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

FORWARD Consortium

Research output: Contribution to journalArticlepeer-review

2 Scopus citations

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.

Original languageEnglish (US)
Pages (from-to)725-737
Number of pages13
JournalJournal of Autism and Developmental Disorders
Volume54
Issue number2
DOIs
StatePublished - Feb 2024

Bibliographical note

Funding Information:
The present study was supported by cooperative agreements #U01DD000231, #U19DD000753, and # U01DD001189, and contract 75D30120F09737, funded by the Centers for Disease Control and Prevention (CDC). Its contents are solely the responsibility of the authors and do not necessarily represent the official views of the CDC or the Department of Health and Human Services.

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

Keywords

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

PubMed: MeSH publication types

  • Journal Article

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