This RO1 application requests funds to develop and validate a novel outcome measure targeting the behavioral inflexibility in children with intellectual and developmental disabilities (IDD). This behavioral domain encompasses a host of closely related clinical sequelae that can be profoundly disabling and are common in IDD such as (a) an overall inflexible temperament or behavioral style that underlies discrete idiosyncratic forms of repetitive and rigid behavior manifestations (e.g., inability to tolerate changes in the environment, stereotyped uses of objects); (b) associated mood and behavior problems that arise when this inflexible temperament and its manifestations cannot be accommodated; and (c) interference with learning and adaptation that stems from rigidity / inflexibility as even treatmen itself can represent intrusion and change for an inflexible child (e.g., a new pill, a new therapy exercise). In recent years there have been large-scale drug trials and small behavioral trials targeting discrete behaviors that fall under the domain of repetitive inflexible behavior. However, the measures included in many of these clinical trials have little to no evidence of their ability o detect change over time. In fact, while several instruments exist that measure the discrete behavioral endpoints of this overall construct of rigidity / inflexibility, no instrument exists tht adequately extends much beyond a behavior checklist approach. The overall objective of this application is to develop and validate a new measure, the Repetitive and Inflexible Behavior Inventory (RIBI), with the primary goal of developing a measure that is sensitive to change over time. The RIBI is designed as a parent report measure for children (ages 3 -18) with IDD. The rationale is that a novel measure is needed to (a) capture the range of repetitive inflexible behaviors that are found in children with IDD across a variety of idiopathic and genetic disorders, (b) measure the functional impact of these behaviors on the family's daily life, and (c) detect change over time when these behaviors are the target of intervention. Thus, the specific aims of this project are to (1) examine the psychometric properties of the RIBI for children and adolescents with autism spectrum disorder (ASD), establishing reliability, factor structure, content and construct validity; (2) evaluate the RIBI's ability to measure change over time in children with ASD; and (3) examine the psychometric stability of the RIBI when applied to children and adolescents with Fragile-X and Prader-Willi syndromes. To accomplish these aims, a multisite study will be conducted using recommended instrument development methodologies, such as focus groups, expert panels, and online surveys (n=1500). This team is in an ideal position to develop the RIBI because of our prior experience with measure development, and the conduct of clinical trials targeting repetitive inflexible behavior. The expected deliverable i an outcome measure that is appropriate for IDD populations and minimally burdensome to complete in order to be useful for multisite clinical trials.

Public Health Relevance

There is a lack of appropriate and validated instruments in the field of Intellectual and Developmental Disabilities that can be used to measure intervention efficacy. This can lead to useful interventions being wrongly deemed ineffective. Outcome measures are particularly needed for the domain of repetitive and inflexible behaviors because of the impact these behaviors have on the person's, and their families', overall quality of life.

Agency
National Institute of Health (NIH)
Institute
Eunice Kennedy Shriver National Institute of Child Health & Human Development (NICHD)
Type
Research Project (R01)
Project #
5R01HD082127-06
Application #
9609526
Study Section
Special Emphasis Panel (ZRG1)
Program Officer
Kau, Alice S
Project Start
2014-12-10
Project End
2019-11-30
Budget Start
2018-12-01
Budget End
2019-11-30
Support Year
6
Fiscal Year
2019
Total Cost
Indirect Cost
Name
University of Kansas Lawrence
Department
Miscellaneous
Type
Organized Research Units
DUNS #
076248616
City
Lawrence
State
KS
Country
United States
Zip Code
66045
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Sethi, Chetna; Harrop, Clare; Zhang, Wanqing et al. (2018) Parent and professional perspectives on behavioral inflexibility in autism spectrum disorders: A qualitative study. Autism :1362361318810217
Shafer, Robin L; Newell, Karl M; Lewis, Mark H et al. (2017) A Cohesive Framework for Motor Stereotypy in Typical and Atypical Development: The Role of Sensorimotor Integration. Front Integr Neurosci 11:19