AIMS: To investigate the potential associations between daily bedtime routines, evening fluctuations in parent mood, and sleep patterns for a sample of children with fragile X syndrome (FXS). This study also investigated whether the association among these factors varies dependent upon the severity of the genetic mutation. SAMPLE: Children ages 3 years, 0 months to 17 years, 11 months with a full or partial mutation of FXS (N = 95). METHOD: In-home assessments were completed including a demographic form, bedtime routine information sheet, and abbreviated Child Sleep Habits Questionnaire (CSHQ), followed by a 14-day sleep diary. Actigraphy was added to the protocol for the local sample of participants (n = 7) from the Fragile X Center of Childrens Hospital of Pittsburgh. RESULTS: Parents reported that 48% of their children with FXS have sleep problems at a level worthy of a sleep clinic referral; 20% of participants received medication to induce sleep, but 58% continued to score in the clinical range on the CSHQ despite receiving medications. Actigraphy showed children with FXS had significantly different sleep parameters than a control group (n =14). Caregiver mood was a significant correlate of sleep disturbances; children with FXS whose caregivers reported feeling more overwhelmed or poorer mood had more sleep disturbances. Inconsistent bedtime routine was also associated with sleep disturbances. CONCLUSIONS: These data strongly suggest that routine clinical care of children with FXS should include careful screening of sleep. Interventions to assist parents in establishing and/or maintaining bedtime routines and managing their mood may be warranted. Effective use of medication to enhance sleep requires further investigation. Additional research is needed to identify specific problems with sleep so that clinical trials focusing on key parameters can be initiated.
| Identifer | oai:union.ndltd.org:PITT/oai:PITTETD:etd-04082008-111106 |
| Date | 25 April 2008 |
| Creators | Kronk, Rebecca Ann |
| Contributors | Ron Dahl, MD, Joan Vondra PhD, Stephen Bagnato, Ed.D, Robert Noll, PhD |
| Publisher | University of Pittsburgh |
| Source Sets | University of Pittsburgh |
| Language | English |
| Detected Language | English |
| Type | text |
| Format | application/pdf |
| Source | http://etd.library.pitt.edu/ETD/available/etd-04082008-111106/ |
| Rights | unrestricted, I hereby certify that, if appropriate, I have obtained and attached hereto a written permission statement from the owner(s) of each third party copyrighted matter to be included in my thesis, dissertation, or project report, allowing distribution as specified below. I certify that the version I submitted is the same as that approved by my advisory committee. I hereby grant to University of Pittsburgh or its agents the non-exclusive license to archive and make accessible, under the conditions specified below, my thesis, dissertation, or project report in whole or in part in all forms of media, now or hereafter known. I retain all other ownership rights to the copyright of the thesis, dissertation or project report. I also retain the right to use in future works (such as articles or books) all or part of this thesis, dissertation, or project report. |
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