| Literature DB >> 32189229 |
Katherine Ellis1,2,3, Chris Oliver4, Chrysi Stefanidou1,5, Ian Apperly6, Jo Moss1,3.
Abstract
We directly assessed the broader aspects of sociability (social enjoyment, social motivation, social interaction skills and social discomfort) in individuals with Cornelia de Lange (CdLS), fragile X (FXS) and Rubinstein-Taybi syndromes (RTS), and their association with autism characteristics and chronological age in these groups. Individuals with FXS (p < 0.01) and RTS (p < 0.01) showed poorer quality of eye contact compared to individuals with CdLS. Individuals with FXS showed less person and more object attention than individuals with CdLS (p < 0.01). Associations between sociability and autism characteristics and chronological age differed between groups, which may indicate divergence in the development and aetiology of different components of sociability across these groups. Findings indicate that individuals with CdLS, FXS and RTS show unique profiles of sociability.Entities:
Keywords: Eye gaze; Fragile X syndrome; Genetics behavioural; Neurodevelopmental disorders; Social behavior
Mesh:
Year: 2020 PMID: 32189229 PMCID: PMC7560922 DOI: 10.1007/s10803-020-04440-4
Source DB: PubMed Journal: J Autism Dev Disord ISSN: 0162-3257
Participant characteristics for all participants with CdLS, FXS and RTS
| CdLS | FXS | RTS | Post-hoc tests ( | ||
|---|---|---|---|---|---|
| Mean chronological age in years (SD) | 12.42 (10.27) | 15.24 (12.59) | 15.22 (13.78) | 0.57 | |
| Gender % female | FXS < CdLS, RTS | ||||
| Non-verbal mental age in years (SD) | 3.82 (2.15)a | 3.45 (1.11)b | 3.35 (1.35)c | 0.79 | |
| Median Developmental Quotient (IQR) | 37.87 (29.95) | 37.96 (34.79) | 27.65 (26.55) | 0.44 | |
| ADOS-2 Social Affect CSS (SD) | CdLS, RTS < FXS |
Variables with significant differences between syndrome groups are highlighted in bold
aInformation not available for three participants due to non-completion of the relevant measure
bInformation not available for three participants due either to floor/ceiling performance (one participant)
cInformation not available for four participants due either to (1) floor/ceiling performance (one participant) or (2) non-completion of non-verbal scales of a cognitive assessment
Fig. 1Median item scores on each CSRS item per syndrome
Kendall Tau correlations for mean CSRS item scores, and ADOS-II Social Affect (SA) CSS and chronological age for each syndrome
| CdLS | FXS ( | RTS ( | ||||
|---|---|---|---|---|---|---|
| ADOS-II SA CSS ( | Chronological age ( | ADOS-II SA CSS ( | Chronological age ( | ADOS-II SA CSS ( | Chronological age ( | |
| Positive emotional affect | 0.24 (0.06) | − 0.08 (0.56) | − 0.05 (0.70) | − | − 0.34 (0.02) | |
| Social responsiveness | − 0.31 (0.02) | 0.28 (0.02) | 0.04 (0.76) | − 0.29 (0.06) | 0.05 (0.76) | |
| Spontaneous initiation of interaction | 0.30 (0.02) | 0.07 (0.55) | 0.04 (0.76) | 0.03 (0.84) | − 0.11 (0.48) | 0.04 (0.81) |
| Focus of attention | − 0.14 (0.26) | − 0.09 (0.50) | 0.04 (0.75) | − 0.35 (0.03) | 0.06 (0.69) | |
| Motivation for adult engagement | − | − 0.04 (0.77) | − 0.09 (0.48) | − 0.18 (0.15) | − 0.16 (0.31) | − 0.06 (0.70) |
| Eye contact | − 0.24 (0.09) | 0.30 (0.03) | − 0.21 (0.12) | − 00.26 (0.04) | − | − 0.21 (0.12) |
| Social communication skills | − 0.33 (0.02) | 0.16 (0.30) | − 0.22 (0.21) | 0.03 (0.87) | ||
| Social anxiety | 0.28 (0.04) | 0.21 (0.21) | 0.12 (0.49) | |||
| Avoidance of social interaction | 0.15 (0.28) | − | 0.01 (0.93) | 0.03 (0.81) | 0.17 (0.31) | 0.13 (0.40) |
Significant correlations are highlighted in bold