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Effects of spouses on distress experienced by BRCA1 mutation carriers over time.

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American Journal of Medical Genetics Part C (Semin. Med. Genet.) 119C:35 – 44 (2003)
A R T I C L E
Effects of Spouses on Distress Experienced
by BRCA1 Mutation Carriers Over Time
JEAN E. WYLIE,* KEN R. SMITH, AND JEFFREY R. BOTKIN
Concerns about psychological distress have arisen regarding genetic testing for susceptibility to late-onset diseases
such as breast and/or ovarian cancer. Early results do not show large-scale psychological distress among those tested;
therefore, research is now focusing on identifying subgroups that may be at risk for negative outcomes. Social support
has been shown to buffer both negative physical and psychological outcomes in health research. The role of spouses as
part of the tested person’s social support system is shown to be significant in a sample of 57 BRCA1 mutation carriers.
Separately, the tested person’s perception of his/her spouse’s anxiety and his/her perception of the spouse’s support at
the time of testing are predictive of the tested person’s psychological distress up to 2 years after testing. The interaction
of the two variables is even more predictive. For those tested who perceived their spouse to be both anxious and
nonsupportive at the time of testing, distress levels reached clinically significant levels 1 week after results were
received and remained above clinical threshold measured 4 months, 1 year, and 2 years after testing. While the effects
were greatest for women, they were significant for both male and female carriers. These findings are an important
addition to the literature and will augment clinicians’ ability to identify individuals potentially at risk for negative
responses to adverse genetic test results. ß 2003 Wiley-Liss, Inc.
KEY WORDS: cancer; genetic testing; social support
INTRODUCTION
The discovery of the first breast and/
or ovarian gene that is associated with
breast and ovarian cancer susceptibility
(BRCA1) occurred in 1994 [Miki et al.,
1994]. This development added to an
ongoing discussion of concerns regarding the possible negative effects of presymptomatic knowledge of carrier status
for late-onset diseases such as cancer
[Huggins et al., 1992; Botkin et al.,
Jean E. Wylie is a Director of the in
Resource for Genetic and Epidemiologic
Research at the University of Utah in Salt
Lake City, Utah.
Ken R. Smith, Ph.D., is a Professor in the
Department of Family and Consumer Studies
at the University of Utah in Salt Lake City,
Utah.
Jeffrey R. Botkin, M.D., is a Professor in
the Department of Pediatrics at the University of Utah in Salt Lake City, Utah.
Grant sponsor: The National Institutes of
Health; Grant number: CA63681.
*Correspondence to: Jean E. Wylie,
Resource for Genetic and Epidemiologic
Research, 1C133 School of Medicine,
University of Utah, 30 North 1900 East, Salt
Lake City, UT 84132.
E-mail: jean.wylie@hsc.utah.edu
DOI 10.1002/ajmg.c.10002
ß 2003 Wiley-Liss, Inc.
1996; Tibben et al., 1997; DudokdeWit
et al., 1998a,b; Freedman, 1998]. Concerns were raised about employment
and insurance discrimination and about
possible negative psychological consequences of knowing one’s carrier status.
We report here on distress among carriers
of a BRCA1 mutation in a kindred where
the mutation confers an 88% risk of either
breast or ovarian cancer by age 70 [Smith
et al., 1999].
This analysis focused on the role
of social support (as a resource) in moderating the effects of positive genetic
test results for a BRCA1 mutation (as a
stressor) on psychological distress. Thoits
[1995] defined social support as a fund
from which people may draw. In this
definition, social support usually refers
to the functions performed for the
individual by significant others, such as
family members, friends, and coworkers.
Wethington and Kessler [1986] note,
however, that it may not be so much that
those functions are actually performed for
the stressed person as that the social fund
is perceived to be available to be drawn on
by that person.
In discussing the role of social support, Thoits [1995] notes that ‘‘the
simplest and most powerful measure of
social support appears to be whether a
person has an intimate, confiding relationship. . . . Having a confidant significantly reduces the effects of stress
experiences on both physical and psychological outcomes.’’ Among all possible sources of social support, spouses are
generally considered to be individuals’
single most important sources of support
[Wethington and Kessler, 1986; Thoits,
1995; Beach et al., 1996].
Yet, by virtue of being close to the
stressed person, a supporter often participates in the stressful situation directly.
In the case of genetic testing, when the
support person is also the spouse of the
tested person, he or she is also affected
by the stressor beyond the effect on the
tested (stressed) person [Kash, 1995]. For
example, the husband of a carrier wife
will not be genetically affected as she
may, but his children could be as a result
of inheriting an excess risk from their
mother. That is, the effect of his wife’s
test results on him are not limited to his
role as her support person but may also
extend to his role as father of their
children. In sum, we hypothesize that
spouses are also directly affected by the
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AMERICAN JOURNAL OF MEDICAL GENETICS (SEMIN. MED. GENET.)
stressor(vis-à-vischildren) and thisimpact
may have an important effect on how the
tested person copes with the effects of
testing [Marteau and Croyle, 1998].
Previous analyses suggest that the
adverse psychological consequences of
positive genetic test results for cancer
susceptibility are modest at best [Coyne
et al., 2000]. There are data, however,
that indicate that some individuals may
be more prone to negative sequelae
[Croyle et al., 1997; West, 1997; Smith
Previous analyses suggest that
the adverse psychological
consequences of positive genetic
test results for cancer
susceptibility are modest at best.
There are data, however,
that indicate that some
individuals may be more prone
to negative sequelae.
et al., 1999]. Accordingly, current
research in this area has begun to focus
on identifying subgroups within those
populations eligible for testing that may
be at greater risk for negative psychological consequences.
Most studies have found that although adverse consequences of genetic
testing are not as large as originally
feared, a positive relationship between
carrier status and psychological outcome
exists, with carriers having more postresult distress than noncarriers. Given
the potential health consequences for
women, it is not surprising that female
carriers show greater distress than male
carriers. One recent study found that
high pretest anxiety in mutation carriers was associated with high anxiety
at posttest [Lodder et al., 2001].
Manne et al. [2001] found that nonsupportive spouses exacerbate the psychological consequences for female
carriers [Manne et al., 2001]. Vernon
et al. [1997] found that increased anxiety was associated with less formal
education, fewer social contacts, and less
satisfaction with them.
Despite fears about the potential
negative psychological effects of genetic
test results and the acknowledged role of
social support as a coping resource for
individuals undergoing stress [Thoits,
1995], little attention has been paid to
the role of spouses in the genetic testing
context. Since there are few data yet
about the specific effects of spousal
support on a person tested for a BRCA1
mutation, we turn to extant research on
the role of spouses in cancer adjustment.
In discussing the effects of spousal
support on cancer patients’ adjustment
to disease and treatment, Manne and
Glassman [2000] point out that ‘‘the
relationship between the cancer patient
and his or her spouse appears to be a
particularly important determinant of
the patient’s psychological adjustment.’’
In a study of the effects of spousal support
on adjustment to breast cancer, Bolger
et al. [1996] found that psychological
support eroded over time and did not
alleviate distress. Indeed, they found that
support was withdrawn in response to
emotional distress, while it was provided in response to physical impairment.
Others have found that husbands have
a major influence on the wellbeing of
women at high risk for breast and
ovarian cancer [Pistrang and Barker,
1995; Coyne and Anderson, 1999].
Social support is acknowledged as
a factor in psychological outcomes of
cancer genetic testing [Baum et al.,
1997] and, as noted above, Vernon
et al. [1997] found that social support
was inversely related to depression and
anxiety postresults. To date, the only
study that examines the role of spouses in
an individual’s ability to cope with the
results of genetic testing is Manne et al.
[2001]. They found spouse support and
encouragement to be consistently inversely associated with participant distress,
although their sample was a mixture of
presymptomatic women and women
already diagnosed with cancer.
The implications of spouses for
genetic testing for a BRCA1 mutation
are profound [Richards, 1998]. The
spouse of a tested person is in a significantly different position from the
ARTICLE
spouse of the person with cancer: at
the time of testing, most tested persons
have no physical impairment (and may
never have). Psychological (emotional)
distress of the tested person is, however, a
real possibility for the spouse at that time.
Male carriers are not at risk of breast or
ovarian cancer, but can transmit the risk
to their daughters, as can female carriers.
Thus, test results have the potential to
create anxiety not only in the tested
person, but in male and female spouses as
well due to concerns for their children.
This direct participation in the stress
situation by both the tested person and
spouse may alter the tested person’s
support system as well as perceptions of
support availability, or both.
In an effort to identify specific
spousal characteristics that may predict
elevated test-related distress among mutation carriers, this study examined the
relationship of the tested person’s perceptions of spousal anxiety and support
at the time of testing to the tested
person’s distress over a 2-year time
period following the receipt of test
results. Our primary hypothesis is that
tested persons who are mutation carriers
and who perceived their spouses to be
unsupportive and anxious will have
higher levels of posttest distress than
carriers whose spouses were perceived as
supportive or not anxious.
Our primary hypothesis is
that tested persons who are
mutation carriers and
who perceived their spouses to
be unsupportive and anxious
will have higher levels of
posttest distress than carriers
whose spouses were perceived as
supportive or not anxious.
MATERIALS AND METHODS
Sample and Procedures
Data were gathered during a study of
the behavioral and psychosocial effects
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AMERICAN JOURNAL OF MEDICAL GENETICS (SEMIN. MED. GENET.)
of testing for a BRCA1 mutation in a
single extended kindred [Botkin et al.,
1996]. Members of the kindred (K2082)
are primarily Caucasian and of North
European descent. The majority live in
Utah and are members of the Church
of Jesus Christ of Latter-day Saints
(Mormon). Excluded from the study
were individuals under 18, those not
competent to give consent, and those
living too far from the University of
Utah to attend the two genetic counseling sessions. Because of a top-down
(within a family pedigree), rolling recruitment of subjects, some potential
subjects who were known not to be
at risk of carrying the mutation due to
knowledge of their parent’s or grandparent’s test results were not invited into
the study. However, some subjects
whose parent/grandparent was found
to be a noncarrier were recruited into
the study prior to that information
being available. The study protocol was
approved by the University of Utah
Institutional Review Board.
Complete details of the study protocol for contact and consent are presented elsewhere [Botkin et al., 1996]
and thus are only briefly reviewed here.
Initial contact letters were sent to 759
individual members of the kindred;
408 completed a baseline interview. Of
those, 296 had genetic counseling and
269 were tested. The results were 89
carriers, 154 noncarriers, and 16 people
who did not receive their results. Those
who received their results from a genetic
counselor were interviewed 1 to 2 weeks
postresults. (Some individuals had blood
drawn for testing, but found out that
their parent was negative and chose not
to return for results. Interviews were
conducted with those individuals 1 to
2 weeks after receipt of their parent’s
information when possible.) All subjects
were interviewed 4 months, 1 year, and
2 years after results (or after the baseline
interview for those individuals who did
not have testing).
Our analysis was based first on
203 married individuals who completed the baseline, 1-year, and 2-year
follow-up interview. Our analyses
focused on 57 married carriers who fit
these criteria. Most but not all of this
sample also completed the 1-week
interview. Subjects who learned their
genetic status by virtue of their parent’s
or grandparent’s negative carrier status
did not participate in the 1-week
interview.
Measures
Table I shows the measures that were
used and at which point in the study they
were administered. We thus test our
hypotheses regarding the effects of perceived spousal support and anxiety on
TABLE I. Measures Used in Analyses
All married tested persons (n ¼ 203)
Baseline
Dependent variable
IES
Independent variables
Age
Sex
Education
Number of children
BRCA1 mutation status
Anxiety of spouse before results
Anxiety of spouse after results
Support of spouse before results
Support of spouse after results
1 week
4 months
1 year
2 years
X
X
X
X
X
X
X
X
X
X
X
X
X
X
X
X
X
X
37
distress of all married tested persons,
where we rely on data provided by the
person tested.
Dependent variable
Distress related to testing was measured by the Impact of Events Scale (IES)
[Horowitz et al., 1979]. The IES is a 15item scale that measures event-specific
distress. The IES was chosen for this
study for its established reliability; it has
been used extensively in many domains of the social sciences, including
genetic testing for late-onset diseases [Lerman et al., 1995, 1997; Tibben
et al., 1997; DudokdeWit et al., 1998a,b;
Lodder et al., 2001]. It was measured at
all postresults interviews. The Cronbach
alpha at 2 years was 0.903.
Independent variables
Carrier status. DNA testing was
done by the DNA Diagnostic Laboratory at the University of Utah using
blood samples provided by the subjects at
the first genetic counseling session
[Botkin et al., 1996]. Results were
provided to the project, and the subject
was provided the results in the second
genetic counseling session. It was possible, however, for some subjects to learn
that they were not carriers by virtue of
their parent’s (or grandparent’s) carrier
status being found to be negative. In this
case, some subjects opted not to have
both genetic counseling sessions, or just
the results session. In follow-up interviews, subjects were asked a series of
questions to determine their knowledge
of their mutation status. (Subject’s genetic test results were returned to the
project in a sealed envelope. Those envelopes were only opened immediately
prior to being given to the genetic
counselor in preparation for the results
session. The counselors had asked to
be advised of any inconclusive results
before the results session. No record was
kept of the results until the counselor
reported them back to the project staff
after the results session. Therefore, the
results of those subjects who did not
return for a results session are still unknown to project staff.) Thus, although
DNA test results that had been provided
to subjects were available to project staff,
38
AMERICAN JOURNAL OF MEDICAL GENETICS (SEMIN. MED. GENET.)
all analyses are based on the subject’s
perception of his or her genetic status
(or of the genetic status of the spouse).
The subject and project versions of
results were divergent in only one case.
Spousal anxiety/distress. Perceptions
of spouse anxiety were measured by two
seven-point Likert scale questions asked
at the 1-week postresults interview: ‘‘On
a scale from 1 to 7, how anxious was your
(husband/wife) between the time you
were tested and when you learned your
results, where 1 is not very anxious and
7 is very anxious?’’ and ‘‘On a scale
from 1 to 7, how anxious was your
(husband/wife) between the time you
learned your results and now, where 1 is
not very anxious and 7 is very anxious?’’
These two time periods will be referred to as the before-results and the
after-results. Our analyses take advantage of both sets of data, but focus on the
after-results, as assessed at the 1-week
postresults interview.
Similar to the questions for perceived spousal anxiety, perceived spousal
support was assessed with two sevenpoint Likert scale questions: ‘‘On a scale
from 1 to 7, how supportive was your
(husband/wife) between the time you
were tested and when you learned your
results, where 1 is not very supportive
and 7 is very supportive?’’ and ‘‘On a
scale from 1 to 7, how supportive was
your (husband/wife) between the time
you learned your results and now, where
1 is not very supportive and 7 is very supportive?’’ As above, our analyses focus
on the after-results.
Family cancer history. Subjects were
asked about the number of first- and
second-degree female relatives they had
who had had breast and/or ovarian
cancer. Others have reported that awareness of risk may affect the tested person’s
distress subsequent to testing [Baum
et al., 1997; Marteau and Croyle,
1998]. We considered this measure in
our analyses to control for the chance
that the effect of spousal anxiety and
support on distress in mutation carriers may be confounded by family
cancer history.
Demographic variables. These variables included age (in years), gender,
number of children, and education
ARTICLE
(in years). Age was considered in the
analysis because the disease, while occurring as early as the late 20s in this
family, is more likely to appear after
40; accordingly, distress related to being
a carrier may be associated with age.
Age is also associated with perceptions
of and satisfaction with one’s spouse.
Gender was incorporated into the model
due to the different health effects for
women versus men and differences
by gender of psychological distress.
Children were considered due to their
potential role in spousal responses to test
results. Education was considered given
that less formal education may be associated with higher scores on depression
measures among persons undergoing
genetic testing [Vernon et al., 1997].
Education is also positively associated
with marriage and spouse satisfaction.
Analyses
Statistical Analysis Systems (SAS) software was used for all statistical analyses.
Sex and carrier status (carrier or noncarrier) were used as dichotomous variables while education, age, and family
cancer history were analyzed as continuous variables.
Perceived spousal support and anxiety were evaluated as direct (main)
effects and then in combination with
each other (interaction effects). Given
that the distributions for perceived
spousal anxiety and support variables
were skewed toward extremes of the
scale (i.e., little anxiety and ample support), we categorized these two variables
into high/low dummy variables. When
perceived anxiety is 3 or higher (1 is
lowest and 7 highest), it was labeled high
and all others were labeled low. When
perceived support equals 7 (1 is lowest
and 7 highest), it was designated as high
and all others were labeled low. Fortythree percent of the subjects reported
spouse anxiety as 3 or higher (high), and
49% reported spouse support as 6 or
lower (low).
Finally, we created a spouse response profile that grouped persons into
one of four categories derived from the
combination of high and low perceived
spousal anxiety and support. These four
categories are defined in Table II.
We hypothesized that the ‘‘burdensome’’ spouse would be associated with
the highest levels of distress in the tested
person, whereas the ‘‘supportive’’spouse
would be associated with the lowest
levels of posttest distress. The other two
groups are predicted to have intermediate levels of distress. In the results,
we compare the first three groups with
the ‘‘supportive’’ group.
Ordinary least squares linear (OLS)
regressions were used to assess the effects
of the independent variables on distress, as measured by the IES. Due to the
relatively small sample size, special care
was taken to keep the model as parsimonious as possible. Preliminary analyses (not shown) indicated that age,
education, children, and family cancer
history did not show any effects and
were thus removed from the model.
Gender was an important predictor
and was retained in all models. Finally,
in order to determine how stable the
results were over time, all analyses
were run using the IES of the tested
person on all four postresults questionnaires, at 1 week, 4 months, 1 year, and
2 years posttesting.
TABLE II. Spouse Response Profile
Spouse response profile typology
Perceived anxiety
High
Low
High
Low
Perceived support
Descriptive label
n
Low
Low
High
High
‘‘Burdensome’’
‘‘Detached’’
‘‘Involved’’
‘‘Supportive’’
9
16
16
16
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AMERICAN JOURNAL OF MEDICAL GENETICS (SEMIN. MED. GENET.)
Several sets of siblings and a few
parent-child clusters are represented
in the sample. This raises a question
about the possible dependence in responses among relatives. We took into
account these dependencies by reestimating our regression models using
generalized estimating equations (GEE)
[Zeger and Liang, 1986]. Our results
based on the GEE approach were nearly
identical to those using the OLS regression approach. Accordingly, we report
results based on the OLS estimates.
Four basic linear regression models
were estimated to predict distress levels
among married persons who were tested. Table III summarizes the specification of these linear regression models,
which vary by the manner in which
the spouse perception variables are included. Model 1 estimated main and
interaction effects of support and anxiety on distress. Model 2 includes the
main effects of support and anxiety and
their interaction with gender. Model
3 includes gender and the spouse response profile dummies, and model 4
adds their interaction terms. For the
main effects, we include the predicted
association with test-related distress in
Table III.
RESULTS
Table IV shows the means, standard
deviations (SD), and ranges for all variables used in the analyses according to
each interview. The combined sample
of married carriers and noncarriers
(n ¼ 203) was 60% female, with a mean
age of 45.27 (range, 20–77) and an
average of slightly more than 2 years
of post-high school education. Twenty
eight per cent of the sample of married tested persons (n ¼ 57 of 203) were
mutation carriers; it is these carriers that
are the focus of our analyses. The mean
number of children per tested person
was almost four. Persons in the sample
had an average of 1.55 first- and/or
second-degree female relatives with
breast and/or ovarian cancer.
Results of the first regression
model, for married carriers who reported their perceptions of their spouses’
anxiety and support in the period between receiving their results and the
1-week interview (after-results), were as
hypothesized. The effects of perceived spouse anxiety are in the direction
predicted: higher perceived spousal anxiety significantly leads to higher distress of the tested person (P < 0.023 at
all time points). High perceived support also shows the predicted inverse
relationship with distress of the tested person (P < 0.05 for all time points).
The effects of perceived spouse
anxiety are in the
direction predicted: higher
perceived spousal anxiety
significantly leads to higher
distress of the tested
person. High perceived support
also shows the predicted
inverse relationship with
distress of the tested person.
The interaction term is not significant at
any measurement point.
An examination of the spouse response profile (Fig. 1) is revealing. The
effect size of the high-anxiety/lowsupport group (‘‘burdensome’’) is quite
large relative to the low-anxiety/highsupport group (‘‘supportive’’) at all four
time points and exceeds the mean IES
TABLE III. Summary of Linear Regression Models
Independent variables
Main effects
Gender (male)
High/low perceived spouse support
High/low perceived spouse anxiety
Spouse response profile
High anxiety, low support (‘‘burdensome’’)
High anxiety, high support (‘‘involved’’)
Low anxiety, low support (‘‘detached’’)
Low anxiety, high support (‘‘supportive’’), omitted group
Interactions
Spouse support spouse anxiety
Spouse support gender (male)
Spouse anxiety gender (male)
Spouse response profile
Burdensome gender (male)
Involved gender (male)
Detached gender (male)
39
Model 1
Model 2
Model 3
Model 4
X
X
X
X
X
X
X
X
X
X
X
X
X
X
X
X
X
X
X
X
1
77
18
9
45
40
49
40
7
7
7
0
20
7
0
0
0
0
0
0
1
1
0.49
13.66
2
2.24
8.90
8.14
8.69
7.97
1.32
0.84
1.37
0.41
46.14
14.25
4.06
7.98
5.79
7.26
5.69
1.44
1.31
6.32
1
76
18
9
51
40
45
37
5
6
7
0
25
12
0
0
0
0
0
0
1
3
0.49
13.56
1.69
2.09
12.36
11.22
10.82
10.64
1.24
1.52
1.25
0.39
43.04
13.91
3.68
13.98
12.21
13.11
11.16
1.82
2.54
6.12
1
77
18
9
51
40
49
40
7
7
7
0
20
7
0
0
0
0
0
0
1
1
0.40
45.27
14.16
3.96
10.0
7.64
8.94
7.23
1.55
1.73
6.25
Male
Age
Education (years)
Number of children
IES1 week
IES4 months
IES1 year
IES2 years
Number of female relatives with cancer
Perceived spouse anxiety immediately after results
Perceived spouse support immediately after results
0.50
13.67
1.92
2.20
10.54
9.56
9.70
9.22
1.31
1.26
1.33
Maximum
Minimum
SD
Minimum
SD
Minimum
SD
Mean
Variables
Total (n ¼ 203)
Maximum
Mean
All carriers (n ¼ 57)
Maximum
Mean
All noncarriers (n ¼ 146)
AMERICAN JOURNAL OF MEDICAL GENETICS (SEMIN. MED. GENET.)
TABLE IV. Means, Standard Deviations, and Ranges for All Variables for Married Subjects
40
ARTICLE
scores of individuals recently diagnosed with cancer [Epping-Jordan et al.,
1994]. The differences between the
other two groups (high-anxiety/highsupport or ‘‘involved’’ and low-anxiety/
low-support or ‘‘detached’’), relative to
the ‘‘supportive’’ group, are generally
significant at P < 0.05, although they do
not reach the level of distress associated
with a recent diagnosis of cancer.
The interaction between gender
and perceived spouse anxiety is significant for all four time points (P < 0.07).
In general, carrier women with anxious husbands experience more distress than carrier men with anxious
wives. The spouse response profile
shows that the distress differences between the women who perceive their
spouses as high anxiety/low support
(‘‘burdensome’’) and women who perceive their sspouses as low anxiety/
high support (‘‘supportive,’’ the omitted
group) are significant (P < 0.001) at the
1-week interview (25.4 vs. 4.0) and remain so at the 2-year measurement
period (32.8 vs. 6.5). A similar pattern
is detected for males but the differences
are attenuated relative to those reported for women. The other two groups
of women (those with ‘‘detached’’ or
‘‘involved’’ husbands) show smaller effect
sizes over time.
Figure 2 shows the differences in
mean IES scores for men and women
over the study period. With the exception of those with ‘‘supportive’’ spouses,
none of the mean scores for the male
grouping reach even the lowest mean
score for the groups for women. Furthermore, the IES scores of women who
perceive their spouses to be anxious and
unsupportive generally increase over the
2 years. Scores for the other women, as
well as for all the male groups, generally
decline over time.
Most importantly, scores for
women in ‘‘burdensome’’ couples, at
all four time points, exceed the mean IES
scores of individuals recently diagnosed
with cancer [Epping-Jordan et al.,
1994]. Epping-Jordan et al. [1994] used
the IES to measure distress approximately 10 weeks after cancer diagnosis;
the mean IES score for their sample was
22.92. The women who perceive their
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AMERICAN JOURNAL OF MEDICAL GENETICS (SEMIN. MED. GENET.)
41
just indirect measures of the tested
person’s own distress, the effects of
perceived spousal anxiety and support
would be greatly diminished, if not
eliminated, by the addition of the IES
at 1 week. We find that the effect of
having a ‘‘burdensome’’ spouse continues to elevate the distress level of the
tested person. This suggests that perceptions of spousal anxiety and support are
imparting an influence on posttest distress levels distinct from the tested
person’s own distress level.
Figure 1. Mean IES scores by spouse profile and time. Light-gray bar, 1 week; darkgray bar, 4 months; white bar, 1 year; black bar, 2 years.
spouses to be highly anxious and nonsupportive exceed this threshold at every
measurement point; at the 1-year mark,
they exceed it by 50%.
It is possible that what has been
assessed by the tested person’s perception
at the 1-week interview of the anxiety/
support of a spouse is merely a reflection
of the tested person’s own anxiety about
the results. We were able to address this
possibility with two further analyses
(results not shown). First, we analyzed
the tested person’s perception of his or
her spouse’s anxiety/support in the time
before results were available (i.e., after
blood draw but before the results
had been received). The one important
difference is that the effect sizes of the
before-result assessment by the tested
person are smaller than those associated
with their assessment of spousal anxiety/
support after-results. The fact that the
tested person’s assessment of his or her
spouse’s support at the two time periods
yields different effects lends credence
to the prediction that it is indeed the
perception of spousal anxiety/support
that is having the effect. If the tested persons’ own response to the test
results colored their perception of
their spouses, then this should have
made their views of their spouses similar,
not different.
Our appraisal that it is indeed the
effects of perception that we are measuring is further born out by regressions that
added the tested person’s distress, measured by the IES at 1 week, to a model
predicting distress at 2 years postresults.
If perceptions of spouses’ responses were
Figure 2. Mean IES scores by spouse profile and gender. Light-gray bar, 1 week; darkgray bar, 4 months; white bar, 1 year; black bar, 2 years.
DISCUSSION
In this analysis, we tested the effects of
BRCA1 mutation carriers’ perceptions
of their spouse’s supportiveness on the
carrier’s long-term distress. We predicted that tested persons who perceived
their spouses to be anxious would themselves have greater distress levels than
those who did not perceive their spouses as anxious. We also predicted that
perceptions of spousal support would
have the opposite effect: higher perceived spousal support would be associated with lower levels of distress
experienced by carriers. Our analyses
are consistent with these predictions.
Moreover, the effect of these perceptions
is long-term and relatively stable; perceptions of spousal anxiety and support
are predictive of the tested person’s
distress levels at every time point up to
2 years posttesting.
Stress theory predicts that the effects
on a person of a stressful situation will be
buffered by the presence of a spouse.
However, this study is concerned with
variations in supportiveness of spouses
within marriages. Furthermore, spouses
Stress theory predicts that the
effects on a person of a stressful
situation will be buffered by the
presence of a spouse. However,
this study is concerned with
variations in supportiveness
of spouses within marriages.
42
AMERICAN JOURNAL OF MEDICAL GENETICS (SEMIN. MED. GENET.)
in the genetic testing context are not in
the same situation as spouses in other
stressful circumstances; they are directly
involved in the stress itself by virtue of
the potential heritability of the mutation
by their children. As Kash [1995] has
pointed out, in genetic testing, spouses’
participation is more than that of supporter. Our results indicate that the
tested person’s perception of the impact
of the genetic test results directly on the
spouse does indeed have an effect on the
spouse’s utility as support giver.
One of the goals of this analysis
was to try to identify subgroups within
the testing population who might be at
increased risk of posttesting distress.
Once the fact that the effects of perceived spousal support and anxiety are
predictive of the tested persons’ distress
levels had been established, we proceeded to examine combinations of
those factors that might identify those
spouse characteristics that might elevate the level of adverse psychological outcomes. Using the possible
combinations of high/low perceived
anxiety and high/low perceived support,
a spouse response profile was created to
define four subgroups. The prediction
that the effect of perceived high anxiety/
low support (i.e., the ‘‘burdensome’’
spouse) would be associated with the
greatest level of posttesting distress,
compared to low-anxiety/high-support
(‘‘supportive’’) spouses, was supported
by the analyses.
The means for carriers in the lowanxiety/high-support (‘‘supportive’’)
group are considerably lower than any
of the other three groups and are roughly
comparable to the means of similar tested
persons who are noncarriers (data not
shown). We conclude that those carriers
who perceive their spouses to have low
anxiety and are supportive are roughly
equal in distress, 2 years posttesting, to
noncarriers. These findings are suggested by stress theory: the perception
that one is supported by one’s spouse
buffers the effects of the stressor [Thoits,
1982, 1995; Wethington and Kessler,
1986; Beach et al., 1996].
Two results stand out for the highanxiety/low-support (‘‘burdensome’’)
group. First, the means at the 1- and
2-year postresults interviews are comparable to those reported by EppingJordan et al. [1994] in their interviews
of cancer patients 10 weeks after diagnosis. The mean IES score for that
sample was 22.92. The adverse effect of
being a carrier with a ‘‘burdensome’’
spouse is both significant and enduring.
These carriers respond 2 years after
genetic testing results as if they had recently received a diagnosis of cancer.
Second, the means for women in
this group at all four posttesting interviews conspicuously exceed those reported by Epping-Jordan et al. [1994].
Women’s distress not only endures over
the 2-year study, it actually increases.
It is important not to underestimate
the effect of positive carrier status on
men. The lower levels of distress posttesting reported by men with ‘‘burdensome’’ wives may only reflect the
cultural fact that men generally report
lower levels of psychological distress
than women. Tested husbands have
wives who may themselves be adversely
affected through their children. When
asked if anyone had had a particularly
negative response to test results, two
kindred members mentioned wives of
carrier men. One noted, ‘‘My sister-inlaw has had a terrible [time]. . . . I think
she blames her husband for having it.’’
Furthermore, examination of wives’
responses to questions about whether
or not having the information about
their husband’s status was positive or
negative yields comments such as ‘‘I received this information . . . 2 years ago.
I had a pretty new baby girl and [it]
was upsetting for me to look at her
and wonder if cancer is going to kill
her before her life’s in full bloom.’’ Interestingly, though, anxious wives of carrier men did not appear to elevate
distress levels among these men. Since
previous research on the psychosocial
effects of being tested for BRCA1
mutations has focused almost exclusively
on women as the tested person, the
effects on males remain an important
area of future research.
These findings have important
implications for future genetic testing.
We show that the tested person’s perceptions of his or her spouse’s responses may
ARTICLE
have potent and persistent effects on
the response to testing. The data indicate
that clinicians should consider the patient’s relationship with their spouse in
order to anticipate potential problems
and provide more effective support for
individuals who undergo genetic testing.
At the least, our results support the
current research practice of encouraging
spouses to be involved in the genetic
education process prior to the decision to
be tested. Effective clinical management
of genetic testing of married individuals may depend on recommendations
for suitable interventions when necessary. Since the results are limited to the
tested person’s perception of his or her
spouse’s response, interventions that
affect each member of the pair’s ability
to communicate and effectively interpret what is communicated may be most
useful in reducing postresults distress
over time. Thus, health care practitioners providing genetic information
about susceptibility to late-onset disease
to married individuals should be aware
that the information they are providing
has direct effects on both members of the
pair, which may affect the spouse’s ability
to act as support person to the tested
individual.
Care must be taken when extrapolating these findings to clinical practice. The size of the subgroups is small
enough to require caution in interpretation. Also, our analyses are based on the
tested person’s perceptions of his or her
spouse’s anxiety or support, or both. It is
unknown if the tested person’s perceptions are consistent with actual spousal
anxiety and support.
The sample in this analysis is largely
made up of members of the Church
of Jesus Christ of Latter-day Saints
(Mormons). Religious characteristics
may make this sample somewhat different from other populations, though the
potential support available to members
of this church suggests that our results
are conservative. Also, the subjects had
extensive genetic counseling prior to
testing as well as after learning their
genetic test results, which may not be
available in future nonresearch settings.
The effects of cancer family history
may limit the utility of studies addressing
ARTICLE
the psychosocial effects of genetic testing. Although we cannot rule out the
possibility that individuals whose families have more cancer than is the norm
(and who generally form the basis for
research studies of the psychosocial
sequelae of genetic testing) have differing responses, we did not find that
having first- or second-degree female
relatives with breast and/or ovarian
cancer affected the responses of subjects
in this analysis.
There have been few reports of the
effects of spouses on the responses of
individuals to genetic testing. Lodder
et al. [2001] report that spouses of
carriers have higher posttest anxiety
than spouses of noncarriers, but no
interaction of that anxiety with distress
of the tested person is indicated. The
data of Manne et al. [2001] indicate that
women who test positive for the mutation and who have supportive spouses
fare better than those who do not have
supportive spouses. Both of these studies
were limited to women tested for the
mutation and their husbands. In addition, that analysis was based on a sample
with a larger proportion of women
who had already had cancer, unlike this
sample.
A significant strength of this analysis is that it examines both males and
females. The effects of positive mutation status are not limited solely to
women, since male carriers may also
pass the mutation to their children.
Thus, although the effect on women is
demonstrably greater than on men, it is
not limited to women. As our analysis
shows, male carriers who perceive their
spouses to be anxious and nonsupportive at the time of testing may be
distressed by their results up to 2 years
posttesting. Focusing clinical intervention on females may deprive male
carriers of much needed intercession
and support. The differences between
males’ and females’ responses to genetic
testing should be elucidated by further study.
In conclusion, we have been able to
identify a subset of carriers who are at
risk for clinically elevated distress levels
up to 2 years posttesting. Our results
suggest that women with ‘‘burdensome’’
AMERICAN JOURNAL OF MEDICAL GENETICS (SEMIN. MED. GENET.)
spouses in particular may be at risk for
elevated levels of distress that grow
with time. Such information may allow
greater focus of genetic counseling on
the spouse of the potential tested person
than has been the case to date.
We note that we have not yet
looked at the effect of testing on spouses. The concordance between actual
and perceived effects of testing on
spouses may assist clinicians in management of the effects of genetic testing.
Additionally, given the rise in singleparent families, more attention may
need to be paid to children as possible support givers. They, like spouses,
have dual roles in this situation.However,
Given the rise in single-parent
families, more attention may
need to be paid to children as
possible support givers. They,
like spouses, have dual roles in
this situation.
since children are potentially directly
affected by their parent’s genetic status,
their relationship may be more complex. Truly effective interventions to
minimize test-related distress will require further investigation and understanding of the complex psychosocial
dynamics of genetic testing for lateonset disorders.
ACKNOWLEDGMENTS
The authors thank Cathleen Zick for her
insightful comments when the project
was being formulated.
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