Brief Report: Modest but Clinically Meaningful Effects of Early Behavioral Intervention in Twins with Rett Syndrome—A Case Study
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A growing evidence base supports early intensive behavioral intervention (EIBI) for young children with autism spectrum disorder. We have found only one study exploring the outcome of EIBI for children with Rett syndrome, which reported little effect. It suggested that future studies should employ more fine-grained outcome measures. We provided EIBI for twin 3-year-old girls with Rett syndrome for a period of 3 years. We analyzed raw scores and standard scores from a measure of adaptive behavior and a detailed assessment of skills across 25 areas. We detected moderate but clinically meaningful gains in areas such as communication and self-help. Gains are discussed from a quality of life perspective and whether the moderate effects can justify the resources required in EIBI.
KeywordsRett syndrome Early intensive behavioral intervention Case study Clinical significance
We thank the girls, the girls’ parents, the staff in the pre-school and the psychologist at the local pedagogical-psychological service that were involved in the behavioral intervention program. A special thanks to our colleges at Department of Autism.
KBT and SE have made substantial contributions to conception and design, acquisition of data, analysis and interpretation of data. They both have been involved in drafting the manuscript and have given final approval of the version to be published.
This study was not funded. In Norway, where this study took place, the approach Early Behavioral Intervention for Children is publicly funded. All children are enrolled in community-based kindergartens and supervision is delivered by community agencies.
Compliance with Ethical Standards
Conflict of interest
The authors declare that they have no conflict of interest.
- Bondy, A., & Frost, L. (2011). A picture’s worth: PECS and other visual communication strategies in autism. Bethesda: Routledge.Google Scholar
- Eikeseth, S., Klintwall, L., Jahr, E., & Karlsson, P. (2012). Outcome for children with autism receiving early and intensive behavioral intervention in mainstream preschool and kindergarten settings. Research in Autism Spectrum Disorders,6(2), 829–835. https://doi.org/10.1016/j.rasd.2011.09.002.CrossRefGoogle Scholar
- Eldevik, S., Jahr, E., Eikeseth, S., Hastings, R. P., & Hughes, C. J. (2010). Cognitive and adaptive behavior outcomes of behavioral intervention for young children with intellectual disability. Behavior Modification,34(1), 16–34. https://doi.org/10.1177/0145445509351961.CrossRefPubMedGoogle Scholar
- Fukumizu, M., Hayashi, M., Miyajima, T., Ishizaki, A., Tanaka, H., & Kohyama, J. (2015). A nationwide survey on the uses of melatonin and ramelteon in Japanese children. No to Hattatsu [Brain & Development],47(1), 23–27.Google Scholar
- Green, G. (2011). Early intensive behavior analytic intervention for autism spectrum disorders. In E. A. Mayville & J. A. Mulick (Eds.), Behavioral foundations of effective autism treatment (pp. 183–199). Cornwall-on-Hudson: Sloan Publishing.Google Scholar
- Kerr, A., & Witt Engerström, I. (2001). Rett disorder and the developing brain. Oxford: Oxford University Press.Google Scholar
- Maffei, S., De Felice, C., Cannarile, P., Leoncini, S., Signorini, C., Pecorelli, A., et al. (2014). Effects of omega-3 PUFAs supplementation on myocardial function and oxidative stress markers in typical Rett syndrome. Mediators of Inflammation,2014, 983178. https://doi.org/10.1155/2014/983178.CrossRefPubMedPubMedCentralGoogle Scholar
- Makrygianni, M. K., Gena, A., Katoudi, S., & Galanis, P. (2018). The effectiveness of applied behavior analytic interventions for children with Autism Spectrum Disorder: A meta-analytic study. Research in Autism Spectrum Disorders,51, 18–31. https://doi.org/10.1016/j.rasd.2018.03.006.CrossRefGoogle Scholar
- Partington, J. (2008). The assessment of basic language and learning skills-revised (the ABLLS-R): An assessment, curriculum guide, and skills tracking system for children with autism or other developmental disabilities. Pleasant Hill: Behavior Analysts Inc.Google Scholar
- Reichow, B., Barton, E., Boyd, B., & Hume, K. (2014). Early intensive behavioral intervention (EIBI) for young children with autism spectrum disorders (ASD). Campbell Collaboration, 10. Retrieved from https://www.campbellcollaboration.org/library/early-intensive-behavioural-intervention-children-autism.html
- Schultz, R. J., Glaze, D. G., Patterson, M. C., Firth, H., & Dashe, J. F. (2018). Rett syndrome: Genetics, clinical features, and diagnosis. Retrieved from https://www.uptodate.com/contents/rett-syndrome-genetics-clinical-features-and-diagnosis?source=search_result&search=rett%20syndrome&selectedTitle=1~29
- Sigafoos, J., Kagohara, D., van der Meer, L., Green, V. A., O’Reilly, M. F., Lancioni, G. E., et al. (2011). Communication assessment for individuals with Rett syndrome: A systematic review. Research in Autism Spectrum Disorders,5(2), 692–700. https://doi.org/10.1016/j.rasd.2010.10.006.CrossRefGoogle Scholar
- Sparrow, S. S., Cicchetti, D. V., & Balla, D. A. (2005). Vineland Adaptive Behavior Scales, (Vineland™-II) (2nd ed.). San Antonio: PEARSON.Google Scholar
- Stasolla, F., De Pace, C., Damiani, R., Di Leone, A., Albano, V., & Perilli, V. (2014). Comparing PECS and VOCA to promote communication opportunities and to reduce stereotyped behaviors by three girls with Rett syndrome. Research in Autism Spectrum Disorders,8(10), 1269–1278. https://doi.org/10.1016/j.rasd.2014.06.009.CrossRefGoogle Scholar
- Weissman, L., Bridgemohan, C., Augustyn, M., Patterson, M., & Torchia, M. (2018). Autism spectrum disorder in children and adolescents: Overview of management. UpToDate [Internet]. Version, 19.Google Scholar