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唐氏综合征和威廉姆斯综合征的空间探索和导航。

Spatial exploration and navigation in Down syndrome and Williams syndrome.

机构信息

Univ. Lille, ULR 4072 - PSITEC - Psychologie: Interactions Temps Émotions Cognition, Lille, France.

Department of Psychology, University of Sheffield, UK.

出版信息

Cortex. 2024 Sep;178:32-50. doi: 10.1016/j.cortex.2024.06.004. Epub 2024 Jun 24.

DOI:10.1016/j.cortex.2024.06.004
PMID:38964151
Abstract

We know little about the ability to explore and navigate large-scale space for people with intellectual disability (ID). In this cross-syndrome study, individuals with Down syndrome (DS), individuals with Williams syndrome (WS) and typically developing children (TD; aged 5-11 years) explored virtual environments with the goal of learning where everything was within the environment (Experiment 1) or to find six stars (Experiment 2). There was little difference between the WS and DS groups when the goal was simply to learn about the environment with no specific destination to be reached (Experiment 1); both groups performed at a level akin to a subset of TD children of a similar level of non-verbal ability. The difference became evident when the goal of the task was to locate targets in the environment (Experiment 2). The DS group showed the weakest performance, performing at or below the level of a subset of TD children at a similar level of non-verbal ability, whilst the WS group performed at the level of the TD subset group. The DS, WS and TD group also demonstrated different patterns of exploration behavior. Exploration behaviour in DS was weak and did not improve across trials. In WS, exploration behavior changed across trials but was atypical (the number of revisits increased with repeated trials). Moreover, transdiagnostic individual difference analysis (Latent Profile Analysis) revealed five profiles of exploration and navigation variables, none of which were uniquely specific to DS or to WS. Only the most extreme profile of very poor navigators was specific to participants with DS and WS. Interestingly, all other profiles contained at least one individual with DS and at least one individual with WS. This highlights the importance of investigating heterogeneity in the performance of individuals with intellectual disability and the usefulness of a data-driven transdiagnostic approach to identifying behavioral profiles.

摘要

我们对智力障碍(ID)患者探索和导航大规模空间的能力知之甚少。在这项跨综合征研究中,唐氏综合征(DS)患者、威廉姆斯综合征(WS)患者和典型发育儿童(TD;年龄 5-11 岁)探索了虚拟环境,目标是了解环境中的所有内容(实验 1)或找到六颗星(实验 2)。当目标只是了解环境而无需到达特定目的地时,WS 和 DS 组之间几乎没有差异(实验 1);两组的表现都与具有相似非言语能力水平的 TD 儿童子集相当。当任务的目标是在环境中定位目标时(实验 2),差异变得明显。DS 组表现最差,表现水平与具有相似非言语能力水平的 TD 儿童子集相当,而 WS 组的表现与 TD 子集组相当。DS、WS 和 TD 组还表现出不同的探索行为模式。DS 组的探索行为较弱,且在试验中没有改善。在 WS 中,探索行为在试验中发生变化,但不典型(随着重复试验,回访次数增加)。此外,跨诊断个体差异分析(潜在剖面分析)揭示了探索和导航变量的五个剖面,没有一个是 DS 或 WS 特有的。只有非常差的导航者的最极端剖面是 DS 和 WS 参与者特有的。有趣的是,所有其他剖面都至少包含一个 DS 患者和至少一个 WS 患者。这突出表明研究智力障碍个体表现的异质性的重要性,以及采用数据驱动的跨诊断方法识别行为剖面的有用性。

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