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Introduction: Agenesis of the corpus callosum (ACC) is a congenital brain malformation resulting in partial to complete absence of the brain’s major transverse commissure. ACC is identified via prenatal ultrasound, and offers a unique opportunity for prospective study during infancy and toddlerhood. Studying ACC tells us about the role of the corpus callosum, and brain plasticity more broadly. Additionally, there is an increased incidence of Autism Spectrum Disorders (ASD) in children with ACC (~10%, as compared to 2.3% in general population). While differences in adaptive functioning present early in development in children with ASD, at this time it is unclear whether children with ACC show a similar pattern. We used a prospective, longitudinal design to examine adaptive behavior from 6-24 months in children with ACC and compared trajectories to those at high and low likelihood for ASD.
Methods: Our sample included children with primary ACC (n= 27-47 depending on time point) whose caregivers completed the Vineland Adaptive Behavior Scales-Interview 3rd Edition, at 6, 12, 18 and 24 months. Comparison samples (using the Vineland-2) included children at high familial likelihood for ASD (HL-; n=192-280), and low likelihood (LL; no family history of autism and no developmental/behavioral diagnosis; n = 111-196). A subset of the HL children received an ASD diagnosis (HL+; n = 48-74).
Results: A series of linear mixed model analyses (using maximum likelihood) for repeated measures was used to compare groups on Vineland domains at 6, 12, 18 and 24 month timepoints). Fixed factors (diagnostic group, group X timepoint interaction) accounted for significant variance on all Vineland domains (p < .001). Age did not account for variance in Social or Communication sub/domains, and Sex did not account for significant variance in Motor sub/domains.
Post hoc comparisons with Bonferroni-correction examined ACC Vineland scores compared to the other diagnostic groups at each timepoint. By 12 months parents rated the ACC group as having weaker Communication, Daily Living and Motor skills than the HL- and LL groups. ACC also showed weaker expressive language skills than the HL+ group at 18 and 24 months. Within the Socialization domain, at 6 months the ACC group had stronger interpersonal skills, but weaker play skills than the HL+, HL - and LL groups. However, by 24 months the ACC group was indistinguishable from the HL- and LL groups, and all had stronger Socialization (play and interpersonal) skills than the HL+ group.
Conclusions: Compared to children with low likelihood of ASD, children with primary ACC reportedly have weaker Communication, Daily Living, and Motor skills from 12 to 24 months. In general, the ACC scores were consistent with the HL+ sample, except the ACC group had stronger Social skills at 18 and 24 months. Additionally, at 6 months, the ACC group showed stronger Interpersonal skills, but weaker Play skills on Vineland subdomains, potentially related to motor weaknesses. The results clearly inform the need for early intervention in the domains of motor and language skills. Additionally, their strength in early social skills offers a window of opportunity to capitalize on early strengths to promote long term social skills.
Lauren Haisley, University of Minnesota - Twin Cities
Presenting Author
Lana Hantzsch, University of Minnesota - Twin Cities
Jasmin Turner, California Institute of Technology
Kelly Botteron, Washington University in St. Louis
Stephen Dager, University Of Washington
Annette Mercer Estes, Uniiversity of Washington
Lisa Flake, Washington University School of Medicine
Heather Cody Hazlett, University of North Carolina at Chapel Hill
Robert Schultz, Children's Hospital of Philadelphia
Joseph Piven, University of North Carolina at Chapel Hill
Lynn Kerlin Paul
Jed T. Elison, University of Minnesota - Twin Cities