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

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

Walter E Kaufmann, Melissa Raspa, Carla M Bann, Julia M Gable, Holly K Harris, Dejan B Budimirovic, Reymundo Lozano, FORWARD Consortium
Author Information
  1. Walter E Kaufmann: Department of Human Genetics, Emory University School of Medicine, 615 Michael Street, Atlanta, GA, 30322, USA. walter.e.kaufmann@emory.edu. ORCID
  2. Melissa Raspa: RTI International, 3040 Cornwallis Road, Research Triangle Park, NC, 27709, USA.
  3. Carla M Bann: RTI International, 3040 Cornwallis Road, Research Triangle Park, NC, 27709, USA.
  4. Julia M Gable: RTI International, 3040 Cornwallis Road, Research Triangle Park, NC, 27709, USA.
  5. Holly K Harris: Texas Children's Hospital, 8080 North Stadium Drive, Houston, TX, 77054, USA.
  6. Dejan B Budimirovic: Kennedy Krieger Institute, 1741 Ashland Avenue, Baltimore, MD, 21205, USA.
  7. Reymundo Lozano: Icahn School of Medicine at Mount Sinai, 1 Gustave L. Levy Place, New York, NY, 10029, USA.
PMID: 36441429 DOI: 10.1007/s10803-022-05821-7

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

Keywords

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

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Grants

  1. U01DD001189/ACL HHS
  2. U01 DD000231/NCBDD CDC HHS
  3. U19 DD000753/NCBDD CDC HHS
  4. #U19DD000753/NCBDD CDC HHS
  5. K01 NS126736/NINDS NIH HHS
  6. contract 75D30120F09737/NCBDD CDC HHS
  7. # U01DD001189/NCBDD CDC HHS
  8. U01 DD001189/NCBDD CDC HHS
  9. U01DD000231/NCBDD CDC HHS

MeSH Term

Male
Female
Child
Humans
Fragile X Syndrome
Autism Spectrum Disorder
Latent Class Analysis
Cross-Sectional Studies
Social Behavior
Journal Article
Article has an altmetric score of 3

Word Cloud

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