This proposed research addresses fundamental questions about the effects of parenting on the development and behavior of adolescents with fragile X syndrome (FXS). It examines the transition to this life stage and investigates variables that may have a significant and continuing influence on adolescent behavior. As such, it takes a natural history approach to the study of FXS during this critical stage. We investigate the continuing impact of early parenting as well as parenting during adolescence on the development and behavior of 55 mother-child dyads that we have studied since early childhood. Our research to date has revealed the significant role that maternal responsivity, a key element of parent-child interaction, plays in the development of children with FXS. Our findings have underscored the fact that FXS is not just the product of innate biology, but is attributable to the dynamic and cumulative interaction of biology, behavior, and environment over time. Moreover, because it is a single gene disorder, FXS represents a rare opportunity to study the forces that shape adolescent development through the use of a number of fundamentally important child and parent variables. These variables include language, cognition, adaptive, social and problem behavior, autism symptoms, gender, and parenting provided by carrier mothers with the FXS premutation. This project will be led by Drs. Steven F. Warren and Nancy Brady at the University of Kansas Institute for Life Span Studies in collaboration with Dr. Elizabeth Berry-Kravis at Rush University Medical Center in Chicago, Dr. Leann Smith at the University of Wisconsin Waisman Center, and Dr. Kandace Fleming of KU. Building on 10 years of prior work with a fully intact longitudinal sample of 55 biological mothers and their full mutation children who are now adolescents, we propose to explore the impact of key behavioral variables on development and behavior through four aims.
The first aim i s to examine trajectories across all domains of child and mother behavior from early childhood through adolescence, and the relationship of gender, autism status, and child molecular measures to these trajectories.
The second aim i s to determine the effects of parenting before and during adolescence on a set of key adolescent outcomes.
The third aim i s to examine the effects of child (i.e., severity of behavior problems and pubertal status) and maternal (i.e., IQ, depression, maternal FXS premutation size and activation ratio, executive functioning, and family income) variables on parenting and mother-child relationship quality in adolescence.
The fourth aim i s to examine sequential relationships between specific adolescent behaviors and maternal behaviors during interactions. Reliable scientific knowledge of the dynamic interplay of children with FXS and their parenting experiences before and during the critical period of adolescence has important implications for the scientific understanding and clinical treatment of individuals with FXS and related conditions.

Public Health Relevance

The proposed research addresses fundamental questions about the effects of parenting on the development and behavior of adolescents with fragile X syndrome (FXS). It will continue a 10 year longitudinal study of 55 mother-child dyads that began during early childhood. The knowledge generated by this study will have important implications for the scientific understanding and clinical treatment of individuals with FXS and related conditions.

Agency
National Institute of Health (NIH)
Institute
Eunice Kennedy Shriver National Institute of Child Health & Human Development (NICHD)
Type
Research Project (R01)
Project #
5R01HD084563-02
Application #
9250202
Study Section
Child Psychopathology and Developmental Disabilities Study Section (CPDD)
Program Officer
King, Tracy
Project Start
2016-04-01
Project End
2021-01-31
Budget Start
2017-02-01
Budget End
2018-01-31
Support Year
2
Fiscal Year
2017
Total Cost
$479,250
Indirect Cost
$135,513
Name
University of Kansas Lawrence
Department
Type
Organized Research Units
DUNS #
076248616
City
Lawrence
State
KS
Country
United States
Zip Code
66045
Bredin-Oja, Shelley L; Fielding, Heather; Fleming, Kandace K et al. (2018) Clinician vs. Machine: Estimating Vocalizations Rates in Young Children With Developmental Disorders. Am J Speech Lang Pathol 27:1066-1072
Komesidou, Rouzana; Brady, Nancy C; Fleming, Kandace et al. (2017) Growth of Expressive Syntax in Children With Fragile X Syndrome. J Speech Lang Hear Res 60:422-434
Warren, Steven F; Brady, Nancy; Fleming, Kandace K et al. (2017) The Longitudinal Effects of Parenting on Adaptive Behavior in Children with Fragile X Syndrome. J Autism Dev Disord 47:768-784
Hahn, Laura J; Brady, Nancy C; Fleming, Kandace K et al. (2016) Joint Engagement and Early Language in Young Children With Fragile X Syndrome. J Speech Lang Hear Res 59:1087-1098