Journal of the American Academy of Child & Adolescent Psychiatry
ARTICLESGenetic and Environmental Influences on the Cognitive Outcomes of Children With Fragile X Syndrome
Section snippets
Subjects
Families were recruited from an existing fragile X registry, through advertisements in fragile X association newsletters and web sites, and through referrals from clinicians and families. To determine eligibility, families completed a telephone-screening interview covering basic demographic information, family history of fragile X, and their children's developmental histories. Subjects were excluded because of other known medical problems or signs of current illness. Confirmatory DNA testing
RESULTS
There were no significant differences between unaffected female siblings and unaffected male siblings on any of the IQ scales. Therefore, the unaffected male and female siblings were combined into one comparison group. A multivariate analysis of variance revealed an overall group effect for the three IQ scales: FSIQ (F3 = 324.35, p < .000), PIQ (F3 = 293.26, p < .000), and VIQ (F3 = 254.52, p < .000). Follow-up Tukey honestly significant difference pairwise tests revealed significant group
DISCUSSION
This study aimed to describe the cognitive profiles of girls and boys with fragile X and to investigate the potential contributions of both biological/genetic and environmental factors to the cognitive outcomes of these children. As predicted, girls with fragile X had somewhat higher cognitive abilities than did boys with fragile X and had relative strengths in verbal domains. Importantly, we also observed that both biological/genetic factors and the quality of the home environment contribute
REFERENCES (25)
- et al.
Rapid antibody test for fragile X syndrome
Lancet
(1995) - et al.
Specification of the neurobehavioral phenotype in males with fragile X syndrome
Pediatrics
(1995) - Bennetto L, Pennington BF (1996), The neuropsychology of fragile X syndrome. In: Fragile X Syndrome: Diagnosis,...
- et al.
Behavior and autonomic nervous system function assessed via heart period measures: the case of hyperarousal in boys with fragile X syndrome
Behav Res Methods Instrum Comput
(2000) Children's home environments, health, behavior, and intervention efforts: a review using the HOME inventory as a marker measure
Genet Soc Gen Psychol Monogr
(1993)- et al.
Home Observation for Measurement of the Environment
(1984) A theoretical analysis of the role of hyperarousal in the learning and behavior of fragile X males
Mental Retardation Dev Disabilities Res Rev
(1995)- et al.
Familial resemblance for specific cognitive abilities
Behav Genet
(1979) - Dyer-Friedman J, Hessl D, Glaser B, Kosaraju A, Reiss AL (2001), The Special Curriculum Opportunity Rating Scale...
- et al.
Trajectories and profiles of adaptive behavior in males with fragile X syndrome: multicenter studies
J Autism Dev Disord
(1996)
Longitudinal study of cognitive abilities and adaptive behavior levels in fragile X males: a prospective multicenter analysis
Am J Med Genet
A neurocognitive phenotype of young males and females with fragile X
Neuropsychiatry Genet
Cited by (0)
This work was supported by NIH grants MHO1142 and MH50047. Further support was received from the Packard Foundation and the Lynda and Scott Canel Fund for Fragile X Research. The authors thank Christine Blasey for statistical consultation.