Responsive Parenting as a Target for Telehealth Language Interventions in Fragile X Syndrome: Implications for Scalability and Best Practices

2021 ◽  
Vol 42 (04) ◽  
pp. 287-300
Author(s):  
Lauren Bullard ◽  
Leonard Abbeduto
Keyword(s):  
Best Practices ◽  
Language Development ◽  
Fragile X Syndrome ◽  
Child Language ◽  
Fragile X ◽  
Future Research ◽  
Dyadic Relationship ◽  
Video Teleconferencing ◽  
Treatment Gains ◽  
Speech And Language Development

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.

scholarly journals Narrating Disability, Narrating Religious Practice: Reconciliation and Fragile X Syndrome

2010 ◽  
Vol 48 (2) ◽  
pp. 99-111 ◽  
Author(s):  
Marsha Michie ◽  
Debra Skinner
Keyword(s):  
Fragile X Syndrome ◽  
Fragile X ◽  
Religious Practice ◽  
Illness Narratives ◽  
Future Research ◽  
Semistructured Interviews ◽  
Religious Perspectives

Abstract This article examines the place of religion in the narratives of mothers of children with fragile X syndrome. In semistructured interviews, a majority of women combined narratives of religious practice with illness narratives, interpreting their children's disabilities within a religious framework. Informed by Arthur Frank's (1995) concept of “wounded storytellers,” the authors articulate a reconciliation narrative that mothers commonly used to describe their transition from viewing disability as a burden or challenge to seeing it as a blessing, or as a part of God's purpose or plan for their lives. The authors discuss the significance of narrative for better understanding religious perspectives on disability and conclude with the implications of these findings for practitioners and future research.

scholarly journals Effect of Recurrent Otitis Media on Language Profile in Children with Fragile X Syndrome

2013 ◽  
Vol 6 ◽  
pp. CMENT.S11157 ◽  
Author(s):  
Hatem Soliman Badran ◽  
Khairy M. Abulnasr ◽  
Sherien Abd El Hameed Nasser
Keyword(s):  
Language Development ◽  
Fragile X Syndrome ◽  
Otitis Media ◽  
Middle Ear ◽  
Fragile X ◽  
Academic Medical Center ◽  
Case Series ◽  
History Of ◽  
Ear Infections ◽  
Recurrent Otitis Media

Objective Language is almost always affected in fragile X syndrome (FXS), and a delay in language acquisition is one of the first characteristics. The aim of this work was to study the effect of recurrent middle ear infections on the language profile in boys with FXS. Study design: Prospective case series. Setting Academic Medical Center. Subjects and Methods The present study was conducted on 30 males, ranging in age from 4–10 years. They were diagnosed as having a full mutation of DNA. The males were divided into two groups: Group A included 15 children with a history of recurrent middle ear infections more than four times per year during the first 4 years of life, and Group B did not have a history of recurrent middle ear infections during the first 4 years of life. Language assessments were done for all participants using the Standardized Arabic Language test. Results Results showed significant delays in language development in children with FXS. Relative strengths in semantics compared to syntax and pragmatics were observed in all boys. The recurrent ear infections of the boys played an important role in the language development delay. The mean of receptive, expressive, and total language age was better and higher among boys without a history of recurrent middle ear infections compared to boys with recurrent middle ear infections. Conclusion Recurrent otitis media in boys with FXS exacerbates the language problems that exist in this syndrome.

scholarly journals Contributions of phonological and verbal working memory to language development in adolescents with fragile X syndrome

2011 ◽  
Vol 3 (4) ◽  
pp. 335-347 ◽  
Author(s):  
Elizabeth I. Pierpont ◽  
Erica Kesin Richmond ◽  
Leonard Abbeduto ◽  
Sara T. Kover ◽  
W. Ted Brown
Keyword(s):  
Working Memory ◽  
Language Development ◽  
Fragile X Syndrome ◽  
Fragile X ◽  
Verbal Working Memory

scholarly journals A Pilot Quantitative Evaluation of Early Life Language Development in Fragile X Syndrome

2019 ◽  
Vol 9 (2) ◽  
pp. 27 ◽  
Author(s):  
Debra Reisinger ◽  
Rebecca Shaffer ◽  
Ernest Pedapati ◽  
Kelli Dominick ◽  
Craig Erickson
Keyword(s):  
Language Development ◽  
Language Learning ◽  
Fragile X Syndrome ◽  
Fragile X ◽  
Language Skills ◽  
Infants And Toddlers ◽  
Early Language ◽  
Early Language Development ◽  
Automated Vocal Analysis ◽  
Analysis System

Language delay and communication deficits are a core characteristic of the fragile X syndrome (FXS) phenotype. To date, the literature examining early language development in FXS is limited potentially due to barriers in language assessment in very young children. The present study is one of the first to examine early language development through vocal production and the language learning environment in infants and toddlers with FXS utilizing an automated vocal analysis system. Child vocalizations, conversational turns, and adult word counts in the home environment were collected and analyzed in a group of nine infants and toddlers with FXS and compared to a typically developing (TD) normative sample. Results suggest infants and toddlers with FXS are exhibiting deficits in their early language skills when compared to their chronological expectations. Despite this, when accounting for overall developmental level, their early language skills appear to be on track. Additionally, FXS caregivers utilize less vocalizations around infants and toddlers with FXS; however, additional research is needed to understand the true gap between FXS caregivers and TD caregivers. These findings provide preliminary information about the early language learning environment and support for the feasibility of utilizing an automated vocal analysis system within the FXS population that could ease data collection and further our understanding of the emergence of language development.

Early Language Intervention Using Distance Video-Teleconferencing: A Pilot Study of Young Boys With Fragile X Syndrome and Their Mothers

2016 ◽  
Vol 25 (1) ◽  
pp. 46-66 ◽  
Author(s):  
Andrea McDuffie ◽  
Ashley Oakes ◽  
Wendy Machalicek ◽  
Monica Ma ◽  
Lauren Bullard ◽  
...  
Keyword(s):  
Fragile X Syndrome ◽  
Parent Education ◽  
Augmentative And Alternative Communication ◽  
Fragile X ◽  
Single Case ◽  
Relative Effectiveness ◽  
Language Intervention ◽  
Intervention Effects ◽  
Video Teleconferencing ◽  
Communication Device

Purpose This study examined the effects of a naturalistic parent-implemented language intervention on the use of verbally responsive language by mothers of 6 young boys with fragile X syndrome. The intervention included parent education sessions and clinician coaching delivered onsite and by distance video-teleconferencing. Method A single-case multiple baseline across participants was used to examine intervention effects on maternal use of language support strategies. A nonparametric analysis was used to evaluate the relative effectiveness of onsite compared with distance coaching sessions. Results Mothers increased their use of utterances that followed into their child's focus of attention and prompted child communication acts. Intervention effects were not observed for maternal contingent responses to child communication, possibly due to the limited number of spontaneous communication acts children produced. Children showed moderate increases in the use of prompted communication acts, whereas intervention effects on spontaneous communication acts were more modest and variable. Comparable increases in maternal strategy use were observed during onsite and distance sessions. Conclusions No previous study has examined a distance-delivered parent-implemented language intervention for young boys with fragile X syndrome. Mothers were able to increase their use of verbally responsive language. Intervention efficacy might be enhanced by incorporating an augmentative and alternative communication device for some children and a more concerted focus on increasing the frequency of child communication acts. Findings provide preliminary support for the efficacy of the distance delivery format.

scholarly journals Language Development in Individuals With Fragile X Syndrome

2009 ◽  
Vol 29 (2) ◽  
pp. 133-148 ◽  
Author(s):  
Lizbeth H. Finestack ◽  
Erica K. Richmond ◽  
Leonard Abbeduto
Keyword(s):  
Language Development ◽  
Fragile X Syndrome ◽  
Fragile X
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