Genetic Predisposition
Conditions
Keywords
genetic testing, genetic counseling, germline testing, digital care plan, cancer predisposition syndrome
Brief summary
Develop and evaluate acceptability, feasibility, and preliminary efficacy of digital care plan and accompanying text message reminders for children and adolescents with a known Cancer Predisposition Syndromes (CPS).
Detailed description
As over 15% of pediatric cancers are associated with a cancer predisposition, it is increasingly becoming standard of care for children with cancer, as well as those with suspected hereditary risk, to be evaluated for germline cancer predisposition. Unfortunately, the increase in pediatric genetic testing has exceeded the pace of research supporting effective cancer surveillance in positive cases. Additionally, guidelines for cancer predisposition management are not easily accessed or understood by families; thus, there is often a disconnect between the understanding and retention of such information relayed to families. In this study, Investigators will develop and evaluate the acceptability, feasibility, and preliminary efficacy of digital care plans for Cancer Predisposition Syndromes (CPS) with accompanying text message reminders for children and adolescents with a known CPS. Digital care plans and text messages will be created for 10 common CPS. Investigators will use a quasi-experimental design with pre and post testing of the same cohort, before and after delivery of care plan and accompanying messages. To evaluate the impact of the digital care plans, Investigators will compare assessments of the same cohorts at 3 and 6 months time points.
Interventions
OTHERDigital Care Plans
Investigators will develop digital care plans for 10 common Cancer Predisposition Syndromes for patients diagnosed within 5 years.
Sponsors
Children's Hospital of Philadelphia
National Institutes of Health (NIH)
National Human Genome Research Institute (NHGRI)
Study design
Allocation
NA
Intervention model
SINGLE_GROUP
Primary purpose
SUPPORTIVE_CARE
Masking
NONE
Intervention model description
Comparison of pre and post testing of same cohort.
Eligibility
Sex/Gender
ALL
Age
12 Years to No maximum
Healthy volunteers
No
Inclusion criteria
Parents 1. Parent or Legal Guardian of a patient with a known cancer predisposition syndrome diagnosed within the last 5 years 2. Received care at the Cancer Predisposition clinic at the Children's Hospital of Philadelphia (CHOP), or other medical institutions (does not need to be receiving follow-up care at CHOP) 3. Appropriate to approach per oncology team/cancer predisposition team 4. No cognitive impairment limiting ability to complete measures 5. Ability to read and speak English fluently Adolescent/Young Adult (AYA) probands 1. Child proband with a known cancer predisposition syndrome diagnosed within the last 5 years 2. Ages 12+ 3. Received care at the Cancer Predisposition clinic at the Children's Hospital of Philadelphia (CHOP), or other medical institutions (does not need to be receiving follow-up care at CHOP) 4. Appropriate to approach per oncology team/cancer predisposition team 5. No cognitive impairment limiting ability to complete measures 6. Ability to read and speak English fluently
Exclusion criteria
* Not meeting any of inclusion criteria
Design outcomes
Primary
| Measure | Time frame | Description |
|---|---|---|
| Change in Knowledge Score | 3 months and 6 months after receipt of digital care plan | The primary outcome is the percentage of correct items on a knowledge survey that was developed for this study and administered via REDCap, measured at baseline (2 weeks prior to receiving the Care Plan/text message intervention), midpoint (3 months post receiving the Care Plan/text message intervention), and final (6 months post receiving the Care Plan/text message intervention) surveys. The change in the mean and standard deviation will be assessed at 3 months and 6 months. Participants evaluated included the caregivers and AYAs aged 12-26. Total scores were generated by summarizing total number of correct responses and generating a percentage. The knowledge measure included a mix of 10-12 multiple choice and true/false questions, CPS dependent. Higher scores indicate higher CPS knowledge and Care Plan content. |
Secondary
| Measure | Time frame | Description |
|---|---|---|
| Acceptability of Use of Digital Care Plans for CPS | 3 Months After Receipt of Digital Care Plan | Acceptability: a 3-question acceptability measure created for this project, scored on a Likert scale from 1=Not at All to 5=Completely. Higher total scores indicate greater acceptability on ease of understanding the Care Plan, new information gained from the Care Plan, and its general usefulness. Scores range from 0-15, with higher scores indicating greater acceptability of the Care Plan. |
| Feasibility of Use of Digital Care Plans for CPS for Patients Diagnosed Within 5 Years | Up to 6 months after receipt of digital care plan | Feasibility will be assessed with absence of mean number of technical difficulties of Care Plan Creation by the Clinical Research Coordinator (CRC) in under 15 minutes in a busy clinical setting. The CRC made note of any technical difficulties at creation of Care Plan via REDCap. |
| Satisfaction With Decision (SDS) to Have Germline Testing | Up to 6 months after receipt of digital care plan | Satisfaction with decision to have germline testing will be measured by participants' completion of electronic questionnaires administered via REDCap. Satisfaction with Decision Scale (SDS): a 6-question decision satisfaction measure, scored on a Likert scale from 1= Strongly Disagree to 5= Strongly Agree. Higher total scores indicate greater satisfaction with decisions for pursuing genetic testing. Scores range from 0-30, with higher scores indicating greater satisfaction. |
| Change in Cancer-Related Anxiety | Up to 6 months after receipt of digital care plan | This 15-item measure assesses cancer-related anxiety through two subscales (intrusive thinking and avoidance). Responses are measured on a 4-point Likert scale with the following anchors: 0 "not at all", 1 "rarely", 3 "sometimes", and 5 "often". Scores range from 0 to 35 for intrusion, 0 to 40 for avoidance, and 0 to 75 for the total IES. Higher scores indicate greater cancer-related anxiety. Cancer-related anxiety will be measured by participants' completion of electronic questionnaires administered via REDCap. |
Countries
United States
Contacts
PRINCIPAL_INVESTIGATORSuzanne MacFarland, MD
Children's Hospital of Philadelphia
Participant flow
Recruitment details
Patients were identified via clinic rosters of the Cancer Predisposition Program and through screening of electronic health records. Patients were approved for eligibility by the attending physician and genetic counselor scheduled to see the patient in the following two weeks. Participants were then approached by phone or in clinic to obtain informed study consent.
Pre-assignment details
Participants enrolled include AYA probands aged 12-26 and their caregiver(s).
Baseline characteristics
| Characteristic | — |
|---|---|
| Age, Continuous | 37.6 Years (1 d.p.) STANDARD_DEVIATION 9.7 |
| Ethnicity (NIH/OMB) Hispanic or Latino | 1 Participants |
| Ethnicity (NIH/OMB) Not Hispanic or Latino | 12 Participants |
| Ethnicity (NIH/OMB) Unknown or Not Reported | 0 Participants |
| Race (NIH/OMB) American Indian or Alaska Native | 0 Participants |
| Race (NIH/OMB) Asian | 6 Participants |
| Race (NIH/OMB) Black or African American | 3 Participants |
| Race (NIH/OMB) More than one race | 1 Participants |
| Race (NIH/OMB) Native Hawaiian or Other Pacific Islander | 0 Participants |
| Race (NIH/OMB) Unknown or Not Reported | 20 Participants |
| Race (NIH/OMB) White | 142 Participants |
| Sex/Gender, Customized Sex Female | 99 Participants |
| Sex/Gender, Customized Sex Male | 2 Participants |
Adverse events
| Event type | EG000 affected / at risk | EG001 affected / at risk |
|---|---|---|
| deaths Total, all-cause mortality | 0 / 164 | 0 / 13 |
| other Total, other adverse events | 0 / 164 | 0 / 13 |
| serious Total, serious adverse events | 0 / 164 | 0 / 13 |
Outcome results
None listed