title: Uncovering the evidence for behavioral interventions with individuals with fragile X syndrome: a systematic review.
abstract: Fragile X syndrome (FXS) is associated with a wide range of cognitive, emotional, and behavioral difficulties. Although there is considerable research on the behavioral phenotype of FXS, few empirical studies of behavioral interventions with this population have been identified. Through a hand search of 34 behavioral journals, we examined the evidence base for behavioral interventions with individuals with FXS and in light of the current state of knowledge regarding the FXS behavioral phenotype. Systematic review procedures were used to identify behavioral intervention studies that included at least one participant with FXS, extract and summarize the data on several relevant dimensions, and rate the methodological quality of the studies. Results revealed 31 intervention studies with a small number of participants with FXS. Overall, results suggest a behavioral approach to intervention with individuals with FXS shows promise. Future research focused on individuals with FXS will be necessary to continue to examine differences in response to intervention and interventions that specifically address phenotypic characteristics.
authors.affiliation: Department of Psychology, St John's University, Queens, NY 11439, United States. Electronic address: MoskowiL@stjohns.edu.
authors.forename: Lauren J
authors.initials: LJ
authors.lastname: Moskowitz
authors.affiliation: Queens College, The Graduate Center of the City University of New York, Queens, NY, United States.
authors.forename: Emily A
authors.initials: EA
authors.lastname: Jones
mesh_terms: D001521:Behavior Therapy / Q000379:methods*; D002659:Child Development Disorders, Pervasive / Q000534:rehabilitation; D055317:Evidence-Based Practice; D005600:Fragile X Syndrome / Q000523:psychology / Q000534:rehabilitation*; D006801:Humans; D008607:Intellectual Disability / Q000534:rehabilitation; D010641:Phenotype
keywords: Behavioral intervention; Fragile X syndrome; Systematic review