TY - JOUR
T1 - Latent Class Analysis Identifies Distinctive Behavioral Subtypes in Children with Fragile X Syndrome
AU - FORWARD Consortium
AU - Kaufmann, Walter E.
AU - Raspa, Melissa
AU - Bann, Carla M.
AU - Gable, Julia M.
AU - Harris, Holly K.
AU - Budimirovic, Dejan B.
AU - Lozano, Reymundo
AU - Berry-Kravis, Elizabeth
AU - Velinov, Milen
AU - Talboy, Amy L.
AU - Sherman, Stephanie L.
AU - Kaufmann, Walter E.
AU - Schuster, Marcy
AU - Tartaglia, Nicole
AU - Filipink, Robyn A.
AU - Budimirovic, Dejan B.
AU - Barbouth, Deborah
AU - Lightbody, Amy
AU - Reiss, Allan
AU - Delahunty, Carol M.
AU - Hagerman, Randi J.
AU - Hessl, David
AU - Erickson, Craig A.
AU - Feldman, Gary
AU - Picker, Jonathan D.
AU - Lachiewicz, Ave M.
AU - Harris, Holly K.
AU - Esler, Amy
AU - Frye, Richard E.
AU - Evans, Patricia A.
AU - Morris, Mary Ann
AU - Haas-Givler, Barbara A.
AU - Gropman, Andrea L.
AU - Uy, Ryan S.
AU - Buchanan, Carrie
AU - Frazier, Jean A.
AU - Morris, Stephanie M.
N1 - 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.
PY - 2022
Y1 - 2022
N2 - 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.
AB - 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.
KW - Autism symptomatology
KW - Fragile X syndrome
KW - Intellectual disability
KW - Latent class analysis
KW - Maladaptive behavior
KW - Phenotypes
UR - http://www.scopus.com/inward/record.url?scp=85142938089&partnerID=8YFLogxK
UR - http://www.scopus.com/inward/citedby.url?scp=85142938089&partnerID=8YFLogxK
U2 - 10.1007/s10803-022-05821-7
DO - 10.1007/s10803-022-05821-7
M3 - Article
C2 - 36441429
AN - SCOPUS:85142938089
SN - 0162-3257
JO - Journal of Autism and Developmental Disorders
JF - Journal of Autism and Developmental Disorders
ER -