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Advance Care Planning for Children With Rare Diseases: A Pilot RCT
Maureen E Lyon1,2, Jamie L Fraser1,2, Jessica D Thompkins1
1Children's National Hospital, Washington, District of Columbia.
Insights
The FAmily CEntered (FACE)-Rare pediatric advance care planning intervention improved caregiver well-being and feelings of peace in families with rare pediatric diseases. Poverty was found to negatively impact family well-being.
Area of Science:
- Pediatric rare diseases
- Advance care planning
- Family quality of life
Background:
- Pediatric rare diseases often present life-limiting challenges and necessitate continuous caregiving.
- Assessing interventions to improve quality of life for these families is crucial.
Purpose of the Study:
- To evaluate the initial efficacy of the FAmily CEntered (FACE)-Rare pediatric advance care planning (pACP) intervention.
- To determine the intervention's impact on family quality of life.
Main Methods:
- A pilot-phase, single-blinded, randomized controlled trial was conducted.
- Families were randomized to either the FACE-Rare pACP intervention or treatment as usual (TAU).
- Outcomes included anxiety, caregiver appraisal, spirituality, and healthcare utilization, analyzed using generalized mixed effect models.
Main Results:
- Caregivers in the FACE-Rare group reported higher levels of meaning and peace compared to TAU.
- Black caregivers experienced significantly less caregiver distress than non-Black caregivers.
- Families living in poverty reported increased anxiety and caregiver strain, alongside decreased family well-being.
Conclusions:
- The FACE-Rare intervention is feasible, acceptable, and safe, showing initial promise in enhancing caregiver well-being.
- Socioeconomic factors, particularly poverty, significantly affect family well-being in this population.
- Further multi-site trials are recommended to establish generalizability.
Background And Objective:
Pediatric rare diseases are often life-limiting conditions and/or require constant caregiving. Investigators assessed the initial efficacy of the FAmily CEntered (FACE) pediatric advance care planning (pACP), FACE-Rare, intervention on families' quality of life.
Methods:
A pilot-phase, single-blinded, intent-to-treat, randomized controlled clinical trial enrolled families from 1 pediatric quaternary hospital between 2021 and 2023. Intervention families received 3 weekly 60-minute (FACE-Rare pACP) sessions: (1) Carer Support Needs Assessment Tool or Action Plan, (2) Carer Support Needs Assessment Tol Action Plan Review, and (3) Pediatric Next Steps: Respecting Choices pACP. Controls received treatment as usual (TAU). Outcome measures were Beck Anxiety Inventory, Family Appraisal of Caregiving, Functional Assessment of Chronic Illness Therapy (FACIT)-Spirituality, and health care utilization. Generalized mixed effect models with γ response assessed the intervention effect at 3-month follow-up.
Results:
Children (n = 21) were aged 1 to 10 years, 48% male, 24% Black; and 100% technology dependent. Primary family caregivers (n = 21) were aged 30 to 43 years, 19% male, 19% Black; and 27% household income below the Federal poverty level. Dyads underwent 1:1 randomization: 9 to FACE-Rare and 12 to TAU. TAU caregivers reported statistically lower meaning and peace than FACE-Rare caregivers (0.9, P = .03, confidence interval [CI]: 0.75-0.99). Black caregivers reported significantly less caregiver distress (0.7, P = .04, CI: 0.47-0.98) than non-Black caregivers. Poor families reported more anxiety (3.5, P = .002, CI: 1.62-7.94), more caregiver strain (1.2, P = .006, CI: 1.07-1.42); and less family well-being (0.8, P = .02, CI: 0.64-0.95).
Conclusions:
FACE®-Rare was feasible, acceptable, safe, and demonstrated initial efficacy, providing greater feelings of meaning and peace to caregivers. Poverty impacted well-being. A multisite trial is needed to determine generalizability.
