Related Experiment Video
Updated: Jun 22, 2025
Author Spotlight: Unveiling the Connection Between Sleep Disorders and Cognitive Symptoms in Depression
Published on: April 26, 2024
Modification of a parent-report sleep scale for individuals with CDKL5 deficiency disorder: a psychometric study
Jacinta M Saldaris1, Scott Demarest2, Peter Jacoby1
1Telethon Kids Institute, Centre for Child Health Research, The University of Western Australia, Perth, Western Australia, Australia.
Insights
Sleep difficulties in CDKL5 deficiency disorder are common. Modified sleep scales (DIMS and DOES) show promise for assessing insomnia and related impairments in this condition.
Area of Science:
- Neurology
- Pediatrics
- Sleep Medicine
Background:
- CDKL5 deficiency disorder is a severe neurodevelopmental condition characterized by frequent seizures.
- Sleep disturbances are a prevalent and significant challenge for individuals with CDKL5 deficiency disorder.
- Existing sleep assessment tools require validation for this specific population.
Purpose of the Study:
- To evaluate the psychometric properties of the Sleep Disturbance Scale for Children (SDSC) in individuals with CDKL5 deficiency disorder.
- To assess the factor structure of the Disorders of Initiating and Maintaining Sleep (DIMS) and Disorders of Excessive Somnolence (DOES) domains of the SDSC.
- To determine the suitability of modified SDSC domains as clinical outcome assessments.
Main Methods:
- A cross-sectional psychometric study design was employed.
- Data were collected from 125 individuals (aged 3+ years) with CDKL5 deficiency disorder.
- Confirmatory factor analysis was used to examine the factor structure and internal consistency of the DIMS and DOES domains.
Main Results:
- Analysis focused on the DIMS and DOES domains due to low response rates for Sleep Breathing Disorders items.
- After item refinement, both DIMS and DOES domains demonstrated good item loading and internal consistency (DIMS: .78, DOES: .76).
- The modified domains exhibited good discriminant validity, though overall model fit indices were slightly below recommended thresholds.
Conclusions:
- The modified DIMS and DOES domains of the SDSC are potentially suitable for assessing insomnia and related impairments in CDKL5 deficiency disorder.
- These validated scales could serve as valuable clinical outcome assessments in research and clinical practice.
- The findings may extend to other developmental and epileptic encephalopathy conditions with similar sleep challenges.
Study Objectives:
Sleep difficulties are common in CDKL5 deficiency disorder, a developmental and epileptic encephalopathy. This study evaluated the factor structure of the Disorders of Initiating and Maintaining Sleep (DIMS), Disorders of Excessive Somnolence (DOES), and Sleep Breathing Disorders domains of the Sleep Disturbance Scale for Children for CDKL5 deficiency disorder.
Methods:
A cross-sectional psychometric study design was used. Data were collected for 125 individuals aged 3 years or older who attended a United States Centers of Excellence clinic or registered with the International CDKL5 Disorder Database.
Results:
The median age was 10.3 years (range 3.2-40.7 years) and 105 (84%) were female. Two of the 3 Sleep Breathing Disorders items were not observed by most respondents and analysis was restricted to the DIMS and DOES domains. Using all items in the initial confirmatory factor analysis, 2 items in the DIMS domain and 1 item in the DOES domain loaded poorly. After deleting these items and repeating the analysis, item loading (.524-.814) and internal consistency (DIMS: .78, DOES: .76) statistics were good. The square of the interdomain correlation coefficient was .17, less than average variance extracted values for both domains and indicating good discriminant validity. The Tucker-Lewis and Comparative Fit indices were slightly lower than the threshold of > .9 for establishing goodness of fit.
Conclusions:
The modified DIMS and DOES domains from the Sleep Disturbance Scale for Children could be suitable clinical outcome assessments of insomnia and related impairments in CDKL5 deficiency disorder and potentially other developmental and epileptic encephalopathy conditions.
Citation:
Saldaris JM, Demarest S, Jacoby P, et al. Modification of a parent-report sleep scale for individuals with CDKL5 deficiency disorder: a psychometric study. J Clin Sleep Med. 2024;20 (12):1887-1893.
