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.
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.
Authors: Marsha Mailick Seltzer; Erin T Barker; Jan S Greenberg; Jinkuk Hong; Christopher Coe; David Almeida Journal: Health Psychol Date: 2011-12-12 Impact factor: 4.267
Authors: Marinus H van Ijzendoorn; Anna H Rutgers; Marian J Bakermans-Kranenburg; Sophie H N Swinkels; Emma van Daalen; Claudine Dietz; Fabienne B A Naber; Jan K Buitelaar; Herman van Engeland Journal: Child Dev Date: 2007 Mar-Apr
Authors: Molly Losh; Jessica Klusek; Gary E Martin; John Sideris; Morgan Parlier; Joseph Piven Journal: Am J Med Genet B Neuropsychiatr Genet Date: 2012-06-12 Impact factor: 3.568
Authors: Nell Maltman; Janna Guilfoyle; Kritika Nayar; Gary E Martin; Molly Winston; Joseph C Y Lau; Lauren Bush; Shivani Patel; Michelle Lee; John Sideris; Deborah A Hall; Lili Zhou; Kevin Sharp; Elizabeth Berry-Kravis; Molly Losh Journal: Front Psychiatry Date: 2021-08-06 Impact factor: 5.435
Authors: Jessica Klusek; Joseph Schmidt; Amanda J Fairchild; Anna Porter; Jane E Roberts Journal: J Neurodev Disord Date: 2017-08-24 Impact factor: 4.025
Authors: Jessica Klusek; Giuseppe LaFauci; Tatyana Adayev; W Ted Brown; Flora Tassone; Jane E Roberts Journal: J Neurodev Disord Date: 2017-05-02 Impact factor: 4.025
Authors: Jessica Klusek; Anna Porter; Leonard Abbeduto; Tatyana Adayev; Flora Tassone; Marsha R Mailick; Anne Glicksman; Bridgette L Tonnsen; Jane E Roberts Journal: Front Genet Date: 2018-08-24 Impact factor: 4.599