Reporting Adult-Onset Genomic Results to Pediatric Biobank Participants and Parents
概览
- 阶段
- 早期 1 期
- 干预措施
- Child(ren) receive an adult-onset result
- 疾病 / 适应症
- Hereditary Breast and Ovarian Cancer Syndrome
- 发起方
- Geisinger Clinic
- 入组人数
- 162
- 试验地点
- 2
- 主要终点
- Initiation of risk reduction behavior
- 状态
- 已完成
- 最后更新
- 23天前
概览
简要总结
The Investigators will conduct a longitudinal, mixed-methods cohort study to assess primary and secondary psychosocial outcomes among MyCode adolescent participants and their parents, and health behaviors of children who received an adult- or pediatric-onset genomic result. Data will be gathered via quantitative surveys using validated measures of distress, family functioning, quality of life, body image, perceived cancer/heart disease risk, genetic counseling satisfaction, genomics knowledge, and adjustment to genetic information; qualitative interviews with adolescents and parents; and electronic health records review of children's initiation of risk reduction behaviors. The investigators will also conduct empirical and theoretical legal research to examine the loss of chance doctrine and its applicability to genomic research.
详细描述
The Investigators propose a longitudinal, observational cohort study using mixed methods to compare change in psychosocial outcomes and health behaviors among three study groups of MyCode adolescents and three groups of their parents: 1. Group 1 - parents of child(ren) with a pathogenic variant in a gene associated with adult onset of disease 2. Group 2 - parents of child(ren) with a pathogenic variant in a gene associated with pediatric onset of disease or with risk reduction interventions that begin in childhood 3. Group 3 - parents of child(ren) who tested negative for the familial genetic variant 4. Group 4 - adolescents with a pathogenic variant in a gene associated with adult onset of disease 5. Group 5 - adolescents with a pathogenic variant in a gene associated with pediatric onset of disease or with risk reduction interventions that begin in childhood 3\. Group 6 - adolescents who tested negative for the familial genetic variant The Investigators will use the current existing MyCode list of actionable genes designated as actionable by the American College of Medical Genetics and Genomics. Parents of pediatric MyCode participants will be offered the opportunity to participate in the study prior to learning to which group they belong. Consistent with Geisinger policy, children ages 7-17 will be asked to give assent to participate. If a child does not want to assent to participate, he or she will not be enrolled into the study (regardless of their parents' preference regarding enrollment). Parents of children who do not give assent will be ineligible to participate. Parents who decline participation when their child is suspected to have a pathogenic adult-onset result will have their child's sample held for clinical confirmation until the child reaches age 18 years. Parents who decline participation when their child is suspected to have a pathogenic pediatric-onset result will proceed to clinical confirmation of the result and, if confirmed, follow the established clinical return procedure. This recruitment approach is consistent with the MyCode philosophy of notifying participants of actionable findings. Parent-participants will be asked to assess psychosocial outcomes for themselves and for their children. Consistent with co-investigator Angela Bradbury's research on the experience of adolescent girls from families at increased risk for breast cancer, pediatric participants ages 11-17 years at enrollment (i.e., adolescents) will also participate in quantitative surveys and qualitative interviews. Psychosocial variables such as anxiety and depression will be assessed among parents and adolescents at enrollment (T1), after which those suspected of having a pathogenic variant will proceed to clinical confirmation of that variant. Those whose variant is confirmed clinically as pathogenic or likely pathogenic will then be scheduled for a disclosure appointment. These appointments will be conducted by a genetic counselor and psychologist, who will perform psychosocial assessment, conduct therapeutic consults as needed, and conduct periodic psychosocial assessments of adolescent participants with adult-onset results. Participants with suspected pathogenic variants that are not confirmed clinically and participants without a suspected pathogenic variant will be scheduled for a study visit to notify them of their group status and remind them to follow up with their pediatrician if they have significant personal or family history of cancer or heart disease. Validated surveys will be used to measure outcomes in each study group at 1 month (T3), 6 months (T4) and 12 months (T5) post-disclosure visit. The investigators will conduct qualitative interviews with a subset of at least 45 participants in each of the two study groups who receive a genomic result to better understand the lived experience of adolescents with an actionable genomic finding and their parents. Data collection will continue after the grant funding ends because of Geisinger Research Division's commitment to following the study cohort. To address the legal specific aim, Dr. Wagner will lead the study team's legal experts in examining and monitoring the loss of chance doctrine in medical malpractice cases in federal and state courts across the United States and in monitoring legislative developments relating to the loss of chance doctrine as it applies to returning adult-onset genomic results to children.
研究者
Adam Buchanan
Associate Professor and Chair
Geisinger Clinic
入排标准
入选标准
- •Any pediatric MyCode participant (ages 0-17) OR
- •Parent of a pediatric MyCode participant who has given assent to participate in this study.
排除标准
- •Individuals who have already had genetic counseling for any of the actionable target conditions as part of their routine clinical care.
- •Individuals who have already had genetic counseling for any of the actionable target conditions through their participation in another research study.
研究组 & 干预措施
Group 1 - Parents of child(ren) who receive an adult-onset result
Compare change in psychosocial outcomes of parents of child(ren) with a pathogenic variant in a gene associated with adult onset of disease.
干预措施: Child(ren) receive an adult-onset result
Group 2 - Parents of child(ren) who receive a pediatric-onset result
Compare change in psychosocial outcomes among parents of child(ren) with a pathogenic variant in a gene associated with pediatric onset of disease or with risk reduction interventions that begin in childhood.
干预措施: Child(ren) received a pediatric-onset result
Group 3 - Parents of child(ren) negative for familial variant
Parents of child(ren) who tested negative for the familial genetic variant
干预措施: Control - Negative Result
Group 4 - Adolescents with adult-onset variant
Adolescents with adult-onset genetic variant
干预措施: Adolescents who received adult-onset result
Group 5 - Adolescents with pediatric-onset variant
Adolescents with pediatric-onset genetic variant
干预措施: Adolescents who received a pediatric-onset result
Group 6 - Adolescents negative for familial variant
Adolescents who tested negative for familial genetic variant
干预措施: Adolescent controls - negative for familial variant
结局指标
主要结局
Initiation of risk reduction behavior
时间窗: 12 months post-disclosure to pediatric proband
Initiation of risk reduction behavior (yes/no) among parents with familial gene variant.
The General Functioning 12-item subscale (GF12) of The McMaster Family Assessment Device (FAD)
时间窗: Baseline, 1, 6, & 12 months post-disclosure
The GF12 subscale is made up of 12 items, six items that reflect healthy family functioning and the other six items reflecting unhealthy functioning. Scoring is on a 4-point scale (from 1 for strongly agree to 4 for strongly disagree) with the scale for the negatively worded items reversed. The total score is then divided by the number of items on the subscale giving a total score ranging from 1.0 (best functioning) to 4.0 (worse functioning)
The Hospital Anxiety and Depression Scale (HADS)
时间窗: Baseline, 1, 6, & 12 months post-disclosure
The HADS questionnaire is a 14-item scale comprised of seven questions for anxiety and seven questions for depression. Each item is scored from 0-3. The total scoring is as follows: 8-10 = Mild, 11-14 = Moderate, 15-21 = Severe. Scoring for anxiety and depression are to be completed separately. For both scales, a total score of less than 7 indicates non-cases.
Uptake of cascade testing
时间窗: 12 months post-disclosure to pediatric proband
Uptake of cascade testing (yes/no) among parents.
Health-Related Quality of Life (HRQOL)
时间窗: Baseline, 1, 6, & 12 months post-disclosure
Healthy days are the positive complementary form of unhealthy days. Healthy days estimates the number of recent days when a person's physical and mental health was good (or better) and is calculated by subtracting the number of unhealthy days from 30 days. Unhealthy days are an estimate of the overall number of days during the previous 30 days when the respondent felt that either his or her physical or mental health was not good. To obtain this estimate, responses to questions 2 and 3 are combined to calculate a summary index of overall unhealthy days, with a logical maximum of 30 unhealthy days. For example, a person who reports 4 physically unhealthy days and 2 mentally unhealthy days is assigned a value of 6 unhealthy days, and someone who reports 30 physically unhealthy days and 30 mentally unhealthy days is assigned the maximum of 30 unhealthy days.
The Hospital Anxiety and Depression Scale (HADS) - Anxiety Subscale
时间窗: Baseline
The HADS questionnaire is a 14-item scale comprised of seven questions for anxiety and seven questions for depression. Each item is scored from 0-3. The total scoring is as follows: 8-10 = Mild, 11-14 = Moderate, 15-21 = Severe. Scoring for anxiety and depression are to be completed separately. For both scales, a total score of less than 7 indicates non-cases. Total range is 0 (lowest anxiety) - 21 (most severe anxiety).
The Hospital Anxiety and Depression Scale (HADS) - Depression Subscale
时间窗: Baseline
The HADS questionnaire is a 14-item scale comprised of seven questions for anxiety and seven questions for depression. Each item is scored from 0-3. The total scoring is as follows: 8-10 = Mild, 11-14 = Moderate, 15-21 = Severe. Scoring for anxiety and depression are to be completed separately. For both scales, a total score of less than 7 indicates non-cases. Total range is 0 (lowest) - 21 (most severe).
Health-Related Quality of Life (HRQOL)
时间窗: 1-month post
Healthy days are the positive complementary form of unhealthy days. Healthy days estimates the number of recent days when a person's physical and mental health was good (or better) and is calculated by subtracting the number of unhealthy days from 30 days. Unhealthy days are an estimate of the overall number of days during the previous 30 days when the respondent felt that either his or her physical or mental health was not good. To obtain this estimate, responses to questions 2 and 3 are combined to calculate a summary index of overall unhealthy days, with a logical maximum of 30 unhealthy days. For example, a person who reports 4 physically unhealthy days and 2 mentally unhealthy days is assigned a value of 6 unhealthy days, and someone who reports 30 physically unhealthy days and 30 mentally unhealthy days is assigned the maximum of 30 unhealthy days. Here we report the number and % of participants who reported fair or poor HRQoL.
The General Functioning 12-item Subscale (GF12) of The McMaster Family Assessment Device (FAD)
时间窗: Baseline
The GF12 subscale is made up of 12 items, six items that reflect healthy family functioning and the other six items reflecting unhealthy functioning. Scoring is on a 4-point scale (from 1 for strongly agree to 4 for strongly disagree) with the scale for the negatively worded items reversed. The total score is then divided by the number of items on the subscale giving a total score ranging from 1.0 (best functioning) to 4.0 (worse functioning)
Health-Related Quality of Life (HRQOL)
时间窗: Baseline
Healthy days are the positive complementary form of unhealthy days. Healthy days estimates the number of recent days when a person's physical and mental health was good (or better) and is calculated by subtracting the number of unhealthy days from 30 days. Unhealthy days are an estimate of the overall number of days during the previous 30 days when the respondent felt that either his or her physical or mental health was not good. To obtain this estimate, responses to questions 2 and 3 are combined to calculate a summary index of overall unhealthy days, with a logical maximum of 30 unhealthy days. For example, a person who reports 4 physically unhealthy days and 2 mentally unhealthy days is assigned a value of 6 unhealthy days, and someone who reports 30 physically unhealthy days and 30 mentally unhealthy days is assigned the maximum of 30 unhealthy days. Here we report the number and % of participants who reported fair or poor HRQoL.
Initiation of Risk Reduction Behavior
时间窗: 6+ months post-disclosure to pediatric proband
Initiation of risk reduction behavior (yes/no) among children with familial gene variant. Not that this is among children of all ages (not just adolescents). Counts are of participants who initiated a risk reduction behavior. Data were collected via chart review for pre-selected risk reduction procedures specific to each genetic condition. Time in months from results disclosure date to date of risk reduction behavior was tracked.
The Hospital Anxiety and Depression Scale (HADS) - Anxiety Subscale
时间窗: 1-month post-disclosure
The HADS questionnaire is a 14-item scale comprised of seven questions for anxiety and seven questions for depression. Each item is scored from 0-3. The total scoring is as follows: 8-10 = Mild, 11-14 = Moderate, 15-21 = Severe. Scoring for anxiety and depression are to be completed separately. For both scales, a total score of less than 7 indicates non-cases.Total range is 0 (lowest anxiety) - 21 (most severe anxiety).
The Hospital Anxiety and Depression Scale (HADS) - Anxiety Subscale
时间窗: 6-month post-disclosure
The HADS questionnaire is a 14-item scale comprised of seven questions for anxiety and seven questions for depression. Each item is scored from 0-3. The total scoring is as follows: 8-10 = Mild, 11-14 = Moderate, 15-21 = Severe. Scoring for anxiety and depression are to be completed separately. For both scales, a total score of less than 7 indicates non-cases. Total range is 0 (lowest anxiety) - 21 (most severe anxiety).
The Hospital Anxiety and Depression Scale (HADS) - Anxiety Subscale
时间窗: 12-month post-disclosure
The HADS questionnaire is a 14-item scale comprised of seven questions for anxiety and seven questions for depression. Each item is scored from 0-3. The total scoring is as follows: 8-10 = Mild, 11-14 = Moderate, 15-21 = Severe. Scoring for anxiety and depression are to be completed separately. For both scales, a total score of less than 7 indicates non-cases. Total range is 0 (lowest anxiety) - 21 (most severe anxiety).
The Hospital Anxiety and Depression Scale (HADS) - Depression Subscale
时间窗: 1-month post-disclosure
The HADS questionnaire is a 14-item scale comprised of seven questions for anxiety and seven questions for depression. Each item is scored from 0-3. The total scoring is as follows: 8-10 = Mild, 11-14 = Moderate, 15-21 = Severe. Scoring for anxiety and depression are to be completed separately. For both scales, a total score of less than 7 indicates non-cases. Total range is 0 (lowest) - 21 (most severe).
The Hospital Anxiety and Depression Scale (HADS) - Depression Subscale
时间窗: 6-month post-disclosure
The HADS questionnaire is a 14-item scale comprised of seven questions for anxiety and seven questions for depression. Each item is scored from 0-3. The total scoring is as follows: 8-10 = Mild, 11-14 = Moderate, 15-21 = Severe. Scoring for anxiety and depression are to be completed separately. For both scales, a total score of less than 7 indicates non-cases. Total range is 0 (lowest) - 21 (most severe).
The Hospital Anxiety and Depression Scale (HADS) - Depression Subscale
时间窗: 12-month post
The HADS questionnaire is a 14-item scale comprised of seven questions for anxiety and seven questions for depression. Each item is scored from 0-3. The total scoring is as follows: 8-10 = Mild, 11-14 = Moderate, 15-21 = Severe. Scoring for anxiety and depression are to be completed separately. For both scales, a total score of less than 7 indicates non-cases. Total range is 0 (lowest) - 21 (most severe).
The General Functioning 12-item Subscale (GF12) of The McMaster Family Assessment Device (FAD)
时间窗: 1-month post
The GF12 subscale is made up of 12 items, six items that reflect healthy family functioning and the other six items reflecting unhealthy functioning. Scoring is on a 4-point scale (from 1 for strongly agree to 4 for strongly disagree) with the scale for the negatively worded items reversed. The total score is then divided by the number of items on the subscale giving a total score ranging from 1.0 (best functioning) to 4.0 (worse functioning)
The General Functioning 12-item Subscale (GF12) of The McMaster Family Assessment Device (FAD)
时间窗: 6-month post
The GF12 subscale is made up of 12 items, six items that reflect healthy family functioning and the other six items reflecting unhealthy functioning. Scoring is on a 4-point scale (from 1 for strongly agree to 4 for strongly disagree) with the scale for the negatively worded items reversed. The total score is then divided by the number of items on the subscale giving a total score ranging from 1.0 (best functioning) to 4.0 (worse functioning)
The General Functioning 12-item Subscale (GF12) of The McMaster Family Assessment Device (FAD)
时间窗: 12-month post
The GF12 subscale is made up of 12 items, six items that reflect healthy family functioning and the other six items reflecting unhealthy functioning. Scoring is on a 4-point scale (from 1 for strongly agree to 4 for strongly disagree) with the scale for the negatively worded items reversed. The total score is then divided by the number of items on the subscale giving a total score ranging from 1.0 (best functioning) to 4.0 (worse functioning)
Health-Related Quality of Life (HRQOL)
时间窗: 6-month post
Healthy days are the positive complementary form of unhealthy days. Healthy days estimates the number of recent days when a person's physical and mental health was good (or better) and is calculated by subtracting the number of unhealthy days from 30 days. Unhealthy days are an estimate of the overall number of days during the previous 30 days when the respondent felt that either his or her physical or mental health was not good. To obtain this estimate, responses to questions 2 and 3 are combined to calculate a summary index of overall unhealthy days, with a logical maximum of 30 unhealthy days. For example, a person who reports 4 physically unhealthy days and 2 mentally unhealthy days is assigned a value of 6 unhealthy days, and someone who reports 30 physically unhealthy days and 30 mentally unhealthy days is assigned the maximum of 30 unhealthy days. Here we report the number and % of participants who reported fair or poor HRQoL.
Health-Related Quality of Life (HRQOL)
时间窗: 12-month post
Healthy days are the positive complementary form of unhealthy days. Healthy days estimates the number of recent days when a person's physical and mental health was good (or better) and is calculated by subtracting the number of unhealthy days from 30 days. Unhealthy days are an estimate of the overall number of days during the previous 30 days when the respondent felt that either his or her physical or mental health was not good. To obtain this estimate, responses to questions 2 and 3 are combined to calculate a summary index of overall unhealthy days, with a logical maximum of 30 unhealthy days. For example, a person who reports 4 physically unhealthy days and 2 mentally unhealthy days is assigned a value of 6 unhealthy days, and someone who reports 30 physically unhealthy days and 30 mentally unhealthy days is assigned the maximum of 30 unhealthy days. Here we report the number and % of participants who reported fair or poor HRQoL.
Revised Children's Anxiety and Depression Scale - Anxiety Subscale
时间窗: baseline
Anxiety subscale of the brief (25-item) version of Revised Children's Anxiety and Depression Scale. Items are scored from 0 (never) to 3 (always) and cumulative scores are converted to T scores per measure guidelines based on participants' school grade and sex. RCADS raw scores were converted to age- and sex-normed T-scores, where a T-score of 48 represents the population mean and the standard deviation is 14 at baseline. T score of greater than or equal to 70 exceeds the clinical threshold (i.e., higher T score = higher anxiety).
Revised Children's Anxiety and Depression Scale - Anxiety Subscale
时间窗: 1-month (T2)
Anxiety subscale of the brief (25-item) version of Revised Children's Anxiety and Depression Scale. Items are scored from 0 (never) to 3 (always) and cumulative scores are converted to T scores per measure guidelines based on participants' school grade and sex. RCADS raw scores were converted to age- and sex-normed T-scores, where a T-score of 48 represents the population mean and the standard deviation is 14 at baseline. T score of greater than or equal to 70 exceeds the clinical threshold (i.e., higher T score = higher anxiety).
Revised Children's Anxiety and Depression Scale - Anxiety Subscale
时间窗: 6-month (T3)
Anxiety subscale of the brief (25-item) version of Revised Children's Anxiety and Depression Scale. Items are scored from 0 (never) to 3 (always) and cumulative scores are converted to T scores per measure guidelines based on participants' school grade and sex. RCADS raw scores were converted to age- and sex-normed T-scores, where a T-score of 48 represents the population mean and the standard deviation is 14 at baseline. T score of greater than or equal to 70 exceeds the clinical threshold (i.e., higher T score = higher anxiety).
Revised Children's Anxiety and Depression Scale - Anxiety Subscale
时间窗: 12-month (T4)
Anxiety subscale of the brief (25-item) version of Revised Children's Anxiety and Depression Scale. Items are scored from 0 (never) to 3 (always) and cumulative scores are converted to T scores per measure guidelines based on participants' school grade and sex. RCADS raw scores were converted to age- and sex-normed T-scores, where a T-score of 48 represents the population mean and the standard deviation is 14 at baseline. T score of greater than or equal to 70 exceeds the clinical threshold (i.e., higher T score = higher anxiety).
Revised Children's Anxiety and Depression Scale - Depression Subscale
时间窗: Baseline
Depression subscale of the brief (25-item) version of Revised Children's Anxiety and Depression Scale. Items are scored from 0 (never) to 3 (always) and cumulative scores are converted to T scores per measure guidelines based on participants' school grade and sex. RCADS raw scores were converted to age- and sex-normed T-scores, where a T-score of 48 represents the population mean and the standard deviation is 14 at baseline. T score of greater than or equal to 70 exceeds the clinical threshold (i.e., higher T score = higher depression).
Revised Children's Anxiety and Depression Scale - Depression Subscale
时间窗: 1-month (T2)
Depression subscale of the brief (25-item) version of Revised Children's Anxiety and Depression Scale. Items are scored from 0 (never) to 3 (always) and cumulative scores are converted to T scores per measure guidelines based on participants' school grade and sex. RCADS raw scores were converted to age- and sex-normed T-scores, where a T-score of 48 represents the population mean and the standard deviation is 14 at baseline. T score of greater than or equal to 70 exceeds the clinical threshold (i.e., higher T score = higher depression).
Revised Children's Anxiety and Depression Scale - Depression Subscale
时间窗: 6-month (T3)
Depression subscale of the brief (25-item) version of Revised Children's Anxiety and Depression Scale. Items are scored from 0 (never) to 3 (always) and cumulative scores are converted to T scores per measure guidelines based on participants' school grade and sex. RCADS raw scores were converted to age- and sex-normed T-scores, where a T-score of 48 represents the population mean and the standard deviation is 14 at baseline. T score of greater than or equal to 70 exceeds the clinical threshold (i.e., higher T score = higher depression).
Revised Children's Anxiety and Depression Scale - Depression Subscale
时间窗: 12-month (T4)
Depression subscale of the brief (25-item) version of Revised Children's Anxiety and Depression Scale. Items are scored from 0 (never) to 3 (always) and cumulative scores are converted to T scores per measure guidelines based on participants' school grade and sex. RCADS raw scores were converted to age- and sex-normed T-scores, where a T-score of 48 represents the population mean and the standard deviation is 14 at baseline. T score of greater than or equal to 70 exceeds the clinical threshold (i.e., higher T score = higher depression).
General Functioning 12-item Subscale (GF12) of the McMaster Family Assessment Device (FAD)
时间窗: baseline
The GF12 subscale is made up of 12 items, six items that reflect healthy family functioning and the other six items reflecting unhealthy functioning. Scoring is on a 4-point scale (from 1 for strongly agree to 4 for strongly disagree) with the scale for the negatively worded items reversed. The total score is then divided by the number of items on the subscale giving a total score ranging from 1.0 (best functioning) to 4.0 (worse functioning)
General Functioning 12-item Subscale (GF12) of the McMaster Family Assessment Device (FAD)
时间窗: 1-month (T2)
The GF12 subscale is made up of 12 items, six items that reflect healthy family functioning and the other six items reflecting unhealthy functioning. Scoring is on a 4-point scale (from 1 for strongly agree to 4 for strongly disagree) with the scale for the negatively worded items reversed. The total score is then divided by the number of items on the subscale giving a total score ranging from 1.0 (best functioning) to 4.0 (worse functioning)
General Functioning 12-item Subscale (GF12) of the McMaster Family Assessment Device (FAD)
时间窗: 6-month (T2)
The GF12 subscale is made up of 12 items, six items that reflect healthy family functioning and the other six items reflecting unhealthy functioning. Scoring is on a 4-point scale (from 1 for strongly agree to 4 for strongly disagree) with the scale for the negatively worded items reversed. The total score is then divided by the number of items on the subscale giving a total score ranging from 1.0 (best functioning) to 4.0 (worse functioning)
General Functioning 12-item Subscale (GF12) of the McMaster Family Assessment Device (FAD)
时间窗: 12-month (T2)
The GF12 subscale is made up of 12 items, six items that reflect healthy family functioning and the other six items reflecting unhealthy functioning. Scoring is on a 4-point scale (from 1 for strongly agree to 4 for strongly disagree) with the scale for the negatively worded items reversed. The total score is then divided by the number of items on the subscale giving a total score ranging from 1.0 (best functioning) to 4.0 (worse functioning)
Health-Related Quality of Life (HRQOL)
时间窗: 1-month (T2)
Healthy days are the positive complementary form of unhealthy days. Healthy days estimates the number of recent days when a person's physical and mental health was good (or better) and is calculated by subtracting the number of unhealthy days from 30 days. Unhealthy days are an estimate of the overall number of days during the previous 30 days when the respondent felt that either his or her physical or mental health was not good. To obtain this estimate, responses to questions 2 and 3 are combined to calculate a summary index of overall unhealthy days, with a logical maximum of 30 unhealthy days. For example, a person who reports 4 physically unhealthy days and 2 mentally unhealthy days is assigned a value of 6 unhealthy days, and someone who reports 30 physically unhealthy days and 30 mentally unhealthy days is assigned the maximum of 30 unhealthy days. Here we report the number and % of participants who reported fair or poor HRQoL.
Health-Related Quality of Life (HRQOL)
时间窗: 6-month (T3)
Healthy days are the positive complementary form of unhealthy days. Healthy days estimates the number of recent days when a person's physical and mental health was good (or better) and is calculated by subtracting the number of unhealthy days from 30 days. Unhealthy days are an estimate of the overall number of days during the previous 30 days when the respondent felt that either his or her physical or mental health was not good. To obtain this estimate, responses to questions 2 and 3 are combined to calculate a summary index of overall unhealthy days, with a logical maximum of 30 unhealthy days. For example, a person who reports 4 physically unhealthy days and 2 mentally unhealthy days is assigned a value of 6 unhealthy days, and someone who reports 30 physically unhealthy days and 30 mentally unhealthy days is assigned the maximum of 30 unhealthy days. Here we report the number and % of participants who reported fair or poor HRQoL.
Health-Related Quality of Life (HRQOL)
时间窗: 12-month (T3)
Healthy days are the positive complementary form of unhealthy days. Healthy days estimates the number of recent days when a person's physical and mental health was good (or better) and is calculated by subtracting the number of unhealthy days from 30 days. Unhealthy days are an estimate of the overall number of days during the previous 30 days when the respondent felt that either his or her physical or mental health was not good. To obtain this estimate, responses to questions 2 and 3 are combined to calculate a summary index of overall unhealthy days, with a logical maximum of 30 unhealthy days. For example, a person who reports 4 physically unhealthy days and 2 mentally unhealthy days is assigned a value of 6 unhealthy days, and someone who reports 30 physically unhealthy days and 30 mentally unhealthy days is assigned the maximum of 30 unhealthy days. Here we report the number and % of participants who reported fair or poor HRQoL.
次要结局
- Genetic Counseling Satisfaction Scale (GCSS)(1 month post-disclosure)
- Body Image instrument(Baseline, 1, 6, & 12 months post-disclosure)
- Decision Regret scale(1 & 12 months post-disclosure)
- Children's Revised Impact of Events scale(1, 6, &12 months post-disclosure)
- Psychological Adaptation to Genetic Information scale (PAGIS)(1, 6, & 12 months post-disclosure)
- Self-Esteem Scale(Baseline, 1, 6, & 12 months post-disclosure)
- Health Education Impact Questionnaire (heiQ)(1, 6, & 12 months post-disclosure)
- Decision Regret Scale(1- month post-disclosure)
- Decision Regret Scale(12- month post-disclosure)