TY - JOUR
T1 - Disentangling autism spectrum and attention-deficit/hyperactivity symptoms over development in fragile X syndrome
AU - Doherty, Brianna Ruth
AU - Longhi, Elena
AU - Cole, Victoria
AU - Karmiloff-Smith, Annette
AU - Cornish, Kim
AU - Scerif, Gaia
N1 - Funding Information:
This research was supported by a Rhodes Scholarship and Clarendon Scholarship to BR Doherty, an Understanding Human Cognition James S. McDonnell Foundation Scholar Award to G Scerif, and a project grant from The Wellcome Trust, UK to G Scerif, K Cornish and A Karmiloff-Smith ( WT079326AIA ). We express our deepest thanks to all families who participated in the research and to the Fragile X Society for their continued support. We also acknowledge Justin Cowan’s contributions to questionnaire selection and the initial stages of data collection. *Sadly Prof Annette Karmiloff-Smith died before this manuscript could be completed. However, her intellectual contribution to the project was central to what we report here.
Funding Information:
This research was supported by a Rhodes Scholarship and Clarendon Scholarship to BR Doherty, an Understanding Human Cognition James S. McDonnell Foundation Scholar Award to G Scerif, and a project grant from The Wellcome Trust, UK to G Scerif, K Cornish and A Karmiloff-Smith (WT079326AIA). We express our deepest thanks to all families who participated in the research and to the Fragile X Society for their continued support. We also acknowledge Justin Cowan's contributions to questionnaire selection and the initial stages of data collection. *Sadly Prof Annette Karmiloff-Smith died before this manuscript could be completed. However, her intellectual contribution to the project was central to what we report here.
Publisher Copyright:
© 2020
PY - 2020/9
Y1 - 2020/9
N2 - Even genetic disorders associated with monogenic aetiologies are characterized by complex and variable risk for poor outcomes, highlighting the need to follow trajectories longitudinally. Here, we investigated the longitudinal relationships between attention deficit hyperactivity disorder (ADHD) and autism spectrum disorder (ASD) symptoms in a population at high risk for both: boys with fragile X syndrome. 59 boys with fragile X syndrome aged 3–10 years old at entry participated in this study, and were followed up one and two years after their first visit. As expected, we found strong relationships over three timepoints for ADHD symptoms (as measured by the parent-rated Conners scale) and ASD symptoms (as measured by the Social Communication Questionnaire [SCQ]). In addition, using structural equation modeling (SEM) we found that ADHD symptoms at time 2 predicted ASD symptoms at time 3, suggestive of a causal relationship. Importantly, these relationships hold when including chronological age at entry to the study, as well as when including severity of impairment as measured by IQ, and their effects on both ASD and ADHD symptoms do not reach significance. This result highlights the need to study outcomes longitudinally and it informs the comorbidity of the two symptom domains in FXS as well as their potential directionality, both of which have been little researched. In addition, our findings may suggest a future need to study how ADHD symptoms and their treatment impact individuals with ASD.
AB - Even genetic disorders associated with monogenic aetiologies are characterized by complex and variable risk for poor outcomes, highlighting the need to follow trajectories longitudinally. Here, we investigated the longitudinal relationships between attention deficit hyperactivity disorder (ADHD) and autism spectrum disorder (ASD) symptoms in a population at high risk for both: boys with fragile X syndrome. 59 boys with fragile X syndrome aged 3–10 years old at entry participated in this study, and were followed up one and two years after their first visit. As expected, we found strong relationships over three timepoints for ADHD symptoms (as measured by the parent-rated Conners scale) and ASD symptoms (as measured by the Social Communication Questionnaire [SCQ]). In addition, using structural equation modeling (SEM) we found that ADHD symptoms at time 2 predicted ASD symptoms at time 3, suggestive of a causal relationship. Importantly, these relationships hold when including chronological age at entry to the study, as well as when including severity of impairment as measured by IQ, and their effects on both ASD and ADHD symptoms do not reach significance. This result highlights the need to study outcomes longitudinally and it informs the comorbidity of the two symptom domains in FXS as well as their potential directionality, both of which have been little researched. In addition, our findings may suggest a future need to study how ADHD symptoms and their treatment impact individuals with ASD.
KW - ADHD symptoms
KW - ASD symptoms
KW - Co-development
KW - Fragile X syndrome
KW - Longitudinal changes
UR - https://www.scopus.com/pages/publications/85085658282
U2 - 10.1016/j.ridd.2020.103692
DO - 10.1016/j.ridd.2020.103692
M3 - Article
C2 - 32505083
AN - SCOPUS:85085658282
SN - 0891-4222
VL - 104
JO - Research in Developmental Disabilities
JF - Research in Developmental Disabilities
M1 - 103692
ER -