Scientific Institute, IRCCS E. Medea, Neuropsychiatry and Neurorehabilitation Unit, Bosisio Parini, Lecco, Italy.
PhD Program in Neural and Cognitive Sciences, Department of Life Sciences, University of Trieste, Trieste, Italy.
Cerebellum. 2024 Apr;23(2):579-588. doi: 10.1007/s12311-023-01580-y. Epub 2023 Jun 23.
The neuropsychological characteristics of the cerebellar cognitive affective syndrome (CCAS) in congenital, non-progressive malformations of the cerebellum have been scarcely investigated, and even less is known for Joubert syndrome (JS), an inherited, non-progressive cerebellar ataxia characterized by the so-called molar tooth sign. The few studies on this topic reported inconsistent results about intellectual functioning and specific neuropsychological impairments. The aim of this research is to examine the neuropsychological profile of JS compared to other congenital cerebellar malformations (CM), considering individual variability of intellectual quotient (IQ) in the two groups. Fourteen patients with JS and 15 patients with CM aged 6-25 years were tested through a comprehensive, standardized neuropsychological battery. Their scores in the neuropsychological domains were inspected through descriptive analysis and compared by mean of MANOVA and ANOVA models, then replicated inserting IQ as covariate. The two groups showed a largely overlapping neuropsychological profile, consistent with CCAS. However, the JS group showed worse performance in visual-spatial memory compared to CM patients, although this difference was mitigated when considering IQ. These findings highlight a divergence between JS and other CM in visual-spatial memory, which might suggest a critical role of the cerebellum in recalling task-relevant memories and might inform rehabilitative interventions.
小脑认知情感综合征(CCAS)在先天性、非进行性小脑畸形中的神经心理学特征研究甚少,而对于杰伯特综合征(JS)更是知之甚少,JS 是一种遗传性、非进行性小脑共济失调,其特征是所谓的磨牙征。关于这一主题的少数研究报告了关于智力功能和特定神经心理学损伤的不一致结果。本研究旨在比较 JS 与其他先天性小脑畸形(CM)的神经心理学特征,同时考虑两组个体智商(IQ)的可变性。14 名 JS 患者和 15 名 CM 患者年龄在 6-25 岁之间,通过全面、标准化的神经心理学测试进行了测试。通过描述性分析检查他们在神经心理学领域的得分,并通过 MANOVA 和 ANOVA 模型进行比较,然后通过插入 IQ 作为协变量进行复制。这两组表现出与 CCAS 非常相似的神经心理学特征。然而,与 CM 患者相比,JS 组在视觉空间记忆方面的表现更差,尽管当考虑 IQ 时,这种差异有所减轻。这些发现突出了 JS 和其他 CM 在视觉空间记忆方面的差异,这可能表明小脑在回忆与任务相关的记忆方面起着关键作用,并为康复干预提供了信息。