Abstracts

Rationale: To characterize the role of consanguinity in the lifetime rates of education services (EDS), attention deficit disorder (ADHD); and parent reported school competence, behavioral problems and regulation, and planning and organization ability in children with recent onset epilepsy, their unaffected siblings, and first-degree cousin controls. We hypothesized that unaffected siblings of children with epilepsy would have higher rates of ADHD and EDS, and would score lower on parent reported measures of behavior compared to first-degree cousin controls.Methods: 179 children with recent onset idiopathic epilepsies (CWE), 66 full biological siblings, and 98 first-degree cousin controls were compared on parent reported measures of CBCL, BRIEF, rates of ADHD, and EDS. Lifetime history of EDS was determined via structured interview with parents. ADHD rates were assessed via the Kiddie Schedule for Affective Disorders and Schizophrenia (K-SADS). Children were 8-18 years old and attended regular school at the time of the assessment. Data analyses for EDS and ADHD were conducted by Pearson’s Chi-Square; CBCL and BRIEF measures were evaluated using Multivariate Analyses of Variance.Results: CWE had the highest rate of EDS (51.4%) compared to siblings (31.8%) and cousin controls (18.4%), p<0.001. Rates of ADHD were also highest for CWE (29.6%) compared to siblings (10.9%) and control cousins (8.2%), p<0.001. MANOVA for CBCL Total Competence and Total Problems by group yielded a significant overall main effect, F(2,340)=19.76, p<0.001. Univariate analyses indicated significant group differences for both Total Competence and Total Problems, p<0.001. Pairwise comparisons revealed that Total Competence was significantly higher for cousin controls compared to both CWE and siblings, p<0.001, with no significant difference between CWE and siblings. Total Problems were significantly higher in CWE compared to cousin controls and siblings, p<0.001, with no significant difference between siblings and cousin controls. MANOVA for BRIEF Meta Cognition Index (MIT) and Behavior Regulation Index (BRIT) yielded a significant overall main effect for group, F(2,340) = 11.41, p<0.001. Univariate analyses indicated significant group differences for MIT and BRIT, p<0.001. Pairwise comparisons revealed that CWE had significantly higher scores than siblings and cousin controls on MIT and BRIT, p<0.001, no significant differences were found between siblings and cousin controls.Conclusions: CWE had significantly higher rates of academic, competence and behavioral problems than unaffected siblings and cousin controls. Siblings of CWE had higher rates of school related problems, ADHD, lower Total Competence and higher Total Problems than their cousin controls. No significant differences were found between siblings of CWE and cousin controls on BRIEF measures. The results suggest that unaffected siblings of CWE have an added risk for common neurobehavioral comorbidities as a result of shared genetics/environment and that this risk is greater for siblings of CWE than for more distant relatives such as cousins.