Measures of attention in Rett syndrome: Internal consistency reliability.

Susan A. Rose, Sam V. Wass, Jeffery J. Jankowski, Aleksandra Djukic

Research output: Contribution to journalArticlepeer-review

Abstract

Objective: Rett syndrome (RTT), an x-linked neurodevelopmental disorder caused by spontaneous mutations in the MECP2 gene, is characterized by profound impairments in expressive language and purposeful hand use. We have pioneered the use of gaze-based tasks to by-pass these limitations and developed measures suitable for clinical trials with RTT. Here we estimated internal consistency reliability for three aspects of attention that are key to cognitive growth and that we previously identified as impaired in RTT. Method: Using a sample of 66 children with RTT (2–19 years), we assessed Sustained Attention (butterfly task: Butterfly traverses the screen only when fixated and distractors are ignored); Disengaging/Shifting Attention (“gap/overlap” task: Shifts of gaze from central to peripheral targets are compared in conditions where the central stimulus remains or disappears at the onset of the peripheral target); Selective Attention (search task: the target is embedded in arrays differing in size and distractor type). Results: Reliability was acceptable to excellent on almost all key measures from tasks assessing Sustained Attention and Disengaging/Shifting Attention, with split-half coefficients and Cronbach alphas ranging from .70 to .93. Reliability increased as more trials were aggregated, with acceptable levels often reached with as few as six to nine trials. Measures from Selective Attention showed only limited reliability. Conclusion: Finding that critical aspects of attention can be reliably assessed in RTT with gaze-based tasks constitutes a major advance in the development of cognitive measures appropriate for clinical and translational work. Key Points—Question: We sought to determine whether measures of three aspects of attention that are key to cognitive growth, and that we previously identified as impaired in children in Rett syndrome (RTT), would show internal consistency reliability. Findings: Reliability was acceptable to excellent on almost all key measures from tasks assessing both sustained attention and disengaging/shifting attention, but limited for measures assessing selective attention. Importance: These data provide the first evidence that measures of critical aspects of cognition, assessed in RTT with gaze-based measures, show good psychometric reliability; this represents an important step in establishing objective behavioral outcome measures for use in assessing the effects of therapeutic interventions. Next Steps: These findings point the way to improving the reliability for measures of Sustain Attention and, more generally, for developing objective, performance-based assessments for other cognitive domains and children and with other disorders. (PsycInfo Database Record (c) 2021 APA, all rights reserved)

Original languageEnglish (US)
JournalNeuropsychology
DOIs
StateAccepted/In press - 2021

Keywords

  • attention
  • eye-tracking
  • reliability
  • Rett syndrome

ASJC Scopus subject areas

  • Neuropsychology and Physiological Psychology

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