Psychometric Assessment of the Rett Syndrome Caregiver Assessment of Symptom Severity (RCASS) / Melissa RASPA in Journal of Autism and Developmental Disorders, 55-3 (March 2025)  

Public ISBD [article]  inJournal of Autism and Developmental Disorders > 55-3 (March 2025) . - p.997-1009 Titre : Psychometric Assessment of the Rett Syndrome Caregiver Assessment of Symptom Severity (RCASS) Type de document : Texte imprimé et/ou numérique Auteurs : Melissa RASPA, Auteur ; Angela GWALTNEY, Auteur ; Carla BANN, Auteur ; Jana VON HEHN, Auteur ; Timothy A. BENKE, Auteur ; Eric D. MARSH, Auteur ; Sarika U. PETERS, Auteur ; Amitha ANANTH, Auteur ; Alan K. PERCY, Auteur ; Jeffrey L. NEUL, Auteur Article en page(s) : p.997-1009 Langues : Anglais (eng) Index. décimale : PER Périodiques Résumé : Rett syndrome is a severe neurodevelopmental disorder that affects about 1 in 10,000 females. Clinical trials of disease modifying therapies are on the rise, but there are few psychometrically sound caregiver-reported outcome measures available to assess treatment benefit. We report on a new caregiver-reported outcome measure, the Rett Caregiver Assessment of Symptom Severity (RCASS). Using data from the Rett Natural History Study (n = 649), we examined the factor structure, using both exploratory and confirmatory factor analysis, and the reliability and validity of the RCASS. The four-factor model had the best overall fit, which covered movement, communication, behavior, and Rett-specific symptoms. The RCASS had moderate internal consistency. Strong face validity was found with age and mutation type, and convergent validity was established with other similar measures, including the Revised Motor-Behavior Assessment Scale, Clinical Severity Scale, Clinical Global Impression Scale, and the Child Health Questionnaire. These data provide initial evidence that the RCASS is a viable caregiver-outcome measure for use in clinical trials in Rett syndrome. Future work to assess sensitivity to change and other measures of reliability, such as test-retest and inter-rater agreement, are needed. En ligne : https://doi.org/10.1007/s10803-024-06238-0 Permalink : https://www.cra-rhone-alpes.org/cid/opac_css/index.php?lvl=notice_display&id=548 [article] Psychometric Assessment of the Rett Syndrome Caregiver Assessment of Symptom Severity (RCASS) [Texte imprimé et/ou numérique] / Melissa RASPA, Auteur ; Angela GWALTNEY, Auteur ; Carla BANN, Auteur ; Jana VON HEHN, Auteur ; Timothy A. BENKE, Auteur ; Eric D. MARSH, Auteur ; Sarika U. PETERS, Auteur ; Amitha ANANTH, Auteur ; Alan K. PERCY, Auteur ; Jeffrey L. NEUL, Auteur . - p.997-1009. Langues : Anglais (eng) in Journal of Autism and Developmental Disorders > 55-3 (March 2025) . - p.997-1009 Index. décimale : PER Périodiques Résumé : Rett syndrome is a severe neurodevelopmental disorder that affects about 1 in 10,000 females. Clinical trials of disease modifying therapies are on the rise, but there are few psychometrically sound caregiver-reported outcome measures available to assess treatment benefit. We report on a new caregiver-reported outcome measure, the Rett Caregiver Assessment of Symptom Severity (RCASS). Using data from the Rett Natural History Study (n = 649), we examined the factor structure, using both exploratory and confirmatory factor analysis, and the reliability and validity of the RCASS. The four-factor model had the best overall fit, which covered movement, communication, behavior, and Rett-specific symptoms. The RCASS had moderate internal consistency. Strong face validity was found with age and mutation type, and convergent validity was established with other similar measures, including the Revised Motor-Behavior Assessment Scale, Clinical Severity Scale, Clinical Global Impression Scale, and the Child Health Questionnaire. These data provide initial evidence that the RCASS is a viable caregiver-outcome measure for use in clinical trials in Rett syndrome. Future work to assess sensitivity to change and other measures of reliability, such as test-retest and inter-rater agreement, are needed. En ligne : https://doi.org/10.1007/s10803-024-06238-0 Permalink : https://www.cra-rhone-alpes.org/cid/opac_css/index.php?lvl=notice_display&id=548

Validating the Communication and Symbolic Behavior Scales-Developmental Profile Infant-Toddler Checklist (CSBS-DP ITC) Beyond Infancy in the CDKL5 Deficiency Disorder / Helen LEONARD ; Kingsley WONG ; Peter JACOBY ; Mary SPENCE ; Eric D. MARSH ; Tim A. BENKE ; Scott DEMAREST ; Jenny DOWNS in Journal of Autism and Developmental Disorders, 54-7 (July 2024)  

Public ISBD [article]  inJournal of Autism and Developmental Disorders > 54-7 (July 2024) . - p.2526-2535 Titre : Validating the Communication and Symbolic Behavior Scales-Developmental Profile Infant-Toddler Checklist (CSBS-DP ITC) Beyond Infancy in the CDKL5 Deficiency Disorder Type de document : Texte imprimé et/ou numérique Auteurs : Helen LEONARD, Auteur ; Kingsley WONG, Auteur ; Peter JACOBY, Auteur ; Mary SPENCE, Auteur ; Eric D. MARSH, Auteur ; Tim A. BENKE, Auteur ; Scott DEMAREST, Auteur ; Jenny DOWNS, Auteur Article en page(s) : p.2526-2535 Langues : Anglais (eng) Index. décimale : PER Périodiques Résumé : 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. En ligne : https://doi.org/10.1007/s10803-023-06002-w Permalink : https://www.cra-rhone-alpes.org/cid/opac_css/index.php?lvl=notice_display&id=533 [article] Validating the Communication and Symbolic Behavior Scales-Developmental Profile Infant-Toddler Checklist (CSBS-DP ITC) Beyond Infancy in the CDKL5 Deficiency Disorder [Texte imprimé et/ou numérique] / Helen LEONARD, Auteur ; Kingsley WONG, Auteur ; Peter JACOBY, Auteur ; Mary SPENCE, Auteur ; Eric D. MARSH, Auteur ; Tim A. BENKE, Auteur ; Scott DEMAREST, Auteur ; Jenny DOWNS, Auteur . - p.2526-2535. Langues : Anglais (eng) in Journal of Autism and Developmental Disorders > 54-7 (July 2024) . - p.2526-2535 Index. décimale : PER Périodiques Résumé : 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. En ligne : https://doi.org/10.1007/s10803-023-06002-w Permalink : https://www.cra-rhone-alpes.org/cid/opac_css/index.php?lvl=notice_display&id=533

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