Ignite Creation Date:
2025-12-25 @ 2:27 AM
Ignite Modification Date:
2025-12-31 @ 12:59 PM
Study NCT ID:
NCT04855734
Status:
COMPLETED
Last Update Posted:
2025-05-04
First Post:
2021-02-22
Is NOT Gene Therapy:
True
Has Adverse Events:
True
Brief Title:
FACE for Children With Rare Diseases
Sponsor:
Organization:
Raw JSON
{'hasResults': True, 'derivedSection': {'miscInfoModule': {'versionHolder': '2025-12-24'}, 'conditionBrowseModule': {'meshes': [{'id': 'D035583', 'term': 'Rare Diseases'}, {'id': 'D000092862', 'term': 'Psychological Well-Being'}, {'id': 'D002908', 'term': 'Chronic Disease'}, {'id': 'D000084802', 'term': 'Caregiver Burden'}, {'id': 'D001008', 'term': 'Anxiety Disorders'}], 'ancestors': [{'id': 'D020969', 'term': 'Disease Attributes'}, {'id': 'D010335', 'term': 'Pathologic Processes'}, {'id': 'D013568', 'term': 'Pathological Conditions, Signs and Symptoms'}, {'id': 'D010549', 'term': 'Personal Satisfaction'}, {'id': 'D001519', 'term': 'Behavior'}, {'id': 'D013315', 'term': 'Stress, Psychological'}, {'id': 'D001526', 'term': 'Behavioral Symptoms'}, {'id': 'D001523', 'term': 'Mental Disorders'}]}}, 'resultsSection': {'moreInfoModule': {'pointOfContact': {'email': 'mlyon@childrensnational.org', 'phone': '703-346-2873', 'title': 'Dr. Maureen E. Lyon, Clinical Health Psychologist, Professor of Pediatrics', 'organization': "Children's National Hospital"}, 'certainAgreement': {'piSponsorEmployee': True, 'restrictiveAgreement': False}, 'limitationsAndCaveats': {'description': 'Small sample size increased the risk of Type II error. We did not have enough power to detect differences in most of the outcome measures. Single site limits generalizability. Social desirability bias could have occurred with remote, but face-to-face, administration of study questionnaires.'}}, 'adverseEventsModule': {'timeFrame': 'From enrollment through study completion up to 3 months post-intervention (+/- 1 month study window).', 'description': 'Potential adverse event data were collected over the 2 year trial period and reported to annual meetings with the Safety Monitoring Committee. Weekly team meetings at this single site consistently reviewed potential adverse events, which included review of all study participant interactions. No patients or family caregivers died during the pilot trial.', 'eventGroups': [{'id': 'EG000', 'title': 'FACE-Rare Intervention-Children', 'description': "FACE-Rare is a behavioral intervention that combines the CSNAT Pediatric Approach and the Respecting Choices® Next Steps ACP over 3 sessions.\n\nSessions 1\\&2: CSNAT is an evidence-based process of family caregiver assessment and support in specialized medical (palliative) care. The CSNAT tool is structured around 16 categories of family caregiver support. With the goal to decrease caregiver burden, this process consists of 5 stages wherein a nurse or practitioner works with the caregiver to create a shared support plan for the child.\n\nSession 3: Respecting Choices® Next Steps- This advanced care planning (pACP) conversation engages families in a process for how to make future medical decisions consistent with their goals and values. The interview is structured in 6 stages to achieve 2 main goals: to facilitate conversations with the family about their child's medical condition, history, fears, values, beliefs, and hopes; and to set the stage for the family's future healthcare decisions.\n\nFAmily CEntered (FACE) pACP Intervention: The (approximately) weekly 3-session FACE-Rare intervention of approximately 45-60 minutes each is comprised of the CSNAT approach \\[Sessions 1 \\& 2\\] and Respecting Choices \\[Session 3\\].", 'otherNumAtRisk': 9, 'deathsNumAtRisk': 9, 'otherNumAffected': 0, 'seriousNumAtRisk': 9, 'deathsNumAffected': 0, 'seriousNumAffected': 0}, {'id': 'EG001', 'title': 'Treatment-as-Usual (TAU) Control- Children', 'description': 'To minimize the burden to families, we have chosen a Treatment-as-Usual (TAU) comparison condition, where patients will receive their normal standard of care. Both study arms will receive palliative (specialized medical) care information at enrollment and complete questionnaires before and after the intervention or TAU period. Current practice for minors with life-limiting illnesses is to defer initial discussions of advanced care planning (pACP) until a medical crisis, so this is what the TAU control arm condition will consist of.', 'otherNumAtRisk': 12, 'deathsNumAtRisk': 12, 'otherNumAffected': 0, 'seriousNumAtRisk': 12, 'deathsNumAffected': 0, 'seriousNumAffected': 0}, {'id': 'EG002', 'title': 'FACE-Rare Intervention-Family Caregiver', 'description': 'FACE-Rare is a behavioral intervention that combines the CSNAT Pediatric Approach and the Respecting Choices® Next Steps ACP over 3 sessions.\n\nSessions 1\\&2: CSNAT is an evidence-based process of family caregiver assessment and support in specialized medical (palliative) care. The CSNAT tool is structured around 16 categories of family caregiver support. With the goal to decrease caregiver burden, this process consists of 5 stages wherein a nurse or practitioner works with the caregiver to create a shared support plan for the child.\n\nSession 3: Respecting Choices® Next Steps- This advanced care planning (pACP) conversation engages families in a process for how to make future medical decisions', 'otherNumAtRisk': 9, 'deathsNumAtRisk': 9, 'otherNumAffected': 0, 'seriousNumAtRisk': 9, 'deathsNumAffected': 0, 'seriousNumAffected': 0}, {'id': 'EG003', 'title': 'TAU-control-Family Caregiver', 'description': 'To minimize the burden to families, we have chosen a Treatment-as-Usual (TAU) comparison condition, where patients will receive their normal standard of care. Both study arms will receive palliative (specialized medical) care information at enrollment and complete questionnaires before and after the intervention or TAU period. Current practice for minors with life-limiting illnesses is to defer initial discussions of advanced care planning (pACP) until a medical crisis, so this is what the TAU control arm condition will consist of.', 'otherNumAtRisk': 12, 'deathsNumAtRisk': 12, 'otherNumAffected': 0, 'seriousNumAtRisk': 12, 'deathsNumAffected': 0, 'seriousNumAffected': 0}], 'frequencyThreshold': '1'}, 'outcomeMeasuresModule': {'outcomeMeasures': [{'type': 'PRIMARY', 'title': 'Beck Anxiety Inventory', 'denoms': [{'units': 'Participants', 'counts': [{'value': '9', 'groupId': 'OG000'}, {'value': '12', 'groupId': 'OG001'}]}], 'groups': [{'id': 'OG000', 'title': 'FACE-Rare Intervention', 'description': "FACE-Rare is a behavioral intervention that combines the CSNAT Pediatric Approach and the Respecting Choices® Next Steps ACP over 3 sessions.\n\nSessions 1\\&2: CSNAT is an evidence-based process of family caregiver assessment and support in specialized medical (palliative) care. The CSNAT tool is structured around 16 categories of family caregiver support. With the goal to decrease caregiver burden, this process consists of 5 stages wherein a nurse or practitioner works with the caregiver to create a shared support plan for the child.\n\nSession 3: Respecting Choices® Next Steps- This advanced care planning (pACP) conversation engages families in a process for how to make future medical decisions consistent with their goals and values. The interview is structured in 6 stages to achieve 2 main goals: to facilitate conversations with the family about their child's medical condition, history, fears, values, beliefs, and hopes; and to set the stage for the family's future healthcare decisions.\n\nFAmily CEntered (FACE) pACP Intervention: The (approximately) weekly 3-session FACE-Rare intervention of approximately 45-60 minutes each is comprised of the CSNAT approach \\[Sessions 1 \\& 2\\] and Respecting Choices \\[Session 3\\]."}, {'id': 'OG001', 'title': 'Treatment-as-Usual (TAU) Control', 'description': 'To minimize the burden to families, we have chosen a Treatment-as-Usual (TAU) comparison condition, where patients will receive their normal standard of care. Both study arms will receive palliative (specialized medical) care information at enrollment and complete questionnaires before and after the intervention or TAU period. Current practice for minors with life-limiting illnesses is to defer initial discussions of advanced care planning (pACP) until a medical crisis, so this is what the TAU control arm condition will consist of.'}], 'classes': [{'title': 'Baseline Total score', 'categories': [{'measurements': [{'value': '10.4', 'spread': '8.5', 'groupId': 'OG000'}, {'value': '9.5', 'spread': '9.5', 'groupId': 'OG001'}]}]}, {'title': '3 month follow-up Total Score', 'categories': [{'measurements': [{'value': '11.1', 'spread': '8.0', 'groupId': 'OG000'}, {'value': '8.5', 'spread': '9.6', 'groupId': 'OG001'}]}]}], 'analyses': [{'pValue': '0.835', 'groupIds': ['OG000', 'OG001'], 'paramType': 'Mean ratio', 'ciNumSides': 'TWO_SIDED', 'ciPctValue': '95', 'paramValue': '0.962', 'ciLowerLimit': '0.661', 'ciUpperLimit': '1.396', 'pValueComment': 'Threshold for significance was \\<0.05.', 'statisticalMethod': 'Mixed Models Analysis', 'nonInferiorityType': 'SUPERIORITY'}], 'paramType': 'MEAN', 'timeFrame': 'Change from Baseline Anxiety at 3 Months Post-Enrollment', 'description': 'The Beck Anxiety Inventory (BAI) is a 21-item measure of anxiety rated on a 4-point Likert scale of symptoms over the past week. The BAI has demonstrated reliability and validity to assess anxiety in individuals aged 17-80 years. The BAI has good reliability and validity for family caregivers of seriously ill children. It is a measure of Quality of life: emotional health. Higher scores mean more anxiety symptoms. Total score was used in analysis. Scores range from 0-63.', 'unitOfMeasure': 'score on a scale', 'dispersionType': 'Standard Deviation', 'reportingStatus': 'POSTED', 'populationDescription': 'Data analysis was with the family caregivers.'}, {'type': 'PRIMARY', 'title': 'Functional Assessment of Chronic Illness Therapy (FACIT)- Spiritual Well-Being Scale, Version 4', 'denoms': [{'units': 'Participants', 'counts': [{'value': '9', 'groupId': 'OG000'}, {'value': '12', 'groupId': 'OG001'}]}], 'groups': [{'id': 'OG000', 'title': 'FACE-Rare Intervention', 'description': "FACE-Rare is a behavioral intervention that combines the CSNAT Pediatric Approach and the Respecting Choices® Next Steps ACP over 3 sessions.\n\nSessions 1\\&2: CSNAT is an evidence-based process of family caregiver assessment and support in specialized medical (palliative) care. The CSNAT tool is structured around 16 categories of family caregiver support. With the goal to decrease caregiver burden, this process consists of 5 stages wherein a nurse or practitioner works with the caregiver to create a shared support plan for the child.\n\nSession 3: Respecting Choices® Next Steps- This advanced care planning (pACP) conversation engages families in a process for how to make future medical decisions consistent with their goals and values. The interview is structured in 6 stages to achieve 2 main goals: to facilitate conversations with the family about their child's medical condition, history, fears, values, beliefs, and hopes; and to set the stage for the family's future healthcare decisions.\n\nFAmily CEntered (FACE) pACP Intervention: The (approximately) weekly 3-session FACE-Rare intervention of approximately 45-60 minutes each is comprised of the CSNAT approach \\[Sessions 1 \\& 2\\] and Respecting Choices \\[Session 3\\]."}, {'id': 'OG001', 'title': 'Treatment-as-Usual (TAU) Control', 'description': 'To minimize the burden to families, we have chosen a Treatment-as-Usual (TAU) comparison condition, where patients will receive their normal standard of care. Both study arms will receive palliative (specialized medical) care information at enrollment and complete questionnaires before and after the intervention or TAU period. Current practice for minors with life-limiting illnesses is to defer initial discussions of advanced care planning (pACP) until a medical crisis, so this is what the TAU control arm condition will consist of.'}], 'classes': [{'title': 'Baseline Meaning/Peace', 'categories': [{'measurements': [{'value': '24.0', 'groupId': 'OG000', 'lowerLimit': '23.0', 'upperLimit': '27.0'}, {'value': '25.0', 'groupId': 'OG001', 'lowerLimit': '23.5', 'upperLimit': '27.0'}]}]}, {'title': '3 month Meaning/Peace', 'categories': [{'measurements': [{'value': '26.0', 'groupId': 'OG000', 'lowerLimit': '24.0', 'upperLimit': '30.0'}, {'value': '26.0', 'groupId': 'OG001', 'lowerLimit': '21.8', 'upperLimit': '29.0'}]}]}, {'title': 'Baseline Faith', 'categories': [{'measurements': [{'value': '11.0', 'groupId': 'OG000', 'lowerLimit': '10.0', 'upperLimit': '14.0'}, {'value': '6.0', 'groupId': 'OG001', 'lowerLimit': '3.8', 'upperLimit': '7.5'}]}]}, {'title': '3 month Faith', 'categories': [{'measurements': [{'value': '9.0', 'groupId': 'OG000', 'lowerLimit': '7.0', 'upperLimit': '14.0'}, {'value': '7.0', 'groupId': 'OG001', 'lowerLimit': '4.8', 'upperLimit': '11.0'}]}]}], 'analyses': [{'pValue': '0.032', 'groupIds': ['OG000', 'OG001'], 'paramType': 'Mean ratio', 'ciNumSides': 'TWO_SIDED', 'ciPctValue': '95', 'paramValue': '0.826', 'ciLowerLimit': '0.75', 'ciUpperLimit': '0.986', 'pValueComment': '\\<0.05 was threshold for significance level.', 'groupDescription': 'Meaning/Peace subscale which was a primary outcome.', 'statisticalMethod': 'Mixed Models Analysis', 'nonInferiorityType': 'SUPERIORITY'}], 'paramType': 'MEDIAN', 'timeFrame': 'Change from Baseline Spiritual Well-Being at 3 Months Post-Enrollment', 'description': 'Assessed construct of spiritual well-being. Two subscales Meaning/Peace (7 items) and Faith (5 items) and Total score (12 items) were calculated. on a 5-point Likert scale from 0=not at all to 5=very much. Some items are reverse scored. See www.facit.org Meaning/Peace subscale score range from minimum value of 0 to maximum value of 32. Higher scores indicate better meaning/peace. Faith subscale score range from 0 minimum value to maximum value of 16. Higher scores indicate better meaning/peace. Higher scores indicate better spiritual well-being.', 'unitOfMeasure': 'score on a scale', 'dispersionType': 'Inter-Quartile Range', 'reportingStatus': 'POSTED', 'populationDescription': 'Family caregivers of children with rare diseases.'}, {'type': 'PRIMARY', 'title': 'Advance Care Plan Document for Children With Rare Diseases', 'denoms': [{'units': 'Participants', 'counts': [{'value': '9', 'groupId': 'OG000'}, {'value': '12', 'groupId': 'OG001'}]}], 'groups': [{'id': 'OG000', 'title': 'FACE-Rare Intervention', 'description': "FACE-Rare is a behavioral intervention that combines the CSNAT Pediatric Approach and the Respecting Choices® Next Steps ACP over 3 sessions.\n\nSessions 1\\&2: CSNAT is an evidence-based process of family caregiver assessment and support in specialized medical (palliative) care. The CSNAT tool is structured around 16 categories of family caregiver support. With the goal to decrease caregiver burden, this process consists of 5 stages wherein a nurse or practitioner works with the caregiver to create a shared support plan for the child.\n\nSession 3: Respecting Choices® Next Steps- This advanced care planning (pACP) conversation engages families in a process for how to make future medical decisions consistent with their goals and values. The interview is structured in 6 stages to achieve 2 main goals: to facilitate conversations with the family about their child's medical condition, history, fears, values, beliefs, and hopes; and to set the stage for the family's future healthcare decisions.\n\nFAmily CEntered (FACE) pACP Intervention: The (approximately) weekly 3-session FACE-Rare intervention of approximately 45-60 minutes each is comprised of the CSNAT approach \\[Sessions 1 \\& 2\\] and Respecting Choices \\[Session 3\\]."}, {'id': 'OG001', 'title': 'Treatment-as-Usual (TAU) Control', 'description': 'To minimize the burden to families, we have chosen a Treatment-as-Usual (TAU) comparison condition, where patients will receive their normal standard of care. Both study arms will receive palliative (specialized medical) care information at enrollment and complete questionnaires before and after the intervention or TAU period. Current practice for minors with life-limiting illnesses is to defer initial discussions of advanced care planning (pACP) until a medical crisis, so this is what the TAU control arm condition will consist of.'}], 'classes': [{'title': 'Baseline', 'categories': [{'title': 'No', 'measurements': [{'value': '8', 'groupId': 'OG000'}, {'value': '12', 'groupId': 'OG001'}]}, {'title': 'Yes', 'measurements': [{'value': '1', 'groupId': 'OG000'}, {'value': '0', 'groupId': 'OG001'}]}]}, {'title': '3-month follow-up', 'categories': [{'title': 'No', 'measurements': [{'value': '6', 'groupId': 'OG000'}, {'value': '12', 'groupId': 'OG001'}]}, {'title': 'Yes', 'measurements': [{'value': '3', 'groupId': 'OG000'}, {'value': '0', 'groupId': 'OG001'}]}]}], 'paramType': 'COUNT_OF_PARTICIPANTS', 'timeFrame': 'Change from Baseline ACP Documentation in the Electronic Health Record at 12 weeks Post-Enrollment', 'description': 'Advanced Care Planning (ACP) Documentation in Electronic Health Records (EHR). Score will be present in EHR or absent from EHR. Evidence of any advance care document (yes/no) will count.', 'unitOfMeasure': 'Participants', 'reportingStatus': 'POSTED', 'populationDescription': 'Advance care plan for child documented in the Electronic Health Record.'}, {'type': 'PRIMARY', 'title': 'Days in Palliative Care Prior to Death', 'denoms': [{'units': 'Participants', 'counts': [{'value': '0', 'groupId': 'OG000'}, {'value': '0', 'groupId': 'OG001'}]}], 'groups': [{'id': 'OG000', 'title': 'FACE-Rare Intervention', 'description': "FACE-Rare is a behavioral intervention that combines the CSNAT Pediatric Approach and the Respecting Choices® Next Steps ACP over 3 sessions.\n\nSessions 1\\&2: CSNAT is an evidence-based process of family caregiver assessment and support in specialized medical (palliative) care. The CSNAT tool is structured around 16 categories of family caregiver support. With the goal to decrease caregiver burden, this process consists of 5 stages wherein a nurse or practitioner works with the caregiver to create a shared support plan for the child.\n\nSession 3: Respecting Choices® Next Steps- This advanced care planning (pACP) conversation engages families in a process for how to make future medical decisions consistent with their goals and values. The interview is structured in 6 stages to achieve 2 main goals: to facilitate conversations with the family about their child's medical condition, history, fears, values, beliefs, and hopes; and to set the stage for the family's future healthcare decisions.\n\nFAmily CEntered (FACE) pACP Intervention: The (approximately) weekly 3-session FACE-Rare intervention of approximately 45-60 minutes each is comprised of the CSNAT approach \\[Sessions 1 \\& 2\\] and Respecting Choices \\[Session 3\\]."}, {'id': 'OG001', 'title': 'Treatment-as-Usual (TAU) Control', 'description': 'To minimize the burden to families, we have chosen a Treatment-as-Usual (TAU) comparison condition, where patients will receive their normal standard of care. Both study arms will receive palliative (specialized medical) care information at enrollment and complete questionnaires before and after the intervention or TAU period. Current practice for minors with life-limiting illnesses is to defer initial discussions of advanced care planning (pACP) until a medical crisis, so this is what the TAU control arm condition will consist of.'}], 'timeFrame': '12 weeks Post-Enrollment', 'description': 'Score is number of days patient was enrolled in a palliative care program in the 30 days prior to death. Applicable only to patients who have deceased since study enrollment. Used to standardize child healthcare utilization from data abstraction based on retrospective medical chart review. Minimum value is 0 with no maximum. Higher score indicates higher level of healthcare utilization.', 'reportingStatus': 'POSTED', 'populationDescription': 'Child with rare disease. No child with a rare disease died during the study, so data for Days in Palliative Care Prior to death was not collected.'}, {'type': 'PRIMARY', 'title': 'Hospitalizations', 'denoms': [{'units': 'Participants', 'counts': [{'value': '9', 'groupId': 'OG000'}, {'value': '12', 'groupId': 'OG001'}]}], 'groups': [{'id': 'OG000', 'title': 'FACE-Rare Intervention', 'description': "FACE-Rare is a behavioral intervention that combines the CSNAT Pediatric Approach and the Respecting Choices® Next Steps ACP over 3 sessions.\n\nSessions 1\\&2: CSNAT is an evidence-based process of family caregiver assessment and support in specialized medical (palliative) care. The CSNAT tool is structured around 16 categories of family caregiver support. With the goal to decrease caregiver burden, this process consists of 5 stages wherein a nurse or practitioner works with the caregiver to create a shared support plan for the child.\n\nSession 3: Respecting Choices® Next Steps- This advanced care planning (pACP) conversation engages families in a process for how to make future medical decisions consistent with their goals and values. The interview is structured in 6 stages to achieve 2 main goals: to facilitate conversations with the family about their child's medical condition, history, fears, values, beliefs, and hopes; and to set the stage for the family's future healthcare decisions.\n\nFAmily CEntered (FACE) pACP Intervention: The (approximately) weekly 3-session FACE-Rare intervention of approximately 45-60 minutes each is comprised of the CSNAT approach \\[Sessions 1 \\& 2\\] and Respecting Choices \\[Session 3\\]."}, {'id': 'OG001', 'title': 'Treatment-as-Usual (TAU) Control', 'description': 'To minimize the burden to families, we have chosen a Treatment-as-Usual (TAU) comparison condition, where patients will receive their normal standard of care. Both study arms will receive palliative (specialized medical) care information at enrollment and complete questionnaires before and after the intervention or TAU period. Current practice for minors with life-limiting illnesses is to defer initial discussions of advanced care planning (pACP) until a medical crisis, so this is what the TAU control arm condition will consist of.'}], 'classes': [{'title': 'Baseline', 'categories': [{'title': 'No', 'measurements': [{'value': '7', 'groupId': 'OG000'}, {'value': '7', 'groupId': 'OG001'}]}, {'title': 'Yes', 'measurements': [{'value': '2', 'groupId': 'OG000'}, {'value': '5', 'groupId': 'OG001'}]}]}, {'title': '3-month follow-up', 'categories': [{'title': 'No', 'measurements': [{'value': '7', 'groupId': 'OG000'}, {'value': '8', 'groupId': 'OG001'}]}, {'title': 'Yes', 'measurements': [{'value': '2', 'groupId': 'OG000'}, {'value': '4', 'groupId': 'OG001'}]}]}], 'paramType': 'COUNT_OF_PARTICIPANTS', 'timeFrame': 'Change from Baseline to Frequency at 12 weeks Post-Enrollment', 'description': 'Used to standardize child healthcare utilization from data abstraction by the research nurse based on medical chart review in Electronic Health Record. Score is recorded number of inpatient hospital admissions for clinical treatment for the child with a rare disease. Minimum value is 0 with no maximum. Higher score indicates higher level of healthcare utilization. Questions was "Hospitalization in the last 3 months?"', 'unitOfMeasure': 'Participants', 'reportingStatus': 'POSTED', 'populationDescription': 'Children with rare diseases.'}, {'type': 'PRIMARY', 'title': 'Place of Death', 'denoms': [{'units': 'Participants', 'counts': [{'value': '0', 'groupId': 'OG000'}, {'value': '0', 'groupId': 'OG001'}]}], 'groups': [{'id': 'OG000', 'title': 'FACE-Rare Intervention', 'description': "FACE-Rare is a behavioral intervention that combines the CSNAT Pediatric Approach and the Respecting Choices® Next Steps ACP over 3 sessions.\n\nSessions 1\\&2: CSNAT is an evidence-based process of family caregiver assessment and support in specialized medical (palliative) care. The CSNAT tool is structured around 16 categories of family caregiver support. With the goal to decrease caregiver burden, this process consists of 5 stages wherein a nurse or practitioner works with the caregiver to create a shared support plan for the child.\n\nSession 3: Respecting Choices® Next Steps- This advanced care planning (pACP) conversation engages families in a process for how to make future medical decisions consistent with their goals and values. The interview is structured in 6 stages to achieve 2 main goals: to facilitate conversations with the family about their child's medical condition, history, fears, values, beliefs, and hopes; and to set the stage for the family's future healthcare decisions.\n\nFAmily CEntered (FACE) pACP Intervention: The (approximately) weekly 3-session FACE-Rare intervention of approximately 45-60 minutes each is comprised of the CSNAT approach \\[Sessions 1 \\& 2\\] and Respecting Choices \\[Session 3\\]."}, {'id': 'OG001', 'title': 'Treatment-as-Usual (TAU) Control', 'description': 'To minimize the burden to families, we have chosen a Treatment-as-Usual (TAU) comparison condition, where patients will receive their normal standard of care. Both study arms will receive palliative (specialized medical) care information at enrollment and complete questionnaires before and after the intervention or TAU period. Current practice for minors with life-limiting illnesses is to defer initial discussions of advanced care planning (pACP) until a medical crisis, so this is what the TAU control arm condition will consist of.'}], 'timeFrame': '12 weeks Post-Enrollment', 'description': 'Location where the patient was pronounced dead, as reported in their EMR. Applicable only to patients who have deceased since study enrollment. Categories are Inpatient hospice setting, Home with hospice, Home without hospice, Hospital ICU, Hospital-Not ICU, Other, or Unknown. Used to standardize child healthcare utilization from data abstraction based on retrospective medical chart review.', 'reportingStatus': 'POSTED', 'populationDescription': 'Child with a rare disease. No child with a rare disease died during the study, so Place of Death was not collected.'}, {'type': 'SECONDARY', 'title': 'Brief-Multidimensional Measure of Religion and Spirituality - Responses to Question 1', 'denoms': [{'units': 'Participants', 'counts': [{'value': '9', 'groupId': 'OG000'}, {'value': '12', 'groupId': 'OG001'}]}], 'groups': [{'id': 'OG000', 'title': 'FACE-Rare Intervention', 'description': "FACE-Rare is a behavioral intervention that combines the CSNAT Pediatric Approach and the Respecting Choices® Next Steps ACP over 3 sessions.\n\nSessions 1\\&2: CSNAT is an evidence-based process of family caregiver assessment and support in specialized medical (palliative) care. The CSNAT tool is structured around 16 categories of family caregiver support. With the goal to decrease caregiver burden, this process consists of 5 stages wherein a nurse or practitioner works with the caregiver to create a shared support plan for the child.\n\nSession 3: Respecting Choices® Next Steps- This advanced care planning (pACP) conversation engages families in a process for how to make future medical decisions consistent with their goals and values. The interview is structured in 6 stages to achieve 2 main goals: to facilitate conversations with the family about their child's medical condition, history, fears, values, beliefs, and hopes; and to set the stage for the family's future healthcare decisions.\n\nFAmily CEntered (FACE) pACP Intervention: The (approximately) weekly 3-session FACE-Rare intervention of approximately 45-60 minutes each is comprised of the CSNAT approach \\[Sessions 1 \\& 2\\] and Respecting Choices \\[Session 3\\]."}, {'id': 'OG001', 'title': 'Treatment-as-Usual (TAU) Control', 'description': 'To minimize the burden to families, we have chosen a Treatment-as-Usual (TAU) comparison condition, where patients will receive their normal standard of care. Both study arms will receive palliative (specialized medical) care information at enrollment and complete questionnaires before and after the intervention or TAU period. Current practice for minors with life-limiting illnesses is to defer initial discussions of advanced care planning (pACP) until a medical crisis, so this is what the TAU control arm condition will consist of.'}], 'classes': [{'title': "Baseline How often do you feel God's presence?", 'categories': [{'title': 'Declined', 'measurements': [{'value': '1', 'groupId': 'OG000'}, {'value': '0', 'groupId': 'OG001'}]}, {'title': 'Every day', 'measurements': [{'value': '3', 'groupId': 'OG000'}, {'value': '1', 'groupId': 'OG001'}]}, {'title': 'Most days', 'measurements': [{'value': '1', 'groupId': 'OG000'}, {'value': '3', 'groupId': 'OG001'}]}, {'title': 'Once in a while', 'measurements': [{'value': '3', 'groupId': 'OG000'}, {'value': '5', 'groupId': 'OG001'}]}, {'title': 'Some days', 'measurements': [{'value': '1', 'groupId': 'OG000'}, {'value': '2', 'groupId': 'OG001'}]}, {'title': 'Many times a day.', 'measurements': [{'value': '0', 'groupId': 'OG000'}, {'value': '0', 'groupId': 'OG001'}]}, {'title': 'Never or almost never', 'measurements': [{'value': '0', 'groupId': 'OG000'}, {'value': '1', 'groupId': 'OG001'}]}]}, {'title': "3-month follow up -How often do you feel God's presence?", 'categories': [{'title': 'Declined', 'measurements': [{'value': '0', 'groupId': 'OG000'}, {'value': '0', 'groupId': 'OG001'}]}, {'title': 'Every day', 'measurements': [{'value': '2', 'groupId': 'OG000'}, {'value': '4', 'groupId': 'OG001'}]}, {'title': 'Most days', 'measurements': [{'value': '2', 'groupId': 'OG000'}, {'value': '2', 'groupId': 'OG001'}]}, {'title': 'Once in a while', 'measurements': [{'value': '1', 'groupId': 'OG000'}, {'value': '5', 'groupId': 'OG001'}]}, {'title': 'Some days', 'measurements': [{'value': '1', 'groupId': 'OG000'}, {'value': '0', 'groupId': 'OG001'}]}, {'title': 'Many times a day.', 'measurements': [{'value': '1', 'groupId': 'OG000'}, {'value': '1', 'groupId': 'OG001'}]}, {'title': 'Never or almost never', 'measurements': [{'value': '2', 'groupId': 'OG000'}, {'value': '0', 'groupId': 'OG001'}]}]}], 'paramType': 'COUNT_OF_PARTICIPANTS', 'timeFrame': 'Baseline and 3-month follow-up.', 'description': "Survey responses to: 1. How often do you feel God's presence? On 6-point Likert-scale responses range from every day to never or almost never.", 'unitOfMeasure': 'Participants', 'reportingStatus': 'POSTED', 'populationDescription': 'Primary family caregivers of children with rare diseases.'}, {'type': 'SECONDARY', 'title': 'Family Appraisal of Caregiving Questionnaire for Palliative Care for Palliative Care (FACQ-PC)', 'denoms': [{'units': 'Participants', 'counts': [{'value': '9', 'groupId': 'OG000'}, {'value': '12', 'groupId': 'OG001'}]}], 'groups': [{'id': 'OG000', 'title': 'FACE-Rare Intervention', 'description': "FACE-Rare is a behavioral intervention that combines the CSNAT Pediatric Approach and the Respecting Choices® Next Steps ACP over 3 sessions.\n\nSessions 1\\&2: CSNAT is an evidence-based process of family caregiver assessment and support in specialized medical (palliative) care. The CSNAT tool is structured around 16 categories of family caregiver support. With the goal to decrease caregiver burden, this process consists of 5 stages wherein a nurse or practitioner works with the caregiver to create a shared support plan for the child.\n\nSession 3: Respecting Choices® Next Steps- This advanced care planning (pACP) conversation engages families in a process for how to make future medical decisions consistent with their goals and values. The interview is structured in 6 stages to achieve 2 main goals: to facilitate conversations with the family about their child's medical condition, history, fears, values, beliefs, and hopes; and to set the stage for the family's future healthcare decisions.\n\nFAmily CEntered (FACE) pACP Intervention: The (approximately) weekly 3-session FACE-Rare intervention of approximately 45-60 minutes each is comprised of the CSNAT approach \\[Sessions 1 \\& 2\\] and Respecting Choices \\[Session 3\\]."}, {'id': 'OG001', 'title': 'Treatment-as-Usual (TAU) Control', 'description': 'To minimize the burden to families, we have chosen a Treatment-as-Usual (TAU) comparison condition, where patients will receive their normal standard of care. Both study arms will receive palliative (specialized medical) care information at enrollment and complete questionnaires before and after the intervention or TAU period. Current practice for minors with life-limiting illnesses is to defer initial discussions of advanced care planning (pACP) until a medical crisis, so this is what the TAU control arm condition will consist of.'}], 'classes': [{'title': 'Caregiver strain subscale baseline', 'categories': [{'measurements': [{'value': '3.3', 'groupId': 'OG000', 'lowerLimit': '2.9', 'upperLimit': '3.6'}, {'value': '3.0', 'groupId': 'OG001', 'lowerLimit': '2.4', 'upperLimit': '3.7'}]}]}, {'title': 'Positive caregiving appraisals Baseline', 'categories': [{'measurements': [{'value': '4.7', 'groupId': 'OG000', 'lowerLimit': '4.1', 'upperLimit': '4.9'}, {'value': '4.6', 'groupId': 'OG001', 'lowerLimit': '4.3', 'upperLimit': '4.7'}]}]}, {'title': 'Caregiver Distress Baseline', 'categories': [{'measurements': [{'value': '3.3', 'groupId': 'OG000', 'lowerLimit': '2.8', 'upperLimit': '3.8'}, {'value': '3.5', 'groupId': 'OG001', 'lowerLimit': '2.9', 'upperLimit': '4.0'}]}]}, {'title': 'Family well-being baseline', 'categories': [{'measurements': [{'value': '4.2', 'groupId': 'OG000', 'lowerLimit': '3.5', 'upperLimit': '4.7'}, {'value': '4.2', 'groupId': 'OG001', 'lowerLimit': '3.9', 'upperLimit': '4.5'}]}]}, {'title': 'caregiver strain 3 Month follow up', 'categories': [{'measurements': [{'value': '3.5', 'groupId': 'OG000', 'lowerLimit': '2.8', 'upperLimit': '3.8'}, {'value': '2.9', 'groupId': 'OG001', 'lowerLimit': '2.5', 'upperLimit': '3.9'}]}]}, {'title': 'Positive caregiving appraisals 3 month follow up', 'categories': [{'measurements': [{'value': '4.7', 'groupId': 'OG000', 'lowerLimit': '4.3', 'upperLimit': '5.0'}, {'value': '4.6', 'groupId': 'OG001', 'lowerLimit': '4.5', 'upperLimit': '4.9'}]}]}, {'title': 'Caregiver distress 3 month follow up', 'categories': [{'measurements': [{'value': '2.8', 'groupId': 'OG000', 'lowerLimit': '2.5', 'upperLimit': '3.3'}, {'value': '3.4', 'groupId': 'OG001', 'lowerLimit': '2.3', 'upperLimit': '3.8'}]}]}, {'title': 'Family well-being 3 month follow up', 'categories': [{'measurements': [{'value': '4.5', 'groupId': 'OG000', 'lowerLimit': '3.8', 'upperLimit': '4.5'}, {'value': '4.2', 'groupId': 'OG001', 'lowerLimit': '4.0', 'upperLimit': '4.4'}]}]}], 'analyses': [{'pValue': '0.284', 'groupIds': ['OG000', 'OG001'], 'paramType': 'Mean ratio', 'ciNumSides': 'TWO_SIDED', 'ciPctValue': '95', 'paramValue': '1.052', 'ciLowerLimit': '0.959', 'ciUpperLimit': '1.157', 'pValueComment': '\\<0.05 threshold for significance level.', 'groupDescription': 'Caregiver strain subscale scores at 3 months post-intervention comparing intervention to TAU, controlling for baseline levels.', 'statisticalMethod': 'Mixed Models Analysis', 'nonInferiorityType': 'SUPERIORITY'}, {'pValue': '0.228', 'groupIds': ['OG000', 'OG001'], 'paramType': 'Mean ratioj', 'ciNumSides': 'TWO_SIDED', 'ciPctValue': '95', 'paramValue': '0.931', 'ciLowerLimit': '0.827', 'ciUpperLimit': '1.048', 'pValueComment': '\\<0.05 threshold for significance level.', 'groupDescription': 'Caregiver distress subscale controlling for baseline levels.', 'statisticalMethod': 'Mixed Models Analysis', 'nonInferiorityType': 'SUPERIORITY'}, {'pValue': '0.504', 'groupIds': ['OG000', 'OG001'], 'paramType': 'Mean ratio', 'ciNumSides': 'TWO_SIDED', 'ciPctValue': '95', 'paramValue': '0.984', 'ciLowerLimit': '0.94', 'ciUpperLimit': '1.032', 'pValueComment': '\\<0.05 significance level.', 'groupDescription': 'Family well-being subscale controlling for baseline levels.', 'statisticalMethod': 'Mixed Models Analysis', 'nonInferiorityType': 'SUPERIORITY'}, {'pValue': '0.741', 'groupIds': ['OG000', 'OG001'], 'paramType': 'Mean ratio', 'ciNumSides': 'TWO_SIDED', 'ciPctValue': '95', 'paramValue': '0.995', 'ciLowerLimit': '0.967', 'ciUpperLimit': '1.024', 'pValueComment': '\\<0.05 significance level.', 'groupDescription': 'Positive Caregiving Appraisals subscale at 3 month follow up, controlling for baseline levels.', 'statisticalMethod': 'Mixed Models Analysis', 'nonInferiorityType': 'SUPERIORITY'}], 'paramType': 'MEDIAN', 'timeFrame': 'Change from Baseline appraisal of Caregiving at 12 weeks Post-Enrollment', 'description': 'The FACQ-PC is a 25-item measure consists of four theoretically derived subscales: (i) caregiver strain, (ii) positive caregiving appraisals, (iii) caregiver distress, and (iv) family well-being. Scores are from 5 = strongly agree to 1 = strongly disagree. We did not calculate a Total score. We only used the 4 subscale scores. On the subscale scores for positive caregiving appraisals and family well-being, higher scores mean better outcomes, i.e. greater positive caregiving appraisals or family well-being. On the subscale scores for caregiver strain and caregiver distress, higher scores mean worse outcomes, i.e. greater caregiver strain or caregiver distress. The FACQ-PC subscale scores were computed by taking the mean of the items (score range 1-5). Some items were reverse scored, depending on how the item is phrased, so that higher scores = higher amount of the subscale being measured. So the minimum value for each subscale is 1 and the maximum value for each subscale is 5.', 'unitOfMeasure': 'score on a scale', 'dispersionType': 'Inter-Quartile Range', 'reportingStatus': 'POSTED', 'populationDescription': 'Primary family caregivers of children with a rare disease.'}, {'type': 'SECONDARY', 'title': 'Hickman Role Stress Decisional Burden Scale', 'denoms': [{'units': 'Participants', 'counts': [{'value': '9', 'groupId': 'OG000'}, {'value': '12', 'groupId': 'OG001'}]}], 'groups': [{'id': 'OG000', 'title': 'FACE-Rare Intervention', 'description': "FACE-Rare is a behavioral intervention that combines the CSNAT Pediatric Approach and the Respecting Choices® Next Steps ACP over 3 sessions.\n\nSessions 1\\&2: CSNAT is an evidence-based process of family caregiver assessment and support in specialized medical (palliative) care. The CSNAT tool is structured around 16 categories of family caregiver support. With the goal to decrease caregiver burden, this process consists of 5 stages wherein a nurse or practitioner works with the caregiver to create a shared support plan for the child.\n\nSession 3: Respecting Choices® Next Steps- This advanced care planning (pACP) conversation engages families in a process for how to make future medical decisions consistent with their goals and values. The interview is structured in 6 stages to achieve 2 main goals: to facilitate conversations with the family about their child's medical condition, history, fears, values, beliefs, and hopes; and to set the stage for the family's future healthcare decisions.\n\nFAmily CEntered (FACE) pACP Intervention: The (approximately) weekly 3-session FACE-Rare intervention of approximately 45-60 minutes each is comprised of the CSNAT approach \\[Sessions 1 \\& 2\\] and Respecting Choices \\[Session 3\\]."}, {'id': 'OG001', 'title': 'Treatment-as-Usual (TAU) Control', 'description': 'To minimize the burden to families, we have chosen a Treatment-as-Usual (TAU) comparison condition, where patients will receive their normal standard of care. Both study arms will receive palliative (specialized medical) care information at enrollment and complete questionnaires before and after the intervention or TAU period. Current practice for minors with life-limiting illnesses is to defer initial discussions of advanced care planning (pACP) until a medical crisis, so this is what the TAU control arm condition will consist of.'}], 'classes': [{'title': 'Baseline', 'categories': [{'measurements': [{'value': '75', 'groupId': 'OG000', 'lowerLimit': '47.0', 'upperLimit': '90.0'}, {'value': '71.0', 'groupId': 'OG001', 'lowerLimit': '39.3', 'upperLimit': '88.0'}]}]}, {'title': '3-month follow-up', 'categories': [{'measurements': [{'value': '75.0', 'groupId': 'OG000', 'lowerLimit': '75.0', 'upperLimit': '85.0'}, {'value': '72.5', 'groupId': 'OG001', 'lowerLimit': '50.0', 'upperLimit': '81.3'}]}]}], 'paramType': 'MEDIAN', 'timeFrame': 'Up to 5 Weeks Post-Enrollment', 'description': 'Visual analogue scale 0-100. "How stressful is it for you to make medical decisions for your child?" 1 item. Higher score means more stress, lower scores mean less stress.', 'unitOfMeasure': 'raw score units on a scale', 'dispersionType': 'Inter-Quartile Range', 'reportingStatus': 'POSTED', 'populationDescription': 'Primary caregiver of child with rare disease.'}, {'type': 'SECONDARY', 'title': 'Brief Multidimensional Measurement of Religiousness/Spirituality (BMMRS-adapted) Survey Responses to Question 2.', 'denoms': [{'units': 'Participants', 'counts': [{'value': '9', 'groupId': 'OG000'}, {'value': '12', 'groupId': 'OG001'}]}], 'groups': [{'id': 'OG000', 'title': 'FACE-Rare Intervention', 'description': "FACE-Rare is a behavioral intervention that combines the CSNAT Pediatric Approach and the Respecting Choices® Next Steps ACP over 3 sessions.\n\nSessions 1\\&2: CSNAT is an evidence-based process of family caregiver assessment and support in specialized medical (palliative) care. The CSNAT tool is structured around 16 categories of family caregiver support. With the goal to decrease caregiver burden, this process consists of 5 stages wherein a nurse or practitioner works with the caregiver to create a shared support plan for the child.\n\nSession 3: Respecting Choices® Next Steps- This advanced care planning (pACP) conversation engages families in a process for how to make future medical decisions consistent with their goals and values. The interview is structured in 6 stages to achieve 2 main goals: to facilitate conversations with the family about their child's medical condition, history, fears, values, beliefs, and hopes; and to set the stage for the family's future healthcare decisions.\n\nFAmily CEntered (FACE) pACP Intervention: The (approximately) weekly 3-session FACE-Rare intervention of approximately 45-60 minutes each is comprised of the CSNAT approach \\[Sessions 1 \\& 2\\] and Respecting Choices \\[Session 3\\]."}, {'id': 'OG001', 'title': 'Treatment-as-Usual (TAU) Control', 'description': 'To minimize the burden to families, we have chosen a Treatment-as-Usual (TAU) comparison condition, where patients will receive their normal standard of care. Both study arms will receive palliative (specialized medical) care information at enrollment and complete questionnaires before and after the intervention or TAU period. Current practice for minors with life-limiting illnesses is to defer initial discussions of advanced care planning (pACP) until a medical crisis, so this is what the TAU control arm condition will consist of.'}], 'classes': [{'title': 'Baseline How often do you pray privately?', 'categories': [{'title': 'A few times a month', 'measurements': [{'value': '2', 'groupId': 'OG000'}, {'value': '2', 'groupId': 'OG001'}]}, {'title': 'A few times a week', 'measurements': [{'value': '1', 'groupId': 'OG000'}, {'value': '3', 'groupId': 'OG001'}]}, {'title': 'More than once a day', 'measurements': [{'value': '3', 'groupId': 'OG000'}, {'value': '3', 'groupId': 'OG001'}]}, {'title': 'Never', 'measurements': [{'value': '1', 'groupId': 'OG000'}, {'value': '2', 'groupId': 'OG001'}]}, {'title': 'Once a month', 'measurements': [{'value': '2', 'groupId': 'OG000'}, {'value': '0', 'groupId': 'OG001'}]}, {'title': 'Less than once a month', 'measurements': [{'value': '0', 'groupId': 'OG000'}, {'value': '1', 'groupId': 'OG001'}]}, {'title': 'Once a day', 'measurements': [{'value': '0', 'groupId': 'OG000'}, {'value': '1', 'groupId': 'OG001'}]}]}, {'title': '3 month follow-up How often do you pray privately?', 'categories': [{'title': 'A few times a month', 'measurements': [{'value': '2', 'groupId': 'OG000'}, {'value': '2', 'groupId': 'OG001'}]}, {'title': 'A few times a week', 'measurements': [{'value': '2', 'groupId': 'OG000'}, {'value': '4', 'groupId': 'OG001'}]}, {'title': 'More than once a day', 'measurements': [{'value': '3', 'groupId': 'OG000'}, {'value': '3', 'groupId': 'OG001'}]}, {'title': 'Never', 'measurements': [{'value': '1', 'groupId': 'OG000'}, {'value': '1', 'groupId': 'OG001'}]}, {'title': 'Once a month', 'measurements': [{'value': '1', 'groupId': 'OG000'}, {'value': '0', 'groupId': 'OG001'}]}, {'title': 'Less than once a month', 'measurements': [{'value': '0', 'groupId': 'OG000'}, {'value': '1', 'groupId': 'OG001'}]}, {'title': 'Once a day', 'measurements': [{'value': '0', 'groupId': 'OG000'}, {'value': '1', 'groupId': 'OG001'}]}]}], 'paramType': 'COUNT_OF_PARTICIPANTS', 'timeFrame': 'Baseline and 3-month follow-up', 'description': 'Responses to survey question 2, How often do you pray privately? 7 point Likert scale ranging from a few times a month to once a day. Higher scores in pray item indicate more prayer.', 'unitOfMeasure': 'Participants', 'reportingStatus': 'POSTED', 'populationDescription': 'Family caregivers of children with rare diseases.'}, {'type': 'OTHER_PRE_SPECIFIED', 'title': 'Satisfaction Questionnaire (Process Measure)', 'denoms': [{'units': 'Participants', 'counts': [{'value': '9', 'groupId': 'OG000'}, {'value': '12', 'groupId': 'OG001'}]}], 'groups': [{'id': 'OG000', 'title': 'FACE-Rare Intervention', 'description': "FACE-Rare is a behavioral intervention that combines the CSNAT Pediatric Approach and the Respecting Choices® Next Steps ACP over 3 sessions.\n\nSessions 1\\&2: CSNAT is an evidence-based process of family caregiver assessment and support in specialized medical (palliative) care. The CSNAT tool is structured around 16 categories of family caregiver support. With the goal to decrease caregiver burden, this process consists of 5 stages wherein a nurse or practitioner works with the caregiver to create a shared support plan for the child.\n\nSession 3: Respecting Choices® Next Steps- This advanced care planning (pACP) conversation engages families in a process for how to make future medical decisions consistent with their goals and values. The interview is structured in 6 stages to achieve 2 main goals: to facilitate conversations with the family about their child's medical condition, history, fears, values, beliefs, and hopes; and to set the stage for the family's future healthcare decisions.\n\nFAmily CEntered (FACE) pACP Intervention: The (approximately) weekly 3-session FACE-Rare intervention of approximately 45-60 minutes each is comprised of the CSNAT approach \\[Sessions 1 \\& 2\\] and Respecting Choices \\[Session 3\\]."}, {'id': 'OG001', 'title': 'Treatment-as-Usual (TAU) Control', 'description': 'To minimize the burden to families, we have chosen a Treatment-as-Usual (TAU) comparison condition, where patients will receive their normal standard of care. Both study arms will receive palliative (specialized medical) care information at enrollment and complete questionnaires before and after the intervention or TAU period. Current practice for minors with life-limiting illnesses is to defer initial discussions of advanced care planning (pACP) until a medical crisis, so this is what the TAU control arm condition will consist of.'}], 'classes': [{'title': '1. It was useful', 'categories': [{'title': 'Strongly disagree', 'measurements': [{'value': '0', 'groupId': 'OG000'}, {'value': '1', 'groupId': 'OG001'}]}, {'title': 'Disagree', 'measurements': [{'value': '0', 'groupId': 'OG000'}, {'value': '2', 'groupId': 'OG001'}]}, {'title': 'Neither agree or disagree.', 'measurements': [{'value': '3', 'groupId': 'OG000'}, {'value': '4', 'groupId': 'OG001'}]}, {'title': 'Agree', 'measurements': [{'value': '3', 'groupId': 'OG000'}, {'value': '4', 'groupId': 'OG001'}]}, {'title': 'Strongly agree.', 'measurements': [{'value': '3', 'groupId': 'OG000'}, {'value': '1', 'groupId': 'OG001'}]}]}, {'title': '2. It was helpful', 'categories': [{'title': 'Strongly disagree', 'measurements': [{'value': '0', 'groupId': 'OG000'}, {'value': '1', 'groupId': 'OG001'}]}, {'title': 'Disagree', 'measurements': [{'value': '0', 'groupId': 'OG000'}, {'value': '2', 'groupId': 'OG001'}]}, {'title': 'Neither agree or disagree.', 'measurements': [{'value': '3', 'groupId': 'OG000'}, {'value': '4', 'groupId': 'OG001'}]}, {'title': 'Agree', 'measurements': [{'value': '4', 'groupId': 'OG000'}, {'value': '4', 'groupId': 'OG001'}]}, {'title': 'Strongly agree.', 'measurements': [{'value': '2', 'groupId': 'OG000'}, {'value': '1', 'groupId': 'OG001'}]}]}, {'title': '3. I felt scared or afraid', 'categories': [{'title': 'Strongly disagree', 'measurements': [{'value': '4', 'groupId': 'OG000'}, {'value': '9', 'groupId': 'OG001'}]}, {'title': 'Disagree', 'measurements': [{'value': '3', 'groupId': 'OG000'}, {'value': '2', 'groupId': 'OG001'}]}, {'title': 'Neither agree or disagree.', 'measurements': [{'value': '2', 'groupId': 'OG000'}, {'value': '0', 'groupId': 'OG001'}]}, {'title': 'Agree', 'measurements': [{'value': '0', 'groupId': 'OG000'}, {'value': '1', 'groupId': 'OG001'}]}, {'title': 'Strongly agree.', 'measurements': [{'value': '0', 'groupId': 'OG000'}, {'value': '0', 'groupId': 'OG001'}]}]}, {'title': '4. It felt like a load off my mind.', 'categories': [{'title': 'Strongly disagree', 'measurements': [{'value': '0', 'groupId': 'OG000'}, {'value': '1', 'groupId': 'OG001'}]}, {'title': 'Disagree', 'measurements': [{'value': '3', 'groupId': 'OG000'}, {'value': '3', 'groupId': 'OG001'}]}, {'title': 'Neither agree or disagree.', 'measurements': [{'value': '2', 'groupId': 'OG000'}, {'value': '7', 'groupId': 'OG001'}]}, {'title': 'Agree', 'measurements': [{'value': '4', 'groupId': 'OG000'}, {'value': '1', 'groupId': 'OG001'}]}, {'title': 'Strongly agree.', 'measurements': [{'value': '0', 'groupId': 'OG000'}, {'value': '0', 'groupId': 'OG001'}]}]}, {'title': '5. It was too much to handle.', 'categories': [{'title': 'Strongly disagree', 'measurements': [{'value': '3', 'groupId': 'OG000'}, {'value': '5', 'groupId': 'OG001'}]}, {'title': 'Disagree', 'measurements': [{'value': '4', 'groupId': 'OG000'}, {'value': '5', 'groupId': 'OG001'}]}, {'title': 'Neither agree or disagree.', 'measurements': [{'value': '0', 'groupId': 'OG000'}, {'value': '1', 'groupId': 'OG001'}]}, {'title': 'Agree', 'measurements': [{'value': '2', 'groupId': 'OG000'}, {'value': '1', 'groupId': 'OG001'}]}, {'title': 'Strongly agree.', 'measurements': [{'value': '0', 'groupId': 'OG000'}, {'value': '0', 'groupId': 'OG001'}]}]}, {'title': '6. I felt satisfied.', 'categories': [{'title': 'Strongly disagree', 'measurements': [{'value': '0', 'groupId': 'OG000'}, {'value': '0', 'groupId': 'OG001'}]}, {'title': 'Disagree', 'measurements': [{'value': '1', 'groupId': 'OG000'}, {'value': '2', 'groupId': 'OG001'}]}, {'title': 'Neither agree or disagree.', 'measurements': [{'value': '2', 'groupId': 'OG000'}, {'value': '4', 'groupId': 'OG001'}]}, {'title': 'Agree', 'measurements': [{'value': '6', 'groupId': 'OG000'}, {'value': '4', 'groupId': 'OG001'}]}, {'title': 'Strongly agree.', 'measurements': [{'value': '0', 'groupId': 'OG000'}, {'value': '2', 'groupId': 'OG001'}]}]}, {'title': '7. It was harmful.', 'categories': [{'title': 'Strongly disagree', 'measurements': [{'value': '6', 'groupId': 'OG000'}, {'value': '10', 'groupId': 'OG001'}]}, {'title': 'Disagree', 'measurements': [{'value': '3', 'groupId': 'OG000'}, {'value': '1', 'groupId': 'OG001'}]}, {'title': 'Neither agree or disagree.', 'measurements': [{'value': '0', 'groupId': 'OG000'}, {'value': '1', 'groupId': 'OG001'}]}, {'title': 'Agree', 'measurements': [{'value': '0', 'groupId': 'OG000'}, {'value': '0', 'groupId': 'OG001'}]}, {'title': 'Strongly agree.', 'measurements': [{'value': '0', 'groupId': 'OG000'}, {'value': '0', 'groupId': 'OG001'}]}]}, {'title': '8. I felt angry.', 'categories': [{'title': 'Strongly disagree', 'measurements': [{'value': '6', 'groupId': 'OG000'}, {'value': '10', 'groupId': 'OG001'}]}, {'title': 'Disagree', 'measurements': [{'value': '3', 'groupId': 'OG000'}, {'value': '1', 'groupId': 'OG001'}]}, {'title': 'Neither agree or disagree.', 'measurements': [{'value': '0', 'groupId': 'OG000'}, {'value': '1', 'groupId': 'OG001'}]}, {'title': 'Agree', 'measurements': [{'value': '0', 'groupId': 'OG000'}, {'value': '0', 'groupId': 'OG001'}]}, {'title': 'Strongly agree.', 'measurements': [{'value': '0', 'groupId': 'OG000'}, {'value': '0', 'groupId': 'OG001'}]}]}, {'title': '9. It was something I needed to do.', 'categories': [{'title': 'Strongly disagree', 'measurements': [{'value': '0', 'groupId': 'OG000'}, {'value': '1', 'groupId': 'OG001'}]}, {'title': 'Disagree', 'measurements': [{'value': '1', 'groupId': 'OG000'}, {'value': '0', 'groupId': 'OG001'}]}, {'title': 'Neither agree or disagree.', 'measurements': [{'value': '1', 'groupId': 'OG000'}, {'value': '5', 'groupId': 'OG001'}]}, {'title': 'Agree', 'measurements': [{'value': '4', 'groupId': 'OG000'}, {'value': '4', 'groupId': 'OG001'}]}, {'title': 'Strongly agree.', 'measurements': [{'value': '3', 'groupId': 'OG000'}, {'value': '2', 'groupId': 'OG001'}]}]}, {'title': '10. I felt sad.', 'categories': [{'title': 'Strongly disagree', 'measurements': [{'value': '1', 'groupId': 'OG000'}, {'value': '7', 'groupId': 'OG001'}]}, {'title': 'Disagree', 'measurements': [{'value': '2', 'groupId': 'OG000'}, {'value': '3', 'groupId': 'OG001'}]}, {'title': 'Neither agree or disagree.', 'measurements': [{'value': '0', 'groupId': 'OG000'}, {'value': '1', 'groupId': 'OG001'}]}, {'title': 'Agree', 'measurements': [{'value': '5', 'groupId': 'OG000'}, {'value': '1', 'groupId': 'OG001'}]}, {'title': 'Strongly agree.', 'measurements': [{'value': '1', 'groupId': 'OG000'}, {'value': '0', 'groupId': 'OG001'}]}]}, {'title': '111. I felt courageous.', 'categories': [{'title': 'Strongly disagree', 'measurements': [{'value': '1', 'groupId': 'OG000'}, {'value': '1', 'groupId': 'OG001'}]}, {'title': 'Disagree', 'measurements': [{'value': '2', 'groupId': 'OG000'}, {'value': '2', 'groupId': 'OG001'}]}, {'title': 'Neither agree or disagree.', 'measurements': [{'value': '3', 'groupId': 'OG000'}, {'value': '6', 'groupId': 'OG001'}]}, {'title': 'Agree', 'measurements': [{'value': '3', 'groupId': 'OG000'}, {'value': '1', 'groupId': 'OG001'}]}, {'title': 'Strongly agree.', 'measurements': [{'value': '0', 'groupId': 'OG000'}, {'value': '2', 'groupId': 'OG001'}]}]}, {'title': '12. It felt harmful.', 'categories': [{'title': 'Strongly disagree', 'measurements': [{'value': '5', 'groupId': 'OG000'}, {'value': '9', 'groupId': 'OG001'}]}, {'title': 'Disagree', 'measurements': [{'value': '4', 'groupId': 'OG000'}, {'value': '1', 'groupId': 'OG001'}]}, {'title': 'Neither agree or disagree.', 'measurements': [{'value': '0', 'groupId': 'OG000'}, {'value': '2', 'groupId': 'OG001'}]}, {'title': 'Agree', 'measurements': [{'value': '0', 'groupId': 'OG000'}, {'value': '0', 'groupId': 'OG001'}]}, {'title': 'Strongly agree.', 'measurements': [{'value': '0', 'groupId': 'OG000'}, {'value': '0', 'groupId': 'OG001'}]}]}, {'title': '13. It was worthwhile.', 'categories': [{'title': 'Strongly disagree', 'measurements': [{'value': '0', 'groupId': 'OG000'}, {'value': '1', 'groupId': 'OG001'}]}, {'title': 'Disagree', 'measurements': [{'value': '0', 'groupId': 'OG000'}, {'value': '0', 'groupId': 'OG001'}]}, {'title': 'Neither agree or disagree.', 'measurements': [{'value': '0', 'groupId': 'OG000'}, {'value': '2', 'groupId': 'OG001'}]}, {'title': 'Agree', 'measurements': [{'value': '7', 'groupId': 'OG000'}, {'value': '7', 'groupId': 'OG001'}]}, {'title': 'Strongly agree.', 'measurements': [{'value': '2', 'groupId': 'OG000'}, {'value': '2', 'groupId': 'OG001'}]}]}], 'paramType': 'COUNT_OF_PARTICIPANTS', 'timeFrame': 'Up to 5 Weeks Post-Baseline', 'description': 'Items for this 13-item assessment of family satisfaction with the intervention were developed using input from the community about the emotional aspects of participation. Items are on a 5-point Likert scale from strongly disagree to strongly agree. 6 items were negative (felt afraid, too much to handle, harmful, angry, sad hurtful) and 7 items were positive (useful, helpful, load off my mind, satisfied, something I needed to do, courageous, worthwhile). Each item was scored separately. Higher score for positive scale was better outcome. Higher score for negative scale was worse outcome. Study-specific process measure to assess adverse events and benefit/burden of participation. 12 items Total.', 'unitOfMeasure': 'Participants', 'reportingStatus': 'POSTED', 'populationDescription': 'Family caregivers of children with rare diseases'}, {'type': 'OTHER_PRE_SPECIFIED', 'title': 'Quality of Communication Questionnaire', 'denoms': [{'units': 'Participants', 'counts': [{'value': '9', 'groupId': 'OG000'}, {'value': '12', 'groupId': 'OG001'}]}], 'groups': [{'id': 'OG000', 'title': 'FACE-Rare Intervention', 'description': "FACE-Rare is a behavioral intervention that combines the CSNAT Pediatric Approach and the Respecting Choices® Next Steps ACP over 3 sessions.\n\nSessions 1\\&2: CSNAT is an evidence-based process of family caregiver assessment and support in specialized medical (palliative) care. The CSNAT tool is structured around 16 categories of family caregiver support. With the goal to decrease caregiver burden, this process consists of 5 stages wherein a nurse or practitioner works with the caregiver to create a shared support plan for the child.\n\nSession 3: Respecting Choices® Next Steps- This advanced care planning (pACP) conversation engages families in a process for how to make future medical decisions consistent with their goals and values. The interview is structured in 6 stages to achieve 2 main goals: to facilitate conversations with the family about their child's medical condition, history, fears, values, beliefs, and hopes; and to set the stage for the family's future healthcare decisions.\n\nFAmily CEntered (FACE) pACP Intervention: The (approximately) weekly 3-session FACE-Rare intervention of approximately 45-60 minutes each is comprised of the CSNAT approach \\[Sessions 1 \\& 2\\] and Respecting Choices \\[Session 3\\]."}, {'id': 'OG001', 'title': 'Treatment-as-Usual (TAU) Control', 'description': 'To minimize the burden to families, we have chosen a Treatment-as-Usual (TAU) comparison condition, where patients will receive their normal standard of care. Both study arms will receive palliative (specialized medical) care information at enrollment and complete questionnaires before and after the intervention or TAU period. Current practice for minors with life-limiting illnesses is to defer initial discussions of advanced care planning (pACP) until a medical crisis, so this is what the TAU control arm condition will consist of.'}], 'classes': [{'title': '1. Do you think that your attitudes are known by the interviewer?', 'categories': [{'title': 'Definitely yes', 'measurements': [{'value': '4', 'groupId': 'OG000'}, {'value': '1', 'groupId': 'OG001'}]}, {'title': 'Do Not Know', 'measurements': [{'value': '1', 'groupId': 'OG000'}, {'value': '5', 'groupId': 'OG001'}]}, {'title': 'Probably yes', 'measurements': [{'value': '4', 'groupId': 'OG000'}, {'value': '3', 'groupId': 'OG001'}]}, {'title': 'Declined', 'measurements': [{'value': '0', 'groupId': 'OG000'}, {'value': '1', 'groupId': 'OG001'}]}, {'title': 'Definitely no', 'measurements': [{'value': '0', 'groupId': 'OG000'}, {'value': '1', 'groupId': 'OG001'}]}, {'title': 'Missing', 'measurements': [{'value': '0', 'groupId': 'OG000'}, {'value': '1', 'groupId': 'OG001'}]}]}, {'title': '2. Did you feel that the interviewer cared about you as a person?', 'categories': [{'title': 'Definitely yes', 'measurements': [{'value': '8', 'groupId': 'OG000'}, {'value': '6', 'groupId': 'OG001'}]}, {'title': 'Do Not Know', 'measurements': [{'value': '0', 'groupId': 'OG000'}, {'value': '1', 'groupId': 'OG001'}]}, {'title': 'Probably yes', 'measurements': [{'value': '1', 'groupId': 'OG000'}, {'value': '3', 'groupId': 'OG001'}]}, {'title': 'Declined', 'measurements': [{'value': '0', 'groupId': 'OG000'}, {'value': '1', 'groupId': 'OG001'}]}, {'title': 'Definitely no', 'measurements': [{'value': '0', 'groupId': 'OG000'}, {'value': '0', 'groupId': 'OG001'}]}, {'title': 'Missing', 'measurements': [{'value': '0', 'groupId': 'OG000'}, {'value': '1', 'groupId': 'OG001'}]}]}, {'title': '3. Did you feel that the interviewer listened to what you said?', 'categories': [{'title': 'Definitely yes', 'measurements': [{'value': '9', 'groupId': 'OG000'}, {'value': '8', 'groupId': 'OG001'}]}, {'title': 'Do Not Know', 'measurements': [{'value': '0', 'groupId': 'OG000'}, {'value': '0', 'groupId': 'OG001'}]}, {'title': 'Probably yes', 'measurements': [{'value': '0', 'groupId': 'OG000'}, {'value': '2', 'groupId': 'OG001'}]}, {'title': 'Declined', 'measurements': [{'value': '0', 'groupId': 'OG000'}, {'value': '1', 'groupId': 'OG001'}]}, {'title': 'Definitely no', 'measurements': [{'value': '0', 'groupId': 'OG000'}, {'value': '0', 'groupId': 'OG001'}]}, {'title': 'Missing', 'measurements': [{'value': '0', 'groupId': 'OG000'}, {'value': '1', 'groupId': 'OG001'}]}]}, {'title': '4. Did you feel that the interviewer gave you enough attention?', 'categories': [{'title': 'Definitely yes', 'measurements': [{'value': '9', 'groupId': 'OG000'}, {'value': '10', 'groupId': 'OG001'}]}, {'title': 'Do Not Know', 'measurements': [{'value': '0', 'groupId': 'OG000'}, {'value': '0', 'groupId': 'OG001'}]}, {'title': 'Probably yes', 'measurements': [{'value': '0', 'groupId': 'OG000'}, {'value': '0', 'groupId': 'OG001'}]}, {'title': 'Declined', 'measurements': [{'value': '0', 'groupId': 'OG000'}, {'value': '1', 'groupId': 'OG001'}]}, {'title': 'Definitely no', 'measurements': [{'value': '0', 'groupId': 'OG000'}, {'value': '0', 'groupId': 'OG001'}]}, {'title': 'Missing', 'measurements': [{'value': '0', 'groupId': 'OG000'}, {'value': '1', 'groupId': 'OG001'}]}]}], 'paramType': 'COUNT_OF_PARTICIPANTS', 'timeFrame': 'Up to 5 Weeks Post-Baseline', 'description': "This is a process, not an outcome, measure. The participant is asked to rate how good the facilitator was for each of 4 communication skills on a scale from 0, definitely no, to 5, definitely yes. Higher scores mean better communication. Minimum score was 4. Maximum score was 20. We did not calculate a total score, as this was a process measure to assess the facilitator's communication and to provide feedback to the facilitator.", 'unitOfMeasure': 'Participants', 'reportingStatus': 'POSTED', 'populationDescription': 'Family caregivers of children living with rare diseases.'}, {'type': 'OTHER_PRE_SPECIFIED', 'title': 'Brief Multidimensional Measure of Religiousness/Spirituality (Brief MMRS) "How Often do You go to Religious Services?"', 'denoms': [{'units': 'Participants', 'counts': [{'value': '9', 'groupId': 'OG000'}, {'value': '12', 'groupId': 'OG001'}]}], 'groups': [{'id': 'OG000', 'title': 'FACE-Rare Intervention', 'description': "FACE-Rare is a behavioral intervention that combines the CSNAT Pediatric Approach and the Respecting Choices® Next Steps ACP over 3 sessions.\n\nSessions 1\\&2: CSNAT is an evidence-based process of family caregiver assessment and support in specialized medical (palliative) care. The CSNAT tool is structured around 16 categories of family caregiver support. With the goal to decrease caregiver burden, this process consists of 5 stages wherein a nurse or practitioner works with the caregiver to create a shared support plan for the child.\n\nSession 3: Respecting Choices® Next Steps- This advanced care planning (pACP) conversation engages families in a process for how to make future medical decisions consistent with their goals and values. The interview is structured in 6 stages to achieve 2 main goals: to facilitate conversations with the family about their child's medical condition, history, fears, values, beliefs, and hopes; and to set the stage for the family's future healthcare decisions.\n\nFAmily CEntered (FACE) pACP Intervention: The (approximately) weekly 3-session FACE-Rare intervention of approximately 45-60 minutes each is comprised of the CSNAT approach \\[Sessions 1 \\& 2\\] and Respecting Choices \\[Session 3\\]."}, {'id': 'OG001', 'title': 'Treatment-as-Usual (TAU) Control', 'description': 'To minimize the burden to families, we have chosen a Treatment-as-Usual (TAU) comparison condition, where patients will receive their normal standard of care. Both study arms will receive palliative (specialized medical) care information at enrollment and complete questionnaires before and after the intervention or TAU period. Current practice for minors with life-limiting illnesses is to defer initial discussions of advanced care planning (pACP) until a medical crisis, so this is what the TAU control arm condition will consist of.'}], 'classes': [{'title': 'How often do you go to religious services? baseline', 'categories': [{'title': 'Every month or so', 'measurements': [{'value': '1', 'groupId': 'OG000'}, {'value': '2', 'groupId': 'OG001'}]}, {'title': 'Never', 'measurements': [{'value': '3', 'groupId': 'OG000'}, {'value': '3', 'groupId': 'OG001'}]}, {'title': 'Once or twice a month', 'measurements': [{'value': '2', 'groupId': 'OG000'}, {'value': '1', 'groupId': 'OG001'}]}, {'title': 'Once or twice a year', 'measurements': [{'value': '3', 'groupId': 'OG000'}, {'value': '3', 'groupId': 'OG001'}]}, {'title': 'Every week or more often', 'measurements': [{'value': '0', 'groupId': 'OG000'}, {'value': '2', 'groupId': 'OG001'}]}, {'title': 'More than once a week', 'measurements': [{'value': '0', 'groupId': 'OG000'}, {'value': '1', 'groupId': 'OG001'}]}]}, {'title': 'How often do you go to religious services? 3 month follow up', 'categories': [{'title': 'Every month or so', 'measurements': [{'value': '2', 'groupId': 'OG000'}, {'value': '1', 'groupId': 'OG001'}]}, {'title': 'Never', 'measurements': [{'value': '3', 'groupId': 'OG000'}, {'value': '4', 'groupId': 'OG001'}]}, {'title': 'Once or twice a month', 'measurements': [{'value': '3', 'groupId': 'OG000'}, {'value': '1', 'groupId': 'OG001'}]}, {'title': 'Once or twice a year', 'measurements': [{'value': '1', 'groupId': 'OG000'}, {'value': '3', 'groupId': 'OG001'}]}, {'title': 'Every week or more often', 'measurements': [{'value': '0', 'groupId': 'OG000'}, {'value': '3', 'groupId': 'OG001'}]}, {'title': 'More than once a week', 'measurements': [{'value': '0', 'groupId': 'OG000'}, {'value': '0', 'groupId': 'OG001'}]}]}], 'paramType': 'COUNT_OF_PARTICIPANTS', 'timeFrame': 'Baseline to 3 month follow-up', 'description': 'Responses to "How often do you go to religious services?" Responses ranged from "once or twice a year" to "more than once a week." Higher scores indicated greater attendance at religious services. Responses were used as a covariate in the initial Generalized linear mixed effect models. Prior research had indicated that this variable moderated quality of life outcomes. Higher scores indicate more attendance at religious services.', 'unitOfMeasure': 'Participants', 'reportingStatus': 'POSTED', 'populationDescription': 'Family caregivers of children with rare diseases.'}, {'type': 'OTHER_PRE_SPECIFIED', 'title': 'Brief Multidimensional Measure of Religiousness/Spirituality (Brief MMRS): "To What Extent do You Consider Yourself a Religious Person?"', 'denoms': [{'units': 'Participants', 'counts': [{'value': '9', 'groupId': 'OG000'}, {'value': '12', 'groupId': 'OG001'}]}], 'groups': [{'id': 'OG000', 'title': 'FACE-Rare Intervention', 'description': "FACE-Rare is a behavioral intervention that combines the CSNAT Pediatric Approach and the Respecting Choices® Next Steps ACP over 3 sessions.\n\nSessions 1\\&2: CSNAT is an evidence-based process of family caregiver assessment and support in specialized medical (palliative) care. The CSNAT tool is structured around 16 categories of family caregiver support. With the goal to decrease caregiver burden, this process consists of 5 stages wherein a nurse or practitioner works with the caregiver to create a shared support plan for the child.\n\nSession 3: Respecting Choices® Next Steps- This advanced care planning (pACP) conversation engages families in a process for how to make future medical decisions consistent with their goals and values. The interview is structured in 6 stages to achieve 2 main goals: to facilitate conversations with the family about their child's medical condition, history, fears, values, beliefs, and hopes; and to set the stage for the family's future healthcare decisions.\n\nFAmily CEntered (FACE) pACP Intervention: The (approximately) weekly 3-session FACE-Rare intervention of approximately 45-60 minutes each is comprised of the CSNAT approach \\[Sessions 1 \\& 2\\] and Respecting Choices \\[Session 3\\]."}, {'id': 'OG001', 'title': 'Treatment-as-Usual (TAU) Control', 'description': 'To minimize the burden to families, we have chosen a Treatment-as-Usual (TAU) comparison condition, where patients will receive their normal standard of care. Both study arms will receive palliative (specialized medical) care information at enrollment and complete questionnaires before and after the intervention or TAU period. Current practice for minors with life-limiting illnesses is to defer initial discussions of advanced care planning (pACP) until a medical crisis, so this is what the TAU control arm condition will consist of.'}], 'classes': [{'title': 'To what extent do you consider yourself a religious person? Baseline', 'categories': [{'title': 'Moderately religious', 'measurements': [{'value': '3', 'groupId': 'OG000'}, {'value': '4', 'groupId': 'OG001'}]}, {'title': 'Not religious at all', 'measurements': [{'value': '3', 'groupId': 'OG000'}, {'value': '2', 'groupId': 'OG001'}]}, {'title': 'Slightly religious', 'measurements': [{'value': '1', 'groupId': 'OG000'}, {'value': '4', 'groupId': 'OG001'}]}, {'title': 'Very religious', 'measurements': [{'value': '2', 'groupId': 'OG000'}, {'value': '2', 'groupId': 'OG001'}]}]}, {'title': 'To what extent do you consider yourself a religious person? 3 month follow up', 'categories': [{'title': 'Moderately religious', 'measurements': [{'value': '2', 'groupId': 'OG000'}, {'value': '2', 'groupId': 'OG001'}]}, {'title': 'Not religious at all', 'measurements': [{'value': '2', 'groupId': 'OG000'}, {'value': '3', 'groupId': 'OG001'}]}, {'title': 'Slightly religious', 'measurements': [{'value': '3', 'groupId': 'OG000'}, {'value': '6', 'groupId': 'OG001'}]}, {'title': 'Very religious', 'measurements': [{'value': '2', 'groupId': 'OG000'}, {'value': '1', 'groupId': 'OG001'}]}]}], 'paramType': 'COUNT_OF_PARTICIPANTS', 'timeFrame': 'Baseline and 3 month follow-up', 'description': 'Responses to "To what extent do you consider yourself a religious person?" Responses ranged from "moderately religious" to "not religious at all." Higher scores indicated higher religiousness. Responses were collected as a covariate in the initial Generalized linear mixed effect models. Prior research had indicated that this variable moderated quality of life outcomes. Higher scores in the religious indicate self-perception as a more religious person.', 'unitOfMeasure': 'Participants', 'reportingStatus': 'POSTED', 'populationDescription': 'Family caregivers of children with rare diseases.'}, {'type': 'OTHER_PRE_SPECIFIED', 'title': 'Brief Multidimensional Measurement of Religiousness/Spirituality (BMMRS-adapted): To What Extent do You Consider Yourself a Spiritual Person?', 'denoms': [{'units': 'Participants', 'counts': [{'value': '9', 'groupId': 'OG000'}, {'value': '12', 'groupId': 'OG001'}]}], 'groups': [{'id': 'OG000', 'title': 'FACE-Rare Intervention', 'description': "FACE-Rare is a behavioral intervention that combines the CSNAT Pediatric Approach and the Respecting Choices® Next Steps ACP over 3 sessions.\n\nSessions 1\\&2: CSNAT is an evidence-based process of family caregiver assessment and support in specialized medical (palliative) care. The CSNAT tool is structured around 16 categories of family caregiver support. With the goal to decrease caregiver burden, this process consists of 5 stages wherein a nurse or practitioner works with the caregiver to create a shared support plan for the child.\n\nSession 3: Respecting Choices® Next Steps- This advanced care planning (pACP) conversation engages families in a process for how to make future medical decisions consistent with their goals and values. The interview is structured in 6 stages to achieve 2 main goals: to facilitate conversations with the family about their child's medical condition, history, fears, values, beliefs, and hopes; and to set the stage for the family's future healthcare decisions.\n\nFAmily CEntered (FACE) pACP Intervention: The (approximately) weekly 3-session FACE-Rare intervention of approximately 45-60 minutes each is comprised of the CSNAT approach \\[Sessions 1 \\& 2\\] and Respecting Choices \\[Session 3\\]."}, {'id': 'OG001', 'title': 'Treatment-as-Usual (TAU) Control', 'description': 'To minimize the burden to families, we have chosen a Treatment-as-Usual (TAU) comparison condition, where patients will receive their normal standard of care. Both study arms will receive palliative (specialized medical) care information at enrollment and complete questionnaires before and after the intervention or TAU period. Current practice for minors with life-limiting illnesses is to defer initial discussions of advanced care planning (pACP) until a medical crisis, so this is what the TAU control arm condition will consist of.'}], 'classes': [{'title': 'To what extent do you consider yourself a spiritual person? Baseline', 'categories': [{'title': 'Moderately spiritual', 'measurements': [{'value': '3', 'groupId': 'OG000'}, {'value': '7', 'groupId': 'OG001'}]}, {'title': 'Not spiritual at all', 'measurements': [{'value': '1', 'groupId': 'OG000'}, {'value': '0', 'groupId': 'OG001'}]}, {'title': 'Slightly spiritual', 'measurements': [{'value': '2', 'groupId': 'OG000'}, {'value': '5', 'groupId': 'OG001'}]}, {'title': 'Very spiritual', 'measurements': [{'value': '3', 'groupId': 'OG000'}, {'value': '0', 'groupId': 'OG001'}]}]}, {'title': 'To what extent do you consider yourself a spiritual person? 3 month follow up', 'categories': [{'title': 'Moderately spiritual', 'measurements': [{'value': '2', 'groupId': 'OG000'}, {'value': '4', 'groupId': 'OG001'}]}, {'title': 'Not spiritual at all', 'measurements': [{'value': '1', 'groupId': 'OG000'}, {'value': '0', 'groupId': 'OG001'}]}, {'title': 'Slightly spiritual', 'measurements': [{'value': '3', 'groupId': 'OG000'}, {'value': '4', 'groupId': 'OG001'}]}, {'title': 'Very spiritual', 'measurements': [{'value': '3', 'groupId': 'OG000'}, {'value': '4', 'groupId': 'OG001'}]}]}], 'paramType': 'COUNT_OF_PARTICIPANTS', 'timeFrame': 'Baseline to 3 month outcomes.', 'description': 'Responses to "To What Extent do You Consider Yourself a spiritual person?" Responses ranged from "not spiritual at all" to "very spiritual." Higher score meant more spiritual. Reported are frequency distributions and percentages. A higher score on the spiritual person item indicates self-perception as more spiritual.', 'unitOfMeasure': 'Participants', 'reportingStatus': 'POSTED', 'populationDescription': 'Family caregivers of children with rare diseases.'}]}, 'participantFlowModule': {'groups': [{'id': 'FG000', 'title': 'FACE-Rare Intervention', 'description': "FACE-Rare is a behavioral intervention that combines the CSNAT Pediatric Approach and the Respecting Choices® Next Steps ACP over 3 sessions.\n\nSessions 1\\&2: CSNAT is an evidence-based process of family caregiver assessment and support in specialized medical (palliative) care. The CSNAT tool is structured around 16 categories of family caregiver support. With the goal to decrease caregiver burden, this process consists of 5 stages wherein a nurse or practitioner works with the caregiver to create a shared support plan for the child.\n\nSession 3: Respecting Choices® Next Steps- This advanced care planning (pACP) conversation engages families in a process for how to make future medical decisions consistent with their goals and values. The interview is structured in 6 stages to achieve 2 main goals: to facilitate conversations with the family about their child's medical condition, history, fears, values, beliefs, and hopes; and to set the stage for the family's future healthcare decisions.\n\nFAmily CEntered (FACE) pACP Intervention: The (approximately) weekly 3-session FACE-Rare intervention of approximately 45-60 minutes each is comprised of the CSNAT approach \\[Sessions 1 \\& 2\\] and Respecting Choices \\[Session 3\\]."}, {'id': 'FG001', 'title': 'Treatment-as-Usual (TAU) Control', 'description': 'To minimize the burden to families, we have chosen a Treatment-as-Usual (TAU) comparison condition, where patients will receive their normal standard of care. Both study arms will receive palliative (specialized medical) care information at enrollment and complete questionnaires before and after the intervention or TAU period. Current practice for minors with life-limiting illnesses is to defer initial discussions of advanced care planning (pACP) until a medical crisis, so this is what the TAU control arm condition will consist of.'}], 'periods': [{'title': 'Overall Study', 'milestones': [{'type': 'STARTED', 'comment': 'Randomization at a ratio of 1:1. Randomization was at the level of the child/caregiver dyad: 9 dyads in intervention, 12 dyads in TAU.', 'achievements': [{'comment': '9 child/caregiver dyads', 'groupId': 'FG000', 'numSubjects': '18'}, {'comment': '12 child/caregiver dyads', 'groupId': 'FG001', 'numSubjects': '24'}]}, {'type': 'Children', 'achievements': [{'groupId': 'FG000', 'numSubjects': '9'}, {'groupId': 'FG001', 'numSubjects': '12'}]}, {'type': 'Caregivers', 'achievements': [{'groupId': 'FG000', 'numSubjects': '9'}, {'groupId': 'FG001', 'numSubjects': '12'}]}, {'type': 'COMPLETED', 'comment': 'Completed are child/caregiver dyads.', 'achievements': [{'comment': '8 child/caregiver dyads completed intervention. One child/family dyad was lost to follow-up after randomization but before beginning the 3-session intervention.', 'groupId': 'FG000', 'numSubjects': '16'}, {'comment': '11 child/caregiver dyads completed TAU. One child/family dyad completed the process measures at 3 weeks post-baseline but was lost to 3-month follow-up.', 'groupId': 'FG001', 'numSubjects': '22'}]}, {'type': 'NOT COMPLETED', 'achievements': [{'groupId': 'FG000', 'numSubjects': '2'}, {'groupId': 'FG001', 'numSubjects': '2'}]}], 'dropWithdraws': [{'type': 'Lost to Follow-up', 'reasons': [{'groupId': 'FG000', 'numSubjects': '2'}, {'groupId': 'FG001', 'numSubjects': '2'}]}]}], 'recruitmentDetails': "Family caregivers were recruited from outpatient clinics at Children's National Hospital, a quaternary pediatric hospital. There were 21 children and 21 primary family caregivers enrolled in the study. Randomization was at the level of the dyad (child with a rare disease and the primary family caregiver). Thus, 42 participants (21 child/family dyads) completed the baseline assessment and were then randomized. The flow chart is reported as adolescent/family dyads."}, 'baselineCharacteristicsModule': {'denoms': [{'units': 'Participants', 'counts': [{'value': '18', 'groupId': 'BG000'}, {'value': '24', 'groupId': 'BG001'}, {'value': '42', 'groupId': 'BG002'}]}], 'groups': [{'id': 'BG000', 'title': 'FACE-Rare Intervention', 'description': "FACE-Rare is a behavioral intervention that combines the CSNAT Pediatric Approach and the Respecting Choices® Next Steps ACP over 3 sessions.\n\nSessions 1\\&2: CSNAT is an evidence-based process of family caregiver assessment and support in specialized medical (palliative) care. The CSNAT tool is structured around 16 categories of family caregiver support. With the goal to decrease caregiver burden, this process consists of 5 stages wherein a nurse or practitioner works with the caregiver to create a shared support plan for the child.\n\nSession 3: Respecting Choices® Next Steps- This advanced care planning (pACP) conversation engages families in a process for how to make future medical decisions consistent with their goals and values. The interview is structured in 6 stages to achieve 2 main goals: to facilitate conversations with the family about their child's medical condition, history, fears, values, beliefs, and hopes; and to set the stage for the family's future healthcare decisions.\n\nFAmily CEntered (FACE) pACP Intervention: The (approximately) weekly 3-session FACE-Rare intervention of approximately 45-60 minutes each is comprised of the CSNAT approach \\[Sessions 1 \\& 2\\] and Respecting Choices \\[Session 3\\]."}, {'id': 'BG001', 'title': 'Treatment-as-Usual (TAU) Control', 'description': 'To minimize the burden to families, we have chosen a Treatment-as-Usual (TAU) comparison condition, where patients will receive their normal standard of care. Both study arms will receive palliative (specialized medical) care information at enrollment and complete questionnaires before and after the intervention or TAU period. Current practice for minors with life-limiting illnesses is to defer initial discussions of advanced care planning (pACP) until a medical crisis, so this is what the TAU control arm condition will consist of.'}, {'id': 'BG002', 'title': 'Total', 'description': 'Total of all reporting groups'}], 'measures': [{'title': 'Age, Categorical', 'classes': [{'title': 'Child with rare disease', 'denoms': [{'units': 'Participants', 'counts': [{'value': '9', 'groupId': 'BG000'}, {'value': '12', 'groupId': 'BG001'}, {'value': '21', 'groupId': 'BG002'}]}], 'categories': [{'title': '<=18 years', 'measurements': [{'value': '9', 'groupId': 'BG000'}, {'value': '12', 'groupId': 'BG001'}, {'value': '21', 'groupId': 'BG002'}]}, {'title': 'Between 18 and 65 years', 'measurements': [{'value': '0', 'groupId': 'BG000'}, {'value': '0', 'groupId': 'BG001'}, {'value': '0', 'groupId': 'BG002'}]}, {'title': '>=65 years', 'measurements': [{'value': '0', 'groupId': 'BG000'}, {'value': '0', 'groupId': 'BG001'}, {'value': '0', 'groupId': 'BG002'}]}]}, {'title': 'Primary family caregiver', 'denoms': [{'units': 'Participants', 'counts': [{'value': '9', 'groupId': 'BG000'}, {'value': '12', 'groupId': 'BG001'}, {'value': '21', 'groupId': 'BG002'}]}], 'categories': [{'title': '<=18 years', 'measurements': [{'value': '0', 'groupId': 'BG000'}, {'value': '0', 'groupId': 'BG001'}, {'value': '0', 'groupId': 'BG002'}]}, {'title': 'Between 18 and 65 years', 'measurements': [{'value': '9', 'groupId': 'BG000'}, {'value': '12', 'groupId': 'BG001'}, {'value': '21', 'groupId': 'BG002'}]}, {'title': '>=65 years', 'measurements': [{'value': '0', 'groupId': 'BG000'}, {'value': '0', 'groupId': 'BG001'}, {'value': '0', 'groupId': 'BG002'}]}]}], 'paramType': 'COUNT_OF_PARTICIPANTS', 'description': "Demographic data were collected on a standardized form. Child's age was measured by family caregiver report and family caregiver age was measured by self-report.", 'unitOfMeasure': 'Participants', 'populationDescription': 'Baseline analysis was at level of participant with separate analysis for children with rare diseases and the primary family caregiver.'}, {'title': 'Sex: Female, Male', 'classes': [{'title': 'Child with rare disease', 'denoms': [{'units': 'Participants', 'counts': [{'value': '9', 'groupId': 'BG000'}, {'value': '12', 'groupId': 'BG001'}, {'value': '21', 'groupId': 'BG002'}]}], 'categories': [{'title': 'Female', 'measurements': [{'value': '5', 'groupId': 'BG000'}, {'value': '6', 'groupId': 'BG001'}, {'value': '11', 'groupId': 'BG002'}]}, {'title': 'Male', 'measurements': [{'value': '4', 'groupId': 'BG000'}, {'value': '6', 'groupId': 'BG001'}, {'value': '10', 'groupId': 'BG002'}]}]}, {'title': 'Primary family caregiver', 'denoms': [{'units': 'Participants', 'counts': [{'value': '9', 'groupId': 'BG000'}, {'value': '12', 'groupId': 'BG001'}, {'value': '21', 'groupId': 'BG002'}]}], 'categories': [{'title': 'Female', 'measurements': [{'value': '7', 'groupId': 'BG000'}, {'value': '10', 'groupId': 'BG001'}, {'value': '17', 'groupId': 'BG002'}]}, {'title': 'Male', 'measurements': [{'value': '2', 'groupId': 'BG000'}, {'value': '2', 'groupId': 'BG001'}, {'value': '4', 'groupId': 'BG002'}]}]}], 'paramType': 'COUNT_OF_PARTICIPANTS', 'description': "Demographic information was collected on a standardized form. Sex of child was based on child's family caregiver report. Sex of primary family caregiver was based on primary family caregiver self-report.", 'unitOfMeasure': 'Participants', 'populationDescription': 'Sex as a biological variable was analyzed as a separate variable for child and primary family caregiver.'}, {'title': 'Race (NIH/OMB)', 'classes': [{'title': 'Child with rare disease', 'denoms': [{'units': 'Participants', 'counts': [{'value': '9', 'groupId': 'BG000'}, {'value': '12', 'groupId': 'BG001'}, {'value': '21', 'groupId': 'BG002'}]}], 'categories': [{'title': 'American Indian or Alaska Native', 'measurements': [{'value': '0', 'groupId': 'BG000'}, {'value': '0', 'groupId': 'BG001'}, {'value': '0', 'groupId': 'BG002'}]}, {'title': 'Asian', 'measurements': [{'value': '0', 'groupId': 'BG000'}, {'value': '0', 'groupId': 'BG001'}, {'value': '0', 'groupId': 'BG002'}]}, {'title': 'Native Hawaiian or Other Pacific Islander', 'measurements': [{'value': '0', 'groupId': 'BG000'}, {'value': '0', 'groupId': 'BG001'}, {'value': '0', 'groupId': 'BG002'}]}, {'title': 'Black or African American', 'measurements': [{'value': '4', 'groupId': 'BG000'}, {'value': '1', 'groupId': 'BG001'}, {'value': '5', 'groupId': 'BG002'}]}, {'title': 'White', 'measurements': [{'value': '3', 'groupId': 'BG000'}, {'value': '10', 'groupId': 'BG001'}, {'value': '13', 'groupId': 'BG002'}]}, {'title': 'More than one race', 'measurements': [{'value': '2', 'groupId': 'BG000'}, {'value': '1', 'groupId': 'BG001'}, {'value': '3', 'groupId': 'BG002'}]}, {'title': 'Unknown or Not Reported', 'measurements': [{'value': '0', 'groupId': 'BG000'}, {'value': '0', 'groupId': 'BG001'}, {'value': '0', 'groupId': 'BG002'}]}]}, {'title': 'Primary family caregiver', 'denoms': [{'units': 'Participants', 'counts': [{'value': '9', 'groupId': 'BG000'}, {'value': '12', 'groupId': 'BG001'}, {'value': '21', 'groupId': 'BG002'}]}], 'categories': [{'title': 'American Indian or Alaska Native', 'measurements': [{'value': '0', 'groupId': 'BG000'}, {'value': '0', 'groupId': 'BG001'}, {'value': '0', 'groupId': 'BG002'}]}, {'title': 'Asian', 'measurements': [{'value': '0', 'groupId': 'BG000'}, {'value': '0', 'groupId': 'BG001'}, {'value': '0', 'groupId': 'BG002'}]}, {'title': 'Native Hawaiian or Other Pacific Islander', 'measurements': [{'value': '0', 'groupId': 'BG000'}, {'value': '0', 'groupId': 'BG001'}, {'value': '0', 'groupId': 'BG002'}]}, {'title': 'Black or African American', 'measurements': [{'value': '3', 'groupId': 'BG000'}, {'value': '1', 'groupId': 'BG001'}, {'value': '4', 'groupId': 'BG002'}]}, {'title': 'White', 'measurements': [{'value': '5', 'groupId': 'BG000'}, {'value': '10', 'groupId': 'BG001'}, {'value': '15', 'groupId': 'BG002'}]}, {'title': 'More than one race', 'measurements': [{'value': '1', 'groupId': 'BG000'}, {'value': '1', 'groupId': 'BG001'}, {'value': '2', 'groupId': 'BG002'}]}, {'title': 'Unknown or Not Reported', 'measurements': [{'value': '0', 'groupId': 'BG000'}, {'value': '0', 'groupId': 'BG001'}, {'value': '0', 'groupId': 'BG002'}]}]}], 'paramType': 'COUNT_OF_PARTICIPANTS', 'description': "Demographic data were collected on a standardized form. Child's race was reported by family caregiver. Family caregiver's race was self-reported.", 'unitOfMeasure': 'Participants', 'populationDescription': 'Analysis of the population was at the level of the individual and not the dyad. We analyzed separately age data for child from age data for primary family caregiver.'}, {'title': 'Region of Enrollment', 'classes': [{'title': 'United States', 'denoms': [{'units': 'Participants', 'counts': [{'value': '18', 'groupId': 'BG000'}, {'value': '24', 'groupId': 'BG001'}, {'value': '42', 'groupId': 'BG002'}]}], 'categories': [{'measurements': [{'value': '18', 'groupId': 'BG000'}, {'value': '24', 'groupId': 'BG001'}, {'value': '42', 'groupId': 'BG002'}]}]}], 'paramType': 'NUMBER', 'description': "All participants were recruited from Children's National Hospital in Washington, DC, in the United States of America. No additional details, such as zip code, were collected from participants.", 'unitOfMeasure': 'participants'}], 'populationDescription': "42 participants (21 child with rare disease and child's primary caregiver dyads) completed baseline assessment. Baseline analysis was at level of participant with separate analysis for child with rare disease and family caregiver."}}, 'documentSection': {'largeDocumentModule': {'largeDocs': [{'date': '2023-05-30', 'size': 790668, 'label': 'Study Protocol and Statistical Analysis Plan', 'hasIcf': False, 'hasSap': True, 'filename': 'Prot_SAP_000.pdf', 'typeAbbrev': 'Prot_SAP', 'uploadDate': '2023-06-06T15:36', 'hasProtocol': True}, {'date': '2023-05-30', 'size': 408616, 'label': 'Informed Consent Form', 'hasIcf': True, 'hasSap': False, 'filename': 'ICF_001.pdf', 'typeAbbrev': 'ICF', 'uploadDate': '2023-06-06T15:35', 'hasProtocol': False}]}}, 'protocolSection': {'designModule': {'phases': ['NA'], 'studyType': 'INTERVENTIONAL', 'designInfo': {'allocation': 'RANDOMIZED', 'maskingInfo': {'masking': 'SINGLE', 'whoMasked': ['OUTCOMES_ASSESSOR'], 'maskingDescription': 'Study arm allocation will be concealed from the Research Assistant (RA) Assessor to prevent bias during the administration of outcome questionnaires, process measures, and chart abstraction.'}, 'primaryPurpose': 'SUPPORTIVE_CARE', 'interventionModel': 'PARALLEL', 'interventionModelDescription': 'Investigators propose a pilot, two-arm, intent-to-treat, single-blinded, single-site, controlled RCT. Family/child dyads (N=30) will be enrolled and randomized to either the FACE-Rare intervention or treatment as usual (TAU) at a 1:1 ratio.'}, 'enrollmentInfo': {'type': 'ACTUAL', 'count': 42}}, 'statusModule': {'overallStatus': 'COMPLETED', 'startDateStruct': {'date': '2021-07-06', 'type': 'ACTUAL'}, 'expandedAccessInfo': {'hasExpandedAccess': False}, 'statusVerifiedDate': '2025-04', 'completionDateStruct': {'date': '2024-06-01', 'type': 'ACTUAL'}, 'lastUpdateSubmitDate': '2025-04-17', 'studyFirstSubmitDate': '2021-02-22', 'resultsFirstSubmitDate': '2025-03-27', 'studyFirstSubmitQcDate': '2021-04-21', 'lastUpdatePostDateStruct': {'date': '2025-05-04', 'type': 'ACTUAL'}, 'resultsFirstSubmitQcDate': '2025-04-17', 'studyFirstPostDateStruct': {'date': '2021-04-22', 'type': 'ACTUAL'}, 'resultsFirstPostDateStruct': {'date': '2025-05-04', 'type': 'ACTUAL'}, 'primaryCompletionDateStruct': {'date': '2023-12-31', 'type': 'ACTUAL'}}, 'outcomesModule': {'otherOutcomes': [{'measure': 'Satisfaction Questionnaire (Process Measure)', 'timeFrame': 'Up to 5 Weeks Post-Baseline', 'description': 'Items for this 13-item assessment of family satisfaction with the intervention were developed using input from the community about the emotional aspects of participation. Items are on a 5-point Likert scale from strongly disagree to strongly agree. 6 items were negative (felt afraid, too much to handle, harmful, angry, sad hurtful) and 7 items were positive (useful, helpful, load off my mind, satisfied, something I needed to do, courageous, worthwhile). Each item was scored separately. Higher score for positive scale was better outcome. Higher score for negative scale was worse outcome. Study-specific process measure to assess adverse events and benefit/burden of participation. 12 items Total.'}, {'measure': 'Quality of Communication Questionnaire', 'timeFrame': 'Up to 5 Weeks Post-Baseline', 'description': "This is a process, not an outcome, measure. The participant is asked to rate how good the facilitator was for each of 4 communication skills on a scale from 0, definitely no, to 5, definitely yes. Higher scores mean better communication. Minimum score was 4. Maximum score was 20. We did not calculate a total score, as this was a process measure to assess the facilitator's communication and to provide feedback to the facilitator."}, {'measure': 'Brief Multidimensional Measure of Religiousness/Spirituality (Brief MMRS) "How Often do You go to Religious Services?"', 'timeFrame': 'Baseline to 3 month follow-up', 'description': 'Responses to "How often do you go to religious services?" Responses ranged from "once or twice a year" to "more than once a week." Higher scores indicated greater attendance at religious services. Responses were used as a covariate in the initial Generalized linear mixed effect models. Prior research had indicated that this variable moderated quality of life outcomes. Higher scores indicate more attendance at religious services.'}, {'measure': 'Brief Multidimensional Measure of Religiousness/Spirituality (Brief MMRS): "To What Extent do You Consider Yourself a Religious Person?"', 'timeFrame': 'Baseline and 3 month follow-up', 'description': 'Responses to "To what extent do you consider yourself a religious person?" Responses ranged from "moderately religious" to "not religious at all." Higher scores indicated higher religiousness. Responses were collected as a covariate in the initial Generalized linear mixed effect models. Prior research had indicated that this variable moderated quality of life outcomes. Higher scores in the religious indicate self-perception as a more religious person.'}, {'measure': 'Brief Multidimensional Measurement of Religiousness/Spirituality (BMMRS-adapted): To What Extent do You Consider Yourself a Spiritual Person?', 'timeFrame': 'Baseline to 3 month outcomes.', 'description': 'Responses to "To What Extent do You Consider Yourself a spiritual person?" Responses ranged from "not spiritual at all" to "very spiritual." Higher score meant more spiritual. Reported are frequency distributions and percentages. A higher score on the spiritual person item indicates self-perception as more spiritual.'}], 'primaryOutcomes': [{'measure': 'Beck Anxiety Inventory', 'timeFrame': 'Change from Baseline Anxiety at 3 Months Post-Enrollment', 'description': 'The Beck Anxiety Inventory (BAI) is a 21-item measure of anxiety rated on a 4-point Likert scale of symptoms over the past week. The BAI has demonstrated reliability and validity to assess anxiety in individuals aged 17-80 years. The BAI has good reliability and validity for family caregivers of seriously ill children. It is a measure of Quality of life: emotional health. Higher scores mean more anxiety symptoms. Total score was used in analysis. Scores range from 0-63.'}, {'measure': 'Functional Assessment of Chronic Illness Therapy (FACIT)- Spiritual Well-Being Scale, Version 4', 'timeFrame': 'Change from Baseline Spiritual Well-Being at 3 Months Post-Enrollment', 'description': 'Assessed construct of spiritual well-being. Two subscales Meaning/Peace (7 items) and Faith (5 items) and Total score (12 items) were calculated. on a 5-point Likert scale from 0=not at all to 5=very much. Some items are reverse scored. See www.facit.org Meaning/Peace subscale score range from minimum value of 0 to maximum value of 32. Higher scores indicate better meaning/peace. Faith subscale score range from 0 minimum value to maximum value of 16. Higher scores indicate better meaning/peace. Higher scores indicate better spiritual well-being.'}, {'measure': 'Advance Care Plan Document for Children With Rare Diseases', 'timeFrame': 'Change from Baseline ACP Documentation in the Electronic Health Record at 12 weeks Post-Enrollment', 'description': 'Advanced Care Planning (ACP) Documentation in Electronic Health Records (EHR). Score will be present in EHR or absent from EHR. Evidence of any advance care document (yes/no) will count.'}, {'measure': 'Days in Palliative Care Prior to Death', 'timeFrame': '12 weeks Post-Enrollment', 'description': 'Score is number of days patient was enrolled in a palliative care program in the 30 days prior to death. Applicable only to patients who have deceased since study enrollment. Used to standardize child healthcare utilization from data abstraction based on retrospective medical chart review. Minimum value is 0 with no maximum. Higher score indicates higher level of healthcare utilization.'}, {'measure': 'Hospitalizations', 'timeFrame': 'Change from Baseline to Frequency at 12 weeks Post-Enrollment', 'description': 'Used to standardize child healthcare utilization from data abstraction by the research nurse based on medical chart review in Electronic Health Record. Score is recorded number of inpatient hospital admissions for clinical treatment for the child with a rare disease. Minimum value is 0 with no maximum. Higher score indicates higher level of healthcare utilization. Questions was "Hospitalization in the last 3 months?"'}, {'measure': 'Place of Death', 'timeFrame': '12 weeks Post-Enrollment', 'description': 'Location where the patient was pronounced dead, as reported in their EMR. Applicable only to patients who have deceased since study enrollment. Categories are Inpatient hospice setting, Home with hospice, Home without hospice, Hospital ICU, Hospital-Not ICU, Other, or Unknown. Used to standardize child healthcare utilization from data abstraction based on retrospective medical chart review.'}], 'secondaryOutcomes': [{'measure': 'Brief-Multidimensional Measure of Religion and Spirituality - Responses to Question 1', 'timeFrame': 'Baseline and 3-month follow-up.', 'description': "Survey responses to: 1. How often do you feel God's presence? On 6-point Likert-scale responses range from every day to never or almost never."}, {'measure': 'Family Appraisal of Caregiving Questionnaire for Palliative Care for Palliative Care (FACQ-PC)', 'timeFrame': 'Change from Baseline appraisal of Caregiving at 12 weeks Post-Enrollment', 'description': 'The FACQ-PC is a 25-item measure consists of four theoretically derived subscales: (i) caregiver strain, (ii) positive caregiving appraisals, (iii) caregiver distress, and (iv) family well-being. Scores are from 5 = strongly agree to 1 = strongly disagree. We did not calculate a Total score. We only used the 4 subscale scores. On the subscale scores for positive caregiving appraisals and family well-being, higher scores mean better outcomes, i.e. greater positive caregiving appraisals or family well-being. On the subscale scores for caregiver strain and caregiver distress, higher scores mean worse outcomes, i.e. greater caregiver strain or caregiver distress. The FACQ-PC subscale scores were computed by taking the mean of the items (score range 1-5). Some items were reverse scored, depending on how the item is phrased, so that higher scores = higher amount of the subscale being measured. So the minimum value for each subscale is 1 and the maximum value for each subscale is 5.'}, {'measure': 'Hickman Role Stress Decisional Burden Scale', 'timeFrame': 'Up to 5 Weeks Post-Enrollment', 'description': 'Visual analogue scale 0-100. "How stressful is it for you to make medical decisions for your child?" 1 item. Higher score means more stress, lower scores mean less stress.'}, {'measure': 'Brief Multidimensional Measurement of Religiousness/Spirituality (BMMRS-adapted) Survey Responses to Question 2.', 'timeFrame': 'Baseline and 3-month follow-up', 'description': 'Responses to survey question 2, How often do you pray privately? 7 point Likert scale ranging from a few times a month to once a day. Higher scores in pray item indicate more prayer.'}]}, 'oversightModule': {'oversightHasDmc': True, 'isFdaRegulatedDrug': False, 'isFdaRegulatedDevice': False}, 'conditionsModule': {'keywords': ['palliative care', 'psychosocial interventions', 'rare diseases', 'family interventions', 'family-centered care', 'patient-centered care', 'pediatrics', 'adolescent', 'end-of-life care', 'public health', 'mental health', 'spirituality', 'children with rare diseases', 'behavioral interventions', 'decisional burden', 'role stress', 'family caregiver', 'person-centered care', 'chronic conditions', 'complex health needs', 'caregiver burden', 'respecting choices', 'evidence-based practice', 'randomized clinical trial (RCT)', 'anxiety'], 'conditions': ['Rare Diseases']}, 'referencesModule': {'references': [{'pmid': '35884083', 'type': 'BACKGROUND', 'citation': 'Lyon ME, Wiener L. Special Issue: Psychosocial Considerations for Children and Adolescents Living with a Rare Disease. Children (Basel). 2022 Jul 21;9(7):1099. doi: 10.3390/children9071099.'}, {'pmid': '35626888', 'type': 'BACKGROUND', 'citation': 'Sandquist M, Davenport T, Monaco J, Lyon ME. The Transition to Adulthood for Youth Living with Rare Diseases. Children (Basel). 2022 May 12;9(5):710. doi: 10.3390/children9050710.'}, {'pmid': '35327817', 'type': 'BACKGROUND', 'citation': 'Fratantoni K, Livingston J, Schellinger SE, Aoun SM, Lyon ME. Family-Centered Advance Care Planning: What Matters Most for Parents of Children with Rare Diseases. Children (Basel). 2022 Mar 21;9(3):445. doi: 10.3390/children9030445.'}, {'pmid': '35327694', 'type': 'BACKGROUND', 'citation': 'Aoun SM, Stegmann R, Deleuil R, Momber S, Cuddeford L, Phillips MB, Lyon ME, Gill FJ. "It Is a Whole Different Life from the Life I Used to Live": Assessing Parents\' Support Needs in Paediatric Palliative Care. Children (Basel). 2022 Mar 1;9(3):322. doi: 10.3390/children9030322.'}, {'pmid': '31345846', 'type': 'BACKGROUND', 'citation': 'Lyon ME, Thompkins JD, Fratantoni K, Fraser JL, Schellinger SE, Briggs L, Friebert S, Aoun S, Cheng YI, Wang J. Family caregivers of children and adolescents with rare diseases: a novel palliative care intervention. BMJ Support Palliat Care. 2022 Nov;12(e5):e705-e714. doi: 10.1136/bmjspcare-2019-001766. Epub 2019 Jul 25.'}, {'pmid': '33033802', 'type': 'BACKGROUND', 'citation': 'Aoun SM, Gill FJ, Phillips MB, Momber S, Cuddeford L, Deleuil R, Stegmann R, Howting D, Lyon ME. The profile and support needs of parents in paediatric palliative care: comparing cancer and non-cancer groups. Palliat Care Soc Pract. 2020 Sep 25;14:2632352420958000. doi: 10.1177/2632352420958000. eCollection 2020.'}, {'type': 'RESULT', 'citation': '- Kreicbergs U (Discussant), Handberg C, Udo C, Thompkins J (presenter) Lyon ME (organizer). Symposium: Lessons Learned during the COVID-19 and Beyond Pandemic for Children Living with Rare Diseases and their Siblings. Lyon Presentation: Family Identified Palliative Care Needs of FAmily Caregivers of Children Living with Rare Diseases during COVID-19-United States 7th Public Health Palliative Care International Conference. September 21, 2022. Bruges, Belgium.'}, {'type': 'RESULT', 'citation': "- Lyon, ME, Fraser J, Thompkins J (presenter). FACE Rare: A novel palliative care intervention for family caregivers of children and adolescents living with a rare disease. Podium Presentation. University of Pittsburgh's National Center on Family Support's Second Biennial Conference on Caregiving Research. Building Bridges: Advancing Family Caregiving Research Across the Lifespan, National Center on Caregiving. Pittsburg, PA. October 1, 2022."}, {'type': 'RESULT', 'citation': 'Lyon ML. Detwiler K, Torres C, Guerrera MF, Thompkins J. FACE-Rare: A Novel Intervention for Family Caregivers of Children Living with Rare Diseases. BMJ Supportive & Palliative Care 2023;13(Suppl 4):A16.'}], 'seeAlsoLinks': [{'url': 'https://doi.org/10.1136/spcare-2023-ACP.39', 'label': 'Lyon ML. Detwiler K, Torres C, Guerrera MF, Thompkins J. FACE-Rare: A Novel Intervention for Family Caregivers of Children Living with Rare Diseases. BMJ Supportive \\& Palliative Care 2023;13(Suppl 4):A16.'}]}, 'descriptionModule': {'briefSummary': 'Children with ultra-rare or complex rare diseases are routinely excluded from research studies because of their conditions, creating a health disparity. However, new statistical techniques make it possible to study small samples of heterogeneous populations. We propose to study the palliative care needs of family caregivers of children with ultra-rare diseases and to pilot test a palliative care needs assessment and advance care planning intervention to facilitate discussions about the future medical care choices families are likely to be asked to make for their child.', 'detailedDescription': "A rare disease is a condition affecting fewer than 200,000 persons. Pediatric patients with rare diseases experience high mortality with 30% not living to see their 5th birthday. Families are likely to be asked to make complex medical decisions for their child. Pediatric advance care planning involves preparation and skill development to help make future medical care choices. Children with rare disorders are a heterogeneous group often with co-morbidities, resulting in their exclusion from research, thereby creating a health disparity for this vulnerable population. Available research on families of children with rare diseases lacks scientific rigor. Although desperately needed, there are few empirically validated interventions to address these issues. We propose to close a gap in our knowledge of families' needs for support in a heterogeneous group of children with rare diseases; and to test an advance care planning intervention.\n\nThe FAmily CEntered (FACE) pediatric advance care planning intervention, proven successful with cancer and HIV, is adapted to families with children who have rare diseases. Theoretically informed and developed by the PI, Dr. Lyon, and colleagues, the proposed intervention will use Respecting Choices Next Steps Pediatric Advance Care Planning™ for families whose child is unable to participate in health care decision-making. Our consultation with families of children with rare disorders and the National Organization for Rare Disorders (NORD) revealed that basic palliative care needs should be addressed first, before an advance care planning intervention. For the study to be able to meet this request, all families randomized to the intervention will first complete the Carer Support Needs Assessment Tool (CSNAT)© which our investigative team adapted for use in pediatrics. In the CSNAT Approach, facilitators assess caregivers' prioritized palliative care needs and develop Shared Action Plans for palliative care support. Thus, we propose an innovative 3-session FACE-Rare intervention, integrating two evidence-based approaches. We will evaluate FACE-Rare using a scientifically rigorous intent-to-treat, single-blinded, randomized controlled trial design. Family/child pairs or dyads (N=30 dyads) will be randomized to FACE-Rare (CSNAT Sessions 1 \\& 2 plus Respecting Choices Sessions 3) or control (Treatment As Usual) groups. Both groups will receive palliative care information. All families will complete questionnaires at baseline and 3-months follow-up. Investigators will evaluate the initial efficacy of FACE-Rare on family quality of life (psychological, spiritual). We will estimate how religiousness and caregiver appraisal influence families' quality of life. We will also explore health care utilization by the children during the study and family satisfaction.\n\nIf the aims of this pilot trial are achieved, a future, large, multi-site trial will test the full theoretical model to improve care for children with rare diseases and their families through family engaged pediatric Advance Care Planning. The ultimate goal is to minimize suffering and enhance the quality of life of family caregivers of children with rare diseases; and through this process to improve the palliative care of their children."}, 'eligibilityModule': {'sex': 'ALL', 'stdAges': ['CHILD', 'ADULT', 'OLDER_ADULT'], 'maximumAge': '99 Years', 'minimumAge': '1 Year', 'healthyVolunteers': False, 'eligibilityCriteria': "Inclusion Criteria:\n\nChild inclusion criteria are\n\n1. ≥1.0 years and \\<18.0 years at enrollment;e\n2. unable to participate in end-of-life care decision-making;\n3. have a rare disease as operationally defined (See Human Subjects);\n4. not under a Do Not Resuscitate Order or Allow a Natural Death Order; and\n5. not in the Intensive Care Unit.\n\nFamily caregiver inclusion criteria are:\n\n1. ≥ 18.0 years at enrollment;\n2. legal guardian of child and child's caregiver;\n3. can speak and understand English; and\n4. not known to be developmentally delayed.\n\nExclusion Criteria:\n\n(1) Family caregiver is actively homicidal, suicidal, or psychotic at the time of enrollment."}, 'identificationModule': {'nctId': 'NCT04855734', 'acronym': 'FACE-Rare', 'briefTitle': 'FACE for Children With Rare Diseases', 'organization': {'class': 'OTHER', 'fullName': "Children's National Research Institute"}, 'officialTitle': 'Palliative Care Needs of Children With Rare Diseases and Their Families', 'orgStudyIdInfo': {'id': '00008808'}, 'secondaryIdInfos': [{'id': '1R21NR019340-01', 'link': 'https://reporter.nih.gov/quickSearch/1R21NR019340-01', 'type': 'NIH'}]}, 'armsInterventionsModule': {'armGroups': [{'type': 'EXPERIMENTAL', 'label': 'FACE-Rare Intervention', 'description': "FACE-Rare is a behavioral intervention that combines the CSNAT Pediatric Approach and the Respecting Choices® Next Steps ACP over 3 sessions.\n\nSessions 1\\&2: CSNAT is an evidence-based process of family caregiver assessment and support in specialized medical (palliative) care. The CSNAT tool is structured around 16 categories of family caregiver support. With the goal to decrease caregiver burden, this process consists of 5 stages wherein a nurse or practitioner works with the caregiver to create a shared support plan for the child.\n\nSession 3: Respecting Choices® Next Steps- This advanced care planning (pACP) conversation engages families in a process for how to make future medical decisions consistent with their goals and values. The interview is structured in 6 stages to achieve 2 main goals: to facilitate conversations with the family about their child's medical condition, history, fears, values, beliefs, and hopes; and to set the stage for the family's future healthcare decisions.", 'interventionNames': ['Behavioral: FAmily CEntered (FACE) pACP Intervention']}, {'type': 'NO_INTERVENTION', 'label': 'Treatment-as-Usual (TAU) Control', 'description': 'To minimize the burden to families, we have chosen a Treatment-as-Usual (TAU) comparison condition, where patients will receive their normal standard of care. Both study arms will receive palliative (specialized medical) care information at enrollment and complete questionnaires before and after the intervention or TAU period. Current practice for minors with life-limiting illnesses is to defer initial discussions of advanced care planning (pACP) until a medical crisis, so this is what the TAU control arm condition will consist of.'}], 'interventions': [{'name': 'FAmily CEntered (FACE) pACP Intervention', 'type': 'BEHAVIORAL', 'otherNames': ['FACE-Rare Intervention'], 'description': 'The (approximately) weekly 3-session FACE-Rare intervention of approximately 45-60 minutes each is comprised of the CSNAT approach \\[Sessions 1 \\& 2\\] and Respecting Choices \\[Session 3\\].', 'armGroupLabels': ['FACE-Rare Intervention']}]}, 'contactsLocationsModule': {'locations': [{'zip': '20010', 'city': 'Washington D.C.', 'state': 'District of Columbia', 'country': 'United States', 'facility': "Children's National Hospital", 'geoPoint': {'lat': 38.89511, 'lon': -77.03637}}], 'overallOfficials': [{'name': 'Maureen E Lyon, PhD', 'role': 'PRINCIPAL_INVESTIGATOR', 'affiliation': "Children's National Research Institute"}]}, 'ipdSharingStatementModule': {'ipdSharing': 'NO', 'description': 'The proposed research will include data from 30 children living with rare diseases who are unable to participate in their own medical care decision-making and their 30 family caregivers. The final dataset will be stripped of individual-level identifiable data prior to release for sharing. Given the vulnerable nature of the subjects (child participants), we will make the data and associated documentation available to researchers under a data-sharing agreement that includes:\n\n1. appropriate human-subjects review and approval; and\n2. a commitment to use the data only for research purposes and that no attempt will be made to identify individual participants.\n\nThe Biostatistics group will perform the mechanics of de-identification and provide the data in an agreed-upon format. De-identification will include stripping the data of PHI; removing dates or adding a random offset to the dates. Only de-identified group data will be shared. This protects participant confidentiality.'}, 'sponsorCollaboratorsModule': {'leadSponsor': {'name': "Children's National Research Institute", 'class': 'OTHER'}, 'collaborators': [{'name': 'Respecting Choices', 'class': 'UNKNOWN'}, {'name': 'National Institutes of Health (NIH)', 'class': 'NIH'}, {'name': 'National Institute of Nursing Research (NINR)', 'class': 'NIH'}], 'responsibleParty': {'type': 'PRINCIPAL_INVESTIGATOR', 'investigatorTitle': 'Dr. Maureen Lyon, Ph.D, ABPP', 'investigatorFullName': 'Maureen Lyon', 'investigatorAffiliation': "Children's National Research Institute"}}}}