Adapting a Behavioral Intervention for Caregivers of Children with Down Syndrome or Fragile X Syndrome: A Pilot Study of RUBI-DD

Challenging behaviors, including noncompliance, aggression, hyperactivity, and impulsivity, are common among individuals with Fragile X Syndrome (FXS) and Down Syndrome (DS). To identify treatment needs specific to these populations, we conducted focus groups with caregivers and educators and used their input to adapt an evidence-based caregiver training program originally designed for caregivers of autistic children (i.e., The Research Units in Behavioral Intervention; RUBI). We then completed a feasibility trial in which five families of children with FXS and four families of children with DS completed a nine-session caregiver training program targeting behavioral principles, syndrome-specific information, and visual supports tailored to the unique needs of FXS or DS (adapted version of RUBI for non-autism developmental disabilities; RUBI-DD). The program demonstrated strong acceptability, with high caregiver satisfaction, 100% retention, and 100% session attendance. Across the combined sample, caregiver reports indicated significant improvements in irritability/aggression (F(2,15.14) = 4.42, p = 0.03), lethargy/social withdrawal (F(2,14.47) = 3.97, p = 0.04), stereotypies (F(2,15.29) = 4.45, p = 0.03), hyperactivity (F(2,15.14) = 6.51, p = 0.009), social inflexibility (F(2,15.43) = 6.33, p = 0.01), demand-based noncompliance (F(2,15.41) = 4.95, p = 0.02), and the impact of behavior on the family (F(2,15.07) = 4.23, p = 0.04) following participation in RUBI-DD. Caregivers of children with FXS reported significant reductions in lethargy/social withdrawal (F(2,8.000) = 6.256, p = 0.023) and hyperactivity (F(2,8.000) = 12.497, p = 0.003) immediately post-treatment and upon 12-week follow-up (g = 1.153, p = 0.044, and g = 1.178, p = 0.003, respectively). Among families of children with DS, caregivers reported reductions in irritability and aggression (F(2,5.047) = 14.073, p = 0.009) and improvements in the impact on the family (F(2,6.000) = 5.489, p = 0.044) immediately post-treatment and at follow-up (g = 1.643, p = 0.016, and g = 0.448, p = 0.045, respectively). These findings support the feasibility, acceptability, and preliminary efficacy of RUBI-DD for children with FXS or DS.