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| ID | Type | Description | Link |
|---|---|---|---|
| R21HG011912 | U.S. NIH Grant/Contract | View source |
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| Name | Class |
|---|---|
| National Institutes of Health (NIH) | NIH |
| National Human Genome Research Institute (NHGRI) | NIH |
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Develop and evaluate the acceptability, feasibility, and preliminary efficacy of an informational video on paired tumor/normal testing for children and adolescents with a new diagnosis of cancer, tumors or other diagnosis.
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 establishing best practices to optimize delivery of care for patients undergoing testing and their families. Tumor/normal genetic testing (testing of both tumor tissue and a paired normal sample) at time of cancer diagnosis or relapse is now widespread in pediatric oncology to improve cancer diagnostics, prognostics, and treatment; this testing also has potential to uncover underlying cancer predisposition syndromes with lifelong implications. Disseminating information at the time of cancer diagnosis is difficult, and is best done by a provider with expertise in cancer genetics. Thus, Investigators will develop an informational video for use prior to tumor/normal genetic testing to augment genetic counseling resources to support patients and families.
In this study, Investigators will develop and evaluate the acceptability, feasibility, and preliminary efficacy of an informational video on paired tumor/normal testing for children and adolescents with a new diagnosis of cancer, tumors or other diagnosis. Investigators will use a non randomized trial whereby a convenience sample of patients/families will be recruited to be controls in Year 1, followed by a convenience sample that will be allocated the video intervention In Year 2. To evaluate the impact of the video intervention, Investigators will compare assessments of two cohorts- an unexposed (no video intervention, Year 1) and exposed (video intervention, Year 2).
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| Label | Type | Description | Intervention Names |
|---|---|---|---|
| Without Video | No Intervention | In year 1, investigators will recruit families of patients receiving the current standard-of-care approach to tumor-normal genetic testing with provider based education. | |
| With Video | Experimental | In year 2, investigators will recruit families of patients receiving the updated standard-of-care approach to tumor-normal genetic testing with provider based education and an educational video. |
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| Name | Type | Description | Arm Group Labels | Other Names |
|---|---|---|---|---|
| Educational Video | Other | Investigators will develop an informational video to be presented within one week of diagnosis of a tumor to those referred for tumor normal paired genetic testing. |
| Measure | Description | Time Frame |
|---|---|---|
| Percentage of Correct Items on Knowledge Questionnaire - Before Results | The primary outcome is the percentage of correct items on a genetic knowledge survey that was developed for this study, measured at baseline and before receipt of genetic test results. Participants evaluated included the primary caregivers, secondary caregivers and probands aged 12+. Total scores were generated by summarizing total number of correct responses and generating a percentage. The Genetic Knowledge measure included 12 true/false questions. Higher scores indicate higher genetic knowledge. | Up to 4 weeks after testing |
| Measure | Description | Time Frame |
|---|---|---|
| Percentage of Correct Items on Knowledge Questionnaire - After Results | A secondary analysis includes data from all caregivers' (primary and secondary) and probands' (over 12 years of age) of percentage of correct items on a genetic knowledge survey that was developed for this study, repeated at a second timepoint (T2) after receipt of genetic test results. The knowledge score is a 12-point true/false questionnaire. Total scores were generated by summarizing total number of correct responses and generating a percentage. The Genetic Knowledge measure included 12 true/false questions. Higher scores indicate higher genetic knowledge. |
| Measure | Description | Time Frame |
|---|---|---|
| Satisfaction With Decision Scale (SDS) - Before Results | 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. | Up to 4 weeks after testing |
Inclusion Criteria:
Parents
Adolescent/Young Adult (AYA) probands
Exclusion Criteria:
1) Not meeting any of inclusion criteria.
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| Name | Affiliation | Role |
|---|---|---|
| Suzanne MacFarland, MD | Children's Hospital of Philadelphia | Principal Investigator |
| Facility | Status | City | State | ZIP | Country | Contacts |
|---|---|---|---|---|---|---|
| Children's Hospital of Philadelphia | Philadelphia | Pennsylvania | 19104 | United States |
Investigators are committed to making all data, protocols, tools, and materials available to the scientific community, in compliance with Children's Hospital of Philadelphia policies and NIH "Sharing Data and Resources" statement, NIH Grants Policy Statement, and the NIH Office of Extramural Research, Division of Extramural Inventions & Technology Resources (DEITR) Intellectual Property Policy. This includes sharing of protocols freely with collaborators and sharing of data through peer-reviewed publication and presentation in scientific meetings. All members of the research team will comply with the NIH policy on sharing data by protecting the privacy and rights of human subjects involved in the research at all times. All members of the research team with comply with the Health Insurance Portability and Accountability Act of 1996 (HIPAA) and its accompanying regulations.
Upon request
Upon request
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Participants enrolled in year 1 were assigned to the control group (without video). Participants enrolled in year 2 were assigned to the intervention group (with video).
Participants enrolled include AYA probands aged 12+ and their caregiver(s).
Patients were identified via existing Cancer Center informatics systems and the Division of Genomic Diagnostics as undergoing paired tumor/normal sequencing. Patients were recruited at least one week after cancer diagnosis and no more than 4 weeks after tumor/normal testing is sent. Families were then approached by phone or in clinic to confirm study eligibility and (if appropriate) obtain informed study consent.
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| ID | Title | Description |
|---|---|---|
| FG000 | Year 1: Without Video - Caregivers | In year 1, investigators will recruit families of patients receiving the current standard-of-care approach to tumor-normal genetic testing with provider-based education. Participants enrolled in this group included caregivers of probands receiving genetic testing consented to participate. |
| FG001 | Year 2: With Video - Caregivers | In year 2, investigators will recruit families of patients receiving the updated standard-of-care approach to tumor-normal genetic testing with provider-based education and an educational video. Participants enrolled in this group included caregivers of probands receiving genetic testing consented to participate. |
| FG002 | Year 1: Without Video - Probands | In year 1, investigators will recruit families of patients receiving the current standard-of-care approach to tumor-normal genetic testing with provider-based education. Participants enrolled in this group included probands aged 12+ that consented/assented to participate. |
| FG003 | Year 2: With Video - Probands | In year 2, investigators will recruit families of patients receiving the updated standard-of-care approach to tumor-normal genetic testing with provider-based education and an educational video. Participants enrolled in this group included Probands aged 12+ that consented/assented to participate. |
| Title | Milestones | Reasons Not Completed | ||||||||||||||||||
|---|---|---|---|---|---|---|---|---|---|---|---|---|---|---|---|---|---|---|---|---|
| Overall Study |
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| ID | Title | Description |
|---|---|---|
| BG000 | Year 1: Without Video - Caregivers | In year 1, investigators will recruit families of patients receiving the current standard-of-care approach to tumor-normal genetic testing with provider-based education. Participants enrolled in this group included caregivers of probands receiving genetic testing consented to participate. |
| BG001 |
| Units | Counts |
|---|---|
| Participants |
|
| Title | Description | Population Description | Parameter Type | Dispersion Type | Unit of Measure | Calculate Percentage | Denominator Units Selected | Denominators | Classes |
|---|---|---|---|---|---|---|---|---|---|
| Age, Continuous | Self-reported Age (extracted from EHR if not reported) |
| Type | Title | Description | Population Description | Reporting Status | Anticipated Posting Date | Parameter Type | Dispersion Type | Unit of Measure | Calculate Percentage | Time Frame | Units Analyzed | Denominator Units Selected | Arm/Group Information | Denominators | Classes | Analyses |
|---|---|---|---|---|---|---|---|---|---|---|---|---|---|---|---|---|
| Primary | Percentage of Correct Items on Knowledge Questionnaire - Before Results | The primary outcome is the percentage of correct items on a genetic knowledge survey that was developed for this study, measured at baseline and before receipt of genetic test results. Participants evaluated included the primary caregivers, secondary caregivers and probands aged 12+. Total scores were generated by summarizing total number of correct responses and generating a percentage. The Genetic Knowledge measure included 12 true/false questions. Higher scores indicate higher genetic knowledge. | Genetic knowledge scores and percentages were generated if participants completed the full measure. N=65 Incomplete Caregiver surveys were due to:
N= 5 Incomplete Proband surveys were due to:
| Posted | Mean | Standard Deviation | Percentage correct (%) | Up to 4 weeks after testing |
|
Up to 8 months
Patient safety was monitored for adverse events related to video intervention only. This was a minimal risk study with no therapeutic intervention; therefore, mortality and other clinical outcomes were not attributable to study participation. Deaths identified reflect the underlying patient population and standard clinical care and were not considered adverse events related to study procedures.
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| ID | Title | Description | Deaths (Affected) | Deaths (At Risk) | Serious Events (Affected) | Serious Events (At Risk) | Other Events (Affected) | Other Events (At Risk) |
|---|---|---|---|---|---|---|---|---|
| EG000 | Year 1: Without Video - Caregivers | In year 1, investigators will recruit families of patients receiving the current standard-of-care approach to tumor-normal genetic testing with provider-based education. Participants enrolled in this group included caregivers probands receiving genetic testing. |
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| Title | Organization | Phone | Extension | |
|---|---|---|---|---|
| Suzanne MacFarland MD | Children's Hospital of Philadelphia | 2153006428 | macfarlands@chop.edu |
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| Type | Includes Protocol | Includes SAP | Includes ICF | Document Label | Document Date | Document Uploaded Date | Document File Name |
|---|---|---|---|---|---|---|---|
| Prot_SAP | Yes | Yes | No | Study Protocol and Statistical Analysis Plan | Jul 26, 2024 | Feb 17, 2026 | Prot_SAP_000.pdf |
| ICF | No | No | Yes | Informed Consent Form: Verbal ICF | Jul 26, 2024 | Feb 17, 2026 | ICF_001.pdf |
| ICF | No | No | Yes | Informed Consent Form: Written ICF | Jul 26, 2024 | Feb 17, 2026 | ICF_002.pdf |
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| ID | Term |
|---|---|
| D030342 | Genetic Diseases, Inborn |
| D009369 | Neoplasms |
| ID | Term |
|---|---|
| D009358 | Congenital, Hereditary, and Neonatal Diseases and Abnormalities |
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Comparison between year 1 and year 2 cohorts, with year 2 cohort receiving intervention
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| Up to 6 months post-genetic test result |
| Acceptability of Use of an Informational Video on Paired Tumor/Normal Testing for Participants With a New Cancer or Other Diagnosis | Acceptability in all participants (primary caregivers, secondary caregivers and probands aged 12+) will be assessed using a questionnaire including the following measures:
Only sub/scales completed entirely were analyzed. | One time, within 6 months after receipt of results |
| Intervention Feasibility - Technical Error | Feasibility will be assessed with absence of mean number of technical difficulties of video delivery reported by research staff from date of receiving genetic testing. | One time, within 6 months after receipt of results |
| Intervention Feasibility - Days to Receive Intervention | Feasibility will be assessed by timeliness of delivery of video after recommendation for testing, defined in average number of days to receive intervention from date of receiving genetic testing. | One time, within 6 months after receipt of results |
| Impact of Events Scale (IES) - Before Results | 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. | Up to 4 weeks after testing |
| Physician Decision |
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| Death |
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| Lost to Follow-up |
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| Became Ineligible |
|
| Year 2: With Video - Caregivers |
In year 2, investigators will recruit families of patients receiving the updated standard-of-care approach to tumor-normal genetic testing with provider-based education and an educational video. Participants enrolled in this group included caregivers of probands receiving genetic testing consented to participate. |
| BG002 | Year 1: Without Video - Probands | In year 1, investigators will recruit families of patients receiving the current standard-of-care approach to tumor-normal genetic testing with provider-based education. Participants enrolled in this group included probands aged 12+ that consented/assented to participate. |
| BG003 | Year 2: With Video - Probands | In year 2, investigators will recruit families of patients receiving the updated standard-of-care approach to tumor-normal genetic testing with provider-based education and an educational video. Participants enrolled in this group included Probands aged 12+ that consented/assented to participate. |
| BG004 | Total | Total of all reporting groups |
| Mean |
| Standard Deviation |
| Years (1 d.p.) |
|
| Sex: Female, Male | Self-reported sex assigned at birth | Count of Participants | Participants |
|
| Race/Ethnicity, Customized | Race and ethnicity were obtained via self-report for caregivers and AYA (supplemented by EHR for AYA if they did not complete the self-report). | N=29 Caregiver participants did not complete measures indicating self-reported race and was not obtainable via EHR, or indicated they did not want to report their race/ethnicity | Count of Participants | Participants |
|
| OG000 |
| Year 1: Without Video - Caregivers |
In year 1, investigators will recruit families of patients receiving the current standard-of-care approach to tumor-normal genetic testing with provider-based education. Participants enrolled in this group included caregivers probands receiving genetic testing. |
| OG001 | Year 2: With Video - Caregivers | In year 2, investigators will recruit families of patients receiving the updated standard-of-care approach to tumor-normal genetic testing with provider-based education and an educational video. Participants enrolled in this group included caregivers of probands receiving genetic testing consented to participate. |
| OG002 | Year 1: Without Video - Probands | In year 1, investigators will recruit families of patients receiving the current standard-of-care approach to tumor-normal genetic testing with provider-based education. Participants enrolled in this group included probands aged 12+ that consented/assented to participate. |
| OG003 | Year 2: With Video - Proband | In year 2, investigators will recruit families of patients receiving the updated standard-of-care approach to tumor-normal genetic testing with provider-based education and an educational video. Participants enrolled in this group included Probands aged 12+ that consented/assented to participate. |
|
|
| Secondary | Percentage of Correct Items on Knowledge Questionnaire - After Results | A secondary analysis includes data from all caregivers' (primary and secondary) and probands' (over 12 years of age) of percentage of correct items on a genetic knowledge survey that was developed for this study, repeated at a second timepoint (T2) after receipt of genetic test results. The knowledge score is a 12-point true/false questionnaire. Total scores were generated by summarizing total number of correct responses and generating a percentage. The Genetic Knowledge measure included 12 true/false questions. Higher scores indicate higher genetic knowledge. | Genetic knowledge scores and percentages were generated if participants completed the full measure. In cases where answers were incomplete or missed, scores were omitted. N=46 Incomplete Caregiver surveys:
N=8 Incomplete proband surveys:
| Posted | Mean | Standard Deviation | Percentage Correct (%) | Up to 6 months post-genetic test result |
|
|
|
|
| Secondary | Acceptability of Use of an Informational Video on Paired Tumor/Normal Testing for Participants With a New Cancer or Other Diagnosis | Acceptability in all participants (primary caregivers, secondary caregivers and probands aged 12+) will be assessed using a questionnaire including the following measures:
Only sub/scales completed entirely were analyzed. | Incomplete caregiver (CG) surveys ranged N=40-44; AYA ranged N=7-8. Incomplete scores across ALL subscales: Caregivers (N=31): Provider W/D N=5, Became ineligible (BI) N=4, Lost to follow up (LTFU) N=22. AYA (N=7): Participant withdrew (PWD) N=2, Provider W/D, N=1, BI N=3, LTFU N=1. Individual subscales incomplete: SDS: CG N=9 (N=1 missed, N=8 PWD); AYA N=1 (missed) IES Intrusion: CG N=12 (N=3 missed, N=9 PWD) IES Avoidance: CG N=9 (PWD) MICRA: CG N=13 (N=4 missed, N=9 PWD) | Posted | Mean | Standard Deviation | Total score | One time, within 6 months after receipt of results |
|
|
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| Other Pre-specified | Satisfaction With Decision Scale (SDS) - Before Results | 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. | SDS Incomplete N=62
| Posted | Mean | Standard Deviation | score on a scale | Up to 4 weeks after testing |
|
|
|
| Other Pre-specified | Impact of Events Scale (IES) - Before Results | 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. | Total incomplete subscales ranged N=63-65. Across All IES scores and subscales incomplete surveys included:
Incomplete IES Total Ns:
Incomplete IES avoidance Ns:
Incomplete IES intrusion Ns:
| Posted | Mean | Standard Deviation | score on a scale | Up to 4 weeks after testing |
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| Secondary | Intervention Feasibility - Technical Error | Feasibility will be assessed with absence of mean number of technical difficulties of video delivery reported by research staff from date of receiving genetic testing. | 204 (N=121 primary caregivers, N=59 secondary caregivers, N= 24 probands) made up 124 family units that were assigned to the intervention arm. Since the intervention arm were the only participants to receive the video intervention, only the Year 2: video received sample were evaluated assessed for intervention feasibility. | Posted | Mean | Standard Deviation | Mean technical errors | One time, within 6 months after receipt of results | Family unit | Family unit |
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|
|
| Secondary | Intervention Feasibility - Days to Receive Intervention | Feasibility will be assessed by timeliness of delivery of video after recommendation for testing, defined in average number of days to receive intervention from date of receiving genetic testing. | 204 (N=121 primary caregivers, N=59 secondary caregivers, N= 24 probands) made up 124 family units participants were assigned to the intervention arm. Since the intervention arm were the only participants to receive the video intervention, only the Year 2: video received sample were evaluated assessed for intervention feasibility. | Posted | Mean | Standard Deviation | Days | One time, within 6 months after receipt of results | Families | Families |
|
|
|
| 0 |
| 137 |
| 0 |
| 137 |
| 0 |
| 137 |
| EG001 | Year 2: With Video - Caregivers | In year 2, investigators will recruit families of patients receiving the updated standard-of-care approach to tumor-normal genetic testing with provider-based education and an educational video. Participants enrolled in this group included caregivers of probands receiving genetic testing consented to participate. Educational Video: Investigators will develop an informational video to be presented within one week of diagnosis of a tumor to those referred for tumor normal paired genetic testing. | 0 | 180 | 0 | 180 | 0 | 180 |
| EG002 | Year 1: Without Video - Probands | In year 1, investigators will recruit families of patients receiving the current standard-of-care approach to tumor-normal genetic testing with provider-based education. Participants enrolled in this group included probands aged 12+ that consented/assented to participate. | 0 | 18 | 0 | 18 | 0 | 18 |
| EG003 | Year 2: With Video - Proband | In year 2, investigators will recruit families of patients receiving the updated standard-of-care approach to tumor-normal genetic testing with provider-based education and an educational video. Participants enrolled in this group included Probands aged 12+ that consented/assented to participate. | 1 | 24 | 0 | 24 | 0 | 24 |
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| Impact of Events Scale Total (IES) Total |
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| Impact of Events Scale (IES) - Avoidance subscale |
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| Impact of Events Scale (IES) Intrusion subscale |
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| Multidimensional Impact of Cancer Risk Assessment (MICRA) - Distress Subscale |
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| IES Avoidance Subscale |
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| IES Intrusion Subscale |
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