Nieto Antonieta, Pérez-Flores Javier, Corral-Juan Marc, Matilla-Dueñas Antoni, Martínez-Burgallo Francisco, Montón Fernando
School of Psychology, Universidad de La Laguna , San Cristóbal de La Laguna , Spain.
Functional and Translational Neurogenetics Unit, Department of Neuroscience, Health Sciences Research Institute Germans Trias i Pujol (IGTP), Universitat Autònoma de Barcelona , Barcelona , Spain.
Neurocase. 2019 Oct;25(5):195-201. doi: 10.1080/13554794.2019.1655064. Epub 2019 Aug 19.
Autosomal recessive spinocerebellar ataxia type 10 (SCAR10) caused by a homozygous c.132dupA mutation in the anoctamin 10 gene is infrequent and little is known about its cognitive profile. Three siblings (1 male) with this mutation were assessed with a neuropsychological battery measuring multiple cognitive domains. The deficits observed in one patient were in executive functions whereas the other two patients showed deficits in practically all the functions. Cognitive impairment seems to be a characteristic of the SCAR10 produced by this mutation, with a range from mild impairment, especially involving prefrontal systems, to a severe cognitive impairment suggesting widespread cerebral involvement.
由anoctamin 10基因纯合c.132dupA突变引起的常染色体隐性遗传性10型脊髓小脑共济失调(SCAR10)较为罕见,人们对其认知特征知之甚少。对三名携带此突变的同胞(1名男性)进行了一系列神经心理学测试,以评估多个认知领域。其中一名患者表现出执行功能缺陷,而另外两名患者几乎在所有功能方面均有缺陷。认知障碍似乎是这种突变导致的SCAR10的一个特征,其范围从轻度障碍(尤其是涉及前额叶系统)到严重认知障碍,提示大脑广泛受累。