Project: Research project

Project Details


DESCRIPTION: This research will investigate the developmental onset and age
related changes in the cognitive, behavioral, social and neuroanatomical
correlates of the neuropsychiatric phenotype associated with the fragile X
condition in young males. Molecular correlates of these changes will also
be investigated. The study uses a multimethod, longitudinal sequential
design for a 2 year evaluation of development in 48 fragile X males compared
to 48 age and IQ matched males with developmental language delay (DLD) of
unknown etiology. All subjects will be between 36 and 71 months old at the
time of enrollment. Two follow up assessments of the child's cognitive,
behavioral, and social functioning will occur at 12 and 24 month intervals.
Neuroanatomical imaging (MRI) will occur with all subjects from both groups
at the initial evaluation and a random subset of 24 subjects from each group
at the 24 month follow up evaluation. A second contrast group of 30 young
males with Duchenne muscular dystrophy (DMD; an X linked genetic disorder),
enrolled at 60 to 95 months, will be used as a single evaluation outcome
comparison group for the fragile X males. Males with DMD will be
individually matched on IQ, developmental age, race and SES with a fragile X
male. Developmental areas specifically addressed include: 1) cognition; 2)
communication; 3) behaviors involving deficits in self regulation (e.g.,
stereotypies, hyperactivity); 4) social interaction; and 5) brain structures
(e.g., caudate, hippocampus, cerebellar vermis). It is hypothesized that
cognitive, communication, behavioral self regulation, and social interaction
deficits will differ among the groups, will increase over time in the
fragile X group but not in the comparison DLD group. It is expected that
age related changes in neuroanatomical structures indicating ongoing,
detrimental effects on the postnatal central nervous system of fragile X but
not control males will be observed. This study will contribute to
understanding the associated effects of a genetically determined
developmental disability during an age period in which a high degree of
developmental change is expected. It will also help identify the multiple
biological and environmental influences on development in young,
developmentally delayed children.
Effective start/end date9/1/978/31/98


  • Eunice Kennedy Shriver National Institute of Child Health and Human Development

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