Anxiety, attention problems, hyperactivity, and the Aberrant Behavior Checklist in fragile X syndrome

Authors

  • Anne Wheeler,

    1. Carolina Institute for Developmental Disabilities, University of North Carolina at Chapel Hill, Chapel Hill, North Carolina
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  • Melissa Raspa,

    1. Social, Statistical, and Environmental Sciences, RTI International, Research Triangle Park, North Carolina
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  • Carla Bann,

    1. Social, Statistical, and Environmental Sciences, RTI International, Research Triangle Park, North Carolina
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  • Ellen Bishop,

    1. Social, Statistical, and Environmental Sciences, RTI International, Research Triangle Park, North Carolina
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  • David Hessl,

    1. MIND Institute, University of California—Davis Medical Center, University of California—Davis School of Medicine, Davis, California
    2. Department of Psychiatry and Behavioral Sciences, University of California—Davis School of Medicine, Davis, California
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  • Pat Sacco,

    1. Novartis Pharmaceuticals Corporation, East Hanover, New Jersey
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  • Donald B. Bailey Jr.

    Corresponding author
    1. Social, Statistical, and Environmental Sciences, RTI International, Research Triangle Park, North Carolina
    • Correspondence to:

      Donald Bailey, Ph.D., Social, Statistical, and Environmental Sciences, RTI International, 3040 Cornwallis Rd., Research Triangle Park, NC 27709.

      E-mail: dbailey@rti.org

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Abstract

Behavior problems are a common challenge for individuals with fragile X syndrome (FXS) and constitute the primary clinical outcome domain in trials testing new FXS medications. However, little is known about the relationship between caregiver-reported behavior problems and co-occurring conditions such as anxiety and attention problems. In this study, 350 caregivers, each with at least one son or daughter with full-mutation FXS, rated one of their children with FXS using the Aberrant Behavior Checklist—Community Version (ABC-C); the Anxiety subscale of the Anxiety, Depression, and Mood Scale; and the Attention/Hyperactivity Items from the Symptom Inventories. In addition to examining family consequences of these behaviors, this study also sought to replicate psychometric findings for the ABC-C in FXS, to provide greater confidence for its use in clinical trials with this population. Psychometric properties and baseline ratings of problem behavior were consistent with other recent studies, further establishing the profile of problem behavior in FXS. Cross-sectional analyses suggest that selected dimensions of problem behavior, anxiety, and hyperactivity are age related; thus, age should serve as an important control in any studies of problem behavior in FXS. Measures of anxiety, attention, and hyperactivity were highly associated with behavior problems, suggesting that these factors at least coincide with problem behavior. However, these problems generally did not add substantially to variance in caregiver burden predicted by elevated behavior problems. The results provide further evidence of the incidence of problem behaviors and co-occurring conditions in FXS and the impact of these behaviors on the family. © 2013 Wiley Periodicals, Inc.

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