Developmental trajectories of attentional control in preschool males with fragile X syndrome

Abstract

Attention problems are among the most impairing features associated with fragile X syndrome (FXS). However, few studies have examined behavioral development of inhibitory control in very young children with FXS. We examined attentional control in 3-6 year boys with FXS using both an experimental inhibitory control paradigm and parent-report of attention problems. Study 1 examined attentional control in FXS compared to comparison groups matched on chronological and mental age. To determine the stability of impairments over time in FXS, Study 2 examined patterns of developmental change in an expanded longitudinal sample. Across studies, males with FXS demonstrated persistent impairments in inhibitory control and parent-reported attention problems. Inhibitory control was related to, but not solely driven by, lower mental age. Although parent-rated attention problems remained stable across ages, inhibitory control improved with time. Children with more severe attention problems often displayed initially poorer inhibitory control. However, these trajectories also improved more rapidly with age. Our findings indicate that despite persistent deficits in attentional control in young children with FXS, multi-method assessment can be used to capture developmental growth that should be further supported through early, targeted intervention.

Brief Report: Autism Symptoms in Infants with Fragile X Syndrome

“…Tonnsen et al 2015). Given the lower developmental ages in FXS (Wilcoxon Z = 4.52, one-tailed p < .001) and ASIB (Z = 2.28, p = .01), and evidence that elevated AOSI scores may be associated with developmental level in ASIBs in some studies (Georgiades et al 2013), Spearman partial correlations were conducted to determine whether mental age should be covaried.…”

“…Specifically, developmental skills are significantly delayed across all major domains in infants with FXS as early as 6 months of age compared to ASIBs, with the deficit between these groups widening through the second year of life. In research focused on behavioral and heart-defined phases of attention in male infants with FXS contrasted to typical controls and ASIBs 6–12 months of age, we identified distinct behavioral and heart-defined attention in ASIBs versus FXS, with both high-risk groups exhibiting abnormalities compared to typical controls (Tonnsen et al 2015). These findings provide novel evidence of distinct cross-group differences, as well as within-group predictors of clinical ASD risk, across ASIB and FXS groups.…”

Noncomprehension Signaling in Males and Females With Fragile X Syndrome

“…For example, social anxiety and/or social avoidance are observed in nearly all individuals with FXS (e.g., Budimirovic et al, 2006;Hall, DeBernardis, & Reiss, 2006;Hessl, Glaser, Dyer-Friedman, & Reiss, 2006;Roberts, Weisenfeld, Hatton, Heath, & Kaufmann, 2007). In addition, symptoms such as gaze avoidance, inattention, hyperactivity, and hyperarousal are commonly associated with FXS (e.g., Chromik et al, 2015;Cornish, Sudhalter, & Turk, 2004;Tonnsen, Grefer, Hatton, & Roberts, 2015;Wisbeck et al, 2000). Collectively, the aforementioned symptoms are often significant enough to draw parallels between the FXS and ASD phenotypes.…”

Behavioral Markers of Emergent Stranger Anxiety in Infants and Toddlers with Fragile X Syndrome

“…displays avoidant eye contact, does not get involved with others or answer when spoken to), constructed based on factor analyses. A number of studies have used the CBCL to examine anxiety, withdrawal and other child problem behaviors in same-aged children with FXS (Hatton et al, 2002; Tonnsen, Grefer, Hatton, & Roberts, 2014; Tonnsen, Malone, Hatton, & Roberts, 2013). Although parents completed the CBCL at multiple time points, participants’ final CBCL prior to 60 months was selected to (1) maintain continuity across participants and (2) capture maximal stability of anxiety symptoms, as anxiety is known to emerge and increase in stability across childhood.…”