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

© 2022. The Author(s), under exclusive licence to Springer Science+Business Media, LLC, part of Springer Nature..

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.

Medienart:

E-Artikel

Erscheinungsjahr:

2024

Erschienen:

2024

Enthalten in:

Zur Gesamtaufnahme - volume:54

Enthalten in:

Journal of autism and developmental disorders - 54(2024), 2 vom: 23. Feb., Seite 725-737

Sprache:

Englisch

Beteiligte Personen:

Kaufmann, Walter E [VerfasserIn]
Raspa, Melissa [VerfasserIn]
Bann, Carla M [VerfasserIn]
Gable, Julia M [VerfasserIn]
Harris, Holly K [VerfasserIn]
Budimirovic, Dejan B [VerfasserIn]
Lozano, Reymundo [VerfasserIn]
FORWARD Consortium [VerfasserIn]
Berry-Kravis, Elizabeth [Sonstige Person]
Velinov, Milen [Sonstige Person]
Talboy, Amy L [Sonstige Person]
Sherman, Stephanie L [Sonstige Person]
Kaufmann, Walter E [Sonstige Person]
Schuster, Marcy [Sonstige Person]
Tartaglia, Nicole [Sonstige Person]
Filipink, Robyn A [Sonstige Person]
Budimirovic, Dejan B [Sonstige Person]
Barbouth, Deborah [Sonstige Person]
Lightbody, Amy [Sonstige Person]
Reiss, Allan [Sonstige Person]
Delahunty, Carol M [Sonstige Person]
Hagerman, Randi J [Sonstige Person]
Hessl, David [Sonstige Person]
Erickson, Craig A [Sonstige Person]
Feldman, Gary [Sonstige Person]
Picker, Jonathan D [Sonstige Person]
Lachiewicz, Ave M [Sonstige Person]
Harris, Holly K [Sonstige Person]
Esler, Amy [Sonstige Person]
Frye, Richard E [Sonstige Person]
Evans, Patricia A [Sonstige Person]
Morris, Mary Ann [Sonstige Person]
Haas-Givler, Barbara A [Sonstige Person]
Gropman, Andrea L [Sonstige Person]
Uy, Ryan S [Sonstige Person]
Buchanan, Carrie [Sonstige Person]
Frazier, Jean A [Sonstige Person]
Morris, Stephanie M [Sonstige Person]

Links:

Volltext

Themen:

Autism symptomatology
Fragile X syndrome
Intellectual disability
Journal Article
Latent class analysis
Maladaptive behavior
Phenotypes

Anmerkungen:

Date Completed 29.01.2024

Date Revised 18.02.2024

published: Print-Electronic

Citation Status MEDLINE

doi:

10.1007/s10803-022-05821-7

funding:

Förderinstitution / Projekttitel:

PPN (Katalog-ID):

NLM34954316X