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

Details

• Alternative Title:
  J Autism Dev Disord
• Personal Author:
  Kaufmann, Walter E. ; Raspa, Melissa ; Bann, Carla M. ; Gable, Julia M. ; Harris, Holly K. ; Budimirovic, Dejan B. ; Lozano, Reymundo
• Description:
  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.
• Subjects:
  Autism Spectrum Disorder Child Cross-Sectional Studies Female Fragile X Syndrome Humans Latent Class Analysis Male Social Behavior
• Keywords:
  Autism Symptomatology Fragile X Syndrome Intellectual Disability Latent Class Analysis Maladaptive Behavior Phenotypes
• Source:
  J Autism Dev Disord. 54(2):725-737
• Pubmed ID:
  36441429
• Pubmed Central ID:
  PMC10258834
• Document Type:
  Journal Article
• Funding:
  U01DD001189/ACL/ACL HHSUnited States/ ; U01 DD000231/DD/NCBDD CDC HHSUnited States/ ; U19 DD000753/DD/NCBDD CDC HHSUnited States/ ; #U19DD000753/DD/NCBDD CDC HHSUnited States/ ; K01 NS126736/NS/NINDS NIH HHSUnited States/ ; contract 75D30120F09737/DD/NCBDD CDC HHSUnited States/ ; # U01DD001189/DD/NCBDD CDC HHSUnited States/ ; U01 DD001189/DD/NCBDD CDC HHSUnited States/ ; U01DD000231/DD/NCBDD CDC HHSUnited States/
• Volume:
  54
• Issue:
  2
• Collection(s):
  CDC Public Access
• Main Document Checksum:
  urn:sha-512:0c0f246419031860ff53f9dcc524330bef779d390b5045f9419807bb8b5e3e23a2a3629f7ab8eeb9f2317ce4bf2f564e2824e67d9dedf98f224216507f950efe
• Download URL:
  https://stacks.cdc.gov/view/cdc/129857/cdc_129857_DS1.pdf
• File Type:
  [PDF - 750.58 KB ]