Distinct attentional and executive profiles in Neurofibromatosis type 1: is there difference with primary attention deficit-hyperactivity disorder? - Université de Picardie Jules Verne Accéder directement au contenu
Article Dans Une Revue European Journal of Paediatric Neurology Année : 2024

Distinct attentional and executive profiles in Neurofibromatosis type 1: is there difference with primary attention deficit-hyperactivity disorder?

Résumé

Purpose Attentional and executive dysfunctions are the most frequent cognitive disorders in neurofibromatosis type 1 (NF1), with a high prevalence of attention deficit-hyperactivity disorder (ADHD). We (i) compared attentional profiles between NF1 children with and without ADHD and children with primary ADHD criteria and (ii) investigated the possible relationship between attentional disorders and "unidentified bright objects" (UBOs) in NF1. Methods This retrospective study included 47 NF1 children, 25 with ADHD criteria (NF1+adhd group), matched for age, sex, and cognitive level with 47 children with primary ADHD (ADHD group). We collected computer task (sustained-attention, visuomotor-decision, inhibition, and cognitive-flexibility tasks) scores normalized for age and sex, and brain magnetic resonance imaging data. Results (i) Working memory was impaired in all groups. (ii) Omissions (p<0.002) and response-time variability (p<0.05) in sustained-attention and visuomotor-decision tasks and errors (p<0.02) in the cognitive-flexibility task were lower for the NFI+adhd and ADHD groups than for the NF1-no-adhd group. (iii) The NF1+adhd group had slower response times (p<0.02) for inhibition and visuomotor-decision tasks than the other groups. (iv) We found no relevant association between cognitive performance and UBOs. Conclusions NF1 children with ADHD have an attentional and executive functions deficit profile similar to that of children with primary ADHD, but with a slower response-time, increasing learning difficulties. The atypical connectivity of fronto-striatal pathways, poorer dopamine homeostasis, and increased GABA inhibition observed in NF1 renders vulnerable the development of the widely distributed neural networks that support attentional, working-memory, and executive functions.
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Dates et versions

hal-04602985 , version 1 (06-06-2024)

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Citer

Laura Routier, Laurent Querné, Cécile Fontaine, Anne-Gaëlle Le Moing, Patrick Berquin. Distinct attentional and executive profiles in Neurofibromatosis type 1: is there difference with primary attention deficit-hyperactivity disorder?. European Journal of Paediatric Neurology, 2024, ⟨10.1016/j.ejpn.2024.05.016⟩. ⟨hal-04602985⟩

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