Assessment & Research

Behavioral Inflexibility Across Two Neurogenetic Conditions: Down Syndrome and Fragile X Syndrome.

Harrop et al. (2021) · American journal on intellectual and developmental disabilities 2021
★ The Verdict

Kids with fragile X plus autism show the greatest caregiver-reported rigidity, so target flexibility early and watch anxiety and social communication alongside.

✓ Read this if BCBAs working with children with fragile X or Down syndrome in clinic or school settings.
✗ Skip if Practitioners who only serve typically developing clients or adult populations.

01Research in Context

01

What this study did

Harrop et al. (2021) asked caregivers to rate how rigid their kids act each day. They compared three groups: children with Down syndrome, fragile X only, and fragile X plus autism.

The survey was done online. Parents answered questions about trouble with change, repeating routines, and sticking to one topic.

02

What they found

Kids with fragile X plus autism scored highest on inflexibility. Kids with fragile X only landed in the middle. Kids with Down syndrome scored lowest.

Across all groups, higher rigidity went hand in hand with more anxiety and weaker social-communication skills.

03

How this fits with other research

Dixon et al. (2008) also used caregiver surveys and found Down syndrome children were more flexible than kids with non-specific ID or ASD. Harrop et al. (2021) adds fragile X groups and shows the same ladder: DS easiest, FXS middle, FXS+ASD hardest.

Lacroix et al. (2022) tested autistic adults with a computer task. They saw flexibility problems only when rule shifts were hidden and emotional. That lab result lines up with the parent reports here: hidden changes cause the most trouble.

Cregenzán-Royo et al. (2018) found that when moms show high expressed emotion, behavior problems rise in both FXS and DS. Clare’s team now links those problems to rigidity, giving you a second lever to watch.

04

Why it matters

If you serve kids with fragile X, screen for autism traits early. When both labels are present, build extra practice with schedule changes, choice boards, and priming. Track anxiety and social goals at the same time; gains in flexibility often pull those skills up with it.

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→ Action — try this Monday

Add a daily ‘change card’ to the visual schedule for FXS+ASD clients and reward calm transitions.

02At a glance

Intervention
not applicable
Design
survey
Sample size
202
Population
down syndrome, autism spectrum disorder, mixed clinical
Finding
not reported

03Original abstract

Behavioral inflexibility (BI) has been highlighted to occur across genetic and neurodevelopmental disorders. This study characterized BI in two common neurogenetic conditions: Fragile X syndrome (FXS) and Down syndrome (DS). Caregivers of children with FXS (N = 56; with ASD = 28; FXS only = 28) and DS (N = 146) completed the Behavioral Inflexibility Scale (BIS) via an online survey. Total BIS scores were higher in FXS+ASD than both FXS only and DS (p <.001). Most endorsed items were similar across the three groups, but scores were higher in the FXS+ASD group. In all groups, BI associated with other clinical variables (receptive behaviors, anxiety, social communication). The current data suggest that BI is variable across neurogenetic conditions and higher in individuals with comorbid ASD.

American journal on intellectual and developmental disabilities, 2021 · doi:10.1352/1944-7558-126.5.409