Extending the Parent-Delivered Early Start Denver Model to Young Children with Fragile X Syndrome
This is the first study to evaluate an autism intervention model, the parent-delivered Early Start Denver Model (P-ESDM), for young children with fragile X syndrome (FXS), a known genetic disorder associated with autism spectrum disorder. Four parent–child dyads participated in a low-intensity, parent coaching model of the P-ESDM to evaluate initial efficacy and acceptability. Parents improved in P-ESDM fidelity, implemented intervention goals to increase child learning, and found the experience moderately to highly acceptable. Visual examination and Baseline Corrected Tau effect sizes revealed mixed results across child measures. Findings suggest a potential therapeutic opportunity in need of larger, well-controlled studies of P-ESDM and other interventions for families of young children with FXS who face limited empirically-supported intervention options.
KeywordsFragile X syndrome Autism spectrum disorder Parent-mediated Early intervention Early Start Denver Model
We gratefully acknowledge the families who participated in this study, as well as Melanie Rothfuss and Patrick Hugunin for their work involved with behavioral coding and the manuscript.
LAV designed and implemented the study, co-assisted in the analyses of data, wrote the bulk of the manuscript, and addressed the reviewers’ comments and revisions made to the manuscript. CEBM conducted the statistical analyses, lead the analysis of the data, wrote the results section of the manuscript, and responded with revisions to the statistical analyses, data, and results section of the manuscript. RS participated in the coding of data. DH provided clinical guidance on the implementation of the study, developed and made changes per reviewers’ requests to the figures representing the data, and assisted with manuscript development and revisions to the manuscript addressing the reviewers’ comments.
This study was funded by the MIND Institute and the National Fragile X Foundation (NFXF) awarded to Laurie A. Vismara and David Hessl.
Compliance with Ethical Standards
Conflict of interest
Potential conflicts of interest for Laurie A. Vismara involve royalties from publications and honoraria received from lectures related to the parent-delivered Early Start Denver Model. David Hessl serves on the Scientific Advisory Board and the Clinical Trials Committee of the NFXF.
All procedures performed in this study involving human participants were in accordance with the ethical standards of the institutional research committees and with the 1964 Helsinki declaration and its later amendments or comparable ethical standards.
Informed consent was obtained from parents or legal guardians of all individual participants for whom identifying information is included in this article. No adverse effects of the study occurred.
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