Response inhibition in Attention deficit disorder and neurofibromatosis type 1 – clinically similar, neurophysiologically different

Bluschke, Annet; Hagen, Maja von der; Papenhagen, Katharina; Roessner, Veit; Beste, Christian

doi:10.1038/srep43929

Cited by 11 publications

(15 citation statements)

References 55 publications

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“…Indeed, executive functioning is associated with a large cerebral network with a preponderant role of the prefrontal cortex and its subcortical connections. These data echo recent studies that observed some differences in neurophysiological processes between adolescents with NF1 and controls in a Go/No-Go task (Bluschke, Von der Hagen, Papenhagen, Roessner, & Beste, 2017b). These findings should encourage future neurophysiological studies to focus on EF studies which will enrich our knowledge of this topic.…”

Section: Discussionsupporting

confidence: 90%

Systematic Review and Meta-analysis of Executive Functions in Preschool and School-Age Children With Neurofibromatosis Type 1

Beaussart

Barbarot²,

Mauger

et al. 2018

J Int Neuropsychol Soc

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Objectives:Neurofibromatosis type 1 (NF1) is a genetic disorder in which the most frequent complication in children is learning disabilities. Over the past decade, growing arguments support the idea that executive dysfunction is a core deficit in children with NF1. However, some data remain inconsistent. The aim of this study was to determine the magnitude of impairment for each executive function (EF) and clarify the impact of methodological choices and participant’s characteristics on EFs.Methods:In this meta-analysis, 19 studies met the selection criteria and were included with data from a total of 805 children with NF1 and 667 controls. Based on the Diamond’s model (2013), EF measures were coded separately according to the following EF components: working memory, inhibitory control, cognitive flexibility, planning/problem solving. The review protocol was registered with PROSPERO (International prospective register of systematic reviews; CRD42017068808).Results:A significant executive dysfunction in children with NF1 is demonstrated. Subgroup analysis showed that the impairment varied as a function of the specific component of executive functioning. The effect size for working memory and planning/problem solving was moderate whereas it was small for inhibitory control and cognitive flexibility. Executive dysfunction seems to be greater with increasing age whereas assessment tool type, intellectual performance, attention deficit hyperactivity disorder and control group composition did not seem to affect EF results.Conclusions:EF deficits are a core feature in children with NF1 and an early identification of executive dysfunctions is essential to limit their impact on the quality of life. (JINS, 2018,24, 977–994)

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Section: Discussionsupporting

confidence: 90%

Systematic Review and Meta-analysis of Executive Functions in Preschool and School-Age Children With Neurofibromatosis Type 1

Beaussart

Barbarot²,

Mauger

et al. 2018

J Int Neuropsychol Soc

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“…Clinically, precisely this slower response speed may be the main contributor to the observed attentional problems and ADHD-like symptoms frequently reported in patients with NF1 ( Kayl and Moore, 2000 , Mautner et al, 2015 , North et al, 1997 , Pride et al, 2012 ). In fact, we found a similar pattern of reduced reaction times and somewhat altered neurophysiological processing mechanisms in patients with NF1 in a GoNogo task ( Bluschke et al, in press ). Here we could show that, although there are clear similarities on the clinical level, neurophysiological processes clearly differ between patients with NF1 and those with ADHD.…”

Section: Discussionsupporting

confidence: 61%

Conflict processing in juvenile patients with neurofibromatosis type 1 (NF1) and healthy controls – Two pathways to success

Bluschke

Hagen

Papenhagen

et al. 2017

NeuroImage: Clinical

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Neurofibromatosis Type 1 (NF1) is a monogenetic autosomal-dominant disorder with a broad spectrum of clinical symptoms and is commonly associated with cognitive deficits. Patients with NF1 frequently exhibit cognitive impairments like attention problems, working memory deficits and dysfunctional inhibitory control. The latter is also relevant for the resolution of cognitive conflicts. However, it is unclear how conflict monitoring processes are modulated in NF1. To examine this question in more detail, we used a system neurophysiological approach combining high-density ERP recordings with source localisation analyses in juvenile patients with NF1 and controls during a flanker task. Behaviourally, patients with NF1 perform significantly slower than controls. Specifically on trials with incompatible flanker-target pairings, however, the patients with NF1 made significantly fewer errors than healthy controls. Yet, importantly, this overall successful conflict resolution was reached via two different routes in the two groups. The healthy controls seem to arrive at a successful conflict monitoring performance through a developing conflict recognition via the N2 accompanied by a selectively enhanced N450 activation in the case of perceived flanker-target conflicts. The presumed dopamine deficiency in the patients with NF1 seems to result in a reduced ability to process conflicts via the N2. However, NF1 patients show an increased N450 irrespective of cognitive conflict. Activation differences in the orbitofrontal cortex (BA11) and anterior cingulate cortex (BA24) underlie these modulations. Taken together, juvenile patients with NF1 and juvenile healthy controls seem to accomplish conflict monitoring via two different cognitive neurophysiological pathways.

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“…On a neurobiological level, these deficits could be due to the deficient synthesis of dopamine on the one hand as well as due to other affected neurotransmitter systems relevant for learning on the other. Tying up with other data (Bluschke, von der Hagen, Papenhagen, Roessner, & Beste, 2017a, 2017b, the results show that despite the strong overlap in clinical phenotype and neuropsychological deficits in NF1 and ADHD, the underlying cognitive mechanisms are different. This is important to consider regarding pharmacological interventions in these diseases.…”

Section: Discussionsupporting

confidence: 51%

Feedback-Based Learning of Timing in Attention-Deficit/Hyperactivity Disorder and Neurofibromatosis Type 1

Prochnow

Bluschke

Novotna

et al. 2021

J Int Neuropsychol Soc

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Objective: Patients with Neurofibromatosis Type 1 (NF1) frequently display symptoms resembling those of Attention Deficit/Hyperactivity Disorder (ADHD). Importantly, these disorders are characterised by distinct changes in the dopaminergic system, which plays an important role in timing performance and feedback-based adjustments in timing performance. In a transdiagnostic approach, we examine how far NF1 and ADHD show distinct or comparable profiles of timing performance and feedback-based adjustments in timing. Method: We examined time estimation and learning processes in healthy control children (HC), children with ADHD with predominantly inattentive symptoms and those with NF1 using a feedback-based time estimation paradigm. Results: Healthy controls consistently responded closer to the correct time window than both patient groups, were less variable in their reaction times and displayed intact learning-based adjustments across time. The patient groups did not differ from each other regarding the number of in-time responses. In ADHD patients, the performance was rather unstable across time. No performance changes could be observed in patients with NF1 across the entire task. Conclusions: Children with ADHD and NF1 differ in feedback learning-based adjustments of time estimation processes. ADHD is characterised by behavioural fluctuations during the learning process. These are likely to be associated with inefficiencies in the dopaminergic system. NF1 is characterised by impairments of feedback learning which could be due to various neurotransmitter alterations occurring in addition to deficits in dopamine synthesis. Results show that despite the strong overlap in clinical phenotype and neuropsychological deficits between NF1 and ADHD, the underlying cognitive mechanisms are different.

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Response inhibition in Attention deficit disorder and neurofibromatosis type 1 – clinically similar, neurophysiologically different

Cited by 11 publications

References 55 publications

Systematic Review and Meta-analysis of Executive Functions in Preschool and School-Age Children With Neurofibromatosis Type 1

Systematic Review and Meta-analysis of Executive Functions in Preschool and School-Age Children With Neurofibromatosis Type 1

Conflict processing in juvenile patients with neurofibromatosis type 1 (NF1) and healthy controls – Two pathways to success

Feedback-Based Learning of Timing in Attention-Deficit/Hyperactivity Disorder and Neurofibromatosis Type 1

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