Quantifying the resolution of spatial and temporal representation in children with 22q11.2 deletion syndrome

Kathryn L. McCabe, Abbie M. Popa, Courtney Durdle, Michele Amato, Margarita H. Cabaral, Joshua Cruz, Ling M. Wong, Danielle Harvey, Nicole Tartaglia, Tony J. Simon

Research output: Contribution to journalArticle

Abstract

Objectives: Our ability to generate mental representation of magnitude from sensory information affects how we perceive and experience the world. Reduced resolution of the mental representations formed from sensory inputs may generate impairment in the proximal and distal information processes that utilize these representations. Impairment of spatial and temporal information processing likely underpins the non-verbal cognitive impairments observed in 22q11.2 deletion syndrome (22q11DS). The present study builds on prior research by seeking to quantify the resolution of spatial and temporal representation in children with 22q11DS, sex chromosome aneuploidy (SCA), and a typically developing (TD) control group. Participants and methods: Children (22q11DS = 70, SCA = 49, TD = 46) responded to visual or auditory stimuli with varying difference ratios. The participant's task was to identify which of two sequentially presented stimuli was of larger magnitude in terms of, size, duration, or auditory frequency. Detection threshold was calculated as the minimum difference ratio between the "standard" and the "target" stimuli required to achieve 75% accuracy in detecting that the two stimuli were different. Results: Children with 22q11DS required larger magnitude difference between spatial stimuli for accurate identification compared with both the SCA and TD groups (% difference from standard: 22q11DS = 14; SCA = 8; TD: 7; F = 8.42, p < 0.001). Temporal detection threshold was also higher for the 22q11DS group to both visual (% difference from standard: 22q11DS = 14; SCA = 8; TD = 7; F = 8.33, p < 0.001) and auditory (% difference from standard: 22q11DS = 23; SCA = 12; TD: 8; F = 8.99, p < 0.001) stimuli compared with both the SCA and TD groups, while the SCA and TD groups displayed equivalent performance on these measures (p's > 0.05). Pitch detection threshold did not differ among the groups (p's > 0.05). Conclusions: The observation of higher detection thresholds to spatial and temporal stimuli indicates further evidence for reduced resolution in both spatial and temporal magnitude representation in 22q11DS, that does not extend to frequency magnitude representation (pitch detection), and which is not explained by generalized cognitive impairment alone. These findings generate further support for the hypothesis that spatiotemporal hypergranularity of mental representations contributes to the non-verbal cognitive impairment seen in 22q11DS.

Original languageEnglish (US)
Article number40
JournalJournal of Neurodevelopmental Disorders
Volume11
Issue number1
DOIs
StatePublished - Dec 20 2019

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DiGeorge Syndrome
Sex Chromosomes
Aneuploidy
Chromosomes, Human, Pair 8
Aptitude
Automatic Data Processing
Observation
Control Groups
Research

Keywords

  • 22q11.2 deletion syndrome (22q11DS)
  • Attention
  • Children
  • Magnitude processing
  • Spatiotemporal attention

ASJC Scopus subject areas

  • Pediatrics, Perinatology, and Child Health
  • Pathology and Forensic Medicine
  • Clinical Neurology
  • Cognitive Neuroscience

Cite this

Quantifying the resolution of spatial and temporal representation in children with 22q11.2 deletion syndrome. / McCabe, Kathryn L.; Popa, Abbie M.; Durdle, Courtney; Amato, Michele; Cabaral, Margarita H.; Cruz, Joshua; Wong, Ling M.; Harvey, Danielle; Tartaglia, Nicole; Simon, Tony J.

In: Journal of Neurodevelopmental Disorders, Vol. 11, No. 1, 40, 20.12.2019.

Research output: Contribution to journalArticle

McCabe, Kathryn L. ; Popa, Abbie M. ; Durdle, Courtney ; Amato, Michele ; Cabaral, Margarita H. ; Cruz, Joshua ; Wong, Ling M. ; Harvey, Danielle ; Tartaglia, Nicole ; Simon, Tony J. / Quantifying the resolution of spatial and temporal representation in children with 22q11.2 deletion syndrome. In: Journal of Neurodevelopmental Disorders. 2019 ; Vol. 11, No. 1.
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AU - Popa, Abbie M.

AU - Durdle, Courtney

AU - Amato, Michele

AU - Cabaral, Margarita H.

AU - Cruz, Joshua

AU - Wong, Ling M.

AU - Harvey, Danielle

AU - Tartaglia, Nicole

AU - Simon, Tony J.

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N2 - Objectives: Our ability to generate mental representation of magnitude from sensory information affects how we perceive and experience the world. Reduced resolution of the mental representations formed from sensory inputs may generate impairment in the proximal and distal information processes that utilize these representations. Impairment of spatial and temporal information processing likely underpins the non-verbal cognitive impairments observed in 22q11.2 deletion syndrome (22q11DS). The present study builds on prior research by seeking to quantify the resolution of spatial and temporal representation in children with 22q11DS, sex chromosome aneuploidy (SCA), and a typically developing (TD) control group. Participants and methods: Children (22q11DS = 70, SCA = 49, TD = 46) responded to visual or auditory stimuli with varying difference ratios. The participant's task was to identify which of two sequentially presented stimuli was of larger magnitude in terms of, size, duration, or auditory frequency. Detection threshold was calculated as the minimum difference ratio between the "standard" and the "target" stimuli required to achieve 75% accuracy in detecting that the two stimuli were different. Results: Children with 22q11DS required larger magnitude difference between spatial stimuli for accurate identification compared with both the SCA and TD groups (% difference from standard: 22q11DS = 14; SCA = 8; TD: 7; F = 8.42, p < 0.001). Temporal detection threshold was also higher for the 22q11DS group to both visual (% difference from standard: 22q11DS = 14; SCA = 8; TD = 7; F = 8.33, p < 0.001) and auditory (% difference from standard: 22q11DS = 23; SCA = 12; TD: 8; F = 8.99, p < 0.001) stimuli compared with both the SCA and TD groups, while the SCA and TD groups displayed equivalent performance on these measures (p's > 0.05). Pitch detection threshold did not differ among the groups (p's > 0.05). Conclusions: The observation of higher detection thresholds to spatial and temporal stimuli indicates further evidence for reduced resolution in both spatial and temporal magnitude representation in 22q11DS, that does not extend to frequency magnitude representation (pitch detection), and which is not explained by generalized cognitive impairment alone. These findings generate further support for the hypothesis that spatiotemporal hypergranularity of mental representations contributes to the non-verbal cognitive impairment seen in 22q11DS.

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