Temperament in Young Children with Fragile X Syndrome

Christina Maile Low

Fragile X Syndrome (FXS) is an x-linked, genetic disorder that represents the most common hereditary cause of mental retardation. While the literature suggests that there are very specific behavioral features associated with FXS in older children and adults, the literature regarding temperament in young children with FXS has been limited and yielded mixed findings. The present study sought to examine early temperament differences between children with FXS (N=28) and normal controls (N=28), matched on age, gender, and race. Child temperament characteristics were assessed via the Child Behavior Questionnaire (CBQ; Putnam & Rothbart, 2006) and behavior problems were evaluated using the age appropriate version of the Child Behavior Checklist (CBCL; Achenbach & Rescorla, 2000, 2001). Independent samples t-tests were performed to detect group differences on the sixteen fine-grained dimensions and three broadband factors of the CBQ. Children with FXS were rated by their parents as displaying significantly greater activity and shyness and significantly less attentional focusing, inhibitory control, soothability, and high intensity pleasure. Children with FXS were also reported as exhibiting less surgency/extraversion and effortful control. Behaviorally, children with FXS were rated by their parents as having significantly more total problems compared to controls. Correlations between temperament and behavior problem scores revealed that broadband temperament factors were not significantly related to the externalizing, internalizing, or total problems scales of the CBCL for children with FXS. Two-way ANOVAs were computed for the three broadband temperament factors to determine if temperament changes over time differed for children with FXS and controls (Tables 7,8,9). A significant interaction between age and fragile x diagnosis (FXS or control) was detected for the temperament factor Negative Affectivity. Our analyses suggest that attentional difficulties commonly found in teens and adults with FXS are also characteristic of young children with this condition. Youngsters with FXS were described as exhibiting lower levels of positive affectivity and greater total problems compared to matched controls. Positive emotionality findings may be of particular significance because this domain of temperament has not been widely examined.

Temperament in Young Children with Fragile X Syndrome

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