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

• Published in: Journal of autism and developmental disorders Vol. 54; no. 2; pp. 725 - 737
• Main Authors: Kaufmann, Walter E., Raspa, Melissa, Bann, Carla M., Gable, Julia M., Harris, Holly K., Budimirovic, Dejan B., Lozano, Reymundo
• Format: Journal Article
• Language: English
• Published: New York Springer US 01.02.2024; Springer Nature B.V
• Subjects: Anxiety; Attention deficit hyperactivity disorder; Autism; Autism Spectrum Disorder; Behavior; Behavioral Science and Psychology; Child; Child and School Psychology; Child development; Cluster analysis; Cognitive ability; Comorbidity; Consortia; Cross-Sectional Studies; Disruptive behaviour; DNA methylation; Down syndrome; Drug therapy; Female; Females; Fragile X Syndrome; Health care management; Humans; Intellectual disabilities; Latent Class Analysis; Male; Medical prognosis; Mutation; Natural history; Neurobehavioral functioning; Neurosciences; Original Paper; Pediatrics; Pharmacology; Psychology; Public Health; Quality of life; Sleep disorders; Social Behavior; Subtypes; Phenotypes; Intellectual disability; Maladaptive behavior; Fragile X syndrome; Autism symptomatology; Latent class analysis
• ISSN: 0162-3257; 1573-3432; 1573-3432
• DOI: 10.1007/s10803-022-05821-7

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 (ABC FX ). A 5-class solution yielded the most clinically meaningful, pharmacotherapy independent behavioral groups with distinctive SCQ, SRS-2, and ABC FX 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.