Latent Class Analysis Identifies Distinctive Behavioral Subtypes in Children with Fragile X Syndrome.

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 natur...

Descripción completa

Detalles Bibliográficos
Publicado en:Journal of Autism & Developmental Disorders Vol. 54; no. 2; pp. 725 - 738
Autores principales: 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
Formato: research tables/charts Journal Article
Publicado: Springer Nature Feb2024
Acceso en línea:Ver este registro en EBSCOhost
Descripción
Sumario: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.