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Executive functions in preschool-aged children with neurofibromatosis type 1: Value for early assessment
Marie-Laure Beaussart-Corbat1, Sébastien Barbarot2,3, Denis Farges4
1Laboratory of Psychology, LPPL EA4638, University of Angers, Angers, France.
Insights
Children with neurofibromatosis type 1 (NF1) show early executive functions (EFs) impairment. Performance tests and parent/teacher ratings reveal significant deficits, highlighting the need for early assessment.
Area of Science:
- Neuroscience
- Pediatrics
- Genetics
Background:
- Executive functions (EFs) impairment is a known vulnerability in neurofibromatosis type 1 (NF1).
- Limited data exists on EFs in preschool-aged children with NF1.
Purpose of the Study:
- To assess executive functions in preschool children with NF1.
- To compare EFs in NF1 children with a healthy control group.
- To investigate the utility of performance-based tests and parent/teacher questionnaires.
Main Methods:
- Evaluated 33 NF1 children (3-5 years) using 7 experimental EF tasks.
- Administered BRIEF-P questionnaires to parents and teachers of NF1 children.
- Compared NF1 children's performance to 52 age, gender, and socio-culturally matched healthy controls.
Main Results:
- NF1 children scored significantly lower on 5 of 7 EF tasks compared to controls.
- Parents and teachers reported significantly higher levels of EF concerns for NF1 children.
- Weak correlations were observed between performance-based tests and questionnaire ratings.
Conclusions:
- Results support early executive dysfunction in children with NF1.
- Early and systematic assessment of EFs in NF1 is recommended.
- Performance tests and questionnaires are complementary for investigating early EFs dysfunction in NF1.
Abstract:
Introduction: Executive functions (EFs) impairment is common in children with neurofibromatosis type 1 (NF1), and could be a significant vulnerability associated with this medical disorder. However, we still know little about EFs in preschool NF1. Our study assessed EFs in NF1 children using performance-based tests and daily life questionnaires, which combined the views of parents and teachers.Method: Seven classic experimental tasks were used to evaluate EFs in 33 NF1 children aged 3 to 5 years old, and BRIEF-P questionnaires were completed by their parents and teachers. These children's performance was compared with a control group of 52 healthy children matched in age, gender and socio-cultural status.Results: NF1 children have significantly lower scores for 5 out of 7 executive tasks than control children and significantly higher levels of EF concerns in the parent and teacher BRIEF-P ratings. The correlations between performance-based tests and questionnaires are weak.Conclusions: Our results support an early executive dysfunction in NF1 children and call for early and systematic assessment of EFs. Both performance-based tests and questionnaires are complementary tools to investigate early EFs dysfunction in children with NF1.
