Validating the Communication and Symbolic Behavior Scales-Developmental Profile Infant-Toddler Checklist (CSBS-DP ITC) Beyond Infancy in the CDKL5 Deficiency Disorder

Jacinta Saldaris; Helen Leonard; Kingsley Wong; Peter Jacoby; Mary Spence; Eric D Marsh; Tim A Benke; Scott Demarest; Jenny Downs

doi:10.1007/s10803-023-06002-w

Validating the Communication and Symbolic Behavior Scales-Developmental Profile Infant-Toddler Checklist (CSBS-DP ITC) Beyond Infancy in the CDKL5 Deficiency Disorder

J Autism Dev Disord. 2023 May 15:10.1007/s10803-023-06002-w. doi: 10.1007/s10803-023-06002-w. Online ahead of print.

Authors

Jacinta Saldaris¹, Helen Leonard¹, Kingsley Wong¹, Peter Jacoby¹, Mary Spence², Eric D Marsh³, Tim A Benke⁴, Scott Demarest⁴, Jenny Downs^{5

6}

Affiliations

¹ Telethon Kids Institute, Centre for Child Health Research, The University of Western Australia, PO Box 855, West Perth, WA, 6872, Australia.
² Children's Hospital Colorado Therapy Care, Highlands Ranch, CO, USA.
³ Division of Neurology, Children's Hospital of Philadelphia, Perelman School of Medicine, University of Pennsylvania, Philadelphia, PA, USA.
⁴ Children's Hospital Colorado, Paediatric Neurology, University of Colorado School of Medicine, Aurora, CO, USA.
⁵ Telethon Kids Institute, Centre for Child Health Research, The University of Western Australia, PO Box 855, West Perth, WA, 6872, Australia. Jenny.Downs@telethonkids.org.au.
⁶ School of Physiotherapy and Exercise Science, Curtin University, Perth, WA, Australia. Jenny.Downs@telethonkids.org.au.

PMID: 37184758
PMCID: PMC10699574 (available on 2024-11-15)
DOI: 10.1007/s10803-023-06002-w

Abstract

CDKL5 deficiency disorder (CDD) results in early-onset epilepsy and lifelong cognitive and motor impairments. With no validated measure for communication in CDD, this study evaluated the psychometric properties of the Communication and Symbolic Behavior Scales-Developmental Profile Infant Toddler Checklist (CSBS-DP ITC). Caregivers (n = 150; affected individuals aged 1-29 years) completed the CSBS-DP ITC. Distribution of scores indicated a floor effect. There was poor divergent validity for the three-factor model but goodness of fit and convergent validity data were satisfactory for the one-factor model. Individuals with poorer overall functional abilities scored lower on the CSBS-DP ITC. Test-retest reliability was excellent. The floor effect could explain the very high reliability, suggesting problems as a sensitive outcome measure in clinical trials for CDD.

Keywords: CDKL5 deficiency disorder; Communication; Outcome measure; Psychometric.

Abstract

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