Rett Syndrome Behaviour Questionnaire in Children and Adults With Rett Syndrome: Psychometric Characterization and Revised Factor Structure
Rett syndrome (RTT) is a severe neurodevelopmental disorder associated with multiple neurobehavioral abnormalities. The Rett Syndrome Behaviour Questionnaire (RSBQ) was developed for pediatric RTT observational studies. Because its application has expanded to adult and interventional studies, we eva...
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| Veröffentlicht in: | American journal on intellectual and developmental disabilities 2023-05, Vol.128 (3), p.237-253 |
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| container_title | American journal on intellectual and developmental disabilities |
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| creator | Oberman, Lindsay M Leonard, Helen Downs, Jenny Cianfaglione, Rina Stahlhut, Michelle Larsen, Jane L Madden, Katherine V Kaufmann, Walter E |
| description | Rett syndrome (RTT) is a severe neurodevelopmental disorder associated with multiple neurobehavioral abnormalities. The Rett Syndrome Behaviour Questionnaire (RSBQ) was developed for pediatric RTT observational studies. Because its application has expanded to adult and interventional studies, we evaluated the RSBQ's psychometric properties in six pediatric (n = 323) and five adult (n = 309) datasets. Total and General Mood subscale scores had good reliability. Clinical severity had no influence on RSBQ scores. Exploratory and confirmatory factor analyses yielded 6 pediatric and 7 adult clinically relevant and psychometrically strong factors including the original Breathing Problems and Fear/Anxiety subscales and the novel Emotional and Disruptive Behavior subscale composed of items from the original General Mood and Nighttime Behaviours subscales. The present findings support additional evaluations and improvements of an important RTT behavioral measure. |
| doi_str_mv | 10.1352/1944-7558-128.3.237 |
| format | Article |
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The Rett Syndrome Behaviour Questionnaire (RSBQ) was developed for pediatric RTT observational studies. Because its application has expanded to adult and interventional studies, we evaluated the RSBQ's psychometric properties in six pediatric (n = 323) and five adult (n = 309) datasets. Total and General Mood subscale scores had good reliability. Clinical severity had no influence on RSBQ scores. Exploratory and confirmatory factor analyses yielded 6 pediatric and 7 adult clinically relevant and psychometrically strong factors including the original Breathing Problems and Fear/Anxiety subscales and the novel Emotional and Disruptive Behavior subscale composed of items from the original General Mood and Nighttime Behaviours subscales. The present findings support additional evaluations and improvements of an important RTT behavioral measure.</description><identifier>ISSN: 1944-7515</identifier><identifier>EISSN: 1944-7558</identifier><identifier>DOI: 10.1352/1944-7558-128.3.237</identifier><identifier>PMID: 37104862</identifier><language>eng</language><publisher>United States: American Association of Intellectual & Developmental Disabilities</publisher><subject>Adult ; Adults ; Behavior disorders ; Child ; Confirmatory factor analysis ; Developmental disabilities ; Disruptive behaviour ; Emotions ; Genetic disorders ; Humans ; Neurobehavioral functioning ; Neurodevelopmental disorders ; Neurological disorders ; Observational studies ; Pediatrics ; Psychometrics ; Quantitative psychology ; Questionnaires ; Reliability ; Reproducibility of Results ; Rett syndrome ; Rett Syndrome - diagnosis ; Surveys and Questionnaires</subject><ispartof>American journal on intellectual and developmental disabilities, 2023-05, Vol.128 (3), p.237-253</ispartof><rights>AAIDD.</rights><rights>Copyright American Association of Intellectual & Developmental Disabilities May 2023</rights><lds50>peer_reviewed</lds50><woscitedreferencessubscribed>false</woscitedreferencessubscribed><citedby>FETCH-LOGICAL-c248t-2dee0dea0b573335d0904e2e0e9d01c6c1f66d13b06b452443d6ac28312ec2d23</citedby><cites>FETCH-LOGICAL-c248t-2dee0dea0b573335d0904e2e0e9d01c6c1f66d13b06b452443d6ac28312ec2d23</cites></display><links><openurl>$$Topenurl_article</openurl><openurlfulltext>$$Topenurlfull_article</openurlfulltext><thumbnail>$$Tsyndetics_thumb_exl</thumbnail><link.rule.ids>314,776,780,12825,27901,27902,30976</link.rule.ids><backlink>$$Uhttps://www.ncbi.nlm.nih.gov/pubmed/37104862$$D View this record in MEDLINE/PubMed$$Hfree_for_read</backlink></links><search><creatorcontrib>Oberman, Lindsay M</creatorcontrib><creatorcontrib>Leonard, Helen</creatorcontrib><creatorcontrib>Downs, Jenny</creatorcontrib><creatorcontrib>Cianfaglione, Rina</creatorcontrib><creatorcontrib>Stahlhut, Michelle</creatorcontrib><creatorcontrib>Larsen, Jane L</creatorcontrib><creatorcontrib>Madden, Katherine V</creatorcontrib><creatorcontrib>Kaufmann, Walter E</creatorcontrib><title>Rett Syndrome Behaviour Questionnaire in Children and Adults With Rett Syndrome: Psychometric Characterization and Revised Factor Structure</title><title>American journal on intellectual and developmental disabilities</title><addtitle>Am J Intellect Dev Disabil</addtitle><description>Rett syndrome (RTT) is a severe neurodevelopmental disorder associated with multiple neurobehavioral abnormalities. 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| ispartof | American journal on intellectual and developmental disabilities, 2023-05, Vol.128 (3), p.237-253 |
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| source | Applied Social Sciences Index & Abstracts (ASSIA); MEDLINE; Education Source |
| subjects | Adult Adults Behavior disorders Child Confirmatory factor analysis Developmental disabilities Disruptive behaviour Emotions Genetic disorders Humans Neurobehavioral functioning Neurodevelopmental disorders Neurological disorders Observational studies Pediatrics Psychometrics Quantitative psychology Questionnaires Reliability Reproducibility of Results Rett syndrome Rett Syndrome - diagnosis Surveys and Questionnaires |
| title | Rett Syndrome Behaviour Questionnaire in Children and Adults With Rett Syndrome: Psychometric Characterization and Revised Factor Structure |
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