TY - JOUR
T1 - Cortical dysplasia and autistic trait severity in children with Tuberous Sclerosis Complex
T2 - a clinical epidemiological study
AU - Mous, Sabine E.
AU - Overwater, Iris E.
AU - Vidal Gato, Rita
AU - Duvekot, Jorieke
AU - Ten Hoopen, Leontine W.
AU - Lequin, Maarten H.
AU - de Wit, Marie Claire Y.
AU - Dieleman, Gwendolyn C.
N1 - Funding Information:
Acknowledgements This research was financially supported by the Sophia Children’s Hospital Fund (Rotterdam, the Netherlands) under grant number SSWO B14-02. Further financial support was provided by the Dutch Brain Foundation (Hersenstichting) and the Dutch Epilepsy Foundation (Epilepsiefonds). Funders were not involved in the design of the study, nor in data collection, analysis, interpretation or writing the manuscript. The authors thank Karen Bindels-de Heus for patient care and data collection, and Simone Eijk, Emma van der Ende and Kimberley Hanemaayer for their help in data collection and cleaning.
Publisher Copyright:
© 2017, The Author(s).
PY - 2018/6
Y1 - 2018/6
N2 - Tuberous Sclerosis Complex (TSC) is characterized by a high prevalence of autism spectrum disorders (ASD). Little is known about the relation between cortical dysplasia and ASD severity in TSC. We assessed ASD severity (using the Autism Diagnostic Observation Scale), tuber and radial migration line (RML) count and location, and cognitive functioning in 52 children with TSC and performed regression and mediation analyses. Tuber and RML count were strongly positively related to ASD severity. However, when correcting for cognitive functioning, the majority of associations became insignificant and only total tuber count remained associated to the severity of restricted/repetitive behaviors. Occipital RML count remained associated with overall ASD severity, and social communication/interaction deficit severity specifically. This study shows the important explanatory role of cognitive functioning in the association between cortical dysplasia and ASD severity, and the relevance of separately studying the two ASD subdomains.
AB - Tuberous Sclerosis Complex (TSC) is characterized by a high prevalence of autism spectrum disorders (ASD). Little is known about the relation between cortical dysplasia and ASD severity in TSC. We assessed ASD severity (using the Autism Diagnostic Observation Scale), tuber and radial migration line (RML) count and location, and cognitive functioning in 52 children with TSC and performed regression and mediation analyses. Tuber and RML count were strongly positively related to ASD severity. However, when correcting for cognitive functioning, the majority of associations became insignificant and only total tuber count remained associated to the severity of restricted/repetitive behaviors. Occipital RML count remained associated with overall ASD severity, and social communication/interaction deficit severity specifically. This study shows the important explanatory role of cognitive functioning in the association between cortical dysplasia and ASD severity, and the relevance of separately studying the two ASD subdomains.
KW - Autism
KW - Cognitive functioning
KW - Intelligence
KW - Quantitative autistic traits
KW - Radial migration lines
KW - Tubers
UR - http://www.scopus.com/inward/record.url?scp=85031892923&partnerID=8YFLogxK
U2 - 10.1007/s00787-017-1066-z
DO - 10.1007/s00787-017-1066-z
M3 - Article
AN - SCOPUS:85031892923
SN - 1018-8827
VL - 27
SP - 753
EP - 765
JO - European child & adolescent psychiatry
JF - European child & adolescent psychiatry
IS - 6
ER -