Assessment of Cognitive Outcome Measures in Teenagers with 15q13.3 Microdeletion Syndrome

Crutcher, Emeline; Ali, May; Harrison, John; Sóvágó, Judit; Gomez‐Mancilla, Baltazar; Schaaf, Christian P.

doi:10.1007/s10803-015-2694-0

Cited by 4 publications

(5 citation statements)

References 17 publications

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“…This is in line with previously published data on individuals with 15q13.3 microdeletion syndrome, showing deficits in standardized cognitive and achievement tests, such as the Differential Ability Scales and impaired performance on cognitive outcome measures, such as the CogState and KiTAP tests (Crutcher et al, 2016 ; Ziats et al, 2016 ). The tasks administered in those tests, rather than purely auditory, required responses based upon both auditory and visual stimuli.…”

Section: Discussionsupporting

confidence: 92%

Brain Network Analysis of EEG Recordings Can Be Used to Assess Cognitive Function in Teenagers With 15q13.3 Microdeletion Syndrome

et al. 2021

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15q13.3 microdeletion syndrome causes a spectrum of cognitive disorders, including intellectual disability and autism. We assessed the ability of the EEG analysis algorithm Brain Network Analysis (BNA) to measure cognitive function in 15q13.3 deletion patients, and to differentiate between patient and control groups. EEG data was collected from 10 individuals with 15q13.3 microdeletion syndrome (14–18 years of age), as well as 30 age-matched healthy controls, as the subjects responded to Auditory Oddball (AOB) and Go/NoGo cognitive tasks. It was determined that BNA can be used to evaluate cognitive function in 15q13.3 microdeletion patients. This analysis also significantly differentiates between patient and control groups using 5 scores, all of which are produced from ERP peaks related to late cortical components that represent higher cognitive functions of attention allocation and response inhibition (P < 0.05).

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Section: Discussionsupporting

confidence: 92%

Brain Network Analysis of EEG Recordings Can Be Used to Assess Cognitive Function in Teenagers With 15q13.3 Microdeletion Syndrome

et al. 2021

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“…Some papers presented studies with populations whose clinical state of ID or ASD was associated to different diagnoses, including diverse genetic syndromes (Borgwardt et al, 2015;Crutcher et al, 2016;Jauregi et al, 2007;Merrill et al, 2014;Rose et al, 2013;Scott & Holfelder, 2015;Trezise et al, 2008;Vinck et al, 2007). A few of the syndromes related to ID or ASD, such as the Rett Syndrome, add testing challenges concerning the significant motor and language impairment in these populations (Rose et al, 2013).…”

Section: Category 1: Tests Attention Abilities Measured and Populatmentioning

confidence: 99%

“…The choice of stimuli was considered a crucial element to foster test´s attractiveness. The selected stimuli included: animal figures (Foster-Owens, 2016; Jiang et al, 2014); figures of objects or symbols (De Vries & Watson, 2008;Garretson et al, 1990, Huguenin, 1997Keehn et al, 2010;Pascualvaca et al, 1998;Rose et al, 2009); fairy tales characters (Aman et al, 1993); and tasks situated in children stories (Crutcher et al, 2016;Foster-Owens, 2016;Knox et al, 2012). Some of the criteria identified for the choice of stimuli included familiarity, emotional neutrality, and level of complexity (Garretson et al, 1990).…”

Section: Category 2: Procedures and Adaptations In The Testing Settingmentioning

confidence: 99%

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Systematic Review of Attention Testing in Allegedly “Untestable” Populations

Rosário¹,

Gomes²,

Loureiro³

2019

IJPRR

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Test use is extremely important, not only for clinical practice, but also for scientific research. Nonetheless, some populations have been considered "untestable". Among the different cognitive abilities assessed using tests, attention is a fundamental one. The present study presents a systematic review of the literature on attention testing in people with Intellectual Disability or Autism Spectrum Disorder, in order to identify: (1) if there are any tests that are fit to assess these populations; (2) which adaptations would be necessary for such tests to become fit; and (3) what limits and needs are involved. Our literature review identified 39 studies (review papers and empirical studies), all of which concern the administration of attention tests for people with Intellectual Disability or Autism Spectrum Disorder. The selected papers are presented and discussed from two analysis categories: (a) tests, attention abilities, and populations under study; (b) procedures and adaptations made to the testing settings. We identified 72 attention tests, where the majority of the groups of participants in the studies that were analyzed presented mild symptoms. The main adaptations done to the tests refer to strategies used to assist the comprehension of tasks, to communicate instructions, to assure engagement during the procedure, and ways to emit answers. The implications of our results are discussed.

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“…To measure cognitive function, the Cogstate Computerized Test Battery ( 2016 ) was included in the JAKE Task Battery and evaluated in this study. Involved are tests of information processing speed and fine motor skills, visual attention, visual recognition memory, working memory, and emotion processing, to assess ASD-associated neurocognitive deficits (Baron-Cohen et al, 2001 ; Surowiecki et al, 2002 ; Bavin et al, 2005 ; Mollica et al, 2005 ; Crutcher et al, 2016 ).…”

Section: Introductionmentioning

confidence: 99%

JAKE® Multimodal Data Capture System: Insights from an Observational Study of Autism Spectrum Disorder

et al. 2017

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Objective: To test usability and optimize the Janssen Autism Knowledge Engine (JAKE®) system's components, biosensors, and procedures used for objective measurement of core and associated symptoms of autism spectrum disorder (ASD) in clinical trials.Methods: A prospective, observational study of 29 children and adolescents with ASD using the JAKE system was conducted at three sites in the United States. This study was designed to establish the feasibility of the JAKE system and to learn practical aspects of its implementation. In addition to information collected by web and mobile components, wearable biosensor data were collected both continuously in natural settings and periodically during a battery of experimental tasks administered in laboratory settings. This study is registered at clinicaltrials.gov, NCT02299700.Results: Feedback collected throughout the study allowed future refinements to be planned for all components of the system. The Autism Behavior Inventory (ABI), a parent-reported measure of ASD core and associated symptoms, performed well. Among biosensors studied, the eye-tracker, sleep monitor, and electrocardiogram were shown to capture high quality data, whereas wireless electroencephalography was difficult to use due to its form factor. On an exit survey, the majority of parents rated their overall reaction to JAKE as positive/very positive. No significant device-related events were reported in the study.Conclusion: The results of this study, with the described changes, demonstrate that the JAKE system is a viable, useful, and safe platform for use in clinical trials of ASD, justifying larger validation and deployment studies of the optimized system.

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Assessment of Cognitive Outcome Measures in Teenagers with 15q13.3 Microdeletion Syndrome

Cited by 4 publications

References 17 publications

Brain Network Analysis of EEG Recordings Can Be Used to Assess Cognitive Function in Teenagers With 15q13.3 Microdeletion Syndrome

Brain Network Analysis of EEG Recordings Can Be Used to Assess Cognitive Function in Teenagers With 15q13.3 Microdeletion Syndrome

Systematic Review of Attention Testing in Allegedly “Untestable” Populations

JAKE® Multimodal Data Capture System: Insights from an Observational Study of Autism Spectrum Disorder

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