Authors: Scott S. Hall, Arlette Bujanda Rodriguez, Booil Jo, and Joy S. Pollard

Summarized by Scott Hall

Summary

The main aim of this study was to determine whether boys with FXS who had participated in our previous 12-week randomized trial of telehealth-enabled behavioral treatments for challenging behaviors were still benefiting from having received the treatment several years later. A second aim of the study was to determine whether administering booster sessions via telehealth could be beneficial for those children who continued to exhibit challenging behaviors at follow-up. The treatment employed a therapy called functional communication training (FCT) that helps the child learn to communicate more effectively, rather than being aggressive or self-injurious. We were able to contact 16 of 24 parents who had been coached to implement the behavioral treatment with their child via telehealth for 12 weeks and 18 of 24 parents who had continued with their child’s usual care for 12 weeks in our previous study.

Data showed that boys who had received behavioral treatment were more likely at follow-up to show continued improvements in caregiver measures of irritability and lethargy/social withdrawal, compared to boys who had been assigned to the treatment-as-usual group. Families who had received behavioral treatment also showed lower levels of parental stress compared to those who had continued with usual care. Six families from the original treatment group opted to receive a 1-hour booster session via telehealth with their child, and 4 of these 6 children exhibited reduced challenging behavior following implementation of the booster session.

The results further underscore the significant impact that behavioral treatments can have on rates of challenging behaviors commonly exhibited by boys with FXS, particularly when parents are coached to implement the intervention with their child via telehealth. Empowering parents to implement behavioral treatments with their child in their own home can therefore have durable effects on minimizing challenging behaviors in boys with FXS.

Why This Matters

The new findings are important because they show that when parents are coached to implement behavioral treatments with their child in their own home, improvements in challenging behaviors can be maintained, even after treatment sessions have ended. This is because the treatment involved teaching the child to communicate what they want more appropriately, rather than using the challenging behavior to get what they want. The results also underscore the feasibility of delivering behavioral treatment for this population via telehealth, which if implemented more broadly, will serve to dramatically increase treatment access for families across the country.

Next Steps

It will be important that future research examine factors that may affect how well the treatment works for individual children, and whether there may be different treatment schedules that best ensure long-term maintenance of improvements in behavior. For example, it may be that older children with FXS may benefit from longer or more frequent booster sessions to prevent challenging behavior from reverting back to its pre-treatment frequency or severity, compared to the schedule for younger children with FXS. Research on family dynamics in the context of caregiver-partnered behavioral care in FXS will also be valuable in designing tailored behavioral treatment protocols.

Acknowledgements

The researchers are very grateful to the participants and families who gave up their time to take part in this study. They would also like to thank the National Fragile X Foundation for their help. This research was supported by a Developmental Disabilities Translational Research Program Award from The John Merck Fund (PI: S. Hall).

FOR MORE DETAILS VISIT:

Read the full publication, Long-term follow-up of telehealth-enabled behavioral treatment for challenging behaviors in boys with fragile X syndrome, in the Journal of Neurodevelopmental Disorders.

Hall, S.S., Rodriguez, A.B., Jo, B. et al. Long-term follow-up of telehealth-enabled behavioral treatment for challenging behaviors in boys with fragile X syndrome. J Neurodevelop Disord 14, 53 (2022). https://doi.org/10.1186/s11689-022-09463-9

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