Psychological issues among children of hereditary breast cancer gene (BRCA1/2) testing participants.

Children growing up in hereditary breast cancer families may experience diminished psychological well-being. In addition to coping with having a cancer-affected parent or close relatives, these children may focus on their own health risks in light of shared genetic information. While knowledge of a parent's BRCA1/2 negative status may allay a subset of children's worries and fears about cancer, others could experience distressing thought patterns over positive test results. The purpose of this preliminary study is to explore conceptions of health, cancer risk, and psychological adjustment among children in families suggestive of carrying BRCA1/2 susceptibility genes. As part of a longitudinal investigation of the outcomes of BRCA1/2 testing in adults, 20 children of a highly select group of 15 mothers (80% previously affected by breast/ovarian cancer) completed a self-report survey of their beliefs and opinions regarding cancer and genetic testing, stress and worry about cancer, and anxiety, depression, and behavior problems. All information was completed at baseline, prior to the mother's receipt of her genetic test result. The data did not suggest unusually elevated cancer worries or psychological adjustment problems in these children at this point in the parental genetic testing process. However, children with more psychological distress symptoms did experience more frequent thoughts of becoming sick and greater cancer worries. To the extent that learning about a parent's positive test result could exacerbate these tendencies, recommendations to promote child psychological and family communications research that monitor such responses are offered.

Effects of coping style and BRCA1 and BRCA2 test results on anxiety among women participating in genetic counseling and testing for breast and ovarian cancer risk.

Using the monitoring process model (MPM), the authors examined the immediate effects of coping style and test results on the psychological distress of women at increased risk for breast and/or ovarian cancers. Cases selected for analysis were 107 probands and relatives of positive probands participating in genetic counseling and testing for heritable cancer risk. Specifically, the authors explored the relationships among coping style (high and low monitoring), test results (BRCA1 and BRCA2 mutation carrier and noncarrier status), and psychological distress (state anxiety). Consistent with the MPM, higher monitoring was associated with greater psychological distress while anticipating genetic test results. After test results were disclosed, greater distress was associated with testing positive for a mutation. The implications of the findings for breast and ovarian cancer patients are discussed.

Psychological response to prenatal genetic counseling and amniocentesis.

The purpose of the present study was to characterize the psychological status (attitudes toward selective abortion, perceived risk, comprehension, patient satisfaction, coping, and state anxiety) of pregnant women at increased risk for fetal genetic anomalies who were referred for prenatal genetic counseling and amniocentesis; to determine which of these factors would predict amniocentesis use; and to identify patient outcomes associated with counseling and testing. Participants were 129 women aged 18 years and older who had one or more fetal genetic risk factors. All were recruited from an urban women's health clinic. The results revealed elevated perceptions of risk and moderate state anxiety despite adequate comprehension of, and patient satisfaction with, the process and content of genetic counseling. Approximately 78% agreed to testing; those who consented were more likely to hold favorable attitudes toward abortion than those who refused. Post-counseling, women experienced decrease in their perceived risk of having a baby born with a birth defect although perceived risk estimates remained higher than actual risks. Anxiety was clinically elevated and highest at the pre-counseling stage, though it dissipated to normal levels over time. Previous experience with prenatal diagnostic testing, increased perceived risk of a birth anomaly, and favorable attitudes toward abortion were independently associated with increased pre-counseling anxiety. Women who were more anxious pre-counseling remained more anxious post-counseling. Coping (high versus low monitoring) was unrelated to anxiety. These findings suggest that women who participate in prenatal counseling and testing may be subject to experience distress and unrealistic perceptions of their risk and may benefit from interventions designed to lessen these states.

Childhood illness-related parenting stress: the pediatric inventory for parents.

OBJECTIVE: To develop a measure of parenting stress related to caring for a child with an illness and to evaluate its psychometric properties with a group of parents of children with cancer. METHODS: One hundred twenty-six parents (105 mothers, 21 fathers) of children (65 boys and 61 girls, M: age: 12.75 years) being followed by an oncology service were assessed using the 42-item self-report Pediatric Inventory for Parents (PIP). Internal consistency was assessed and construct validity was investigated with standardized, general self-report measures of anxiety and parenting stress. RESULTS: Internal consistency reliability for the PIP was high (Cronbach alpha range:80-.96). PIP scores were significantly correlated with a measure of state anxiety and also with parenting stress, demonstrating construct validity. After we controlled for demographic variables and general parenting stress, PIP scores showed strong independent associations with state anxiety. CONCLUSIONS: Preliminary data indicate that the PIP is a reliable and valid tool to assess parenting stress in pediatric oncology populations. As a measure of illness-related parenting stress, the PIP may be used to provide information about parent well-being that extends beyond that obtained from general measures.

Parental communication of BRCA1/2 genetic test results to children.

The aim of this study was to evaluate the likelihood, correlates, and psychological impact of parental communication to children of parents' BRCA1/2 genetic test results for breast cancer risk. Subjects were 133 adult members of high risk families. Sociodemographic, clinical, and psychological distress variables were assessed during a baseline telephone interview conducted prior to patient education and test result notification. Parental communication of test results to children and parental psychological distress and coping efforts were assessed 1 month post-genetic counseling and receipt of test results. Mothers (versus fathers), and persons with higher levels of baseline general distress, were significantly more likely to communicate their test results to children. Post-counseling coping efforts, both active and avoidant, were positively associated with post-counseling distress levels. However, communication of test results to children did not relate to changes in distress. In conclusion, parents with higher levels of pre-counseling general psychological distress may be more likely to communicate their genetic test results to children; however, this act does not minimize their distress and could possibly generate distress in their youngsters. Research is needed to evaluate the process and content of post-test disclosure episodes and the impact on participant, child, and family functioning.

Patient and family reflections on the use of subcutaneous insulin to prevent diabetes: a retrospective evaluation from a pilot prevention trial.

A retrospective, semi-structured telephone interview was used to collect data from 28 of 31 families who participated in a pilot study testing subcutaneous insulin as a means to prevent or delay insulin-dependent diabetes mellitus (IDDM) onset. Interviews were conducted an average of 3 years after the initiation of the subcutaneous insulin protocol. Both the high-risk person (if > or = 8 years of age) and a family member (spouse or parent) were interviewed. Most participants reported that they were distressed to learn that they or a family member were at risk for IDDM, and families readily agreed to initiate subcutaneous insulin therapy. More children than adults reported insulin injections and blood glucose tests were "hard" or "very hard," but noncompliance was more common in adults. Of the high-risk participants interviewed, 58% of children and 100% of adults reported experiencing hypoglycemia, although episodes requiring someone else's assistance were rare, occurring in 38% of the children and 27% of the adults. Participants remained enthusiastic about trial participation; most favored screening programs to identify those at risk for IDDM, believed screening should be conducted regardless of age, believed subcutaneous insulin prevented or delayed IDDM onset, and would recommend subcutaneous insulin therapy to another high-risk individual. In addition, more than 40% of children, parents, and spouses reported that they would have benefited from access to a mental health professional at some point during the trial.

Offering a randomized trial of intensive therapy for IDDM to adolescents. Reasons for refusal, patient characteristics, and recruiter effects.

OBJECTIVE: To identify reasons adolescents refuse to participate in a randomized trial of intensive therapy (IT) for IDDM, to describe the patient characteristics of those who consent and those who refuse to participate, and to examine recruiter effects on trial participation rates. RESEARCH DESIGN AND METHODS: A total of 99 adolescents, age 11-18 years, were provided with the results of the Diabetes Control and Complications Trial and approached for possible study participation by two nurse recruiters. Adolescents refusing the trial were administered a semi-structured interview to describe reasons for study refusal; responses were recorded and later coded into categories. Patient characteristics of consenters and refusers were collected and compared. The differential enrollment rates of the two nurse recruiters were also compared. RESULTS: A total of 56 patients (approximately 57%) agreed to participate; 43 refused. The four most common reasons for study refusal were 1) increased clinic visits (42%), 2) increased insulin injections (30%), 3) increased frequency of self-monitoring of blood glucose (SMBG) (28%), and 4) transportation difficulties (19%). Concerns about randomization to an unwanted treatment condition and fears of hypoglycemia or weight gain were rarely cited. Consenters and refusers did not differ in demographic characteristics, disease status, or family composition. Large differences were found between the two nurse recruiters: one experienced a 60% refusal rate, while the other experienced a 27% refusal rate. CONCLUSIONS: Issues of convenience (increased clinic visits, transportation difficulties) and concerns about the demands of IT (increased injections and SMBG) were the predominant reasons for trial refusal. Patient characteristics did not differentiate consenters from refusers. However, recruiters differed greatly in study refusal rates, suggesting that provider behavior may be an important but understudied aspect of adolescent acceptance of randomized trials in general and IT in particular.

Test-retest reliability for false-belief tasks.

Despite notable variations in children's rate of success on theory of mind tasks and the presumed theoretical implications drawn from a child's success or failure on such tasks, there have been no studies of the test-retest reliability of children's performance on these tasks. Twenty-three children (mean age 49.6 months, SD 8.6) watched three videotaped stories illustrating a false-belief situation: the standard experimenter narrated false-belief task, a minor variant replacing the narration of the story with a dialogue among the characters, and a third version involving a humorous situation. The time elapsed between test and retest was 2-3 weeks and the order of presentation was counterbalanced. Results corroborated previous findings of a developmental trend in the understanding of false-belief questions but, despite a general improvement in children's comprehension of the stories, the test-retest reliability for the false belief questions was poor. Although changes recorded between test-retest sessions frequently occurred in the direction of children answering correctly questions they had previously failed, a subset of children incorrectly answered questions they had initially passed. These findings underscore the need for validation assessments of techniques for studying children's developing theories of mind.

Childhood trichotillomania: clinical phenomenology, comorbidity, and family genetics.

OBJECTIVE: DSM-IV defines trichotillomania as an impulse disorder with rising tension followed by relief or gratification. Alternative formulations view trichotillomania as an internalizing disorder or variant of obsessive-compulsive disorder (OCD). This study addresses this controversy by examining the phenomenology, comorbidity, and family genetics of childhood trichotillomania. METHOD: Fifteen chronic hair-pullers (13 girls), aged 9 through 17 years (mean 12.3 +/- 2.3 years), were systematically assessed. Child Behavior Checklist (CBCL) profiles of the hair-pulling girls were compared with those of 37 girls from a general child psychiatry clinic and of 15 girls with OCD. RESULTS: All the hair-pullers had impairing cosmetic disfigurement; however, 4 subjects (26.7%) denied rising tension or relief. All three groups had comparable global CBCL problem scores. The CBCL symptom profile of the hair-pulling group differed significantly from that of the general clinic group but strongly resembled that of the OCD group. The hair-pulling group, however, had few obsessions or compulsions aside from hair-pulling; two (13%) subjects met criteria for OCD. As a group, hair-pulling subjects had substantial comorbid psychopathology, and a parental history of tics, habits, or obsessive-compulsive symptoms was common. CONCLUSIONS: These findings lend only partial support to the notion of trichotillomania as an OCD-spectrum disorder. Rising tension followed by relief or gratification may not be an appropriate diagnostic criterion for trichotillomania.

Psychological impact of islet cell antibody screening for IDDM on children, adults, and their family members.

OBJECTIVE: To describe the psychological impact of positive islet cell antibody (ICA) screening results in children and adults, as well as their parents and spouses. RESEARCH DESIGN AND METHODS: The psychological impact of ICA screening results was assessed subsequent to subjects' being informed of ICA-positive (ICA+) status and was re-evaluated 4 months later. Impact was measured using the state subscale of the State-Trait Anxiety Inventory (STAI) for adults or the State-Trait Anxiety Inventory for Children (STAIC), as well as structured interviews. A total of 34 ICA+ children, 34 ICA+ adults, 33 parents, and 25 spouses were evaluated. RESULTS: At initial notification of ICA+ status, clinically and statistically significant anxiety was observed in ICA+ children and adults and their family members (P < 0.001). Parents of ICA+ children were more anxious than spouses of ICA+ adults (P < 0.05). Child and parent anxiety were significantly correlated (P < 0.05); more-anxious children lived with more-anxious parents. No significant association was found between ICA+ adults' initial anxiety and their spouses' anxiety. For ICA+ participants and their family members, anxiety dissipated to normal levels in 4 months (P < 0.02). ICA+ children were less likely than parents to believe they would ever develop insulin-dependent diabetes mellitus (IDDM). Nevertheless, 52% of ICA+ children and 24% of ICA+ adults endorsed lifestyle or behavior changes as a result of their IGA+ status. Behavior change was associated with greater initial anxiety in both children and adults (P < 0.05 for both). CONCLUSIONS: These data suggest that notification of ICA+ status has both emotional and behavioral impact. Initial notification of ICA+ status is associated with considerable anxiety in both ICA+ individuals and their family members. In most cases, this initial anxiety appears to dissipate to normal levels over time. However, many ICA+ individuals report initiating lifestyle or health behavior changes in an effort to delay or prevent IDDM onset.