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

Journal of Neurodevelopmental Disorders

Doron Gothelf⁶  Abraham Weizman³  Amos Frisch³  Gil Zalsman¹  Gal Shoval¹  Hadas Zilkha⁵  Ariel Knafo⁴  Maayan Davidov²  Yael Schonherz⁵ 

[1] Child and Adolescent Psychiatry Division, Geha Mental Health Center, Petah Tikva 49202, Israel;School of Social Work and Social Welfare, The Hebrew University of Jerusalem, Mount Scopus, Jerusalem 91905, Israel;Felsenstein Medical Research Center, Petah Tikva 49202, Israel;Department of Psychology, The Hebrew University of Jerusalem, Mount Scopus, Jerusalem 91905, Israel;The Behavioral Neurogenetics Center, The Edmond and Lily Safra Children’s Hospital, Sheba Medical Center, Tel Hashomer 52621, Israel;Sackler Faculty of Medicine, Tel Aviv University, Tel Aviv 69978, Israel

关键词: Williams syndrome;    Velocardiofacial syndrome;    Temperament;    Shyness prediction;    Psychosis;   

Others  :  804559
DOI  :  10.1186/1866-1955-6-3

 received in 2013-04-25, accepted in 2014-02-03,  发布年份 2014

【 摘 要 】

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.

【 授权许可】

   
2014 Schonherz et al.; licensee BioMed Central Ltd.

【 图 表 】

Figure 2.

Figure 2.

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