Pragmatic Language Features of Mothers With the FMR1 Premutation Are Associated With the Language Outcomes of Adolescents and Young Adults With Fragile X Syndrome Purpose Pragmatic language difficulties have been documented as part of the FMR1 premutation phenotype, yet the interplay between these features in mothers and the language outcomes of their children with fragile X syndrome is unknown. This study aimed to determine whether pragmatic language difficulties in mothers with the FMR1 premutation ... Research Article
Research Article  |   February 01, 2016
Pragmatic Language Features of Mothers With the FMR1 Premutation Are Associated With the Language Outcomes of Adolescents and Young Adults With Fragile X Syndrome
 
Author Affiliations & Notes
  • Jessica Klusek
    University of South Carolina, Columbia
  • Sara E. McGrath
    University of South Carolina, Columbia
  • Leonard Abbeduto
    MIND Institute, University of California, Davis
  • Jane E. Roberts
    University of South Carolina, Columbia
  • 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 Jessica Klusek: klusek@mailbox.sc.edu
  • Editor: Rhea Paul
    Editor: Rhea Paul×
  • Associate Editor: Andrew Whitehouse
    Associate Editor: Andrew Whitehouse×
Article Information
Special Populations / Genetic & Congenital Disorders / Language Disorders / Social Communication & Pragmatics Disorders / Language / Research Articles
Research Article   |   February 01, 2016
Pragmatic Language Features of Mothers With the FMR1 Premutation Are Associated With the Language Outcomes of Adolescents and Young Adults With Fragile X Syndrome
Journal of Speech, Language, and Hearing Research, February 2016, Vol. 59, 49-61. doi:10.1044/2015_JSLHR-L-15-0102
History: Received March 16, 2015 , Revised June 26, 2015 , Accepted July 9, 2015
 
Journal of Speech, Language, and Hearing Research, February 2016, Vol. 59, 49-61. doi:10.1044/2015_JSLHR-L-15-0102
History: Received March 16, 2015; Revised June 26, 2015; Accepted July 9, 2015
Web of Science® Times Cited: 2

Purpose Pragmatic language difficulties have been documented as part of the FMR1 premutation phenotype, yet the interplay between these features in mothers and the language outcomes of their children with fragile X syndrome is unknown. This study aimed to determine whether pragmatic language difficulties in mothers with the FMR1 premutation are related to the language development of their children.

Method Twenty-seven mothers with the FMR1 premutation and their adolescent/young adult sons with fragile X syndrome participated. Maternal pragmatic language violations were rated from conversational samples using the Pragmatic Rating Scale (Landa et al., 1992). Children completed standardized assessments of vocabulary, syntax, and reading.

Results Maternal pragmatic language difficulties were significantly associated with poorer child receptive vocabulary and expressive syntax skills, with medium effect sizes.

Conclusions This work contributes to knowledge of the FMR1 premutation phenotype and its consequences at the family level, with the goal of identifying modifiable aspects of the child's language-learning environment that may promote the selection of treatments targeting the specific needs of families affected by fragile X. Findings contribute to our understanding of the multifaceted environment in which children with fragile X syndrome learn language and highlight the importance of family-centered intervention practices for this group.

Acknowledgments
We are grateful for funding support from the National Institutes of Health Grants 5F32DC013934, 5R01MH090194, 5R01HD024356, and 5U54HD079125 and the Research Participant Registry Core of the Carolina Institute for Developmental Disabilities Grant P30 HD03110 and to all of the families who participated.
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