A multicenter study of supportive-expressive group therapy for women with BRCA1/BRCA2 mutations
Women with a BRCA1/BRCA2 mutation experience significant challenges. These include decision-making regarding surgical options and notification to offspring and family, along with a sense of isolation, which may lead to psychological and emotional distress. The current study developed, standardized, and conducted preliminary testing of a supportive-expressive group therapy intervention designed to address these challenges.
Seventy women with a BRCA1/BRCA2 mutation recruited from familial cancer risk clinics participated in 12 sessions of supportive-expressive group therapy that lasted 6 months. Before and after measures of psychosocial functioning, knowledge, and surveillance/surgery activities were completed.
Sixty-seven women completed the intervention. Significant improvements were observed in psychosocial functioning: cancer worries (P = 0.005), anxiety (P = 0.033), and depression (P = 0.015). Knowledge level and surveillance levels were high at baseline and there were no significant changes postintervention. A significant number of women made decisions concerning prophylactic surgery (oophorectomy/mastectomy) during and after the intervention.
The feasibility of a supportive-expressive group for BRCA1/BRCA2 mutation carriers was demonstrated. Findings from the study are consistent with an effective intervention. However, further research is required using a randomized controlled study design. Cancer 2004. © 2004 American Cancer Society.
Significant progress has been achieved in identifying the genetic component of familial breast carcinoma. Two of these susceptibility genes are, BRCA1, localized and cloned in 1994, and BRCA2. Women with a mutation in BRCA1/BRCA2 have a ≤ 87% lifetime risk for developing breast carcinoma and a 15–60% lifetime risk for developing ovarian carcinoma.1, 2
Numerous challenges arise after the identification of a BRCA1/BRCA2 mutation, including the burden of disseminating this knowledge to family members, worry about vulnerability to breast and ovarian carcinoma, and decision-making regarding prophylactic surgery and chemoprevention options.3, 4 Needs assessment surveys indicate that women are interested in psychosocial supports, including support-group models.5 However, to our knowledge to date no studies have been reported on the development or evaluation of psychosocial interventions for the specific population of women who carry genetic mutations for BRCA1/BRCA2. To our knowledge, the current study is the first to report the effects of a group support model designed for BRCA1/BRCA2 carriers.
Psychological Issues Associated with Having a Family History of Breast Carcinoma and Testing Positive for BRCA1/BRCA2
Women at risk for cancer have a wide range of emotions including fear, guilt, anger, isolation, grief, depression, and anxiety.6–9 Identification with family members, notably mothers, evoke recollection of interruptions in family life during the time of the earlier cancer-related experience (e.g., loss of a parent and subsequent disruption of the home).8 This represents a serious stressor that can have a negative psychological impact. In addition, a profound but different sense of loss, associated with the personal experience of an impaired or “shattered” invulnerability to serious illness, has emerged as a major psychological consequence to the recognition of the genetic predisposition to cancer. Many women at high risk for cancer believe it is not a question of “if” but “when” they will develop cancer, and feel strongly identified with their family member who developed cancer.
Women who learn they have a BRCA1 or BRCA2 mutation may experience an increase in distress3 and do not experience the decrease in distress afforded by women who, in contrast, receive a negative test.10 Women who have faced the previous stressor of having a diagnosis of cancer may underestimate their emotional responses to genetic testing.11 Factors known to influence distress levels during genetic testing include mutation status, pretest psychosocial functioning, the number of family members with cancer, the degree of social support, coping style, gender, and the expectation of a “negative” test result.3, 8, 12–16 Psychosocial response can also depend highly on the familial context. For example, if a woman tests positive in a family context of other siblings testing negative, she may experience significant psychosocial distress.14
Currently, women with BRCA mutations are candidates for intensive cancer surveillance.17 Prophylactic surgery may offer the greatest reduction in cancer risk, however, with mastectomy reducing the risk of breast carcinoma by ≥ 90%.18, 19 Oophorectomy significantly reduces the risk of ovarian carcinoma.19 Although these medical options potentially decrease anxiety attributable to cancer risk, they can also impose additional psychologic burden because they often involve the processing of complex and evolving information and choices.20, 21 Decision-making concerning prophylactic surgery, for example, is challenging for a woman who is young and disease free and must weigh the benefits and risks of her choices with regard to the overall impact on her quality of life. Additional stressors that can effect the quality of life among women with BRCA1/BRCA2 mutations include communication challenges within the family to disseminate the genetic information to relatives, fears and worry concerning the health of offspring, concerns regarding insurance issues, potential confusion or misinformation concerning complex genetic knowledge, and ongoing fears of one's own risk for cancer (or for an additional cancer).22–24
We undertook the development of an intervention to address these challenges and the psychological impact of learning that one carries a BRCA1/BRCA2 mutation to help women to cope with their high-risk status. The program we developed is based on a well established model of supportive-expressive group therapy.25–29 Supportive-expressive group therapy provides mutual support and opportunities for emotional expressiveness and confrontation with existential challenges to facilitate coping. Although we recognize that a randomized, controlled trial is the ideal study design, we chose first to conduct a pretrial/posttrial for a number of reasons. First, it was unknown whether a group intervention would be feasible or acceptable in this new clinical population. Second, it was unclear as to the feasibility of combining the two subgroups of women with BRCA1/BRCA2 mutations (i.e., women with and those without a previous diagnosis of cancer). Third, it was unknown whether standard psychotherapy sessions (12 sessions) would produce intervention effects and yet would be tolerable by this new clinical population with minimum dropouts. Fourth the feasibility regarding the training in the delivery of our intervention needed to be demonstrated. Finally, we wanted to identify and address all topics/themes identified by this new clinical population when developing intervention manuals. The objectives of the current study were to conduct preliminary testing of our group intervention on emotional well-being, knowledge, and surveillance behaviors and to explore potential additional benefits.
We hypothesized that a group supportive-expressive intervention that addresses the psychologic impact of being at high risk for breast and ovarian carcinoma would be associated with improved psychosocial functioning (i.e., cancer-specific distress, depression, and anxiety), enhanced knowledge of breast carcinoma genetics, and adherence to recommended screening.
MATERIALS AND METHODS
General Study Design
A prenonrandomized/postnonrandomized intervention study was undertaken.
Women who attended familial cancer risk clinics at participating centers in Canada and Australia, who met inclusion criteria, were invited to participate in the group support study. The institutional ethics review board at the University of Toronto (Toronto, Ontario, Canada) and the research ethics boards at participating centers in Canada and Australia approved the study procedures. Women were recruited from genetic clinic sites at the Centre for Research in Women's Health in Toronto (n = 25), Toronto Sunnybrook Regional Cancer Centre (n = 13), the North York General Hospital in Toronto (n = 4), the Hamilton Regional Cancer Centre in Hamilton, Ontario, Canada (n = 7), the Cross Cancer Centre in Edmonton, Alberta, Canada (n = 8), and the Peter MacCallum Institute in Melbourne, Australia (n = 13). Patients were recruited between May 1999 and April 2002. All women had previously undergone genetic counseling and mutation testing at their institutions. Information concerning women who were eligible for the study was forwarded to research assistants or clinical staff who then informed the women of the study. Informed consent was obtained from interested women. All women underwent a psychosocial assessment and interview to confirm eligibility criteria. They also received information regarding and preparation for the group support program.
The study inclusion criteria were females ages 18–70 years, completion of the genetic testing process indicating a positive test for a BRCA1 or BRCA2 mutation, and living within 1 hour of a participating center. We included women at risk (with no previous diagnosis of cancer) as well as those with a previous diagnosis of breast and/or ovarian carcinoma. Any individual who had a previous diagnosis of breast or ovarian carcinoma must have been ≥ 1 year posttreatment and not undergoing psychologic treatment for a current diagnosis. Individuals were excluded if they met any of the following criteria: a history of a major psychiatric disorder (e.g., psychosis), as classified in the American Psychiatric Association's 4th edition. of the Diagnostic and Statistical Manual of Mental Disorders and identified through a semistructured psychiatric interview; an unwillingness to provide informed consent; current active treatment for any cancer; an inability to converse in English; and a current enrollment in any other psychologic intervention study. Women who developed breast or ovarian carcinoma during the course of the group therapy would be encouraged to continue with the group as an important source of support.
Written informed consent was obtained from all participating women in accordance with approval granted by the human subjects committees of participating centers.
The group therapy intervention was standardized. It was comprised of the principles of supportive-expressive group therapy outlined in the treatment manual by Spiegel and Spira27 and in previous reports by Leszcz and Goodwin30 and Esplen et al.31 Supportive-expressive group interventions have been well established in cancer populations26, 28, 29 for facilitating psychologic adjustment to a life-threatening illness. The version in the current study was comprised of 8 weekly (90-minute) group sessions followed by 4 “booster” monthly sessions for a total of 12 sessions over 6 months. The supportive-expressive therapy addresses the emotional impact associated with having a family history of breast and/or ovarian carcinoma and being at high risk for developing cancer.
A mental health professional (e.g., psychologist, social worker) and a genetic counselor conducted the groups together. Group leaders participated in a 2-day training workshop and attended weekly taped reviews at the Toronto site. In sites outside Toronto, videotapes of each session were monitored for competence and adherence to the intervention by the Toronto investigators (Drs. Esplen and Leszcz) and written feedback was provided on a weekly basis. Because one of the group leaders was a genetic counselor, the opportunity to clarify or correct factual misinformation occurred and was encouraged. The intervention groups combined equal numbers of women with and without a previous diagnosis of breast or ovarian carcinoma. Six to 10 women participated in each group and group membership was closed for the duration of the group to promote group cohesiveness and consistency. The intervention included one family session at which women were invited to bring a family member (or friend) of their choice to visit the group as an opportunity to clarify information relevant to the family impact. Two sessions involved visits by an oncologist or surgeon to further clarify or address concerns regarding participants' decisions and/or gene mutation status.
Features of expressive therapies that are unique to, or strengthened by, the group format include the facilitation of mutual support, the creation of a sense of normalization through shared experience, the encouragement of emotional expressiveness, the promotion of family and social support, the enhancement of an expanded repertoire of coping skills, vicarious learning through others, detoxification of being at risk, and the integration of new meaning.27, 31 A number of topic categories were abstracted by the study team from videotaped reviews of a prestudy pilot group and described in our manual. The topic categories included the following: psychological domains (loss and grief, stress response syndrome, living with uncertainty, changes in self-image, impact on life trajectory); genetic and medical domains (genetic testing and research issues, medical treatment, and surveillance); and social domains (disclosure-related issues, next generation, sociocultural issues, family and social network).
At baseline, demographic and medical variables (e.g., previous diagnosis of cancer, history of surgery) were collected through self-report and review of clinic charts. Before study entry and at postintervention (within 2 weeks of completion of the 12-session intervention) women completed a battery of self-administered psychological questionnaires.
Genetic test-specific distress.
We used the Impact of Event Scale (IES) to measure the specific distress associated with receiving a positive test result for BRCA1/BRCA2.32 The IES is a 15-item scale used to assess the experience of stress for a specific life event (e.g., the stress of a positive genetic test result). The IES has two subscales that measure intrusive thoughts and feelings and avoidance of certain thoughts, feelings, or situations. The IES has good internal consistency (Cronbach α = 0.87 for the total score; α = 0.84 and 0.78 for the intrusion and avoidance subscales, respectively). The IES has been used previously to assess the stress impact among women at increased risk for developing cancer and the intrusion subscale is used frequently to describe the preoccupation with cancer risk in this population or the specific stress associated with a genetic test result.33–36
The Brief Symptom Inventory (BSI),37 a standardized and widely validated shorter version of the Symptom Check List-90, was used to measure global psychologic functioning. The BSI measures nine psychologic symptom dimensions—somatization, obsessive-compulsiveness, interpersonal sensitivity, depression, anxiety, hostility, phobic anxiety, paranoid ideation, and psychotocism—and provides a global severity index (GSI). We report herein on the primary outcomes of depression and anxiety and the total score-GSI. The depression subscale of the BSI was utilized to define women below and above the clinical cutoff point (≥ 60 total score [T score]) for depression.8, 37
Predetermined criteria for intervention success.
“Criteria for success” with the intervention was defined a priori as a reduction of a > 0.5 standard deviation (SD) on the IES intrusion subscale for 50% of the participants or a 4-point reduction on the standardized BSI depression subscale. Previous studies have described success with interventions on the IES with a mean change of 0.5 SD on the IES intrusion subscale, corresponding with a 4–5-point change on the scale.33, 36 For BSI depression, a mean change of 0.5 SD on the depression subscale corresponds with a 4-point reduction in the T score for nonpatient females.37
We utilized The Texas Revised Inventory of Grief (TRIG)—a standardized measure of grief that yields two factors: past behavior and present feelings.38 Subjects who had experienced a loss of a family member to cancer were asked to complete the TRIG. The past behavior subscale contains eight items that ask the subject to “think back to the time the person died,” and provides information concerning a variety of life events that might have been disrupted by grief, thus measuring one's initial adjustment to the death of a loved one. The present feelings subscale includes 13 items focused on current feelings associated with the deceased, such as “I still want to cry when I think about the person who died.” The TRIG is an easily administered self-report and permits evaluation of the extent and nature of an individual's personal reaction to loss. Grief reactions can be compared with normative means and ranges at each of 4 time intervals after the loss: within the first year, 1–5 years after the loss, 5–10 years later, and > 10 years later. In addition, the TRIG contains a set of clinical demographic questions, for example, “How close were you to the person who died?” The TRIG has good psychometric properties including α coefficients of 0.87 and 0.89 for past and present factors, respectively.
Quality of life.
Before study entry and at postintervention. we administered the Quality of Life Index (QLI) developed by Ferrans and Powers.39 The QLI is a multidimensional rating scale that measures satisfaction using 34 items representing 4 life domains (i.e., family, health and functioning, psychologic/spiritual, and socioeconomic), as well as their importance to the respondent. Scores, ranging from 0 to 30, are obtained for the overall scale and for each of the subscales. The QLI has shown good internal consistency and reliability in studies of breast carcinoma.39
Breast carcinoma riskgenetics knowledge.
Before study entry and at postintervention, women completed a self-administered questionnaire, developed for the study, to assess knowledge. The questionnaire was comprised of 12 true or false items, as well as a “don't know” category. Items included questions regarding breast and ovarian carcinoma risk, general population risk levels, the probability of transmission of a genetic mutation to offspring, information concerning risk level for males, and the implications of carrying a genetic mutation for BRCA1/BRCA2. The questionnaire was scored for the number of correct items with a possible range from 0 to 12.
Surveillance activities and surgery.
Before starting group therapy and at postintervention and at 1year after entry, women completed self-reports of surveillance activities including breast self-examinations, clinical breast examinations, mammography, and ovarian ultrasounds. Clinical chart reviews assessed compliance with screening recommendations. Women completed self-reports at baseline, after intervention and follow-up surgery, and medical treatment histories. We report the preintervention and postintervention measures and 9-month follow-up for surgical outcomes.
Satisfaction and other reported benefits.
Women were asked to report on levels of satisfaction with a Likert- type questionnaire and with open-ended questions. They were also asked to identify specific benefits and to indicate whether the group addressed their personal reasons for participating in the intervention.
All data were coded in a standardized format and entered into a SPSS database, Version 11.0 (SPSS, Chicago, IL). All 70 women who participated in the intervention were included in the analysis using intent-to-treat analysis. A descriptive analysis was undertaken of all study variables to examine distributions, means, SD, and proportions.
To address our concerns with regard to the feasibility of combining the two subgroups of women with BRCA1/BRCA2 mutations (i.e., women with and those without a previous diagnosis of cancer), analyses were conducted at baseline between these two groups to compare demographic and psychosocial variables using independent Student t tests for continuous variables, such as age and time since the test result, and chi-square analysis for categoric variables such as marital status and educational level. Parametric statistics were used because the techniques are quite robust in samples comprising > 30 participants. Because of known factors that influence distress levels during genetic testing, such as age, having a previous diagnosis of breast carcinoma, and time since receipt of the genetic test result,3, 8, 12–16 we also conducted a linear regression with baseline measures of psychosocial functioning and these independent variables to determine whether any of the independent variables would influence baseline psychosocial functioning.
First, Student t tests for paired data were conducted to examine the preintervention and postintervention measures of psychosocial functioning (e.g., genetic test specific distress, depression). We performed a linear regression analysis with outcome measures on psychosocial functioning and potential predictive factors, such as age, previous diagnosis of breast carcinoma, and time since genetic testing results, controlling for baseline measures of psychosocial functioning. Using our predetermined criteria for intervention success, we calculated the proportion of participants who achieved a reduction of > 0.5 SD before and after the intervention. For missing data, we used the intent-to-treat analysis recommended by the Cochrane collaborative model,40 which assumes that participants missing postintervention data do not benefit from the intervention and that their baseline score remained unchanged postintervention. This is a conservative estimate of the intervention effect. All P values were 2 tailed and set at significance level of 0.05. No further adjustments were made for multiple comparisons. A content analysis was conducted for the open-ended questions regarding potential additional benefits and satisfaction.
In the current study, 165 eligible women were offered participation in the group therapy. Seventy patients (42%) were enrolled in 11 intervention groups. All groups began between May 1999 and June 2002 and were completed by January 2003. Seventy-five individuals (45%) refused or did not return phone calls inviting them to participate. The main reasons for refusal were being too busy/not enough time to commit to the study (n = 21 [28%]), not being interested in group therapy (n = 18 [24%]), living within 1 hour but feeling that they are too far away from the participating site (n = 16 [21 %]), being unable to participate due to surgical complications (n = 6 [8%]), and being worried about confidentiality (n = 2 [3%]).
Of the 70 women who agreed to participate, 67 (96%) completed either the intensive phase (8 weekly sessions) or both the intensive and booster phase of the intervention (8 weekly sessions and 4 monthly boosters) and only 3 stopped attending group sessions early in the intensive phase. Completers attended a mean of 9.8 sessions (range, 8–12 sessions).
Baseline characteristics of the study population are provided in Table 1. There were no differences among study sites on demographic, psychosocial, and knowledge variables. In addition, comparisons were conducted on demographic variables between women with a previous diagnosis of cancer in addition to their positive mutation status (n = 38) and those (n = 32) with no previous cancer diagnosis. With the exception of a significant difference in age, with the women with a previous diagnosis of cancer being older, there were no statistically significant differences found with regard to demographic variables between the two groups. The women were all recruited from familial cancer risk clinics and reflect the reported profile of women seeking genetic testing described in the literature (i.e., well educated, married, white women with children). At least one-third to one-half of the subgroups had experienced a loss of a family member to breast carcinoma.
Table 1. Demographics (whole group, n = 70)
|Mean age, (yrs) (SD)|| || || |
| ||41.7 (8.4)||48.5 (9.0)||Student t test (68) = 3.27, P = 0.002|
| ||Range 25 to 56||Range 30 to 68|| |
|Marital status|| || || |
| Married or common law||23 (71.9)||30 (78.9)||Chi-square test (1) = 0.47, P = 0.49|
| Single (never married)|| 7 (21.9)|| 6 (15.8)|| |
| Divorced/widowed/single parent|| 2 (6.3)|| 2 (5.2)|| |
|Having children|| || || |
| Yes||20 (62.5)||27 (71.1)||Chi-square test (1) = 0.58, P = 0.45|
| No||12 (37.5)||11 (28.9)|| |
|No. of children: mean (SD) (range)|| || || |
| ||2.4 (0.9) (1–4)||1.93 (0.72) (1–3)||Student t test (46) = −1.9, P = 0.07|
|Highest level of educationa|| || || |
| High school|| 7 (21.9)||13 (34.2)||Chi-square test (3) = 2.0, P = 0.57|
| College|| 8 (25.0)|| 6 (15.8)|| |
| University|| 8 (25.0)||10 (26.3)|| |
| Postgraduate studies|| 9 (27.7)|| 8 (21.1)|| |
|Ethnic backgrounda|| || || |
| Anglo-Saxon||10 (31.3)||22 (57.9)||Chi-square test (2) = 5.0, P = 0.08|
| Ashkenazi||13 (40.6)|| 9 (23.7)|| |
| Other|| 8 (25.0)|| 6 (15.8)|| |
|Mean no. of family members with BC: (SD) (range)|| || || |
| ||2.8 (2.2) (0–12)||2.6 (1.6) (0–6)||Student t test (66) = −0.46, P = 0.65|
|Loss of a family member to BCb|| || || |
| Yes||16 (50.0)||12 (31.6)||Chi-square test (1) = 1.1, P = 0.30|
| No||13 (40.6)||17 (44.7)|| |
Psychosocial variables at baseline
To assess potential predictors of differences in the psychosocial measures at baseline, we conducted a series of linear regression analyses on the dependent variables of IES intrusion and avoidance, BSI anxiety, depression, and GSI, and the TRIG past behavior and present feelings of grief subscales. Each regression analysis included the independent variables of age, previous diagnosis of cancer, and number of months since the receipt of test results, as well as one of the baseline psychosocial functioning variables. None of the independent variables in the model were found to be statistically significant predictors of any baseline psychosocial functioning variables. For instance, for the dependent variable of the GSI of the BSI, the P values for the independent variables were 0.55 for age, 0.20 for previous diagnosis of cancer, 0.51 for time since test result, and 0.24 for group site. together, they explained a very small amount of variance of baseline psychosocial measures (adjusted R2 ranged from 4% to 6%).
Criteria for success
Cancer-specific distress (Impact of Event Scale intrusion).
The intrusion subscale score ranged from 0 to 35. Baseline cancer distress scores for the IES intrusion subscale were consistent with subclinical ranges for 31%41 and 14% met the cutoff score of > 20, which is consistent with symptoms of a posttraumatic stress disorder.32 Thirty participants (43%) demonstrated success in the reduction of intrusion or cancer worries by a > 0.5 SD, or a reduction of > 3.6 points. Eleven women (16%) who had a low baseline score that remained low after the intervention were also considered to have a successful outcome, resulting in an overall success rate of 59% for the study. Six women (9%) had lower scores that increased over time. The difference in the intrusion subscale score before and after the intervention was statistically significant, t(64) degrees of freedom = 2.9, P = 0.005. Nineteen women (27%) did not complete poststudy measures but were included in the analysis using an intent-to-treat analysis. The magnitude of the benefit in the women who completed the measures was greater compared with women who did not complete the measures, with an average reduction of 4 points or 10% of the possible range of scores on the cancer worries scale, which is consistent with previous intervention studies of similar populations of women33, 34 and those of supportive-expressive group therapy.29 Scores for the avoidance subscale of the IES followed a similar course.
Depression and anxiety.
Twenty-four women (34%) scored above the clinical cutoff for depression at baseline (T score ≥ 60). Women demonstrating levels consistent with clinical or subclinical depression benefited most from the intervention. The magnitude of the benefit in women who demonstrated clinical ranges was an average reduction of 4 points, > 0.5 SD, from a standardized T score of 64 to 60—the clinical cutoff point for depression for nonpatient adult females.37 Another 25 women (36%) scored in the normal range for depression before the intervention and remained low after the intervention. The difference in the BSI raw score before and after the intervention was found to be statistically significant using the whole sample, t(69) = 2.5; P = 0.015. Baseline anxiety and GSI scores indicate 32% and 34%, respectively, in the clinical ranges. Statistics for the anxiety and GSI subscales followed a similar course to that of the depression subscale (Table 2).
Table 2. Psychosocial Functioning: IES and BSI Subscales
|IES|| || || || || || || |
| Intrusion or cancer worries||8.8||(8.0)||6.5||(6.7)||2.9||64||0.005|
|BSI|| || || || || || || |
| Depression (raw score)||0.62||(0.74)||0.48||(0.62)||2.5||69||0.015|
| Anxiety (raw score)||0.62||(0.64)||0.51||(0.60)||2.2||68||0.033|
| Global severity index (raw score)||0.59||(0.56)||0.47||(0.46)||2.75||60||0.008|
Impact of baseline characteristics on pos intervention intrusion, depression, and anxiety
Baseline levels of intrusion, depression, and anxiety were found to be significant predictors of corresponding postintervention psychosocial functioning. After controlling for baseline psychosocial variables in the linear regression model, none of the other variables (e.g., age, previous diagnosis of cancer, or number of months since receipt of test results) were found to be statistically significant predictors of intrusion, depression, or anxiety postintervention. For example, for the dependent variable of the intrusion subscale of the IES, the P values for the independent variables were 0.82 for age, 0.59 for previous diagnosis of cancer, 0.39 for time since test result, and 0.005 for baseline intrusion score.
The TRIG was specifically anchored around “loss of family members to cancer” (Table 3). Of 70 participants, 59 (84%) had lost a family member to cancer, 30 of 59 participants (51%) had lost a female family member to breast carcinoma, 20 of 30 participants (67%) had specifically lost a mother, and 10 of 30 (33%) participants had lost a sister or daughter. Only subjects with loss of family members to cancer completed the TRIG. The baseline mean scores for the past behavior and present feelings subscales were 20.1 and 40.4, respectively. These levels are consistent with a pattern of previously unresolved grief or acute prolonged grief when compared with population norms on this measure.38 The intervention reduced the intensity of grief for those who had their test result for < 1 year compared with those who had had their test result for > 1 year, t(18) = 2.5, P = 0.02. For the latter group, the intervention was more effective in reducing the levels of intrusion/cancer worries, t(39) = 2.4, P = 0.023.
Table 3. Texas Revised Inventory of Grief
|Current grief feelings||39.6||(10.8)||38.5||(10.7)||0.6||39||0.54|
|Past grief behavior||19.9||(6.7)||18.8||(7.5)||1.1||35||0.29|
|Current feelings (result ≤ 1 yr)||42.4||(10.7)||38.8||(9.3)||2.5||18||0.02|
|Current feelings (result > 1 yr)||36.6||(10.7)||37.4||(12.2)||0.24||18||0.82|
Quality of life.
There was a mean total score increase in general health-related quality of life (QLI) from 20 at baseline to 22 postintervention. However, the increase was not statistically significant, t(69) = 1.75, P > 0.05.
Breast carcinoma genetics knowledge.
Before the group intervention, women demonstrated a mean total score of 9 correct items of 12 (Table 4). Postintervention, the women demonstrated a mean total score of 10 and this improvement was not statistically significant, t(69) = 1.3, P > 0.05. However, personal knowledge was satisfactory and comprehensive.
Table 4. Hereditary BC and Ovarian Carcinoma Knowledge Levels (Total Score and Percentage Correct Per Item)
|There is one BC gene||90.0|
|All BC is inherited||92.9|
|If no gene mutation, woman will not get BC||94.3|
|Risk of BC changes with age||91.4|
|Men do not develop BC||91.4|
|Can be paternal transmission||74.3|
|One in nine women develop BC||75.7|
|Woman has gene mutation, 100% chance of BC||90.0|
|BC less frequent in Ashkenazi population||70.0|
|Genetic test always shows alterations in gene||42.9|
|If mother has gene mutation, 50% chance children will get BC||31.4|
|One of 20 women will develop ovarian carcinoma (50% unsure)||24.3|
Before the group intervention, 10 of 38 women (26%) with a previous diagnosis of cancer had undergone a mastectomy for treatment of breast carcinoma and 6 of these surgeries included prophylactic removal of the unaffected breast. A similar proportion (11 of 38 [29%]) of women with a previous diagnosis of cancer had undergone an oophorectomy for ovarian and/or breast carcinoma treatment. In comparison, among those without a previous diagnosis of cancer, at baseline, 11 of 32 women (34%) had undergone a prophylactic oophorectomy. In contrast to prophylactic oophorectomy, only 1 of the 32 women (3%) without a previous diagnosis of cancer had opted for a prophylactic mastectomy before the study. Only one woman had undergone both a prophylactic mastectomy and an oophorectomy before the group intervention and she had received a previous diagnosis of breast carcinoma.
Surgical status changed markedly over the course, and subsequent to the intervention. Four women underwent prophylactic oophorectomy during the group intervention phase, two of whom were without a diagnosis of cancer and two of whom had a previous diagnosis of breast carcinoma. At postintervention measurement, an additional six women had undergone a prophylactic oophorectomy, five of whom had no previous diagnosis of cancer.
During the course of the group intervention, only one woman with cancer had a breast removed prophylactically (Table 5). However, at the time of the postintervention measurement point, four additional women without cancer had undergone prophylactic mastectomy and one was scheduled for the surgery. In addition, seven women reported making decisions “not” to undergo surgery at the current time; five chose not to undergo a prophylactic mastectomy and two declined the option of prophylactic oophorectomy. We plan to continue to conduct long-term follow-up on these outcomes over 1 and 2 years.
Table 5. Surgery
|Oophorectomy|| || || |
| Women with previous diagnosis of cancer (n = 38)||11||2||1|
| Women with no previous diagnosis of cancer (n = 32)||11||2||5|
|Mastectomy|| || || |
| Women with previous diagnosis of cancer||16||1||—|
| Women with no previous diagnosis of cancer||1||—||4|
The baseline breast screening activities were fairly consistent with recommendations for a high-risk group of women. Ninety-nine percent (69 of 70) had received ≥ 1 mammogram with the average number of mammograms equaling 7 (range, 1–25 mammograms). All 70 participants received clinical breast examinations an average of 4 times per year (range, 1–4 clinical breast examinations). Fifty-eight women (83%) said that they practiced and felt comfortable with breast self-examination. The remaining participants (17%) reported that they experienced either too much anxiety or did not feel secure enough in their technical abilities to detect a problem during breast self-examination.
At postintervention, details were available for clinical breast examinations and breast self-examinations. There were no significant changes in rates of clinical breast examinations and there was self-reported improvement in feelings of anxiety around self-breast examinations for five women. One-year follow-up will report on mammography compliance. Magnetic resonance imaging (MRI) scans varied by site: 80% of the women at the Toronto site, 40% of the women at the Hamilton site, 20% of the women at the Edmonton site, and no women at the Melbourne site had undergone MRI scans.
Satisfaction and other benefits.
Of the 67 women who completed the intervention, 65 reported a “very high” level of satisfaction with the intervention and 2 reported being “moderately satisfied.” No patient indicated disappointment with the intervention. Additional benefits reported by the women included assistance with medical decision-making (75%); support and information regarding notification of offspring (72%); ongoing support from similar women (69%); opportunities to learn about current medical options/information (97%); a sense of validation concerning fears (96%); increased assertiveness and knowledge regarding medical professional/patient communication (60%); positive feelings related to personal growth, goals, and potential for life (96%); and enhanced quality of life through a sense of mastery over fears (88%). In addition, 97% of the women reported that they enjoyed and appreciated the opportunity to meet the other subgroup and indicated that they agreed with this approach. As we had anticipated, the women with a previous diagnosis of cancer reported finding it helpful to meet with younger women who were at risk (with no personal diagnosis) to understand their daughters' perspectives. Similarly, the younger cohort of women explored the impact of having a cancer diagnosis with the older subgroup, and frequently described them as “role models.”
As hypothesized, our group intervention was associated with improvements in the psychosocial variables of intrusion and depression and we have demonstrated the feasibility of a supportive-expressive group support program for women with BRCA1/BRCA2 mutations. In a nonrandomized study, it is not possible to conclude that these changes occurred as a result of the intervention, as it is possible that they would have occurred in a nonintervention control group. However, changes in these variables during the 6-month intervention are consistent with an effective intervention. In fact, in other illness populations, supportive-expressive group therapy, through its focus on emotional expressiveness and mutual support, has demonstrated effectiveness in facilitating active coping, providing emotional support, and in confronting and dealing with the impact of having a life-threatening illness.26, 29 In these groups for women with BRCA1/BRCA2 mutations, many of the discussions focused on issues such as living with continued uncertainty and the threat of cancer. In a similar fashion to that seen in groups of women with breast carcinoma,27, 28, 30 the women explored the range of their experience in an authentic, open manner. Many of the women explored difficult issues such as the guilt of transmission of the mutation to offspring, their altered body image, sexuality, fear, grief, potential illnesses, and death. After the expression and processing of emotions, the women would often examine their life priorities, engaging the existential threat of cancer to assist them in addressing life goals and values. This difficult process was experienced subjectively as life enhancing. Social support is another key feature of the intervention. Previous studies have demonstrated the impact of social support on adjustment to illness and its role in improving compliance with medical treatments.28, 30
Our recruitment rate of 42.5% reflects a high level of interest in a group support intervention. Ninety-six percent of the women completed the 6-month (12-session) intervention. The dropout rate in this study compares favorably with that of previous studies of group support in medical populations, and group therapy in general, suggesting its acceptability and relevance to the women.29, 42
A significant number of women demonstrated emotional responses consistent with clinical levels of depression and anxiety. The BSI subscale scores ranged from 0.5 to 1 SD above the normal population T scores. The adjustment level of these women may reflect the ongoing dilemmas and burdens of genetic testing as well as unresolved grief issues. A number of different motivations were expressed for group participation. These included decreasing their sense of isolation and obtaining support (unpublished data). Specifically, a significant number of the women (54.6%) without a previous diagnosis of cancer were motivated to participate to facilitate surgical decision-making and approximately one-fourth (25%) of both subgroups also were interested in getting support for dissemination of test results to family members, especially offspring (unpublished data). Women in the current study reported that the opportunity to meet others in a similar situation was a particular appealing feature of the group intervention. The observation that challenges and issues continue to emerge over time emphasizes the relevance and potential benefit that added psychosocial interventions may play in supporting the genetic counseling process.43, 44 Psychosocial supports in general are an important dimension in providing optimal care in genetics clinics, but it is likely that different approaches will be of interest and of benefit. However, the opportunity to facilitate mutual support and decrease isolation through group contact is a unique feature of group psychotherapy and has the added benefit of being cost-effective.30, 45
Limited information was collected on individuals who declined the intervention. This issue of specificity (i.e., which intervention is optimal for which individual) is an important issue for future studies. As we initiated the study, we were uncertain whether the women with and without a previous diagnosis of cancer would be able to be fully open and express their personal situations in the group. We were concerned about inhibitions in self-expression arising from the diversity of experience and the potential for feeling that they would burden, alarm, or offend the other subgroup with their personal perspectives. For example, a woman with a previous experience with a cancer diagnosis may feel inhibited to talk openly about her sense of guilt regarding the transmission of a gene mutation to her daughter. There were a number of universal themes common to the two subgroups, including challenges regarding surgery, disclosure, and the sense of vulnerability associated with being at risk for a first or subsequent cancer. The group provides an ideal opportunity for exchange between the two subgroups of women, and the process of exchange also can be applied as a model to assist women to address personal issues outside the group with families and health care professionals. We also compared the two subgroups with regard to demographics and no statistically significant differences (at P value = 0.05) were found between women with and those without a previous diagnosis of cancer when comparing demographic variables (with the exception of age). A linear regression model was also used to determine whether any particular groups of women would respond better to our intervention. Among potential predictors in the model, having a previous diagnosis of cancer did not appear to predict, in a statistically significant way, neither baseline psychosocial functioning nor psychosocial outcome measures after intervention. Therefore, we were comfortable in considering both affected and nonaffected women in the same intervention group.
An interesting perspective on grief emerged. Women who demonstrated a significant reduction in current feelings of grief had their test results for a shorter time period. This subgroup of women may have been dealing with the more immediate adjustment to their test result. Women anecdotally often experience the test result as a kind of loss, and describe losing peace of mind and may be feeling a greater threat to their security, hope for their future, and sense of immortality. Grief reactions have been found to be more intense, particularly in the early phases of the loss experience (e.g., 1–6 months).46, 47 Women with a family history also identify frequently with and experience a sense of merging with the lost family member to cancer, which can compound the stress response to a loss or life event, such as a medical diagnosis.48, 49 In this situation, the group may have facilitated an adjustment to the more recent loss experienced from receiving the test result. It is interesting to note that women who receive a negative test result anecdotally have reported feeling a loss in the connection they had to a mother who died of breast carcinoma. Perhaps the intervention and its focus on expression and exploration facilitated the processing of grief and adjustment among this subgroup.
For the women who had their test results for > 1 year, the reduction in current feelings of grief was not found to be significant. This finding is similar to that of a brief 6-week group intervention model developed by Wellisch et al.50 for women with a family history of breast carcinoma. We expected that the supportive-expressive group therapy would be particularly helpful in facilitating the emotional expression and processing of grief. A longer duration of intervention50 or individual therapy51 may be required to deal with what may be previously unresolved issues of loss and grief. Alternatively, the postintervention time point may not adequately reflect an ongoing process that is started by the group. For example, it is possible that the group in some way “catalyzed” the expression of the emotional experience of grief by having the women reflect on periods of previous loss and express their feelings, which for some women was the first time. It will be important to further examine this variable in our 1-year follow-up.
The lack of improvement in the knowledge questionnaire is somewhat surprising, especially given the presence of a genetic counselor coleading all of the group sessions. This may reflect a ceiling effect due to the high level of knowledge at baseline. It was encouraging to find that most of the women retained accurate information concerning genetics knowledge after their genetic testing. The knowledge level was found to be lower for items regarding general population risk levels for ovarian and breast carcinoma. For example, the two items “1 in 20 women in the general population will develop ovarian carcinoma in her lifetime” and “1 in 9 women in the general population will develop breast carcinoma during her lifetime” were those most often answered incorrectly. Perhaps women retained the information most relevant to them, or the more general information was less often provided during the genetic counseling sessions. In addition, the supportive-expressive group model is not primarily an “educational” model and therefore, psychoeducational principles were not incorporated because the objective of the group was simply to clarify rather then teach particular topics. However, the current study findings suggest that the intervention facilitated adjustment to information. The lack of improvement in the knowledge measure also may indicate a lack of sensitivity in capturing subtle clarifications or improvement in specific content areas that may have occurred as a result of having the genetic counselors colead the group.
Surveillance and Risk-Reducing Options
In terms of surveillance, the women demonstrated levels of surveillance consistent with recommendations for their high-risk status. Women appeared to be highly motivated to maintain their surveillance. Some women were very anxious about screening and reported being able to decrease their hypervigilance and experience a reduction of screening-related anxiety. Other women continued to conduct breast self-examinations and maintained confidence despite their fears regarding the detection of an abnormality. The range of MRI screening availability among sites reflects the opportunity for women in Canada to participate in an MRI study being offered to women with BRCA1/BRCA2 mutations. MRI screening currently is not a part of standard care for breast screening.
A number of women indicated at baseline that they were interested in exploring their perceptions and decision-making concerning surgery and in meeting other women making this decision (unpublished data). A few women opted to undergo surgery during the course of the intervention. By postintervention, an additional 10 women chose to undergo either prophylactic oophorectomy or mastectomy. This finding suggests that the group was helpful to women's decision-making. Of those women who reported before the intervention that they wished to receive support regarding surgical decision-making, all completed a decision to either undergo prophylactic surgery or to continue with surveillance and opt to decline the option of prophylactic surgery. For these women relying on monitoring, the group may have provided an opportunity to fully examine their feelings and perspectives regarding surgery, and facilitated confidence in their ability to make a decision to not go forward to mastectomy. Although it is possible that these differences would have occurred in a nonintervention control group, the parallel improvements in psychosocial adjustment were significant and are consistent with the experience of the less ambiguous state that occurs with a reduction in decisional conflict.52 Future studies using standardized measures of decision-making and our qualitative analysis of videotaped group sessions will explore more fully the decision-making process that occurred for prophylactic surgery.
The women reported a high level of satisfaction with the intervention and identified a number of important additional benefits. Clearly, social support in facilitating decision-making and the opportunity to explore such important themes as notification to offspring were important features. The additional quality of life benefits such as personal growth and a sense of mastery are consistent with supportive-expressive group therapy, particularly given its attention to the existential issues associated with being faced with a life-threatening illness or risk, and the opportunity to fully explore, openly and in a non-judgmental manner, these highly personal issues and concerns.27, 30 Future studies should include measurement of these additional variables to further our understanding of mechanisms of change and their significance.
Our supportive-expressive group intervention appeared to be relevant for, and highly acceptable to, the women who carry mutations in BRCA1/BRCA2. The study also demonstrated the feasibility of combining women with a previous diagnosis of cancer with those who have not received a diagnosis of cancer. The groups appeared to be an ideal forum for exploring key issues, such as the notification of test results to family, guilt regarding transmission of a mutation, and decision-making regarding risk-reducing options. Preliminary findings were consistent with an effective intervention. Further research using a randomized, controlled study design is necessary to evaluate the contribution of the intervention we have developed to psychologic adjustment, grief work, surgical decision-making, and medical follow-up.
The authors acknowledge the additional group therapists and genetic counselors who participated in the groups at each of the study sites. They also appreciate the valuable feedback received on an earlier draft of the current article from members of the University Health Network Quality of Life Program manuscript review seminar. They offer a special thank you to Ms. Judy Laceby who assisted in data collection. Finally, they express their gratitude to all of the women who participated in the study.