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Adaptive Behavior and Executive Functioning in Children with Neurofibromatosis Type 1 Using a Mixed Design
Danielle M Glad1, Christina L Casnar2, Brianna D Yund1
1Department of Psychology, University of Wisconsin-Milwaukee, Milwaukee, WI.
Insights
Children with neurofibromatosis type 1 (NF1) show declining adaptive skills over time, with poorer outcomes in school-age years. Executive functioning impacts these abilities longitudinally.
Area of Science:
- Neuroscience
- Developmental Psychology
- Genetics
Background:
- Neurofibromatosis type 1 (NF1) is a genetic disorder impacting development.
- Children with NF1 often exhibit challenges in adaptive functioning compared to peers.
- Longitudinal data on adaptive behavior in NF1 is scarce.
Purpose of the Study:
- To investigate the longitudinal trajectory of parent-reported adaptive behavior in children with NF1.
- To explore the relationship between adaptive behavior and executive functioning in NF1.
- To characterize changes in adaptive skills from early childhood to school age.
Main Methods:
- Longitudinal assessment of adaptive functioning using the Scales of Independent Behavior-Revised.
- Evaluation of executive functioning with the Behavior Rating Inventory of Executive Function (BRIEF).
- Participants included children with NF1 assessed in early childhood and/or school age.
Main Results:
- Adaptive behavior in early childhood correlated with school-age adaptive behavior, though social/communication skills varied.
- Children with NF1 showed poorer adaptive functioning at school age compared to early childhood.
- Difficulties in overall adaptive behavior and motor skills increased over time.
- Executive functioning demonstrated cross-sectional associations and longitudinal predictive value for adaptive behavior.
Conclusions:
- This study provides crucial longitudinal insights into adaptive behavior patterns in NF1.
- Findings highlight the evolving challenges in adaptive functioning and the role of executive function.
- Results inform interventions aimed at supporting children with NF1.
Objective:
Children with neurofibromatosis type 1 (NF1) demonstrate poorer adaptive functioning compared with same-aged peers; however, there is limited research about the longitudinal pattern of adaptive behavior. The aim of this investigation was to examine parent-reported adaptive behavior of children with NF1 longitudinally beginning in early childhood and to examine relations with executive functioning.
Method:
Children with NF1 were assessed during early childhood (n = 59; aged 3-7; mean = 4.8, SD = 1.42) or school age (n = 39; aged 9-13; mean = 10.85, SD = 1.58), and a subset was seen at both time points (n = 26). The Scales of Independent Behavior-Revised was used to assess adaptive functioning, and the Behavior Rating Inventory of Executive Function-Preschool Version/Behavior Rating Inventory of Executive Function was used to evaluate everyday executive functioning.
Results:
Adaptive behavior in early childhood was significantly correlated with adaptive behavior at school age (with the exception of social interaction and communication skills) and was significantly poorer at school age. The frequency of difficulties increased over time for overall adaptive behavior and motor skills. Executive functioning was related to adaptive behavior cross-sectionally within early childhood and at school age and showed longitudinal predictive value over time.
Conclusion:
This research contributes to the limited NF1 adaptive behavior literature by characterizing the longitudinal pattern of adaptive behavior and relations with executive abilities.
