Skip to content Skip to navigation
University of Warwick
  • Study
  • |
  • Research
  • |
  • Business
  • |
  • Alumni
  • |
  • News
  • |
  • About

University of Warwick
Publications service & WRAP

Highlight your research

  • WRAP
    • Home
    • Search WRAP
    • Browse by Warwick Author
    • Browse WRAP by Year
    • Browse WRAP by Subject
    • Browse WRAP by Department
    • Browse WRAP by Funder
    • Browse Theses by Department
  • Publications Service
    • Home
    • Search Publications Service
    • Browse by Warwick Author
    • Browse Publications service by Year
    • Browse Publications service by Subject
    • Browse Publications service by Department
    • Browse Publications service by Funder
  • Help & Advice
University of Warwick

The Library

  • Login
  • Admin

Lifespan trajectory of affect in Cornelia de Lange syndrome : towards a neurobiological hypothesis

Tools
- Tools
+ Tools

Groves, Laura, Moss, Joanna, Crawford, Hayley, Nelson, Lisa, Stinton, Chris, Singla, Gursharan and Oliver, Chris (2019) Lifespan trajectory of affect in Cornelia de Lange syndrome : towards a neurobiological hypothesis. Journal of Neurodevelopmental Disorders, 11 . 6. doi:10.1186/s11689-019-9269-x ISSN 1866-1947.

[img]
Preview
PDF
WRAP-lifespan-trajectory-affect-cornelia-de-lange-syndrome-Crawford-2019.pdf - Published Version - Requires a PDF viewer.
Available under License Creative Commons Attribution 4.0.

Download (1080Kb) | Preview
Official URL: http://dx.doi.org/10.1186/s11689-019-9269-x

Request Changes to record.

Abstract

Background
Depressive symptomology and low affect are comparatively common in individuals with genetic disorders such as Cornelia de Lange syndrome. However, lifespan trajectories and associated person characteristics have not been examined. In this study, the trajectories for affect and associated behavioural characteristics were investigated in individuals with Cornelia de Lange syndrome with individuals with fragile X syndrome (FXS) comparable for chronological age and total number of behavioural indicators of ASD included for the purpose of contrast.
Methods
A 7-year longitudinal study of affect (mood, interest and pleasure) was conducted in individuals with CdLS (n = 44) and FXS (n = 95). The trajectories of low affect were explored, as well as associations between Time 1 behavioural characteristics and affect at Time 1 and Time 3 (7 years later).
Results
The CdLS group were lower in mood than the FXS group overall (p < .001). Interest and pleasure scores showed a significant decline over the lifespan for individuals with CdLS (p < .001) but not the FXS group. Lower level of ability at Time 1 was associated with lower mood at Time 1 and Time 3 in the FXS group only. Higher levels of ASD symptomology at Time 1 were associated with low mood and interest and pleasure in both syndrome groups at Time 1 and Time 3. Greater insistence on sameness at Time 1 was associated with lower mood at Time 1 in the FXS group and lower interest and pleasure at Time 1 and Time 3 in the CdLS group.
Conclusions
Low affect in specific genetic syndromes may be associated with differing lifespan trajectories and behavioural profiles. Specifically, individuals with CdLS appear at risk for experiencing declines in levels of interest and pleasure whereas individuals with FXS show no significant change in the level of affect with age.

Item Type: Journal Article
Divisions: Faculty of Science, Engineering and Medicine > Medicine > Warwick Medical School > Health Sciences
Faculty of Science, Engineering and Medicine > Medicine > Warwick Medical School > Health Sciences > Mental Health and Wellbeing
Faculty of Science, Engineering and Medicine > Medicine > Warwick Medical School
Journal or Publication Title: Journal of Neurodevelopmental Disorders
Publisher: BioMed Central Ltd.
ISSN: 1866-1947
Official Date: 7 June 2019
Dates:
DateEvent
7 June 2019Published
23 May 2019Accepted
Volume: 11
Article Number: 6
DOI: 10.1186/s11689-019-9269-x
Status: Peer Reviewed
Publication Status: Published
Access rights to Published version: Open Access (Creative Commons)
Date of first compliant deposit: 23 January 2020
Date of first compliant Open Access: 23 January 2020
Related URLs:
  • Other Repository

Request changes or add full text files to a record

Repository staff actions (login required)

View Item View Item

Downloads

Downloads per month over past year

View more statistics

twitter

Email us: wrap@warwick.ac.uk
Contact Details
About Us