Actigraph-Measured Movement Correlates of Attention-Deficit/Hyperactivity Disorder (ADHD) Symptoms in Young People with Tuberous Sclerosis Complex (TSC) with and without Intellectual Disability and Autism Spectrum Disorder (ASD)

Abstract

Actigraphy, an objective measure of motor activity, reliably indexes increased movement levels in attention-deficit/hyperactivity disorder (ADHD) and may be useful for diagnosis and treatment-monitoring. However, actigraphy has not been examined in complex neurodevelopmental conditions. This study used actigraphy to objectively measure movement levels in individuals with a complex neurodevelopmental genetic disorder, tuberous sclerosis (TSC). Thirty participants with TSC (11–21 years, 20 females, IQ = 35–108) underwent brief (approximately 1 h) daytime actigraph assessment during two settings: movie viewing and cognitive testing. Multiple linear regressions were used to test associations between movement measurements and parent-rated ADHD symptoms. Correlations were used to examine associations between actigraph measures and parent-rated ADHD symptoms and other characteristics of TSC (symptoms of autism spectrum disorder (ASD), intellectual ability (IQ), epilepsy severity, cortical tuber count). Higher movement levels during movies were associated with higher parent-rated ADHD symptoms. Higher ADHD symptoms and actigraph-measured movement levels during movies were positively associated with ASD symptoms and negatively associated with IQ. Inter-individual variability of movement during movies was not associated with parent-rated hyperactivity or IQ but was negatively associated with ASD symptoms. There were no associations with tuber count or epilepsy. Our findings suggest that actigraph-measured movement provides a useful correlate of ADHD in TSC.

The autism spectrum disorder phenotype in children with tuberous sclerosis complex: A systematic review and meta‐analysis

“…Thirty‐four studies, published between 1987 and 2020, including 3160 children with TSC, 30% (914 out of 3060) with ASD, presented data about at least one aspect of the TSC‐ASD phenotype (Table 1). Two studies 28,29 did not report the number of children with TSC‐ASD separately and were excluded from the group ASD percentage calculations. Six of the studies presented an ASD‐no TSC comparator, including 338 children with idiopathic ASD and 49 with other forms of syndromic ASD 18,28,30–33 …”

“…Group 1 and the 2014 study from group 2 are included in the total number of children for this review. d Total TSC‐ASD % was calculated with the Eden et al 28 and Earnest et al 29 studies excluded because the ASD % data were not reported. Abbreviations: ADHD, attention‐deficit/hyperactivity disorder; ASD, autism spectrum disorder; C, community sample; NOS, Newcastle–Ottawa Scale; NR, not reported; T, tertiary sample; TSC, tuberous sclerosis complex; Y, yes.…”

“…Parent rated ADHD symptoms and actigraphy movement were associated with ASD symptoms, measured on the ADI-R. IQ was negatively associated with ADHD symptoms in the whole group (Spearman's correlation coefficient, ρ = −0.597, p = 0.002). 29…”

Epilepsy severity mediates association between mutation type and ADHD symptoms in tuberous sclerosis complex

“…Still it is unclear whether the symptoms measured represent ADHD in TSC, or difficulties associated with ID, and current antiepileptic medication and seizure severity may present as attentional difficulties. Direct comparison of ADHD in syndromic and non-syndromic individuals is required, alongside more objective measurement of activity 14 and neurocognitive impairment associated with ADHD, which may be independent of aetiological factors shared with IQ 15 . Because diagnostic overshadowing of ID may limit reports of additional behavioural challenges, it is also important to consider the role of other TAND manifestations, including autism (Supplementary Material Section 2), mood and anxiety disorders 4 .…”

Exploring an objective measure of overactivity in children with rare genetic syndromes

“…Although previous studies of rare genetic syndromes have measured children’s physical activity using actigraphy, none have rigorously examined the sensitivity of actigraphy to overactivity [ 54 – 58 ]. No published study has directly addressed the convergent validity of actigraphy-defined activity with established overactivity questionnaires used in rare syndrome research.…”