scholarly journals Language Development From Early Childhood to Adolescence in Youths With Fragile X Syndrome

2020 ◽  
Vol 63 (11) ◽  
pp. 3727-3742
Author(s):  
Nancy C. Brady ◽  
Kandace Fleming ◽  
Shelley L. Bredin-Oja ◽  
Heather Fielding-Gebhardt ◽  
Steven F. Warren

Purpose The aim of this study was to investigate language growth in individuals with fragile X syndrome (FXS) from early childhood to adolescence and the influence of maternal responsivity on language growth. Method We conducted a longitudinal analysis of language development in 55 youths (44 males, 11 females) with FXS. Data collection spanned the ages of 11–216 months. We measured expressive and receptive vocabulary with standardized tests. The number of different words and mean length of utterance were obtained from language sample analyses of mother–child interactions. We also measured maternal comments (responsivity indicator) produced during the language samples and child nonverbal IQ. Results Growth models indicated that rates of number of different words and receptive vocabulary were related to maternal commenting. Mean length of utterance did not change significantly over time. Expressive vocabulary measured with a standardized test grew, but the growth was not related to maternal commenting. Nonverbal IQ was related to all language outcomes at age of 10 years and to changes over time in vocabulary. Visual analysis indicated that the highest scores on standardized tests were produced by girls; however, measures derived from language sample analyses appeared similar for boys and girls. Language models for boys only were similar to the total sample models with lower scores at age of 10 years for some outcomes. Conclusion Results of persistent language impairments for most youths with FXS suggest the need for continued, focused interventions aimed at improved language productions in addition to a responsive environment. Supplemental Material https://doi.org/10.23641/asha.13022825

2017 ◽  
Vol 45 (1) ◽  
pp. 72-96 ◽  
Author(s):  
ELIZABETH S. CHE ◽  
PATRICIA J. BROOKS ◽  
MARIA F. ALARCON ◽  
FRANCIS D. YANNACO ◽  
SEAMUS DONNELLY

AbstractWhen engaged in conversation, both parents and children tend to re-use words that their partner has just said. This study explored whether proportions of maternal and/or child utterances that overlapped in content with what their partner had just said contributed to growth in mean length of utterance (MLU), developmental sentence score, and vocabulary diversity over time. We analyzed the New England longitudinal corpus from the CHILDES database, comprising transcripts of mother–child conversations at 14, 20, and 32 months, using the CHIP command to compute proportions of utterances with overlapping content. Rates of maternal overlap, but not child overlap, at earlier time-points predicted child language outcomes at later time-points, after controlling for earlier child MLU. We suggest that maternal overlap plays a formative role in child language development by providing content that is immediately relevant to what the child has in mind.


2017 ◽  
Vol 60 (2) ◽  
pp. 422-434 ◽  
Author(s):  
Rouzana Komesidou ◽  
Nancy C. Brady ◽  
Kandace Fleming ◽  
Amy Esplund ◽  
Steven F. Warren

Purpose This research explored syntactic growth in children with fragile X syndrome (FXS) over a 5-year period, and variability in growth in relation to autism symptoms, nonverbal cognition, maternal responsivity, and gender. Method Language samples at 4 time points from 39 children with FXS, 31 boys and 8 girls, were analyzed using the Index of Productive Syntax (Scarborough, 1990) and mean length of utterance (Brown, 1973). The degree of autism symptoms was evaluated using the Childhood Autism Rating Scale (Schopler, Reichler, & Renner, 1988) at the first time point. Maternal responsivity estimates were averaged across time points. Results Children with FXS showed significant syntactic growth over time and a significant plateau (quadratic trend) in the later observations. Children who exhibited more autism symptoms at Time 1 had significantly lower syntactic abilities over time than children who exhibited fewer autism symptoms. Nonverbal cognition significantly predicted mean length of utterance scores but not Index of Productive Syntax scores. Maternal responsivity was not a significant predictor of syntactic outcomes. Girls with FXS generally demonstrated better expressive syntax than boys with FXS with notable individual differences. Conclusion Despite significant growth over time, expressive syntax is a vulnerable domain for children with FXS, especially for those with severe autism symptoms. Clinical implications arising from the current findings are discussed.


1991 ◽  
Vol 12 (1) ◽  
pp. 23-46 ◽  
Author(s):  
Hollis S. Scarborough ◽  
Leslie Rescorla ◽  
Helen Tager-Flusberg ◽  
Anne E. Fowler ◽  
Vicki Sudhalter

ABSTRACTMean length of utterance (MLU) in morphemes was examined as a predictor of the grammatical complexity of natural language corpora of normal preschoolers and of children and adolescents with delayed language, Fragile X syndrome, Down syndrome, and autism. The Index of Productive Syntax (IPSyn) served as the measure of syntactic and morphological proficiency. For all groups, a strong curvilinear association between measures was found across the MLU range from 1.0 to about 4.5. Correlations were weaker when MLU exceeded 3.0 than during earlier stages of language development, however, confirming previous suggestions that MLU becomes less closely associated with grammatical development as linguistic proficiency increases. For the language-disordered groups, moreover, the curves relating the two measures differed from the curves for the normal preschoolers because MLU frequently overestimated actual IPSyn scores. The results are discussed with respect to the use of MLU in conjunction with other measures of syntactic complexity in the study of atypical language development.


2021 ◽  
Vol 42 (04) ◽  
pp. 287-300
Author(s):  
Lauren Bullard ◽  
Leonard Abbeduto

AbstractThis review highlights the ways in which telehealth procedures can be implemented to help bridge the research-to-practice gap in supporting developmental outcomes for youth with fragile X syndrome (FXS). We review how the literature to date has informed potential treatment targets in the areas of speech and language development with a focus on understanding and supporting the dyadic relationship between the child and their biological mother, who is also impacted biologically. Notably, parental responsivity is an area that is strongly related to child language outcomes, both early and into adolescence, and thus, it is an important treatment target for subsequent interventions. To date, several parent-implemented interventions have been done in FXS across a broad age range (2–17-year-olds) all showing support not only that parents are successful in learning responsive strategies but also that there are subsequent impacts to child language development. Moreover, these interventions were successfully implemented at a distance through telehealth procedures including video teleconferencing and shared recordings of parent–child interactions. This review also addresses potential moderators of treatment gains. Implications for scaling such interventions in the future as well as best practices for incorporating telehealth procedures into future research and intervention programs are also discussed.


Author(s):  
Anne Hoffmann ◽  
Sue Ellen Krause ◽  
Joanne Wuu ◽  
Sue Leurgans ◽  
Stephen J. Guter ◽  
...  

Abstract Background Receptive and expressive vocabulary in adult and adolescent males with fragile X syndrome (FXS) have been shown as significantly lower than their chronological age; however, receptive vocabulary has been considered a strength relative to mental age. This has not been formally examined, however, and data are needed to compare receptive vocabulary with other language skills and with mental age in individuals with FXS. This is especially important as vocabulary measures are sometimes used as a proxy to estimate language ability. Methods This preliminary study examined receptive vocabulary, global language, and cognitive skills in 42 adults (33 males and 9 females) with FXS as a portion of the baseline evaluation prior to randomization in a clinical trial of ampakine CX516. The battery of standardized tests addressed receptive vocabulary with the Peabody Picture Vocabulary Test, Third Edition (PPVT-III), receptive and expressive language (termed henceforth as global language) via the Preschool Language Scale, Fourth Edition or the Clinical Evaluation of Language Fundamentals, Third Edition, and non-verbal cognition via the Stanford-Binet Intelligence Scales, Fourth Edition (SB-IV). Results Results showed (1) significantly higher receptive vocabulary than global language, (2) significantly better receptive vocabulary than non-verbal cognition, (3) equivalent non-verbal cognition and global language, and (4) severity of autism symptomatology was not correlated to receptive vocabulary or global language once non-verbal cognition was removed as factor. The scores from the PPVT-III did not represent the global language skills in our sample of adults with FXS. Conclusions Findings from this investigation strongly suggest that the PPVT-III should not be used as a screening tool for language levels or cognitive function in clinical studies since the scores from the PPVT-III were not representative of global language or non-verbal cognitive skills in adults with intellectual disabilities. This finding is critical in order to understand how to evaluate, as well as to treat, language in individuals with FXS. Development of efficient and appropriate tools to measure language, cognition, and behavior in individuals with FXS is essential.


2014 ◽  
Vol 57 (1) ◽  
pp. 212-226 ◽  
Author(s):  
Nancy Brady ◽  
Steven F. Warren ◽  
Kandace Fleming ◽  
Juliana Keller ◽  
Audra Sterling

Purpose This research explored whether sustained maternal responsivity (a parent–child interaction style characterized by warmth, nurturance, and stability as well as specific behaviors, such as contingent positive responses to child initiations) was a significant variable predicting vocabulary development of children with fragile X syndrome through age 9 years. Method Fifty-five mother–child dyads were followed longitudinally when children were between 2 and 10 years of age. Measures of maternal responsivity and child vocabulary were obtained at regular intervals starting at age 2.9 years. Sustained responsivity was indicated by the average responsivity measured over Observations 2–5. Responsivity at the 1st time period, autism symptoms, and cognitive development were used as control variables. Results After controlling for development and autism symptoms, the authors found significant effects for sustained responsivity on receptive vocabulary, expressive vocabulary, and the rate of different words children produced through age 9. Conclusions Maternal responsivity, which is typically a variable of interest during early childhood, continues to be a significant variable, predicting vocabulary development through the middle childhood period. Thus, responsivity is a potential target for language interventions through this age period.


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