Oliveira Lívia de Fátima Silva, Júlio-Costa Annelise, Dos Santos Fernanda Caroline, Carvalho Maria Raquel Santos, Haase Vitor Geraldi
Laboratório de Neuropsicologia do Desenvolvimento, Departamento de Psicologia, Universidade Federal de Minas Gerais, Belo Horizonte, Brazil.
Programa de Pós-Graduação em Neurociências, Universidade Federal de Minas Gerais, Belo Horizonte, Brazil.
Front Psychol. 2018 Nov 21;9:2193. doi: 10.3389/fpsyg.2018.02193. eCollection 2018.
Although progress has been made, the cognitive, biological and, particularly, the genetic underpinnings of math learning difficulties (MD) remain largely unknown. This difficulty stems from the heterogeneity of MD and from the large contribution of environmental factors to its etiology. Understanding endophenotypes, e.g., the role of the Approximate Number System (ANS), may help understanding the nature of MD. MD associated with ANS impairments has been described in some genetic conditions, e.g., 22q11.2 deletion syndrome (22q11.2DS or Velocardiofacial syndrome, VCFS). Recently, a girl with MD was identified in a school population screening. She has a new syndrome resulting from a microdeletion in 22q11.2 (LCR22-4 to LCR22-5), a region adjacent to but not overlapping with region 22q11.2 (LCR22-2 to LCR22-4), typically deleted in VCFS. Here, we describe her cognitive-neuropsychological and numerical-cognitive profiles. The girl was assessed twice, at 8 and 11 years. Her numerical-cognitive performance at both times was compared to demographically similar girls with normal intelligence in a single-case, quasi-experimental study. Neuropsychological assessment was normal, except for relatively minor impairments in executive functions. She presented severe and persistent difficulties in the simplest single-digit calculations. Difficulties in commutative operations improved from the first to the second assessment. Difficulties in subtraction persisted and were severe. No difficulties were observed in Arabic number writing. Difficulties in single-digit calculation co-occurred with basic numerical processing impairments in symbolic and non-symbolic (single-digit comparison, dot sets size comparison and estimation) tasks. Her difficulties suggest ANS impairment. No difficulties were detected in visuospatial/visuoconstructional and in phonological processing tasks. The main contributions of the present study are: (a) this is the first characterization of the neuropsychological phenotype in 22q11.2DS (LCR22-4 to LCR22.5) with normal intelligence; (b) mild forms of specific genetic conditions contribute to persistent MD in otherwise typical persons; (c) heterogeneity of neurogenetic underpinnings of MD is suggested by poor performance in non-symbolic numerical processing, dissociated from visuospatial/visuoconstructional and phonological impairments; (d) similar to what happens in 22q11.2DS (LCR22-2 to LCR22-4), ANS impairments may also characterize 22q11.2DS (LCR22-4 to LCR22-5).
尽管已取得进展,但数学学习困难(MD)的认知、生物学,尤其是遗传基础在很大程度上仍不清楚。这种困难源于MD的异质性以及环境因素对其病因的巨大影响。了解内表型,例如近似数字系统(ANS)的作用,可能有助于理解MD的本质。与ANS损伤相关的MD已在一些遗传疾病中有所描述,例如22q11.2缺失综合征(22q11.2DS或心脏颜面综合征,VCFS)。最近,在一次学校人群筛查中发现了一名患有MD的女孩。她患有一种新的综合征,是由22q11.2(LCR22 - 4至LCR22 - 5)区域的微缺失导致的,该区域与通常在VCFS中缺失的22q11.2(LCR22 - 2至LCR22 - 4)区域相邻但不重叠。在此,我们描述她的认知神经心理学和数字认知特征。该女孩在8岁和11岁时接受了两次评估。在一项单病例准实验研究中,可以将她两次的数字认知表现与人口统计学特征相似、智力正常的女孩进行比较。除了执行功能方面相对较小的损伤外,神经心理学评估结果正常。她在最简单的一位数计算中表现出严重且持续的困难。从第一次评估到第二次评估,交换运算方面的困难有所改善。减法运算方面的困难仍然存在且很严重。在阿拉伯数字书写方面未观察到困难。一位数计算方面的困难与符号和非符号(一位数比较、点阵大小比较和估计)任务中的基本数字处理损伤同时出现。她的困难表明存在ANS损伤。在视觉空间/视觉构建和语音处理任务中未检测到困难。本研究的主要贡献在于:(a)这是对智力正常的22q11.2DS(LCR22 - 4至LCR22.5)患者神经心理学表型的首次描述;(b)轻度形式的特定遗传疾病会导致其他方面正常的人持续存在MD;(c)MD的神经遗传基础的异质性表现为在非符号数字处理中表现不佳,且与视觉空间/视觉构建和语音损伤无关;(d)与22q11.2DS(LCR22 - 2至LCR22 - 4)的情况类似,ANS损伤也可能是22q11.2DS(LCR22 - 4至LCR22 - 5)的特征。