A Cross-Sectional Analysis of the Development of Response Inhibition in Children with Chromosome 22q11.2 Deletion Syndrome

Ling M. Wong,Tony J. Simon,Heather M. Shapiro

doi:10.3389/fpsyt.2013.00081

Abstract

Chromosome 22q11.2 deletion syndrome (22q11.2DS) is a neurogenetic disorder that is associated with cognitive impairments and significantly elevated risk for developing schizophrenia. While impairments in response inhibition are central to executive dysfunction in schizophrenia, the nature and development of such impairments in children with 22q11.2DS, a group at high risk for the disorder, are not clear. Here we used a classic Go/No-Go paradigm to quantify proactive (anticipatory stopping) and reactive (actual stopping) response inhibition in 47 children with 22q11.2DS and 36 typically developing (TD) children, all ages 7–14. A cross-sectional design was used to examine age-related associations with response inhibition. When compared with TD individuals, children with 22q11.2DS demonstrated typical proactive response inhibition at all ages. By contrast, reactive response inhibition was impaired in children with 22q11.2DS relative to TD children. While older age predicted better reactive response inhibition in TD children, there was no age-related association with reactive response inhibition in children with 22q11.2DS. Closer examination of individual performance data revealed a wide range of performance abilities in older children with 22q11.2DS; some typical and others highly impaired. The results of this cross-sectional analysis suggest an impaired developmental trajectory of reactive response inhibition in some children with 22q11.2DS that might be related to atypical development of neuroanatomical systems underlying this cognitive process. As part of a larger study, this investigation might help identify risk factors for conversion to schizophrenia and lead to early diagnosis and preventive intervention.

Highlights

Adaptive behavior in a dynamic world relies heavily on cognitive control, defined as the ability to suppress irrelevant thoughts and actions while strengthening others [1]
Individuals with schizophrenia perform more poorly on response inhibition tasks when compared to healthy controls [8, 9], and it is believed that aberrant response inhibition might contribute to some of the general neuropsychological impairments seen in the disorder
Diagnostic group, Go trial type, and gender were regressed on accuracy and response time (RT)

Summary

Introduction

Adaptive behavior in a dynamic world relies heavily on cognitive control, defined as the ability to suppress irrelevant thoughts and actions while strengthening others [1]. An integral factor in successful cognitive control is response inhibition, which supports the suppression of a prepotent response that might be irrelevant or inappropriate in a given context. Evidence suggests that response inhibition improves throughout child development and into adulthood [2,3,4], likely due to the development of supporting neural architecture [5, 6]. Individuals with schizophrenia perform more poorly on response inhibition tasks when compared to healthy controls [8, 9], and it is believed that aberrant response inhibition might contribute to some of the general neuropsychological impairments seen in the disorder. Evidence suggests that impairments in control processes might be present in individuals long before conversion to schizophrenia [10,11,12], as well as in their unaffected first-degree relatives [13]. Impairments in inhibitory control are considered a key component of the intermediate phenotype of the disorder [14, 15]

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