Trajectories and Predictors of the Development of Very Young Boys with Fragile X Syndrome

Roberts, Jane; Mankowski, Jean Boswell; Sideris, John; Goldman, Barbara Davis; Hatton, Deborah D.; Mirrett, Penny; Baranek, Grace T.; Reznick, J. Steven; Long, Anna C. J.; Bailey, Donald B.

doi:10.1093/jpepsy/jsn129

Cited by 62 publications

(83 citation statements)

References 28 publications

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“…Most children with FXS experience delays in expressive and receptive language Mirrett, Bailey, Roberts, & Hatton, 2004;Roberts et al, 2009), but there have been a few studies examining the roots of these language deficits. These studies on early communication development suggest that evidence for delays in language can be traced to deficits in early social communication behaviors.…”

Section: Early Language Development In Fxsmentioning

confidence: 99%

Joint Engagement and Early Language in Young Children With Fragile X Syndrome

Hahn

Brady

Fleming

et al. 2016

J Speech Lang Hear Res

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Purpose: In this study, we examine joint engagement (JE) in young children with fragile X syndrome (FXS) and its relationship to language abilities and autism spectrum disorder symptomatology at 24 to 36 months (toddler period) and 59 to 68 months (child period). Method: Participants were 28 children with FXS (24 boys, four girls) and their mothers. Videotaped home observations were conducted during the toddler period and coded for JE. Language abilities were measured at both ages from a developmental assessment, a functional measure, and from a language sample. The Childhood Autism Rating Scale (Schopler, Reichler, & Renner, 1988) was completed at both ages.Results: Children with FXS spent more time in supported JE than in coordinated JE. Using a weighted JE variable, we found that children with FXS who had higher weighted JE scores also had more advanced expressive language skills at both the toddler and child periods. Weighted JE was negatively related to autism symptomatology in the toddler period. Conclusion: This study provides evidence that children with FXS who use more JE also have more advanced expressive language skills in early development. Therefore, existing early interventions that target JE behaviors may be effective for promoting language, social communication, and social interaction in this population.

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Section: Early Language Development In Fxsmentioning

confidence: 99%

Joint Engagement and Early Language in Young Children With Fragile X Syndrome

Hahn

Brady

Fleming

et al. 2016

J Speech Lang Hear Res

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“…Similar patterns have been observed through examiner ratings of naturalistic interactions at the beginning and end of research assessments. Compared with TD controls, individuals with FXS have shown atypical overall social approach behaviors, as measured by facial expression, eye contact, and physical movement (Roberts, Mankowski, et al, 2009; FXS mean age = 3.99 ± 2.25 yeqrs). Consistent with findings in older children (Hall et al, 2009), young children with FXS have shown improvements in approach behaviors over the course of assessments, suggesting a “warm up” effect after initial periods of social anxiety.…”

Section: Anxiety In Fxsmentioning

confidence: 99%

“…Several studies have begun laying the foundation for this work by characterizing early features of the FXS phenotype relevant to anxiety, such as atypical social approach (e.g., Hall, Lightbody, Huffman, Lazzeroni, & Reiss, 2009; Hessl, Glasser, Dyer-Friedman, & Reiss, 2006; Roberts, Mankowski, et al, 2009; Roberts, Weisenfeld, Hatton, Heath, & Kaufmann, 2007) and physiological arousal (e.g., Hall et al, 2009; Roberts, Boccia, Bailey, Hatton, & Skinner, 2001, Roberts, Tonnsen, Robinson, & Shinkareva, 2012). These studies suggest the expression of anxiety and other challenging behaviors in FXS may be partially attributed to atypical arousal modulation (Cornish et al, 2004; Hessl, Rivera, & Reiss, 2004).…”

mentioning

confidence: 99%

Biobehavioral Indicators of Social Fear in Young Children With Fragile X Syndrome

Tonnsen¹,

Shinkareva²,

Deal³

et al. 2013

American Journal on Intellectual and Developmental Disabilities

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Anxiety is among the most impairing conditions associated with Fragile X syndrome (FXS) and is putatively linked to atypical physiological arousal. However, few studies have examined this association in young children with FXS. The authors examined whether patterns of arousal and behavior during an experimental stranger approach paradigm differ between a cross-sectional sample of 21 young children with FXS and 19 controls (12–58 months old). Groups did not differ in mean levels of behavioral fear. Unlike the control group, however, the FXS group demonstrated increased facial fear at older ages, as well as age-dependent changes in associations between heart activity and distress vocalizations. These findings may inform theoretical models of anxiety etiology in FXS and early detection efforts.

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“…Measures of anxiety, attention, and hyperactivity are highly associated with other behavior problems (29). There is evidence that autism scores decreased with time, particularly in communication and social aspects of adaptive behavior (57). However, emotional symptoms, behavioral difficulties, problems with peers and social behaviors may remain relatively stable over time (58).…”

Section: Behavioral Phenotypementioning

confidence: 99%