Shyness discriminates between children with 22q11.2 deletion syndrome and Williams syndrome and predicts emergence of psychosis in 22q11.2 deletion syndrome

Yael Schonherz, Maayan Davidov, Ariel Knafo, Hadas Zilkha, Gal Shoval, Gil Zalsman, Amos Frisch, Abraham Weizman, Doron Gothelf*

*Corresponding author for this work

Research output: Contribution to journalArticlepeer-review

Abstract

Background: 22q11.2 deletion syndrome (22q11.2DS) is a common neurogenetic syndrome associated with high rates of psychosis. The aims of the present study were to identify the unique temperament traits that characterize children with 22q11.2DS compared to children with Williams syndrome (WS) and typically developing (TD) controls, and to examine temperamental predictors of the emergence of psychosis in 22q11.2DS. Methods: The temperament of 55 children with 22q11.2DS, 36 with WS, and 280 TD children was assessed using the Emotionality, Activity, Sociability (EAS) Temperament Survey, Parental Ratings. The presence of a psychotic disorder was evaluated in 49 children and adolescents with 22q11.2DS at baseline and again 5.43 ± 2.23 years after baseline temperament assessment. Results: Children with 22q11.2DS scored higher on Shyness compared to WS and TD controls. Children with 22q11.2DS and WS scored higher on Emotionality and lower on Activity compared to TD controls. Shyness was more severe in older compared to younger children with 22q11.2DS. Baseline Shyness scores significantly predicted the later emergence of a psychotic disorder at follow-up, in children with 22q11.2DS. Conclusions: Our results suggest that shyness is an early marker associated with the later emergence of psychosis in 22q11.2DS.

Original languageEnglish
Article number3
JournalJournal of Neurodevelopmental Disorders
Volume6
Issue number1
DOIs
StatePublished - 11 Feb 2014

Keywords

  • Psychosis
  • Shyness prediction
  • Temperament
  • Velocardiofacial syndrome
  • Williams syndrome

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