학술논문
Latent Class Analysis Identifies Distinctive Behavioral Subtypes in Children with Fragile X Syndrome.
Document Type
Article
Author
Kaufmann, Walter E.; Raspa, Melissa; Bann, Carla M.; Gable, Julia M.; Harris, Holly K.; Budimirovic, Dejan B.; Lozano, Reymundo; Berry-Kravis, Elizabeth; Velinov, Milen; Talboy, Amy L; Sherman, Stephanie L; Kaufmann, Walter E; Schuster, Marcy; Tartaglia, Nicole; Filipink, Robyn A; Budimirovic, Dejan B; Barbouth, Deborah; Lightbody, Amy; Reiss, Allan; Delahunty, Carol M
Source
Subject
*STRUCTURAL equation modeling
*CROSS-sectional method
*SENSORY disorders
*CHILD behavior
*NEUROLOGIC manifestations of general diseases
*SLEEP disorders
*FRAGILE X syndrome
*RESEARCH funding
*AUTISM
*QUESTIONNAIRES
*INTELLECTUAL disabilities
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Language
ISSN
0162-3257
Abstract
Fragile X syndrome (FXS) is characterized by variable neurobehavioral abnormalities, which leads to difficulties in developing and evaluating treatments and in determining accurate prognosis. We employed a pediatric cross-sectional sample (1,072 males, 338 females) from FORWARD, a clinic-based natural history study, to identify behavioral subtypes by latent class analysis. Input included co-occurring behavioral conditions, sleep and sensory problems, autistic behavior scales (SCQ, SRS-2), and the Aberrant Behavior Checklist revised for FXS (ABCFX). A 5-class solution yielded the most clinically meaningful, pharmacotherapy independent behavioral groups with distinctive SCQ, SRS-2, and ABCFX profiles, and adequate non-overlap (≥ 71%): "Mild" (31%), "Moderate without Social Impairment" (32%), "Moderate with Social Impairment" (7%), "Moderate with Disruptive Behavior" (20%), and "Severe" (9%). Our findings support FXS subtyping, for improving clinical management and therapeutic development. [ABSTRACT FROM AUTHOR]