Spatial exploration and navigation in Down syndrome and Williams syndrome.

We know little about the ability to explore and navigate large-scale space for people with intellectual disability (ID). In this cross-syndrome study, individuals with Down syndrome (DS), individuals with Williams syndrome (WS) and typically developing children (TD; aged 5-11 years) explored virtual environments with the goal of learning where everything was within the environment (Experiment 1) or to find six stars (Experiment 2). There was little difference between the WS and DS groups when the goal was simply to learn about the environment with no specific destination to be reached (Experiment 1); both groups performed at a level akin to a subset of TD children of a similar level of non-verbal ability. The difference became evident when the goal of the task was to locate targets in the environment (Experiment 2). The DS group showed the weakest performance, performing at or below the level of a subset of TD children at a similar level of non-verbal ability, whilst the WS group performed at the level of the TD subset group. The DS, WS and TD group also demonstrated different patterns of exploration behavior. Exploration behaviour in DS was weak and did not improve across trials. In WS, exploration behavior changed across trials but was atypical (the number of revisits increased with repeated trials). Moreover, transdiagnostic individual difference analysis (Latent Profile Analysis) revealed five profiles of exploration and navigation variables, none of which were uniquely specific to DS or to WS. Only the most extreme profile of very poor navigators was specific to participants with DS and WS. Interestingly, all other profiles contained at least one individual with DS and at least one individual with WS. This highlights the importance of investigating heterogeneity in the performance of individuals with intellectual disability and the usefulness of a data-driven transdiagnostic approach to identifying behavioral profiles.

Exploring relative strengths in people with Down syndrome: Spatial thinking and its role in mathematics.

There is convincing evidence that training spatial abilities leads to improved mathematics performance in typically developing (TD) children. However, a lack of information on mathematical development and spatial-mathematical associations in people with Down syndrome (DS) hinders the translation of these interventions. Here, we established developmental trajectories of mathematics and explored whether spatial ability predicts attainment on different mathematics measures in individuals with DS. Participants with DS (n = 36; ages 9-35 years) and TD children (n = 132; ages 4-11 years) completed three groups of tasks: spatial tasks assessing different subdomains of spatial thinking; mathematics tasks assessing early mathematics skills, mathematical reasoning, arithmetic, and geometry; and IQ tasks. The developmental trajectories of mathematics performance against mental age revealed similar starting points of the trajectories and similar rates of development for DS and TD groups. Furthermore, after controlling for verbal skills, spatial skills explained 5.8% to 18.1% of the variation in mathematical performance across different mathematics tasks, and the pattern of spatial-mathematical relations was similar for DS and mental age-matched TD groups. This shows that mathematical development in DS groups appears to mirror that in TD children, indicative of delay only. Strong spatial-mathematical relations were observed for individuals with DS, like those seen for TD participants. This is the vital preliminary knowledge needed to support the design and use of spatial intervention for improving mathematics in individuals with DS.

Assessing the impact of LEGO® construction training on spatial and mathematical skills.

Lego construction ability is associated with a variety of spatial skills and mathematical outcomes. However, it is unknown whether these relations are causal. We aimed to establish the causal impact of Lego construction training on: Lego construction ability; a broad range of spatial skills; and on mathematical outcomes in 7-9-year-olds. We also aimed to identify how this causal impact differs for digital versus physical Lego construction training. One-hundred and ninety-eight children took part in a six-week training programme, delivered twice weekly as a school lunch time club. They completed either physical Lego training (N = 59), digital Lego training (N = 64), or an active control condition (crafts; N = 75). All children completed baseline and follow-up measures of spatial skills (disembedding, visuo-spatial working memory, spatial scaling, mental rotation, and performance on a spatial-numerical task, the number line task), mathematical outcomes (geometry, arithmetic, and overall mathematical skills) and Lego construction ability. Exploratory analyses revealed evidence for near transfer (Lego construction ability) and some evidence for far transfer (arithmetic) of Lego training, but overall transfer was limited. Despite this, we identified key areas for further development (explicit focus on spatial strategies, training for teachers, and embedding the programme within a mathematical context). The findings of this study can be used to inform future development of Lego construction training programmes to support mathematics learning.

Behavioral and Neural Correlates of Visual Working Memory Reveal Metacognitive Aspects of Mental Imagery.

Mental imagery (MI) is the ability to generate visual phenomena in the absence of sensory input. MI is often likened to visual working memory (VWM): the ability to maintain and manipulate visual representations. How MI is recruited during VWM is yet to be established. In a modified orientation change-discrimination task, we examined how behavioral (proportion correct) and neural (contralateral delay activity [CDA]) correlates of precision and capacity map onto subjective ratings of vividness and number of items in MI within a VWM task. During the maintenance period, 17 participants estimated the vividness of their MI or the number of items held in MI while they were instructed to focus on either precision or capacity of their representation and to retain stimuli at varying set sizes (1, 2, and 4). Vividness and number ratings varied over set sizes; however, subjective ratings and behavioral performance correlated only for vividness rating at set size 1. Although CDA responded to set size as was expected, CDA did not reflect subjective reports on high and low vividness and on nondivergent (reported the probed number of items in mind) or divergent (reported number of items diverged from probed) rating trials. Participants were more accurate in low set sizes compared with higher set sizes and in coarse (45°) orientation changes compared with fine (15°) orientation changes. We failed to find evidence for a relationship between the subjective sensory experience of precision and capacity of MI and the precision and capacity of VWM.

Cross-sectional and longitudinal assessment of cognitive development in Williams syndrome.

Williams syndrome (WS) is a rare genetic syndrome. As with all rare syndromes, obtaining adequately powered sample sizes is a challenge. Here we present legacy data from seven UK labs, enabling the characterisation of cross-sectional and longitudinal developmental trajectories of verbal and non-verbal development in the largest sample of individuals with WS to-date. In Study 1, we report cross-sectional data between N = 102 and N = 209 children and adults with WS on measures of verbal and non-verbal ability. In Study 2, we report longitudinal data from N = 17 to N = 54 children and adults with WS who had been tested on at least three timepoints on these measures. Data support the WS characteristic cognitive profile of stronger verbal than non-verbal ability, and shallow developmental progression for both domains. Both cross-sectional and longitudinal data demonstrate steeper rates of development in the child participants than the adolescent and adults in our sample. Cross-sectional data indicate steeper development in verbal than non-verbal ability, and that individual differences in the discrepancy between verbal and non-verbal ability are largely accounted for by level of intellectual functioning. A diverging developmental discrepancy between verbal and non-verbal ability, whilst marginal, is not mirrored statistically in the longitudinal data. Cross-sectional and longitudinal data are discussed with reference to validating cross-sectional developmental patterns using longitudinal data and the importance of individual differences in understanding developmental progression.

Building Numeracy Skills: Associations between DUPLO® Block Construction and Numeracy in Early Childhood.

Research shows that children's block construction skills are positively associated with their concurrent and later mathematics performance. Furthermore, there is evidence that block construction training is particularly beneficial for improving early mathematics skills in children from low-Socio Economic Status (SES) groups who are known to have lower maths performance than their peers. The current study investigates (a) the association between block construction and mathematics in children just before the start of formal schooling (4 years-of-age in the UK) and (b) whether the association between block construction and mathematics differs between children from more compared to less affluent families. Participants in this study included 116 children (M = 3 years 11 months, SD = 3 months) who all completed numeracy, block construction, and receptive vocabulary tasks. Socio-economic status and demographic information (child age, gender, ethnicity) were also obtained from parents. Findings show a strong positive association between block construction and early numeracy skills. Block construction skills explained approximately 5% of the variation in numeracy, even after controlling for age in months, household income, and child receptive vocabulary. When separated by SES group, for children from less affluent families, block construction explained a significant amount of variability (14.5%) in numeracy performance after covariates. For children from more affluent families, block construction did not explain a significant amount of variation in numeracy. These findings suggest that, interventions involving block construction skills may help to reduce SES-based attainment gaps in UK children's mathematics achievement.

Hands-On: Investigating the role of physical manipulatives in spatial training.

Studies show that spatial interventions lead to improvements in mathematics. However, outcomes vary based on whether physical manipulatives (embodied action) are used during training. This study compares the effects of embodied and non-embodied spatial interventions on spatial and mathematics outcomes. The study has a randomized, controlled, pre-post, follow-up, training design (N = 182; mean age 8 years; 49% female; 83.5% White). We show that both embodied and non-embodied spatial training approaches improve spatial skills compared to control. However, we conclude that embodied spatial training using physical manipulatives leads to larger, more consistent gains in mathematics and greater depth of spatial processing than non-embodied training. These findings highlight the potential of spatial activities, particularly those that use physical materials, for improving children's mathematics skills.

Associations and indirect effects between LEGO® construction and mathematics performance.

There is a known association between LEGO® construction ability and mathematics achievement, yet the mechanisms which drive this association are largely unknown. This study investigated the spatial mechanisms underlying this association, and whether this differs for concrete versus digital construction. Between January 2020 and July 2021, children aged 7-9 years (N = 358, 189 female, ethnicity not recorded) completed spatial and mathematics tasks, and either a concrete or digital Lego construction task. Mediation analyses examining direct and indirect pathways (through spatial skills) between Lego construction ability and mathematics explained 8.4% to 26.6% of variance in mathematics scores. Exploratory moderated mediation analyses revealed that only the indirect path through mental rotation differed between Lego conditions. Findings are discussed in relation to theories of spatial-numerical associations and the potential of Lego training for mathematics improvement.

Examining the prevalence and type of technology-use in people with Down syndrome: Perspectives from parents and caregivers.

Familiarity with technology has become a requirement for independent living, however there is limited information on technology access and use for people with Down syndrome (DS). The aim of this study is to describe technology, gaming, and social-media use in people with DS. Parents/caregivers (N = 220) of individuals with DS aged 5-35 years (49% female) completed an online questionnaire. They felt that technology and social media use, and to a lesser extent gaming, played an important role in their son/daughter's life. However, many had concerns about their son/daughter's safety online, and identified challenges they faced with using technology, such as using a mouse and speech recognition. We also found substantial parental interest in learning more about technology-use in DS, particularly the impact of using social media. This paper summarises important details about technology-use in people with DS, providing foundational information for the design of effective technology-based activities and support.

Practitioners' perspectives on spatial reasoning in educational practice from birth to 7 years.

BACKGROUND: There is a growing evidence base for the importance of spatial reasoning for the development of mathematics. However, the extent to which this translates into practice is unknown. AIMS: We aimed to understand practitioners' perspectives on their understanding of spatial reasoning, the extent to which they recognize and implement spatial activities in their practice, and the barriers and opportunities to support spatial reasoning in the practice setting. SAMPLE: Study 1 (questionnaire) included 94 participants and Study 2 (focus groups) consisted of nine participants. Participants were educational practitioners working with children from birth to 7 years. METHODS: The study was mixed methods and included a questionnaire (Study 1) and a series of focus groups (Study 2). RESULTS: We found that whilst practitioners engage in a variety of activities that support spatial reasoning, most practitioners reported little confidence in their understanding of what spatial reasoning is. CONCLUSION: Informative and accessible resources are needed to broaden understanding of the definition of spatial reasoning and to outline opportunities to support spatial reasoning.

How Do Executive Functions Influence Children's Reasoning About Counterintuitive Concepts in Mathematics and Science?

Many scientific and mathematical concepts are counterintuitive because they conflict with misleading perceptual cues or incorrect naive theories that we build from our everyday experiences of the world. Executive functions (EFs) influence mathematics and science achievement, and inhibitory control (IC), in particular, might facilitate counterintuitive reasoning. Stop & Think (S&T) is a computerised learning activity that trains IC skills. It has been found effective in improving primary children's mathematics and science academic performance in a large scale RCT trial (Palak et al., 2019; Wilkinson et al., Journal of Cognitive Enhancement, 4, 296-314, 2020). The current study aimed to investigate the role of EFs and the moderating effects of S&T training on counterintuitive mathematics and science reasoning. A sample of 372 children in school Years 3 (7- to 8-year-olds) and 5 (9- to 10-year-olds) were allocated to S&T, active control or teaching as usual conditions, and completed tasks assessing verbal and visuospatial working memory (WM), IC, IQ, and counterintuitive reasoning, before and after training. Cross-sectional associations between counterintuitive reasoning and EF were found in Year 5 children, with evidence of a specific role of verbal WM. The intervention benefited counterintuitive reasoning in Year 3 children only and EF measures were not found to predict which children would most benefit from the intervention. Combined with previous research, these results suggest that individual differences in EF play a lesser role in counterintuitive reasoning in younger children, while older children show a greater association between EFs and counterintuitive reasoning and are able to apply the strategies developed during the S&T training to mathematics and science subjects. This work contributes to understanding why specifically the S&T intervention is effective. This work was preregistered with the ISRCTN registry (TRN: 54726482) on 10/10/2017. Supplementary Information: The online version contains supplementary material available at 10.1007/s41465-023-00271-0.

Block Design Performance in Williams Syndrome: Visuospatial Abilities or Task Approach Skills?

The block design task (BDT) is a visuospatial measure that individuals with Williams syndrome (WS) perform poorly on. However, it is unclear what underlies their impaired performance. This study investigated whether poorer performance is a result of visuospatial difficulties, executive function (EF) difficulties, atypical looking strategies, or a combination of these. Eleven individuals with WS participated alongside mental age (MA)- and chronological age (CA)-matched control groups. Eye movements were recorded while they took the BDT. Dwell times and visits to areas of interest in WS differed from CA, but not MA, groups. Findings suggest that BDT abilities of individuals with WS are delayed, but not atypical. Delays result from visuospatial and attention-switching difficulties rather than atypical looking strategies.

Anxiety and Spatial Navigation in Williams Syndrome and Down Syndrome.

Individuals with Down Syndrome (DS) and individuals with Williams syndrome (WS) present with poor navigation and elevated anxiety. The aim of this study was to determine the relationship between these two characteristics. Parent report questionnaires measured navigation abilities and anxiety in WS (N = 55) and DS (N = 42) as follows. Anxiety: Spence Children's Anxiety Scale and a novel measure of navigation anxiety. Navigation: Santa Barbara Sense of Direction Scale (SBSOD) and a novel measure of navigation competence. Most individuals were not permitted to travel independently. A relationship between navigation anxiety and SBSOD scores (but not navigation competence) was observed for both groups.

Improving research quality: the view from the UK Reproducibility Network institutional leads for research improvement.

The adoption and incentivisation of open and transparent research practices is critical in addressing issues around research reproducibility and research integrity. These practices will require training and funding. Individuals need to be incentivised to adopt open and transparent research practices (e.g., added as desirable criteria in hiring, probation, and promotion decisions, recognition that funded research should be conducted openly and transparently, the importance of publishers mandating the publication of research workflows and appropriately curated data associated with each research output). Similarly, institutions need to be incentivised to encourage the adoption of open and transparent practices by researchers. Research quality should be prioritised over research quantity. As research transparency will look different for different disciplines, there can be no one-size-fits-all approach. An outward looking and joined up UK research strategy is needed that places openness and transparency at the heart of research activity. This should involve key stakeholders (institutions, research organisations, funders, publishers, and Government) and crucially should be focused on action. Failure to do this will have negative consequences not just for UK research, but also for our ability to innovate and subsequently commercialise UK-led discovery.

Spatial Cognition in Children With Physical Disability; What Is the Impact of Restricted Independent Exploration?

Given the developmental inter-relationship between motor ability and spatial skills, we investigated the impact of physical disability (PD) on spatial cognition. Fifty-three children with special educational needs including PD were divided into those who were wheelchair users (n = 34) and those with independent locomotion ability (n = 19). This division additionally enabled us to determine the impact of limited independent physical exploration (i.e., required wheelchair use) on spatial competence. We compared the spatial performance of children in these two PD groups to that of typically developing (TD) children who spanned the range of non-verbal ability of the PD groups. Participants completed three spatial tasks; a mental rotation task, a spatial programming task and a desktop virtual reality (VR) navigation task. Levels of impairment of the PD groups were broadly commensurate with their overall level of non-verbal ability. The exception to this was the performance of the PD wheelchair group on the mental rotation task, which was below that expected for their level of non-verbal ability. Group differences in approach to the spatial programming task were evident in that both PD groups showed a different error pattern from the TD group. These findings suggested that for children with both learning difficulties and PD, the unique developmental impact on spatial ability of having physical disabilities, over and above the impact of any learning difficulties, is minimal.

The developmental trajectories of spatial skills in middle childhood.

The multidimensional structure of spatial ability remains a debated issue. However, the developmental trajectories of spatial skills have yet to be investigated as a source of evidence within this debate. We tested the intrinsic versus extrinsic and static versus dynamic dimensions of the Uttal et al. (2013, Psychol. Bull., 139, 352) typology in relation to spatial development. Participants (N = 184) aged 6-11 completed spatial tasks chosen to measure these spatial dimensions. The results indicated that the developmental trajectories of intrinsic versus extrinsic skills differed significantly. Intrinsic skills improved more between 6 and 8 years, and 7 and 8 years, than extrinsic skills. Extrinsic skills increased more between 8 and 10 years than intrinsic skills. The trajectories of static versus dynamic skills did not differ significantly. The findings support the intrinsic versus extrinsic, but not the static versus dynamic dimension, of the Uttal et al. (2013, Psychol. Bull., 139, 352) typology.

The relationship between motor milestone achievement and childhood motor deficits in children with Attention Deficit Hyperactivity Disorder (ADHD) and children with Developmental Coordination Disorder.

BACKGROUND: The prevalence of motor impairment is high in ADHD, but we do not know if this stems from infancy. AIMS: 1) to compare the acquisition of motor milestones across three groups: Typically Developing (TD), Attention Deficit Hyperactivity Disorder (ADHD) and Developmental Coordination Disorder (DCD); 2) to determine the relationship between current motor ability and ADHD characteristics in children with ADHD. METHODS AND PROCEDURES: The parents of children aged 8-16 years (ADHD, N = 100; DCD, N = 66; TD, N = 40) completed three online questionnaires: Motor milestone questionnaire; Developmental Coordination Disorder Questionnaire (DCDQ'07) (concurrent motor ability); Conners 3 Parent Rating Scale Long Form (ADHD characteristics). OUTCOME AND RESULTS: When considered as a group, the ADHD group achieved motor milestones within a typical timeframe, despite concurrent motor impairments. Motor ability was not associated with ADHD characteristics. Latent Profile Analysis demonstrated that 56 % of the ADHD group and 48 % of the DCD group shared the same profile of motor milestone achievement, concurrent motor ability and ADHD characteristics. OUTCOMES AND CONCLUSIONS: Unlike children with DCD, the motor impairment often observed in ADHD is not evident from infancy. It is also not part of the ADHD phenotype. Individual differences analysis demonstrated the broad heterogeneity of the ADHD phenotype.

Responses to Navon tasks differ across development and between tasks with differing attentional demands.

Navon hierarchical stimuli are designed to measure responses to the global level (grouped local elements, e.g. a forest) and the local level (individuated local elements, e.g. trees) of a visual scene. Cross-sectional evidence suggests that there are developmental changes in global and local processing. We examined global and local processing in 135 typically developing children in Year 1 (aged 5-6 year), Year 3 (aged 7-8 years), and Year 5 (aged 9-10 years). Participants completed a range of Navon tasks, each with different attentional demands. The design of the Navon stimuli remained constant across the tasks, ensuring that any task-related differences were not due to stimulus characteristics. Sixty children from Years 1 and 3 repeated the testing session two years later. Linear mixed model analyses combined longitudinal and cross-sectional data to assess developmental changes and the influence of attentional task demands on responses. The results revealed differing patterns of global and local processing responses according to Year group and attentional task demands. We found some evidence of developmental change in responses from a relatively more local advantage to a relatively more global advantage, which is consistent with the literature. However, the age at which this transition occurred varied across the tasks. We conclude that responses to hierarchical Navon stimuli are modulated by attentional task characteristics which mask any underlying global or local processing advantage.

Domain-Specific Inhibitory Control Training to Improve Children's Learning of Counterintuitive Concepts in Mathematics and Science.

Evidence from cognitive neuroscience suggests that learning counterintuitive concepts in mathematics and science requires inhibitory control (IC). This prevents interference from misleading perceptual cues and naïve theories children have built from their experiences of the world. Here, we (1) investigate associations between IC, counterintuitive reasoning, and academic achievement and (2) evaluate a classroom-based computerised intervention, called Stop & Think, designed to embed IC training within the learning domain (i.e. mathematics and science content from the school curricula). Cross-sectional analyses of data from 627 children in Years 3 and 5 (7- to 10-year-olds) demonstrated that IC, measured on a Stroop-like task, was associated with counterintuitive reasoning and mathematics and science achievement. A subsample (n = 456) participated either in Stop & Think as a whole-class activity (teacher-led, STT) or using individual computers (pupil-led, STP), or had teaching as usual (TAU). For Year 3 children (but not Year 5), Stop & Think led to better counterintuitive reasoning (i.e. near transfer) in STT (p < .001, ηp 2 = .067) and STP (p < .01, ηp 2 = .041) compared to TAU. Achievement data was not available for Year 3 STP or Year 5 STT. For Year 3, STT led to better science achievement (i.e. far transfer) compared to TAU (p < .05, ηp 2 = .077). There was no transfer to the Stroop-like measure of IC. Overall, these findings support the idea that IC may contribute to counterintuitive reasoning and mathematics and science achievement. Further, we provide preliminary evidence of a domain-specific IC intervention with transferable benefits to academic achievement for Year 3 children.

Characterizing the neural signature of face processing in Williams syndrome via multivariate pattern analysis and event related potentials.

Face recognition ability is often reported to be a relative strength in Williams syndrome (WS). Yet methodological issues associated with the supporting research, and evidence that atypical face processing mechanisms may drive outcomes 'in the typical range', challenge these simplistic characterisations of this important social ability. Detailed investigations of face processing abilities in WS both at a behavioural and neural level provide critical insights. Here, we behaviourally characterised face recognition ability in 18 individuals with WS comparatively to typically developing children and adult control groups. A subset of 11 participants with WS as well as chronologically age matched typical adults further took part in an EEG task where they were asked to attentively view a series of upright and inverted faces and houses. State-of-the-art multivariate pattern analysis (MVPA) was used alongside standard ERP analysis to obtain a detailed characterisation of the neural profile associated with 1) viewing faces as an overall category (by examining neural activity associated with upright faces and houses), and to 2) the canonical upright configuration of a face, critically associated with expertise in typical development and often linked with holistic processing (upright and inverted faces). Our results show that while face recognition ability is not on average at a chronological age-appropriate level in individuals with WS, it nonetheless appears to be a relative strength within their cognitive profile. Furthermore, all participants with WS revealed a differential pattern of neural activity to faces compared to objects, showing a distinct response to faces as a category, as well as a differential neural pattern for upright vs. inverted faces. Nonetheless, an atypical profile of face orientation classification was found in WS, suggesting that this group differs from typical individuals in their face processing mechanisms. Through this innovative application of MVPA, alongside the high temporal resolution of EEG, we provide important new insights into the neural processing of faces in WS.