Salivary testosterone in relation to social cognition and social anxiety in children and adolescents with 47,XXY (Klinefelter syndrome).

Sophie van Rijn,Andrew Wolfe

doi:10.1371/journal.pone.0200882

Abstract

BackgroundApproximately 1 in 650 boys are born with an extra X chromosome. Boys and men with 47,XXY (Klinefelter syndrome) are at risk for neurodevelopmental disorders and specific cognitive impairments. This study was focused on social anxiety and social cognition. The aim was to assess if these aspects of the phenotype are related to testosterone deficiency, which is typically seen in 47,XXY from puberty onwards.MethodsIn the study 20 boys with 47,XXY and 25 non-clinical controls between 8 and 19 years participated. None had ever used testosterone supplements. Cognitive tests measuring the labeling of facial expressions and perspective taking (Theory of Mind) were administered. Self-report questionnaires were used to assess social anxiety. Testosterone was measured in saliva.ResultsWithin the 47,XXY group lower levels of salivary testosterone were significantly associated with higher levels of social anxiety. The correlation was strong, andindependent of age and pubertal development. However, salivary levels of testosterone were uncorrelated to social cognitive skills.DiscussionThese findings point out that lower testosterone levels might contribute to high social anxiety in 47,XXY, suggesting that anxiety should be monitored in pubertal boys with XXY presenting with testosterone deficiency. This should be done in addition to exploring cognitive behavioral therapy or psychopharmacologic treatments targeting anxiety, which are more evidence based. In contrast, testosterone levels were not associated with social cognitive functioning, suggesting that other mechanisms are driving vulnerabilities in this domain.

Highlights

Within the 47,XXY group lower levels of salivary testosterone were significantly associated with higher levels of social anxiety
These findings point out that lower testosterone levels might contribute to high social anxiety in 47,XXY, suggesting that anxiety should be monitored in pubertal boys with XXY presenting with testosterone deficiency
There is increasing interest in the phenotype of individuals with sex chromosome aneuploidies, because this is crucial for optimizing clinical care, and because by we may learn about factors that drive risk for psychopathology

Summary

Introduction

There is increasing interest in the phenotype of individuals with sex chromosome aneuploidies, because this is crucial for optimizing clinical care, and because by we may learn about factors that drive risk for psychopathology. Among these aneuploidies is the 47, XXY (Klinefelter syndrome) variant, which is present in approximately 1 in 650 boys and men [1]. Social adaptive dysfunction is often seen in boys and men with 47,XXY [4, 5] This includes social withdrawal, social anxiety, depressed social skills, and in a proportion (an estimated 20–25%) of the individuals high levels of autism traits or symptoms [6, 7]. The aim was to assess if these aspects of the phenotype are related to testosterone deficiency, which is typically seen in 47,XXY from puberty onwards

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