OBJECTIVE: A controlled clinical study was designed to identify the neurobehavioral profile that is specific to males with fragile X syndrome. DESIGN: Thirty-one males with fragile X syndrome and 30 age and IQ-matched male controls were evaluated with instruments that assess multiple domains of adaptive functioning and problem behaviors. The Vineland Adaptive Behavior Scales and the Aberrant Behavior Checklist were selected for their dimensional scaling of behavioral ratings. RESULTS: Parent and Teacher versions of the Aberrant Behavior Checklist demonstrated a profile of behaviors specific to males with fragile X syndrome characterized by significantly higher levels of hyperactivity, stereotypic movements, and unusual speech. The Vineland Adaptive Behavior Scales revealed no fragile X-specific profile of adaptive skills development. CONCLUSIONS: The distinct pattern of aberrant behavior observed among males with fragile X emphasizes the importance of drawing subtype distinctions within the classification of individuals with mental retardation on the basis of underlying etiology. For clinical research, specifying the fragile X phenotype is a vital part in the effort to elucidate the neurodevelopmental pathways of normal behavior and psychopathology. Understanding the fragile X symptom pattern is essential for designing symptom-specific treatment interventions, as well as for research into the efficacy of interventions strategies.
OBJECTIVE: A controlled clinical study was designed to identify the neurobehavioral profile that is specific to males with fragile X syndrome. DESIGN: Thirty-one males with fragile X syndrome and 30 age and IQ-matched male controls were evaluated with instruments that assess multiple domains of adaptive functioning and problem behaviors. The Vineland Adaptive Behavior Scales and the Aberrant Behavior Checklist were selected for their dimensional scaling of behavioral ratings. RESULTS: Parent and Teacher versions of the Aberrant Behavior Checklist demonstrated a profile of behaviors specific to males with fragile X syndrome characterized by significantly higher levels of hyperactivity, stereotypic movements, and unusual speech. The Vineland Adaptive Behavior Scales revealed no fragile X-specific profile of adaptive skills development. CONCLUSIONS: The distinct pattern of aberrant behavior observed among males with fragile X emphasizes the importance of drawing subtype distinctions within the classification of individuals with mental retardation on the basis of underlying etiology. For clinical research, specifying the fragile X phenotype is a vital part in the effort to elucidate the neurodevelopmental pathways of normal behavior and psychopathology. Understanding the fragile X symptom pattern is essential for designing symptom-specific treatment interventions, as well as for research into the efficacy of interventions strategies.
Authors: Wenzhong William Min; Christopher J Yuskaitis; Qijiang Yan; Christopher Sikorski; Shengqiang Chen; Richard S Jope; Robert P Bauchwitz Journal: Neuropharmacology Date: 2008-10-14 Impact factor: 5.250
Authors: Gisela M Sandoval; Sehoon Shim; David S Hong; Amy S Garrett; Eve-Marie Quintin; Matthew J Marzelli; Swetapadma Patnaik; Amy A Lightbody; Allan L Reiss Journal: J Psychiatr Res Date: 2018-10-25 Impact factor: 4.791
Authors: Elizabeth M Berry-Kravis; Lothar Lindemann; Aia E Jønch; George Apostol; Mark F Bear; Randall L Carpenter; Jacqueline N Crawley; Aurore Curie; Vincent Des Portes; Farah Hossain; Fabrizio Gasparini; Baltazar Gomez-Mancilla; David Hessl; Eva Loth; Sebastian H Scharf; Paul P Wang; Florian Von Raison; Randi Hagerman; Will Spooren; Sébastien Jacquemont Journal: Nat Rev Drug Discov Date: 2017-12-08 Impact factor: 84.694
Authors: R Nick Hernandez; Rachel L Feinberg; Rebecca Vaurio; Natalie M Passanante; Richard E Thompson; Walter E Kaufmann Journal: Am J Med Genet A Date: 2009-06 Impact factor: 2.802