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Autor/inn/en | Raspa, Melissa; Bann, Carla M.; Gwaltney, Angela; Benke, Timothy A.; Fu, Cary; Glaze, Daniel G.; Haas, Richard; Heydemann, Peter; Jones, Mary; Kaufmann, Walter E.; Lieberman, David; Marsh, Eric; Peters, Sarika; Ryther, Robin; Standridge, Shannon; Skinner, Steven A.; Percy, Alan K.; Neul, Jeffrey L. |
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Titel | A Psychometric Evaluation of the Motor-Behavioral Assessment Scale for Use as an Outcome Measure in Rett Syndrome Clinical Trials |
Quelle | In: American Journal on Intellectual and Developmental Disabilities, 125 (2020) 6, S.493-509 (17 Seiten)Infoseite zur Zeitschrift
PDF als Volltext |
Sprache | englisch |
Dokumenttyp | gedruckt; online; Zeitschriftenaufsatz |
ISSN | 1944-7515 |
Schlagwörter | Genetic Disorders; Psychometrics; Psychomotor Skills; Physical Disabilities; Interpersonal Competence; Daily Living Skills; Social Behavior; Antisocial Behavior; Physical Health; Test Items; Item Response Theory; Age Differences; Parent Attitudes; Severity (of Disability); Factor Structure; Test Validity Psychometry; Psychometrie; Psychomotorische Aktivität; Physical handicap; Körperbehinderung; Interpersonale Kompetenz; Alltagsfertigkeit; Social behaviour; Soziales Verhalten; Gesundheitszustand; Test content; Testaufgabe; Item-Response-Theorie; Age; Difference; Age difference; Altersunterschied; Elternverhalten; Schweregrad; Faktorenstruktur; Testvalidität |
Abstract | Rett syndrome (RTT) is a neurodevelopmental disorder that primarily affects females. Recent work indicates the potential for disease modifying therapies. However, there remains a need to develop outcome measures for use in clinical trials. Using data from a natural history study (n = 1,075), we examined the factor structure, internal consistency, and validity of the clinician-reported Motor Behavior Assessment scale (MBA). The analysis resulted in a five-factor model: (1) motor dysfunction, (2) functional skills, (3) social skills, (4) aberrant behavior, and (5) respiratory behaviors. Item Response Theory (IRT) analyses demonstrated that all items had acceptable discrimination. The revised MBA subscales showed a positive relationship with parent reported items, age, and a commonly used measure of clinical severity in RTT, and mutation type. Further work is needed to evaluate this measure longitudinally and to add items related to the RTT phenotype. (As Provided). |
Anmerkungen | American Association on Intellectual and Developmental Disabilities. P.O. Box 1897, Lawrence, KS 66044-1897. Tel: 785-843-1235; Fax: 785-843-1274; e-mail: AJMR@allenpress.com; Web site: https://meridian.allenpress.com/aaidd |
Erfasst von | ERIC (Education Resources Information Center), Washington, DC |
Update | 2024/1/01 |