Rett Syndrome behaviour questionnaire in children and adults with Rett Syndrome: psychometric characterization and revised factor structure
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 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.
Adult, Child, Emotions, Humans, Psychometrics, Reproducibility of Results, Rett Syndrome/diagnosis, Surveys and Questionnaires
237-253
Leonard, Helen
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Oberman, Lindsay M.
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Downs, Jenny
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Cianfaglione, Rina
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Stahlhut, Michelle
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Larsen, Jane L.
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Madden, Katherine V.
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Kaufmann, Walter E.
adf4f856-003b-4b75-9645-35aad741424d
1 May 2023
Leonard, Helen
761634de-9bbd-4101-8d3e-a041f6abd526
Oberman, Lindsay M.
408d9b3c-5528-4a87-afcd-994e5186fe1b
Downs, Jenny
1d205734-590a-4725-b7da-b5f8abdd856c
Cianfaglione, Rina
bf9b4507-4a79-4f72-b7e2-7244b9dea9ef
Stahlhut, Michelle
18c19555-252c-48b3-801e-75f712346f3e
Larsen, Jane L.
f2b8be65-6dea-4824-bfef-940ccc2e5b83
Madden, Katherine V.
b1bce590-e97c-4b2a-8110-b0ca3a85525d
Kaufmann, Walter E.
adf4f856-003b-4b75-9645-35aad741424d
Leonard, Helen, Oberman, Lindsay M., Cianfaglione, Rina and Stahlhut, Michelle
,
et al.
(2023)
Rett Syndrome behaviour questionnaire in children and adults with Rett Syndrome: psychometric characterization and revised factor structure.
American journal on intellectual and developmental disabilities, 128 (3), .
(doi:10.1352/1944-7558-128.3.237).
Abstract
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.
Text
Manuscript_RSBQ_Characterization_clean_blinded_04OCT2022
- Accepted Manuscript
More information
Accepted/In Press date: 18 October 2022
e-pub ahead of print date: 28 April 2023
Published date: 1 May 2023
Additional Information:
Publisher Copyright:
© 2023 American Association on Mental Retardation. All rights reserved.
Keywords:
Adult, Child, Emotions, Humans, Psychometrics, Reproducibility of Results, Rett Syndrome/diagnosis, Surveys and Questionnaires
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Local EPrints ID: 479993
URI: http://eprints.soton.ac.uk/id/eprint/479993
PURE UUID: 3331df51-d125-4e18-bd00-7a3405292859
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Date deposited: 31 Jul 2023 17:04
Last modified: 28 Apr 2024 04:01
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Contributors
Author:
Helen Leonard
Author:
Lindsay M. Oberman
Author:
Jenny Downs
Author:
Michelle Stahlhut
Author:
Jane L. Larsen
Author:
Katherine V. Madden
Author:
Walter E. Kaufmann
Corporate Author: et al.
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