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 - Publisher Copyright:
© 2022, The Author(s), under exclusive licence to Springer Science+Business Media, LLC, part of Springer Nature.
PY - 2024/2
Y1 - 2024/2
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
VL - 54
SP - 725
EP - 737
JO - Journal of Autism and Developmental Disorders
JF - Journal of Autism and Developmental Disorders
IS - 2
ER -