation status in childhoodis not considered medically necessary for disease pre-vention or early detection.

BACKGROUND

The issue of testing children for BRCA1/2 mutationscan be placed within the broader context of testingasymptomatic minors for genetic conditions that aretraditionally manifested in adulthood. The ethics ofsuch procedures were debated when testing for Hun-tington disease (an adult-onset neurodegenerative dis-order) became available, and have now been applied todiscussions of cancer susceptibility testing. It has beenargued that in the absence of medical benefit, offeringpredisposition genetic testing to a child could compro-mise his or her freedom to choose whether or not toobtain this genetic information in adulthood [see Wertzet al., 1994, for discussion]. In addition, there is con-cern that the dissemination of mutation status willcompromise a child’s access to insurance and/or educa-tion. Testing could also cause adverse psychologicalconsequences for children, such as impaired self-esteem, stigmatization within the family, and anxietyrelated to hypervigilant symptom monitoring [Clayton,1995; Michie and Marteau, 1996].

On the other hand, several arguments can be pro-posed in favor of testing children for adult-onset con-ditions. Some have cited research in developmentalpsychology and decision-making to argue that olderadolescents (ages 15–17) generally have the compe-tence and maturity to make testing decisions for them-selves and should not be forced to wait until adulthoodto seek testing [Binedell et al., 1996]. One might alsoargue that minors should not be denied potential psy-chological benefits of genetic testing, such as knowl-edge to prepare for the future and the relief of anxietyassociated with learning that one is not a gene muta-tion carrier [Michie and Marteau, 1996].

The importance of legal and legislative factors in de-cisions about testing children for adult-onset condi-tions has been debated in the literature, and some haveargued that medical and legal standards support thedecision-making authority of parents. In the Americanmedical system, parents traditionally have had broaddiscretion in making decisions about their children’smedical treatment [Wertz et al., 1994; ASHG/ACMG,1995]. One point of view is that the law gives parentsprimary authority to obtain genetic tests for their chil-dren, and physicians may be found legally liable if theyrefuse a parent’s request [Pelias, 1994]. However, Clay-

ton [1995] argued that the law does not dictate thatphysicians have a responsibility to perform genetictests on children solely at parents’ requests. Clayton[1995] notes that as long as the potential informationgained from a genetic test does not affect medical man-agement, physicians can decline to test a child withoutrisk of liability. Furthermore, medical providers havethe right to refuse requested treatments when profes-sional judgment indicates that the costs and risks out-weigh the benefits [Clarke, 1994; Clayton, 1995].

A number of statements by medical organizationshave addressed the controversy. In 1995, the Board ofDirectors of the American Society of Human Geneticsand the American College of Medical Genetics issued ajoint report addressing the issue of genetic testing forchildren under the age of 18 [ASHG/ACMG, 1995].While they acknowledged that genetic testing for a“competent adolescent” may be justified based on po-tential psychosocial benefits, they also stated that, “Ifthe medical or psychosocial benefits of a genetic testwill not accrue until adulthood, as in the case of carrierstatus or adult-onset diseases, genetic testing gener-ally should be deferred” [ASHG/ACMG, 1995]. In theUnited Kingdom, the Working Party of the Clinical Ge-netics Society, based on internal consultations and sur-veys of health professionals, indicated that “predictivetesting for an adult onset disorder should generally notbe undertaken if the child is healthy and there are nomedical interventions established as useful that can beoffered in the event of a positive test result” [Clarke,1994]. However, not all professional organizations havesupported the general view that predispositional test-ing should not be performed on children in the absenceof medical benefit. For example, in 1995 the NationalSociety of Genetic Counselors (NSGC) passed a resolu-tion stating that with counseling, proper information,and caution, parents may be able to make testing de-cisions on behalf of their children [NSGC, 1995a,b].

In addition to statements by medical professional or-ganizations, a government-sponsored task force hasaddressed the controversy. The Task Force on GeneticTesting, which was commissioned by the National In-stitutes of Health — Department of Energy WorkingGroup on Ethical, Legal, and Social Implications of Hu-man Genome Research, issued a document entitled“Promoting safe and effective genetic testing in theUnited States.” In this statement, the Task Force de-termined that, “Genetic testing of children for adultonset diseases should not be undertaken unless directmedical benefit will accrue to the child and this benefitwould be lost by waiting until the child has reachedadulthood” [Task Force on Genetic Testing, 1997].

While statements by professional organizations andgovernment task forces are influential, such recom-mendations may not parallel medical professionals’and consumers’ beliefs and practices. Several research-ers have interviewed relevant groups about testing mi-nors for adult-onset genetic diseases, with varying re-sults. For example, a study by Wertz and Reilly [1997]of genetic laboratories found that 28% had testedasymptomatic minors for Huntington disease. In addi-tion, 26% of the laboratory respondents indicated thatparents should have the right to request tests on behalf

26 Hamann et al.

of minor (under age 18) children even if the childrenobject. In a study of individuals at risk for Huntingtondisease, 35% of the participants indicated that theywould want at-risk minors to be tested [Meissen andBerchek, 1987]. Patenaude et al. [1996] asked mothersof pediatric oncology patients whether they would wantan unaffected child tested for cancer susceptibility. Theresearchers did not specify the age of onset or type ofcancer about which their hypothesized test would pro-vide information. Their results indicated that 42% ofthe mothers would want their healthy children tested,even in the absence of medical benefit for the child. Ina study of high-risk women, physicians, and nursepractitioners, Geller et al. [1998] found that approxi-mately one-third of each surveyed group would agree totesting a 13-year-old daughter of a woman with an in-herited breast cancer susceptibility gene. Benkendorfet al. [1997] surveyed unaffected first-degree femalerelatives of women with breast or ovarian cancer andfound that 88% thought parents should be able to con-sent to BRCA1 and BRCA2 genetic testing for theirchildren. Specifically, African American women andwomen who reported higher levels of optimism weremore likely to favor parents’ rights to consent for thegenetic testing of minors.

Although the previous research is important for de-lineating attitudes of high-risk individuals, health careproviders, and laboratories toward genetic testing foradult-onset conditions, most of the findings are basedon responses to hypothetical scenarios presented to in-dividuals who have not experienced genetic testing. Toour knowledge, no investigators have inquired aboutthis issue to individuals who have been tested and re-ceived results for cancer susceptibility gene mutations.It is important to survey these individuals, especiallythose who test positive, because they represent a popu-lation for which this controversy is directly relevant. Inaddition, it is important to gauge the ways in which therevelation of one’s own test result influences views ontesting for his or her own children and among minors ingeneral. The following study focuses on attitudes to-ward BRCA1 testing for children among individualswho have received test results for a family-specificBRCA1 mutation.

MATERIALS AND METHODSParticipants

The present study reports on 218 individuals whowere tested for a BRCA1 mutation, learned their mu-tation status from a certified genetic counselor, andparticipated in a telephone follow-up interview ap-proximately 4–7 months after receiving their genetictest results (M 4 137.76 days; range 4 107–225 days).Participants in this study had been recruited as part ofa larger study of an extensive Utah-based kindred(K2082) of Northern European descent with a knowncancer-susceptibility mutation in BRCA1 [Goldgar etal., 1994; Botkin et al., 1996]. Women in this kindredhave a high risk of ovarian cancer (approximately 90%by age 80) and a later age of onset of breast cancer thanthe figures from the BCLC [Miki et al., 1994; Easton etal., 1993; Ford et al., 1994; Botkin et al., 1996].

Of the 218 study participants, 133 were women and85 were men. Ages at the time of the baseline interview(prior to genetic counseling and testing) ranged from 18years to 82 years (M 4 46.92). The majority of theparticipants were married (n 4 181, 83.0%), had atleast some post-high school education (n 4 179,82.1%), and reported incomes of $30,000 or more (n 4149, 68.4%). Most (n 4 207, 95.0%) of the participantsreported their religious affiliation as Church of JesusChrist of Latter-day Saints (Mormon). Nearly half (n 4104, 47.7%) of the participants reported that they hadchildren under the age of 18. Overall, 49 women and 29men were found to carry the deleterious BRCA1 muta-tion.

Procedure

The overall study procedure is detailed elsewhere[Botkin et al., 1996]. To summarize, family membersage 18 and over were contacted by letter and invited toparticipate in a study that offered free genetic counsel-ing and genetic testing. Letters were first sent to theoldest generations of the family in the branches thathad previously participated in genetic linkage re-search. If the older family members consented to par-ticipate, additional members were then contacted. Let-ters were sent to adult children of individuals whodeclined to participate only if consent had been grantedby the parent. The participants who had agreed to par-ticipate were then contacted by telephone, mailed con-sent forms, and scheduled for baseline interviews. Af-ter the baseline interview, individuals met with agenetic counselor and a marriage and family counselor.Participants received structured, detailed genetic edu-cation and counseling as part of the study protocol.Details of the counseling issues and challenges associ-ated with this particular study are described elsewhere[Baty et al., 1997]. Individuals were then given thechoice to have their blood drawn for the mutationanalysis. Those who consented and wanted their re-sults were seen in the results session by both a geneticcounselor and a mental health professional.

For individuals who received genetic test results, thefirst telephone follow-up interview was conducted ap-proximately 1 week after the receipt of test results.Subsequent telephone interviews for all who receivedtest results occurred approximately 4–7 months, 1year, and 2 years after the receipt of test results. Thepresent analysis utilizes information from the baselinemeasurement and the 4–7 month telephone interviewof individuals who were tested for the family-specificBRCA1 mutation and received their results from a cer-tified genetic counselor.

Measures: Dependent Variables

Opinions about genetic testing for minors. Atthe 4–7 month follow-up, participants were queriedabout their attitudes toward BRCA1 genetic testing forminors (children under age 18). Participants wereasked about their opinion regarding testing minors ingeneral: “The current recommendation from the fed-eral government is to limit BRCA1 testing to adults 18years and older. If you were deciding, do you think

Attitudes Toward the Genetic Testing of Children 27

children under 18 should be given the opportunity to betested for the BRCA1 gene?” As a follow-up question,individuals who indicated at the 4–7 month follow-upthat they had children under the age of 18 were asked:“If you were deciding now, would you want your chil-d(ren) to be tested for the BRCA1 gene while they areunder age 18?” The same questions were asked at boththe 1-year and 2-year follow-up intervals, but thesedata are not yet available for analysis.

Measures: Predictor Variables

Demographic variables. The surveys adminis-tered as part of the overall study included an extensiveset of demographic measures, psychological distressscales, perceived cancer risk scales, family information,and health history data. The measures utilized in thecurrent study were taken from both the baseline inter-view (prior to genetic counseling and testing) and theinterview conducted 4–7 months after the receipt ofgenetic test results. Data on age, education, and familyincome were obtained in the baseline survey. Measuresof marital status and the presence of children underthe age of 18 in the household were obtained in the 4–7month follow-up survey.

General attitudes toward genetic testing. Atthe 4–7 month interview, participants were asked, “Ifyou think about all the positive and negative changesthat may happen to families who are tested for theBRCA1 gene, which would you say is larger — the posi-tive changes or the negative changes?”

General psychological distress. The 20 itemState Anxiety scale of the State-Trait Anxiety Inven-tory Form X [Spielberger et al., 1970; Spielberger,1983] was administered at both the baseline and 4–7month follow-up interviews. The STAI is a well-validated and widely utilized measure of current, gen-eral distress. For the data presented here, the Cron-bach’s alpha for the STAI measure was .92 at baselineand .93 at the 4–7 month follow-up.

Test-related distress. The Impact of Event Scale[IES; Horowitz et al., 1979; Zilberg et al., 1982] wasadministered and evaluated in the 4–7 month inter-view. The IES is a 15-item scale that measures event-related distress. The IES yields a total score and sub-scores for two subscales of intrusion and avoidance. Forthe present data, the IES had a Cronbach’s alpha of.88.

Breast/ovarian cancer or cancer-related sur-gery history. Personal history of cancer or relatedsurgery was assessed by self-report at the baseline in-terview. Female study participants were asked if theyhad ever been told by a physician that they had beendiagnosed with breast or ovarian cancer. In addition,they were asked if they had ever had their breasts orovaries removed. Female participants who answeredyes to either question were considered as havingbreast/ovarian cancer or related surgical history. Forsome of the operations reported by women with no re-ported cancer history, it was not known whether theprocedure was prophylactic or was prompted by cancersigns and symptoms. Some oophorectomies may alsohave been performed as part of a complete hysterec-tomy.

Family cancer history. Family cancer historywas assessed by self-report at the baseline interview.Participants reported the number of first- and second-degree family members diagnosed with cancer. Theyalso indicated whether their mothers had been diag-nosed with either breast or ovarian cancer.

Personal BRCA1 mutation status. Mutationstatus was first assessed by participant self-report atthe 4–7 month interview. Each self-reported mutationstatus was then verified by the laboratory genetic test-ing report. Participants were divided into mutationcarriers and mutation noncarriers based on their re-port and laboratory confirmation of test results.

Statistical Analyses

The relationships between the predictor variablesand dependent variable were assessed by bivariateanalyses. Chi-square statistics evaluated categoricalpredictors and t-tests were used for continuous predic-tor variables. Logistic regression was used to modelattitudes toward the testing of children. Predictor vari-ables that were correlated with the given outcome vari-able at P # 0.15 in the bivariate analyses were enteredinto the logistic regression equation. For analyses inwhich there are few related studies to provide theoret-ical or empirical precedent, a more liberal criterion forinclusion in the multivariate analysis is often sug-gested [Tabachnick and Fidell, 1996].

RESULTSAttitudes Toward the Testing of Minors

Of the 218 study participants, 57 (26.1%) agreedwith the statement that children under age 18 shouldhave the opportunity to be tested for BRCA1 gene mu-tations, 133 (61.0%) did not agree, 21 (9.6%) either re-sponded that they were not sure or it depends, and 7(3.2%) either did not know or declined to answer thequestion. To allow analyses with a dichotomous out-come, these groups were divided into those who an-swered “yes” to the question of whether children under18 should be able to be tested for BRCA1 mutations (n4 57), and those who did not answer “yes” (this in-cludes the no, depends, and don’t know/reject groups; n4 161).

As indicated in Table I, results of chi-square analy-ses indicated that male sex, negative mutation status,not having children under 18, and not having a motherwho has had breast cancer were associated with per-mitting testing for children under 18 (P # 0.15). Inaddition, individuals who perceived mostly positivechanges associated with testing were more likely topermit testing for minors. T-tests showed that lowerlevels of test-related distress (measured by the IES) atthe 4–7 month follow-up were also associated with sup-porting the testing of minors (P # 0.15). When thesevariables were entered in a multivariate logistic re-gression, analyses showed that perceiving positivechanges associated with genetic testing, being male,being a noncarrier, and not having a mother withbreast cancer were predictive of permitting testing forchildren under 18 (P # 0.05; Table II). The other pre-dictor variables included in the logistic regression

28 Hamann et al.

equation were not significantly related to the outcomevariable.

Attitudes Toward the Testing of Own Children

Among the 218 study participants, 104 reported hav-ing children under the age of 18. Of these 104 individu-als, only 18 (17.3%) noted that they would want theirown children tested for a BRCA1 mutation, while 86

(82.7%) did not endorse this statement. There was nosignificant difference between carriers and noncarriersin their support of testing for their own children, x2 (1,n 4 104) 4 0.29, P 4 0.59. A McNemar’s test of relateddichotomous variables indicated that, among partici-pants with minor children, there was no significantdifference in support for testing children in general ascompared with support for testing their own children(P 4 0.58). Among the group with minor children, eightindividuals (7.7%) who permitted testing for minors ingeneral did not endorse it for their own children. Fiveindividuals (4.8%) did not support testing for minors ingeneral, but wanted their own children tested. The restof the participants (n 4 91; 87.5%) were consistent intheir responses to both questions.

DISCUSSIONOverall, approximately one-fourth of the genetic test-

ing participants in this study indicated that theythought BRCA1 testing should be available for minors.When compared to the results of the study by Benken-dorf et al. [1997], which surveyed unaffected first-

TABLE II. Logistic Regression Analyses Predicting Support forBRCA1 Genetic Testing in Minors

Variable B SE P

General attitude toward genetictesting (positive) 1.49 0.57 0.01

Sex (male) 0.68 0.34 0.04Absence of children under 18 0.30 0.17 0.08Carrier status (mutation noncarrier) 0.38 0.19 0.05No maternal history of breast cancer 1.39 0.57 0.02IES at 4–7 months −0.01 0.02 0.42

STAI 4 State-Trait Anxiety Inventory; IES 4 Impact of Event Scale; B 4regression coefficient; SE 4 standard error.

TABLE I. Results of Bivariate Analyses for Permitting BRCA1 Genetic Testing in Minors†

Variable

Support for BRCA1genetic testing in minors (n 4 218)

% Endorsing x2 n

General attitude toward testingPositive 29.4 5.82* 180Not Positive 10.5 38

SexMale 32.9 3.33* 85Female 21.8 133

Marital statusMarried 26.0 0.02 181Unmarried 27.0 37

Hx of br/ov ca. or related surgerya (females only)Yes 18.4 0.36 38No 23.2 95

Children under 18 in householdYes 20.2 3.65* 104No 31.6 114

Carrier statusCarrier 16.7 5.65** 78Noncarrier 31.4 140

Maternal history of breast cancerYes 10.5 5.82** 38No 29.4 180

Maternal history of ovarian cancerYes 31.5 1.05 54No 24.4 164

Variable

M ± SD

df tbYes Not yes

Age (in years) 47.2 ± 16.5 46.8 ± 15.2 215 0.18Education (in years) 13.9 ± 2.1 13.9 ± 2.0 216 0.10Family income (in thousands of dollars) 42.4 ± 23.3 45.0 ± 24.4 207 0.69STAI at baseline 31.2 ± 9.4 31.6 ± 9.3 216 0.28STAI at 4–7 month 28.2 ± 8.9 29.1 ± 9.1 216 0.62IES at 4–7 month 7.2 ± 8.8 9.4 ± 10.7 215 1.44*Number of relatives with br/ov ca 1.8 ± 1.3 1.5 ± 1.3 216 1.33

†For all chi-square analyses, df 4 1. IES 4 Impact of Event Scale; STAI 4 State-Trait Anxiety Inventory; br/ovca. 4 breast or ovarian cancer.aHistory of breast/ovarian cancer or related surgery assessed for women only.bAbsolute value.*P # 0.15**P < 0.05.

Attitudes Toward the Genetic Testing of Children 29

degree relatives of breast cancer patients, our findingsshowed significantly less support for testing minors.We found a smaller percentage of respondents in thecurrent study who would permit minors to be testedcompared to the Patenaude et al. [1996] study of moth-ers of pediatric oncology patients and the Geller et al.[1998] study of at-risk women, physicians, and nursepractitioners. One potential explanation for this find-ing is that the individuals who have experienced ge-netic counseling and testing themselves may be moreaware of the possible consequences and limitations oftesting than those who have not been counseled andtested. Benkendorf et al. [1997] assessed attitudes to-ward the genetic testing of minors before individualshad participated in a genetics education session and, toour knowledge, nobody in their sample had receivedgenetic testing. Also, the reports by Patenaude et al.[1996] and Geller et al. [1998] did not indicate that theparticipants had received genetic counseling and/ortesting. In the present study, part of the genetic coun-seling process explored the potential risks and benefitsof genetic testing, including insurance and employmentissues, health care management, and possible psycho-logical consequences [Baty et al., 1997]. In addition,with the act of testing study participants were able toexperience for themselves personal and familial conse-quences of testing. Awareness of and experiences withthese issues, especially limitations and negative conse-quences of testing, may have facilitated a greater hesi-tancy to permit children access to the informationgained from testing.

The actual wording of the research questions mayhave also played a role in differences between studies.While Benkendorf et al. [1997] gauged responses to thestatement, “Parents should be able to decide if theirminor children (under age 18) should have a genetictest,” the present study inquired, “If you were deciding,do you think children under 18 should be given theopportunity to be tested for the BRCA1 gene?” In gen-eral, Benkendorf et al. [1997] addressed the issue ofparental right to make decisions for children while thepresent study asked whether children should be al-lowed to be tested. Both the Geller et al. [1998] andPatenaude et al. [1996] protocols inquired about a spe-cific situation (that of deciding whether to have a 13-year-old girl tested for a breast cancer susceptibilitymutation and having a real, nonaffected child testedfor a hypothetical genetic mutation, respectively). Ingeneral, varying responses to differently worded ques-tions indicate that testing minors is likely to be a mul-tifaceted issue.

As a final point, differences in demographic factorsmay have contributed to divergent findings of the stud-ies. Both the Benkendorf et al. [1997] and Patenaude etal. [1996] study populations were similar to those inthe present study in terms of educational, socioeco-nomic, and marital status. While our sample includedonly individuals of Northern European descent, Benk-endorf et al. [1997] included both African Americanand White women and found that White women weresignificantly less likely to support parental decisionsfor the testing of minors than African Americanwomen. Although more research is needed to investi-

gate this phenomenon, it is likely that complex culturalissues influence attitudes toward the genetic testing ofchildren.

As would be expected, individuals who perceivedmore positive changes associated with genetic testingwere more likely to support testing of children. Thiscorrelation indicates consistency in attitudes towardtesting in general as compared with the more specificquestion of the appropriateness for children. Resultsalso indicated that noncarriers were more likely to per-mit BRCA1 testing availability for minors. In essence,noncarriers received “good” news when told of their sta-tus, and studies focusing on emotional reactions toBRCA1/2 testing have found that noncarriers show sig-nificant decreases in distress and increases in reliefafter learning test results [Lerman et al., 1996; Croyleet al., 1997; Lynch et al., 1997]. Among noncarriers, thepersonal experience of positive emotions may havebeen associated with the belief that others, includingchildren, would also benefit from testing. However, itshould be noted that in the multivariate analysis of thepresent study, psychological distress was not a uniquepredictor of positive attitudes toward testing for mi-nors. Supplemental analyses also indicated no signifi-cant interaction between psychological distress andcarrier status.

Our analyses also found that men were more likely tosupport BRCA1 testing of children. Since BRCA1 mu-tations confer greater risks of cancer on women thanmen, men may not have perceived the informationgained from genetic testing to be as personally threat-ening as women may have. If the men did not identifyas many possible threats from testing, they may havebeen more likely to permit it for others, including chil-dren. In addition, individuals who did not have amother with breast cancer were more likely to supporttesting in children. It is possible that individuals whoobserved their mother suffer from the potentially dev-astating effects of breast cancer may have seen theinformation gained from testing as more threateningand were less willing to allow children to face thatthreat.

Although there has been debate about the ethics oftesting children for cancer susceptibility mutations, itis not clear how large a demand there is or will be forsuch a practice. The findings of our study indicatedthat the majority of tested individuals with minor chil-dren did not want their own children tested. Amongthose respondents with children under the age of 18,less than one-fifth supported testing for their own chil-dren, and most participants were consistent in theirattitudes toward testing children in general and test-ing their own children. Although children of noncarri-ers in our study would not need genetic testing to showthat they were not carriers themselves, test result sur-prisingly did not predict interest in testing one’s ownchildren. It is possible that some noncarriers may havemisunderstood concepts of genetic transmission or ne-glected to take them into account when answering inregards to their own children. This finding underscoresthe need for genetic education and counseling in ad-dressing any request for minor testing.

One limitation of this study is that we did not include

30 Hamann et al.

measures of attitudes toward testing children beforeparticipants were counseled and tested. Thus, al-though we can speculate about the effects of counselingand testing on attitudes toward the testing of minors,we cannot demonstrate that attitudes changed frompre- to post-testing. Another study limitation involvesthe generalizability of our sample to others receivingBRCA1/2 results. The participants are members of alarge kindred of Northern European descent, and themajority identify themselves as members of the Churchof Jesus Christ of Latter Day Saints (LDS or Mor-mons). Therefore, our study participants are not rep-resentative of the United States population in terms ofrace or religion. In regard to other factors, it has beenshown that devout Mormons are more likely to be mar-ried and have larger families and social networks thannon-Mormons [Heaton and Goodman, 1985; Smith andShipman, 1996]. However, as described in comprehen-sive volumes edited by Ludlow [1992], there are notonly many similarities between Mormons and nonMor-mons, but also varying degrees of religiosity and cul-tural practices among those who identify themselves asMormon. We did not evaluate religious or culturalpractices among our sample in the current analyses.Further discussion of religion and its relevance to thecurrent study population may be found in Botkin et al.[1996].

The findings from this study represent a first step inexploring attitudes toward the genetic testing of chil-dren among individuals who have received cancer sus-ceptibility test results. As BRCA1/2 testing becomesmore prevalent in the clinical realm, a greater numberof individuals who have received test results may seektesting for their children. Our results indicate that themajority of tested individuals did not support this prac-tice or desire such testing for their own children. Fu-ture studies are needed to further delineate this inter-est among tested individuals and other populations.Studies might also focus on documented requests forminor testing to examine the paths by which hypotheti-cal inquiries translate into parents’ requests and phy-sicians’ test practices.

ACKNOWLEDGMENTS

The authors thank Jean Nash, Debra Dutson, DianeLane, Tamra Frei, and Georgia Hatch for valuablecontributions to this project. John Ruiz provideduseful comments on an earlier draft of the article.Heidi A. Hamann is sponsored by an NIH GenomeScience Predoctoral Traineeship; grant number5-T32-NG00042-05.

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