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.

中文翻译：

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基于反馈的注意力缺陷/多动障碍和 1 型神经纤维瘤病的时间学习

目的：1 型神经纤维瘤病 (NF1) 患者经常表现出类似于注意力缺陷/多动障碍 (ADHD) 的症状。重要的是，这些疾病的特点是多巴胺能系统的明显变化，多巴胺能系统在计时表现和基于反馈的计时表现调整中起着重要作用。在跨诊断方法中，我们检查了 NF1 和 ADHD 在多大程度上显示出不同或可比较的计时性能和基于反馈的计时调整。方法：我们检查了健康对照儿童 (HC) 的时间估计和学习过程，主要患有 ADHD 的儿童注意力不集中的症状和 NF1 患者使用基于反馈的时间估计范式。结果：与两个患者组相比，健康对照始终更接近正确的时间窗口，他们的反应时间变化较小，并且随着时间的推移显示出完整的基于学习的调整。患者组在及时响应的数量方面没有差异。在 ADHD 患者中，随着时间的推移，表现相当不稳定。NF1 患者在整个任务中没有观察到表现变化。结论：患有 ADHD 和 NF1 的儿童在基于反馈学习的时间估计过程调整方面存在差异。ADHD 的特点是学习过程中的行为波动。这些可能与多巴胺能系统的低效率有关。NF1 的特点是反馈学习障碍，这可能是由于除了多巴胺合成缺陷外还发生了各种神经递质改变。