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ASD Comorbidity in Fragile X Syndrome: Symptom Profile and Predictors of Symptom Severity in Adolescent and Young Adult Males

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Abstract

Many males with FXS meet criteria for ASD. This study was designed to (1) describe ASD symptoms in adolescent and young adult males with FXS (n = 44) and (2) evaluate the contributions to ASD severity of cognitive, language, and psychiatric factors, as well as FMRP (the protein deficient in FXS). A few ASD symptoms on the ADOS-2 were universal in the sample. There was less impairment in restricted and repetitive behaviors (RRB) than in the social affective (SA) domain. The best predictor of overall ASD severity and SA severity was expressive syntactic ability. RRB severity was best predicted by the psychiatric factors. Implications for clinical practice and for understanding the ASD comorbidity in FXS are discussed.

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Acknowledgments

The authors gratefully acknowledge the families who participated in the project. The authors also thank the many research staff involved in the project, but especially Alicia Cox, Joan Gunther, Leona Kelly, Debra Reisinger, Emily Schwoerer, and Stephanie Summers.

Funding

This research was supported by grants R01HD024356 and U54HD079125 from the National Institutes of Health awarded to Leonard Abbeduto, grant F32DC013934 to Jessica Klusek, grants R01MH090194 and R01MH107573 to Jane E. Roberts, and by grant UL1TR001860 to University of California, Davis.

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Authors and Affiliations

  1. University of California, Davis, USA

    Leonard Abbeduto, Angela John Thurman, Andrea McDuffie, Robyn Tempero Feigles & Danielle J. Harvey

  2. University of South Carolina, Columbia, USA

    Jessica Klusek & Jane E. Roberts

  3. New York State Institute for Basic Research in Developmental Disabilities, New York, USA

    W. Ted Brown, Tatyana Adayev, Giuseppe LaFauci & Carl Dobkins

  4. UC Davis MIND Institute, 2825 50th St, Sacramento, CA, 95817, USA

    Leonard Abbeduto

Authors
  1. Leonard Abbeduto
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  2. Angela John Thurman
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  3. Andrea McDuffie
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  4. Jessica Klusek
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  5. Robyn Tempero Feigles
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  6. W. Ted Brown
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  7. Danielle J. Harvey
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  8. Tatyana Adayev
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  9. Giuseppe LaFauci
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  10. Carl Dobkins
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  11. Jane E. Roberts
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Contributions

LA conceived of the study, participated in its design and coordination, performed the statistical analysis, drafted the manuscript, and conceptualized and received funding for the larger project from which the data for this study were drawn; AJT participated in the design, data collection, data preparation, and interpretation of the data; AM participated in the design, data collection, and interpretation of the data; JK participated in the design, data collection, and interpretation of the data; RTF participated in data collection and interpretation of the data; WTB participated in the design of the study and interpretation of the data; DJH participated in the conceptualization of the statistical analysis and the interpretation of the data; TA, GL, and CD all helped perform the analysis of the biological measures and interpretation of the data; and JER helped conceive of the larger study from which the data were drawn and participated in the design, coordination, and interpretation of the data for this study. All authors helped to draft the manuscript, and read and approved the final manuscript.

Corresponding author

Correspondence to Leonard Abbeduto.

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Conflict of interest

Leonard Abbeduto has received funding to develop and implement outcome measures for clinical trials from F. Hoffman-LaRoche, Ltd., Roche TCRC, Inc., Neuren Pharmaceuticals Limited, and Fulcrum Therapeutics, Inc. W. Ted Brown and Giuseppe LaFauci hold a patent for a measure used in this study [System and Method for Quantifying Fragile X Mental 1 Protein in tissue and blood samples (United States Patent # 8628934)], with the assignee for the patent being the Research Foundation for Mental Hygiene, Inc. No other authors have conflicts or financial disclosures to declare.

Ethical Approval

All procedures performed in studies involving human participants were in accordance with the ethical standards of the institutional and/or national research committee and with the 1964 Helsinki declaration and its later amendments or comparable ethical standards. All authors obtained review and approval from the Institutional review boards of their respective universities/institutions. This article does not contain any studies with animals performed by any of the authors.

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was obtained from all individual participants included in the study or from their legally authorized representatives.

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Abbeduto, L., Thurman, A.J., McDuffie, A. et al. ASD Comorbidity in Fragile X Syndrome: Symptom Profile and Predictors of Symptom Severity in Adolescent and Young Adult Males. J Autism Dev Disord 49, 960–977 (2019). https://doi.org/10.1007/s10803-018-3796-2

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