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Principal Investigator: Latha Valluripalli Soorya
Organization: RUSH UNIVERSITY MEDICAL CENTER
Fiscal Year: 2024
Award: $806,933
Funding agency: Eunice Kennedy Shriver National Institute of Child Health and Human Development
Project Summary/Abstract:
The enormous impact of COVID-19 service disruptions on individuals with intellectual and developmental
disabilities (IDDs) has highlighted the critically urgent need to increase access to mental and behavioral health
services. Within IDD populations, genetic syndromes associated with IDDs (“syndromic IDDs”) represent a
particularly vulnerable subgroup. Many patients with syndromic IDDs present with medically complex
phenotypes and remain minimally verbal even into adulthood, creating challenges accessing and benefiting
from community-based interventions. Several reports point to extreme behavior and communication challenges
as the most pressing behavioral health concerns for this population. Our near-term goals seek to identify
effective approaches to target the more severe cognitive and behavioral phenotypes found in syndromic IDDs.
Here, we propose adaptations to function-based treatment (FBT) – an already well-established, person-
centered applied behavior analysis (ABA) model focused on replacing challenging behaviors with prosocial
communication and behavior responses. Using the Planned Adaptation approach, proactive adaptations to
improve the fit of FBT with the syndromic IDD population include syndrome-specific characterizations to inform
phenotype-environment interactions, systematic screening for automatically reinforced behaviors which are
often excluded from published FBT approaches, and adjustments to support minimally verbal individuals. This
proposal draws upon the expertise of the investigative group in syndromic IDDs, conventional and telehealth
behavioral interventions, and implementation sciences to evaluate the adapted, parent-implemented, telehealth
FBT model for syndromic IDDs (FBTsIDD). The goal of this fully remote hybrid type 1 effectiveness-
implementation study is to test FBTsIDD as delivered by non-specialist providers housed in medical hubs serving
individuals with syndromic IDDs. Aim 1 involves a 24-week randomized control trial (RCT) that will randomize
80 children (ages 2 to 12 years) with syndromic IDDs and moderate to severe intellectual disability (ID) and
their caregivers and randomize them into FBTsIDD or positive-parenting treatment (Treatment as Usual, TAU).
Our overarching hypothesis is that FBTsIDD will be associated with significant reductions in challenging
behaviors compared to TAU on independent evaluator ratings using a consumer-driven, Parent Target
Problems (PTP) inventory and standardized measures of behavior and functional communication. Aim 2 seeks
to systematically measure and understand both planned and unplanned adaptions to FBTsIDD using the
Framework for Reporting Adaptations and Modifications-Expanded (FRAME). Together, these aims provide an
innovative model to develop effective, acceptable, and scalable interventions for behavioral and
communication challenges across the diverse, vulnerable population of individuals with syndromic IDDs.
Terms: <0-11 years old><21+ years old><22q13 deletion syndrome><Access to Care><Address><Adult><Adult Human><Affect><Age><Angelman Syndrome><Behavior><Behavior Conditioning Therapy><Behavior Modification><Behavior Therapy><Behavior Treatment><Behavioral><Behavioral Conditioning Therapy><Behavioral Modification><Behavioral Therapy><Behavioral Treatment><Blinded><Bourneville Disease><Bourneville Phakomatosis><Bourneville syndrome><Bourneville-Brissaud disease><Bourneville-Pringle syndrome><COVID crisis><COVID epidemic><COVID pandemic><COVID-19 affected><COVID-19 consequence><COVID-19 crisis><COVID-19 effect><COVID-19 epidemic><COVID-19 era><COVID-19 global health crisis><COVID-19 global pandemic><COVID-19 health crisis><COVID-19 impact><COVID-19 impacted><COVID-19 pandemic><COVID-19 period><COVID-19 public health crisis><COVID-19 years><Care Givers><Care giver support><Caregiver support><Caregivers><Caring><Child><Child Behavior><Child Rearing><Child Youth><Children (0-21)><Clinical><Clinical effectiveness><Cognitive><Communication><Communication challenge><Communication difficulty><Communities><Complex><Conditioning Therapy><Data><Disparities><Disparity><Environment><Epiloia><Equipment and supply inventories><Exclusion><Family><Functional impairment><Genetic><Goals><Happy Puppet Syndrome><Health Inequity><Health Services><Health Services Accessibility><Healthcare><Home><Hybrids><Impairment><Individual><Inequalities in Health><Inequities in Health><Intellectual disability><Intellectual functioning disability><Intellectual limitation><Intervention><Intervention Strategies><Interview><Inventory><Life><Measures><Medical><Mental Health><Mental Hygiene><Methods><Modeling><Modification><Natural History><Neurobiology><Outcome><Parenting><Parenting behavior><Parents><Participant><Patients><Phelan-McDermid syndrome><Phenotype><Population><Pringle disease><Provider><Psychological Health><Publishing><Puppet Children><Questionnaires><Randomized><Randomized, Controlled Trials><Reporting><Research><Research Resources><Resources><SARS-CoV-2 epidemic><SARS-CoV-2 global health crisis><SARS-CoV-2 global pandemic><SARS-CoV-2 pandemic><SARS-coronavirus-2 epidemic><SARS-coronavirus-2 pandemic><Services><Severe Acute Respiratory Syndrome CoV 2 epidemic><Severe Acute Respiratory Syndrome CoV 2 pandemic><Severe acute respiratory syndrome coronavirus 2 epidemic><Severe acute respiratory syndrome coronavirus 2 pandemic><Standardization><Subgroup><Survey Instrument><Surveys><Syndrome><Testing><Tuberous Sclerosis><Vulnerable Populations><Work><access to health services><access to services><access to treatment><accessibility to health services><adenoma sebaceum><adulthood><ages><applied behavior analysis><applied behavioral analysis><assess effectiveness><autistic individuals><autistic people><availability of services><behavior intervention><behavior phenotype><behavior response><behavioral health><behavioral health intervention><behavioral intervention><behavioral phenotyping><behavioral response><care access><care as usual><care services><care systems><cerebral sclerosis><childrearing><community intervention><community level intervention><community-based intervention><coronavirus disease 2019 consequence><coronavirus disease 2019 crisis><coronavirus disease 2019 effect><coronavirus disease 2019 epidemic><coronavirus disease 2019 global health crisis><coronavirus disease 2019 global pandemic><coronavirus disease 2019 health crisis><coronavirus disease 2019 impact><coronavirus disease 2019 pandemic><coronavirus disease 2019 public health crisis><coronavirus disease crisis><coronavirus disease epidemic><coronavirus disease pandemic><coronavirus disease-19 global pandemic><coronavirus disease-19 impact><coronavirus disease-19 pandemic><design><designing><determine effectiveness><effective intervention><effectiveness assessment><effectiveness evaluation><effectiveness-implementation RCT><effectiveness-implementation 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