Thomas Michael S C, Ojinaga Alfageme Olatz, D'Souza Hana, Patkee Prachi A, Rutherford Mary A, Mok Kin Y, Hardy John, Karmiloff-Smith Annette
Centre for Brain and Cognitive Development, Birkbeck, University of London, London WC1E 7HX, United Kingdom.
Centre for Brain and Cognitive Development, Birkbeck, University of London, London WC1E 7HX, United Kingdom; Centre for the Developing Brain, School of Biomedical Engineering and Imaging Sciences, King's College London, St. Thomas's Hospital, London, SE1 7EH, United Kingdom.
Res Dev Disabil. 2020 Sep;104:103638. doi: 10.1016/j.ridd.2020.103638. Epub 2020 Jul 10.
In this article, we focus on the causes of individual differences in Down syndrome (DS), exemplifying the multi-level, multi-method, lifespan developmental approach advocated by Karmiloff-Smith (1998, 2009, 2012, 2016). We evaluate the possibility of linking variations in infant and child development with variations in the (elevated) risk for Alzheimer's disease (AD) in adults with DS. We review the theoretical basis for this argument, considering genetics, epigenetics, brain, behaviour and environment. In studies 1 and 2, we focus on variation in language development. We utilise data from the MacArthur-Bates Communicative Development Inventories (CDI; Fenson et al., 2007), and Mullen Scales of Early Learning (MSEL) receptive and productive language subscales (Mullen, 1995) from 84 infants and children with DS (mean age 2;3, range 0;7 to 5;3). As expected, there was developmental delay in both receptive and expressive vocabulary and wide individual differences. Study 1 examined the influence of an environmental measure (socio-economic status as measured by parental occupation) on the observed variability. SES did not predict a reliable amount of the variation. Study 2 examined the predictive power of a specific genetic measure (apolipoprotein APOE genotype) which modulates risk for AD in adulthood. There was no reliable effect of APOE genotype, though weak evidence that development was faster for the genotype conferring greater AD risk (ε4 carriers), consistent with recent observations in infant attention (D'Souza, Mason et al., 2020). Study 3 considered the concerted effect of the DS genotype on early brain development. We describe new magnetic resonance imaging methods for measuring prenatal and neonatal brain structure in DS (e.g., volumes of supratentorial brain, cortex, cerebellar volume; Patkee et al., 2019). We establish the methodological viability of linking differences in early brain structure to measures of infant cognitive development, measured by the MSEL, as a potential early marker of clinical relevance. Five case studies are presented as proof of concept, but these are as yet too few to discern a pattern.
在本文中,我们聚焦于唐氏综合征(DS)个体差异的成因,以卡米洛夫 - 史密斯(1998年、2009年、2012年、2016年)所倡导的多层次、多方法、毕生发展方法为例。我们评估了将婴幼儿发育差异与成年DS患者患阿尔茨海默病(AD)的(升高的)风险差异相联系的可能性。我们审视了这一论点的理论基础,涉及遗传学、表观遗传学、大脑、行为和环境。在研究1和研究2中,我们聚焦于语言发展的差异。我们使用了来自麦克阿瑟 - 贝茨交流发展量表(CDI;芬森等人,2007年)以及穆伦早期学习量表(MSEL)接受性和表达性语言子量表(穆伦,1995年)的数据,这些数据来自84名DS婴幼儿和儿童(平均年龄2岁3个月,范围从0岁7个月至5岁3个月)。正如预期的那样,接受性和表达性词汇都存在发育延迟,且个体差异很大。研究1考察了一项环境指标(以父母职业衡量的社会经济地位)对所观察到的变异性的影响。社会经济地位并未预测出可靠的变异量。研究2考察了一项特定基因指标(载脂蛋白APOE基因型)的预测能力,该指标调节成年后患AD的风险。APOE基因型没有可靠的影响,不过有微弱证据表明,赋予更高AD风险的基因型(ε4携带者)发育更快,这与近期在婴儿注意力方面的观察结果一致(德索萨、梅森等人,2020年)。研究3考虑了DS基因型对早期大脑发育的协同作用。我们描述了用于测量DS患者产前和新生儿脑结构的新磁共振成像方法(例如,幕上脑体积、皮质、小脑体积;帕特基等人,2019年)。我们确立了将早期脑结构差异与通过MSEL测量的婴儿认知发展指标相联系的方法可行性,将其作为具有临床相关性的潜在早期标志物。呈现了五个案例研究作为概念验证,但目前数量还太少,难以辨别出模式。