Evidence map›Paper›PMID 38963064›Full record

ArticleJournal of clinical sleep medicine : JCSM : official publication of the American Academy of Sleep Medicine2024

Modification of a parent-report sleep scale for individuals with CDKL5 deficiency disorder: a psychometric study.

Jacinta M Saldaris, Scott Demarest, Peter Jacoby, Heather E Olson, Kiran Maski, Elia Pestana-Knight, Dana Price, Rajsekar Rajaraman, Bernhard Suter, Judith Weisenberg and 4 more

Abstract read
In one paragraph

Article in Journal of clinical sleep medicine : JCSM : official publication of the American Academy of Sleep Medicine, 2024. The graph could read no effect estimate from its abstract, so it casts no vote on the map. Cited by 3 papers.

0numbers the graph read from it
0cells of the map it votes in
3citing papers in PubMed
–field-weighted citation impact
1 · What the graph read from it

What it found

Each row is one number read from the abstract, on the scale the paper reported it, with its interval. Left of the dashed line favours the treatment, right favours the comparator. Under each row is the sentence it came from. New to these charts? A ten-minute tutorial.

The abstract states no effect estimate the extractor could read, or names no intervention and outcome on the map, so this paper lights no cell and moves no belief. It is still indexed, cited and linked below.

2 · The registry

The trial behind it

Trials whose registry record cites this paper, or whose number appears in the abstract. A trial that started after this paper was published is citing it as background, not reporting it.

Neither the registry nor the abstract names a trial number. If this is a trial report, that itself is worth knowing.

3 · Its place in the literature

Who cites it

3 citing papers in PubMed.

  1. Exploring sleep challenges in CDKL5 Deficiency Disorder.Journal of neurodevelopmental disorders · 2026
    Article
  2. Observational
  3. Article
4 · The record

Corrections and comments

PubMed lists nothing against this paper. Absence here is not a guarantee, only a check that was made.

5 · Who and what money

Authors and funding

14 authors.

Jacinta M SaldarisTelethon Kids Institute, Centre for Child Health Research, The University of Western Australia, Perth, Western Australia, Australia.
Scott DemarestSchool of Medicine, Depts Pediatrics, Neurology and Pharmacology and Children's Hospital Colorado, University of Colorado, Aurora, Colorado.
Peter JacobyTelethon Kids Institute, Centre for Child Health Research, The University of Western Australia, Perth, Western Australia, Australia.
Heather E OlsonDivision of Epilepsy and Clinical Neurophysiology and Epilepsy Genetics Program, Department of Neurology, Boston Children's Hospital, Boston, Massachusetts.
Kiran MaskiDivision of Epilepsy and Clinical Neurophysiology and Epilepsy Genetics Program, Department of Neurology, Boston Children's Hospital, Boston, Massachusetts.
Elia Pestana-KnightCleveland Clinic, Neurological Institute, Cleveland, Ohio.
Dana PriceNew York University Langone Health and Department of Neurology, New York University, New York, New York.
Rajsekar RajaramanUniversity of California, Los Angeles Mattel Children's Hospital, Los Angeles, California.
Bernhard SuterDepartment of Pediatrics & Neurology, Baylor College of Medicine, Houston, Texas.
Judith WeisenbergSt. Louis Children's Hospital and Washington University School of Medicine, St Louis, Missouri.
Helen LeonardTelethon Kids Institute, Centre for Child Health Research, The University of Western Australia, Perth, Western Australia, Australia.
Eric D MarshDivision of Child Neurology, Children's Hospital of Philadelphia and Departments of Neurology and Pediatrics, University of Pennsylvania Perelman School of Medicine, Philadelphia, Pennsylvania.
Tim A BenkeSchool of Medicine, Depts Pediatrics, Neurology and Pharmacology and Children's Hospital Colorado, University of Colorado, Aurora, Colorado.
Jenny DownsTelethon Kids Institute, Centre for Child Health Research, The University of Western Australia, Perth, Western Australia, Australia.

Funding

Genetic Analysis and Manipulation Core (GAEC)P50HD105351 · NICHD · BOSTON CHILDREN'S HOSPITAL · PI SCOTT Loren POMEROY, MUSTAFA SAHIN · 2021 to 2026
$9.4M
Multi-Site Validation of Biomarkers and Core Clinical Outcome Measures for Clinical Trials Readiness in CDKL5 Deficiency DisorderU01NS114312 · NINDS · UNIVERSITY OF COLORADO DENVER · PI BENKE, TIMOTHY A, DOWNS, JENNY · 2021 to 2025
$4.7M
NICHD NIH HHS P50 HD105351NINDS NIH HHS U01 NS114312
6 · The paper itself

Abstract

STUDY

objectivesSleep 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.

methodsA 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.

resultsThe 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.

conclusionsThe 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.

Indexed as

Epileptic SyndromesParentsPsychometricsSpasms, InfantileAdolescentAdultChildChild, PreschoolCross-Sectional StudiesFemaleHumansMaleReproducibility of ResultsSleep Wake DisordersSurveys and QuestionnairesYoung AdultCDKL5 deficiency disorderclinical outcome assessmentpsychometricsleep

Identifiers

PMID38963064
PMCPMC11609843

What OpenQuestion holds

Textmetadata
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Registered trials

None linked

Read under generation 80e0d062 · epoch 390. Bibliography from PubMed, PubMed Central and OpenAlex; grants from NIH RePORTER; trial links from ClinicalTrials.gov; estimates, votes and beliefs from the OpenQuestion graph.