29711
Toddlers with Tuberous Sclerosis Complex with Comorbid Autism Spectrum Disorder at High Risk for Adaptive Behavior Delay By 36 Months.
Infants born with TSC have a 40-50% risk for developing Autism Spectrum Disorder (ASD), a condition strongly associated with deficits in adaptive behavior development. Previous investigations have noted that children with TSC are at high risk for intellectual and adaptive deficits (e.g., Kopp et al., 2008), and that significant declines in adaptive function occur over time in individuals with TSC (van Eeghen et al, 2011). Although concerns with adaptive behavior dysfunction has been noted in TSC, little is known about the relationship between adaptive function and ASD in very young children (e.g., toddlers) with TSC
Objectives:
In this study, we compare developmental trajectories in adaptive behavior between age 18 and 36 months in toddlers with TSC, to determine if a diagnosis of ASD is associated with a higher risk over time for adaptive deficits.
Methods:
Sample was composed of toddlers who had completed their 36-month TACERN project evaluation. The ASD group consisted of 19 toddlers (15 boys, mean age= 36.5 mo., mean MSEL=53.3). The non-ASD group consisted of 60 toddlers (30 boys, mean age= 36.5 mo., mean MSEL= 85.3). Adaptive function was assessed using the Vineland Adaptive Behavior Scales, 2nd Ed., with parents serving as informants. Time-series analysis techniques (in which each child’s development is tracked over time) were used to compare the developmental trajectories in VABS-II domains standard scores between 18 and 36 months in the two groups.
Results:
At 18 months, the VABS-II scores were lower in TSC+ASD compared to non-ASD (p<0.001). From 18 months to 24 months, the trajectories of adaptive development remained steady in both groups (p>0.05). However, between 24 and 36 months, significant group differences emerged in developmental trajectories (p<0.01). Toddlers with TSC+ASD demonstrated significant standard score decreases in Communication (-8.5), Daily Living Skills (-15.9), Socialization (-13.5), and the Adaptive Behavior Composite (-13.2) (p<0.05 for all). In contrast, for non-ASD group, standard scores decreased by 2.1, 4.4, 2.7 and 7.9--none of which was significant. Examination of subdomain raw scores suggested that while both groups gained new skills between 24 and 36 months, the rate of acquisition of new adaptive skills was much slower in the TSC+ASD group.
Conclusions:
Given that adaptive deficits are associated with suboptimal developmental outcomes, the declining trajectories of adaptive skill development in TSC+ASD suggests that by 36 months, toddlers with TSC+ASD are at significantly higher risk for suboptimal outcomes—similar to ASD-related adaptive concerns seen in the general pediatric population.