Noncomprehension Signaling in Males and Females With Fragile X Syndrome Purpose This study used a prospective longitudinal design to evaluate the trajectory and predictors of noncomprehension signaling in male and female youth with fragile X syndrome (FXS). Method A direction-following task in which some of the directions were inadequate was administered. Participants were 52 youth (36 boys, 16 ... Research Article
Research Article  |   June 10, 2017
Noncomprehension Signaling in Males and Females With Fragile X Syndrome
 
Author Affiliations & Notes
  • Angela John Thurman
    MIND Institute, University of California Davis, Sacramento
    Department of Psychiatry and Behavioral Sciences, University of California Davis, Sacramento
  • Sara T. Kover
    Department of Speech and Hearing Sciences, University of Washington, Seattle
  • W. Ted Brown
    New York State Institute for Basic Research in Developmental Disabilities, Staten Island
  • Danielle J. Harvey
    Department of Public Health Sciences, University of California Davis
  • Leonard Abbeduto
    MIND Institute, University of California Davis, Sacramento
    Department of Psychiatry and Behavioral Sciences, University of California Davis, Sacramento
  • Disclosure: The authors have declared that no competing interests existed at the time of publication.
    Disclosure: The authors have declared that no competing interests existed at the time of publication. ×
  • Correspondence to Angela John Thurman: ajthurman@ucdavis.edu
  • Editor: Rhea Paul
    Editor: Rhea Paul×
  • Associate Editor: Sarita Eisenberg
    Associate Editor: Sarita Eisenberg×
Article Information
Special Populations / Genetic & Congenital Disorders / Attention, Memory & Executive Functions / Language / Research Articles
Research Article   |   June 10, 2017
Noncomprehension Signaling in Males and Females With Fragile X Syndrome
Journal of Speech, Language, and Hearing Research, June 2017, Vol. 60, 1606-1621. doi:10.1044/2016_JSLHR-L-15-0358
History: Received October 15, 2015 , Revised June 10, 2016 , Accepted December 5, 2016
 
Journal of Speech, Language, and Hearing Research, June 2017, Vol. 60, 1606-1621. doi:10.1044/2016_JSLHR-L-15-0358
History: Received October 15, 2015; Revised June 10, 2016; Accepted December 5, 2016

Purpose This study used a prospective longitudinal design to evaluate the trajectory and predictors of noncomprehension signaling in male and female youth with fragile X syndrome (FXS).

Method A direction-following task in which some of the directions were inadequate was administered. Participants were 52 youth (36 boys, 16 girls) with FXS. Upon study entry, participants ranged from 10 to 16 years. The average number of annual assessments per participant was 3.65 (range = 1–4), providing 198 data points for analysis.

Results Participants with FXS were less likely to signal noncomprehension than younger, typically developing, cognitively matched children. The average rate of change in noncomprehension signaling was not significantly different from 0 for either boys or girls, suggesting a plateau. Both FMRP and nonverbal IQ were significant independent predictors of noncomprehension signaling for boys. Variability in noncomprehension signaling among girls was not explained by any of the predictors, but trends similar to those observed for boys were observed.

Conclusions Noncomprehension signaling appears to be an area of weakness for individuals with FXS. Because the failure to signal noncomprehension can have negative, cumulative effects on comprehension, the results suggest a need for interventions targeting the requisite cognitive skills.

Acknowledgments
We wish to thank the families who so graciously participated in this project. This research was supported by Grant R01HD024356 (awarded to Leonard Abbeduto) and by the MIND Institute Intellectual and Developmental Disabilities Research Center (U54 HD079125, awarded to Leonard Abbeduto), both funded by the National Institute of Child Health and Human Development. Note that Leonard Abbeduto has received financial support to develop and implement outcome measures for clinical trials from F. Hoffman-LaRoche Ltd., Roche TCRC, Inc., and Neuren Pharmaceuticals Ltd. No other authors have financial disclosures to make.
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