Laboratory of Experimental Psychology & Neuroscience (LPEN), Institute of Cognitive Neurology (INECO) & Institute of Neuroscience, Favaloro University , Buenos Aires , Argentina . ; National Scientific and Technical Research Council (CONICET) , Buenos Aires , Argentina ; Pontifical Catholic University of Argentina , Buenos Aires , Argentina ; UDP-INECO Foundation Core on Neuroscience (UIFCoN), Diego Portales University , Santiago , Chile.
Front Aging Neurosci. 2013 Nov 26;5:80. doi: 10.3389/fnagi.2013.00080. eCollection 2013.
Cockayne syndrome (CS) is an autosomal recessive disease associated with premature aging, progressive multiorgan degeneration, and nervous system abnormalities including cerebral and cerebellar atrophy, brain calcifications, and white matter abnormalities. Although several clinical descriptions of CS patients have reported developmental delay and cognitive impairment with relative preservation of social skills, no previous studies have carried out a comprehensive neuropsychological and social cognition assessment. Furthermore, no previous research in individuals with CS has examined the relationship between brain atrophy and performance on neuropsychological and social cognition tests. This study describes the case of an atypical late-onset type III CS patient who exceeds the mean life expectancy of individuals with this pathology. The patient and a group of healthy controls underwent a comprehensive assessment that included multiple neuropsychological and social cognition (emotion recognition, theory of mind, and empathy) tasks. In addition, we compared the pattern of atrophy in the patient to controls and to its concordance with ERCC8 gene expression in a healthy brain. The results showed memory, language, and executive deficits that contrast with the relative preservation of social cognition skills. The cognitive profile of the patient was consistent with his pattern of global cerebral and cerebellar loss of gray matter volume (frontal structures, bilateral cerebellum, basal ganglia, temporal lobe, and occipito-temporal/occipito-parietal regions), which in turn was anatomically consistent with the ERCC8 gene expression level in a healthy donor's brain. The study of exceptional cases, such as the one described here, is fundamental to elucidating the processes that affect the brain in premature aging diseases, and such studies provide an important source of information for understanding the problems associated with normal and pathological aging.
科凯恩综合征(CS)是一种常染色体隐性疾病,与过早衰老、进行性多器官退化以及神经系统异常有关,包括大脑和小脑萎缩、脑钙化和白质异常。尽管有几项关于 CS 患者的临床描述报告了发育迟缓、认知障碍,伴有社交技能相对保留,但以前没有研究进行过全面的神经心理学和社会认知评估。此外,以前在 CS 患者中没有研究过脑萎缩与神经心理学和社会认知测试表现之间的关系。本研究描述了一例不典型的迟发性 III 型 CS 患者,其超过了该病理患者的平均预期寿命。患者和一组健康对照者接受了全面评估,包括多项神经心理学和社会认知(情绪识别、心理理论和同理心)任务。此外,我们比较了患者与对照组之间的萎缩模式,以及与健康大脑中 ERCC8 基因表达的一致性。结果显示记忆、语言和执行功能缺陷,与社会认知技能相对保留形成对比。患者的认知特征与他的大脑灰质体积(额叶结构、双侧小脑、基底节、颞叶和枕叶/顶叶区域)整体和小脑丧失模式一致,而这与健康供体大脑中 ERCC8 基因表达水平在解剖学上是一致的。对异常病例(如本文所述)的研究对于阐明影响过早衰老疾病中大脑的过程至关重要,此类研究为理解正常和病理性衰老相关问题提供了重要的信息来源。