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Are Children with Fragile X Syndrome Losing their Zzzz's ... and Y?

Kronk, Rebecca Ann (2008) Are Children with Fragile X Syndrome Losing their Zzzz's ... and Y? Doctoral Dissertation, University of Pittsburgh. (Unpublished)

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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 Children's 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.


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Item Type: University of Pittsburgh ETD
Status: Unpublished
CreatorsEmailPitt UsernameORCID
Kronk, Rebecca
ETD Committee:
TitleMemberEmail AddressPitt UsernameORCID
Committee CoChairVondra,
Committee CoChairNoll,
Committee MemberDahl,
Committee MemberBagnato,
Date: 25 April 2008
Date Type: Completion
Defense Date: 26 March 2008
Approval Date: 25 April 2008
Submission Date: 8 April 2008
Access Restriction: No restriction; Release the ETD for access worldwide immediately.
Institution: University of Pittsburgh
Schools and Programs: School of Education > Psychology in Education
Degree: PhD - Doctor of Philosophy
Thesis Type: Doctoral Dissertation
Refereed: Yes
Uncontrolled Keywords: bedtime routine; Fragile X syndrome; parent mood; sleep
Other ID:, etd-04082008-111106
Date Deposited: 10 Nov 2011 19:35
Last Modified: 15 Nov 2016 13:39


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