Distance delivery of a spoken language intervention for school-aged and adolescent boys with fragile X syndrome

ABSTRACT

A small randomized group design (N = 20) was used to examine a parent-implemented intervention designed to improve the spoken language skills of school-aged and adolescent boys with FXS, the leading cause of inherited intellectual disability. The intervention was implemented by speech-language pathologists who used distance video-teleconferencing to deliver the intervention. The intervention taught mothers to use a set of language facilitation strategies while interacting with their children in the context of shared story-telling. Treatment group mothers significantly improved their use of the targeted intervention strategies. Children in the treatment group increased the duration of engagement in the shared story-telling activity as well as use of utterances that maintained the topic of the story. Children also showed increases in lexical diversity, but not in grammatical complexity.

KEYWORDS:

• Distance teleconferencing
• expressive language sampling
• narrative story-telling
• parent-implemented intervention

Acknowledgements

The authors wish to thank the families who participated in this research. We also thank Dr. Wendy Machalicek for her assistance with videoteleconferencing procedures and formative contributions to conceptualizing the role of the BCBA in this program of intervention research.

Funding

This research was supported by grant U54 HD079125 from the National Institute of Child Health and Human Development to Dr. Leonard Abbeduto. 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 Limited. Randi J. Hagerman has received funding from Marinus, Novartis, Alcobra, Neuren, and Curemark to carry out treatment studies in fragile X syndrome and ASD. She has also consulted with Roche/Genetech, Zynerba, Orid, and Novartis regarding treatment studies in fragile X syndrome. No other authors have financial disclosures or declarations of interests to report.

Declaration of interest

The authors report no conflicts of interest. The authors alone are responsible for the content and writing of the paper.

Additional information

Funding

This research was supported by grant U54 HD079125 from the National Institute of Child Health and Human Development to Dr. Leonard Abbeduto. 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 Limited. Randi J. Hagerman has received funding from Marinus, Novartis, Alcobra, Neuren, and Curemark to carry out treatment studies in fragile X syndrome and ASD. She has also consulted with Roche/Genetech, Zynerba, Orid, and Novartis regarding treatment studies in fragile X syndrome. No other authors have financial disclosures or declarations of interests to report.