Advance Care Planning for Children With Rare Diseases: A Pilot RCT

Authors

Maureen E. Lyon, Children's National Hospital, Washington, District of Columbia.
Jamie L. Fraser, Children's National Hospital, Washington, District of Columbia.
Jessica D. Thompkins, Children's National Hospital, Washington, District of Columbia.
Heidi Clark, Children's National Hospital, Washington, District of Columbia.
Nicola Brodie, Children's National Hospital, Washington, District of Columbia.
Kathryn Detwiler, Children's National Hospital, Washington, District of Columbia.
Clarivet Torres, Children's National Hospital, Washington, District of Columbia.
Michael F. Guerrera, Children's National Hospital, Washington, District of Columbia.
Tamiko Younge, Children's National Hospital, Washington, District of Columbia.
Samar Aoun, Peron Institute, Palliative Care, The Western University of Australia, Perth, Washington, Australia.
Eduardo A. Trujillo Rivera, Children's National Hospital, Washington, District of Columbia.

Document Type

Journal Article

Publication Date

6-1-2024

Journal

Pediatrics

Volume

153

Issue

6

DOI

10.1542/peds.2023-064557

Abstract

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.

APA Citation

Lyon, Maureen E.; Fraser, Jamie L.; Thompkins, Jessica D.; Clark, Heidi; Brodie, Nicola; Detwiler, Kathryn; Torres, Clarivet; Guerrera, Michael F.; Younge, Tamiko; Aoun, Samar; and Trujillo Rivera, Eduardo A., "Advance Care Planning for Children With Rare Diseases: A Pilot RCT" (2024). GW Authored Works. Paper 5195.
https://hsrc.himmelfarb.gwu.edu/gwhpubs/5195

Department

Pediatrics