Exploring Executive Functioning of Adults With Probable Developmental Coordination Disorder Using the Jansari Assessment of Executive Functions.

Many individuals with Developmental Coordination Disorder (DCD) demonstrate executive functioning difficulties on standardized assessments, yet these difficulties have not been investigated using ecologically-valid measures. 26 adults with probable DCD (pDCD), and 26 typically developing (TD) adults completed selected background measures and the Jansari assessment of Executive Functions (JEF©). JEF© is an ecologically-valid measure that provides measures of Planning, Prioritization, Selective-Thinking, Creative-Thinking, Adaptive-Thinking, and Action-, Event- and Time-Based Prospective Memory. pDCD participants were impaired relative to TD participants, with difficulties in Planning, Action-, and Event-Based Prospective Memory. These findings are consistent with self-reported difficulty and provide avenues for research investigating intervention.

The course and prognostic capability of motor difficulties in infants showing early signs of autism.

Delays within the motor domain are often overlooked as an early surveillance marker for autism. The present study evaluated motor difficulties and its potential as an early predictive marker for later autism likelihood in a cohort of infants (N = 96) showing early behavioral signs of autism aged 9-14 months. The motor domain was evaluated using the motor subscales of the Mullen Scales of Early Learning at baseline, and at a 6-month follow-up. The Autism Diagnostic Observation Schedule - Toddler Module (ADOS-T) was completed at follow-up as a measure of autism likelihood. Motor difficulties were common at baseline, with 63/96 (65.6%) infants scoring very low or below average in the gross motor domain and 29/96 (30.2%) in the fine motor domain. At follow-up, gross motor difficulties had resolved for many, with 23/63 (36.5%) infants maintaining these difficulties. Fine motor difficulties resolved in fewer infants, with 20/29 (69.0%) continuing to present with fine motor delays at follow-up. Adjusted linear regression models suggested that fine motor scores at baseline (ß = -0.12, SE = 0.04) and follow-up (ß = -0.17, SE = 0.05) were associated with higher ADOS-T scores; with difficulties across both timepoints (ß = 5.60, SE = 1.35) the strongest (largest in magnitude) association with ADOS-T scores of the predictors examined. Motor difficulties are prominent in children displaying emerging signs of autism, with persistent fine motor difficulties predictive of the developing autism phenotype. The findings indicate the potential clinical value of including evaluation of motor skills within early autism surveillance measures. LAY SUMMARY: This prospective study evaluated motor development over a 6-month period in infants showing early behavioral signs of autism. Atypical motor development was a common feature of infants showing early signs of autism and persistent fine motor difficulties were predictive of the emerging autism phenotype.

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.

Driving Skills of Individuals With and Without Developmental Coordination Disorder (DCD/Dyspraxia).

Learning to drive is a significant event for the transition to adulthood and delay or avoidance may have social, practical, and psychological implications. For those with Developmental Coordination Disorder (DCD/Dyspraxia), driving presents a considerable challenge, and the literature shows that there are differences in driving ability between individuals with and without DCD. The aim of the current research is to further our understanding of the mechanisms underlying the driving experiences of individuals with DCD. Nineteen participants with DCD (10 drivers and 9 non-drivers) and 36 controls (17 drivers and 19 non-drivers) aged 18-57 years took part in this study. Participants completed standardized tests, questionnaires and a driving simulation task designed to measure speed, road positioning, and rate of change of steering in three conditions with increasing perceptual complexity. Results indicate that behaviors for all participants changed as the perceptual demands of the task increased. However, drivers with DCD were more affected than all other groups, driving more slowly, and driving further to the right. These findings illustrate how the impact of both internal and external constraints negatively affect the success of the driving task for individuals with DCD compared to their TD peers.

Error-Related Cognitive Control and Behavioral Adaptation Mechanisms in the Context of Motor Functioning and Anxiety.

Motor proficiency reflects the ability to perform precise and coordinated movements in different contexts. Previous research suggests that different profiles of motor proficiency may be associated with different cognitive functioning characteristics thus suggesting an interaction between cognitive and motor processes. The current study investigated this interaction in the general population of healthy adults with different profiles of motor proficiency by focusing on error-related cognitive control and behavioral adaptation mechanisms. In addition, the impact of these processes was assessed in terms of trait anxiety and worries. Forty healthy adults were divided into high and low motor proficiency groups based on an assessment of their motor skills. Using electroencephalography during a flanker task, error-related negativity (ERN) was measured as the neural indicator of cognitive control. Post-error slowing (PES) was measured to represent behavioral adaptation. Participants also completed an anxiety assessment questionnaire. Participants in the high motor proficiency group achieved better task accuracy and showed relatively enhanced cognitive control through increased ERN. Contrastingly, individuals in the lower motor proficiency group achieved poorer accuracy whilst showing some evidence of compensation through increased PES. Trait anxiety reflecting general worries was found to be correlated with motor functioning, but the study could not provide evidence that this was related to cognitive or behavioral control mechanisms. The interaction between cognitive and motor processes observed in this study is unique for healthy and sub-clinical populations and provides a baseline for the interpretation of similar investigations in individuals with motor disorders.

Internalising symptoms in Developmental Coordination Disorder: The indirect effect of everyday executive function.

BACKGROUND: Individuals with Developmental Coordination Disorder (DCD) report elevated executive function (EF) difficulties and internalising symptoms. Previous research suggests EF is important for wellbeing, yet no research has examined its role in internalising symptoms in DCD. AIMS: To explore an indirect relationship between DCD and internalising symptoms, through everyday EF difficulties. METHOD AND PROCEDURES: Thirty-two children with a DCD diagnosis and 51 typically-developing children (ages 8-15) participated. A cross-sectional survey was conducted to collect parent-reported EF and self-reported internalising symptoms. OUTCOMES AND RESULTS: Internalising symptoms and everyday EF difficulties were significantly higher in the DCD group. A bias-corrected, bootstrapped mediation analysis identified an indirect effect of everyday EF difficulties on the relationship between DCD diagnosis and internalising symptoms. CONCLUSIONS AND IMPLICATIONS: This supports previous research indicating that individuals with DCD experience greater levels of internalising symptoms and EF difficulties than peers. It is the first to suggest an indirect effect of everyday EF difficulties in the pathway between DCD and internalising symptoms. This highlights hypotheses for future research into the role of EFs in understanding mental health in DCD. It suggests benefits from increased awareness, routine screening, and intervention for mental health and EF in people with poor motor skills.

Motor and non-motor sequence prediction is equally affected in children with developmental coordination disorder.

Children with Developmental Coordination Disorder (DCD) are diagnosed based on motor difficulties. However, they also exhibit difficulties in several other cognitive domains, including visuospatial processing, executive functioning and attention. One account of the difficulties seen in DCD proposes an impairment in internal forward modelling, i.e., the ability to (i) detect regularities of a repetitive perceptual or motor pattern, (ii) predict future outcomes of motor actions, and (iii) adapt behaviour accordingly. Using electroencephalographic recordings, the present study aimed to delineate these different aspects of internal forward modelling across several domains. To this end, 24 children with DCD and 23 typically-developing children (aged 7-10 years) completed a serial prediction task in the visual, temporal, spatial and motor domains. This task required them to learn short sequences and to indicate whether a sequence was disrupted towards its end. Analyses revealed that, across all domains, children with DCD showed poorer discrimination between intact and disrupted sequences, accompanied by a delayed late parietal positivity elicited by disrupted sequences. These results indicate an impairment in explicit sequence discrimination in DCD across motor and cognitive domains. However, there is no evidence for an impairment in implicit performance on the visuomotor task in DCD. These results suggest an impairment of the updating of an internal forward model in DCD resulting in a blurred representation of that model and, consequently, in a reduced ability to detect regularities in the environment (e.g., sequences). Such a detailed understanding of internal forward modelling in DCD could help to explain the wide range of co-occurring difficulties experienced by those with a diagnosis of DCD.

Impact of Interdisciplinary Team Care for Children With 22q11.2 Deletion Syndrome.

OBJECTIVE: To evaluate disease-specific guideline adherence among children with 22q11.2 deletion syndrome receiving multidisciplinary team care through a 22q specialty clinic compared to children not receiving team care. DESIGN: Retrospective chart review; quality improvement project. SETTING: Tertiary care pediatric hospital. PATIENTS: One hundred eighty-nine patients with 22q11.2 deletion syndrome were categorized into those receiving team care and those not receiving team care. Guideline adherence was compared between the 2 groups. MAIN OUTCOME MEASURE(S): Percent adherence across 8 disease-specific guidelines. RESULTS: A Welch t test revealed mean adherence among patients receiving team care was significantly higher (83% vs 42%, P < .001) compared those not receiving team care. Among team patients with a single 22q Center visit, a paired samples t test showed that mean adherence increased from 63% before the clinic encounter to 86% six months after the encounter (P < .001). Some guidelines were more likely to be associated with provider nonadherence, whereas others were more likely to be associated with patient nonadherence. CONCLUSIONS: Multidisciplinary team care is associated with significantly higher guideline adherence in children with 22q11DS. Additional research is needed to investigate the effect of team care on long-term health outcomes in children with 22q11DS.

Parenting stress in parents of children with developmental coordination disorder.

BACKGROUND: Developmental Coordination Disorder (DCD) is a neurodevelopmental disorder which affects around 5% of children but is often not detected by clinicians. AIMS: The aim of the current study was to investigate whether parents of children with DCD report clinically-significant levels of parenting stress, and to assess a number of factors contributing to this stress, based on evidence from research into other neurodevelopmental disorders. METHODS AND PROCEDURES: A survey was completed by 174 parents of children aged 5-12 years with DCD (parents age M = 42;3, SD = 4;11), comprising measures of the child's motor difficulties and behavioural problems, the parent's perceived competence in the parenting role and perceived levels of social support, and parenting stress. OUTCOMES AND RESULTS: Two thirds of parents reported clinically-significant levels of stress. Multiple regression analyses revealed that all variables except for level of motor difficulties were unique predictors of parenting stress. CONCLUSIONS AND IMPLICATIONS: The study highlights the need for greater awareness of DCD by bringing attention to the impact it can have on the whole family. Providing targeted interventions, particularly addressing parents' perceived competence in managing their child, could reduce parenting stress and improve parents' wellbeing. This in turn could influence their parenting and, therefore, child outcomes.

Fine Motor Control Underlies the Association Between Response Inhibition and Drawing Skill in Early Development.

Previous research shows that the development of response inhibition and drawing skill are linked. The current research investigated whether this association reflects a more fundamental link between response inhibition and motor control. In Experiment 1, 3- and 4-year-olds (n = 100) were tested on measures of inhibition, fine motor control, and drawing skill. Data revealed an association between inhibition and fine motor control, which was responsible for most of the association observed with drawing skill. Experiment 2 (n = 100) provided evidence that, unlike fine motor control, gross motor control and inhibition were not associated (after controlling for IQ). Alternative explanations for the link between inhibition and fine motor control are outlined, including a consideration of how these cognitive processes may interact during development.

Research Review: Internalising symptoms in developmental coordination disorder: a systematic review and meta-analysis.

BACKGROUND: Developmental coordination disorder (DCD) affects 5%-6% of children. There is growing evidence that DCD is associated with greater levels of internalising symptoms (i.e. depression and anxiety). This is the first systematic review and meta-analysis to explore the magnitude of this effect, the quality of the evidence and potential moderators. METHODS: A systematic search was conducted to identify studies reporting a comparison between individuals with DCD/probable DCD and typically developing (TD) individuals on measures of internalising symptoms. A pooled effect size (Hedges g) was calculated using random-effects meta-analysis. Study quality, publication bias and potential moderators of the effect were explored. RESULTS: Twenty studies, including a total of 23 subsamples, met the inclusion criteria, of which 22 subsamples were included in the meta-analysis (DCD: n = 1123; TD: n = 7346). A significant, moderate effect of DCD on internalising symptoms was found (g = 0.61). This effect remained robust after accounting for publication bias and excluding lower quality studies. The effect was significantly larger in studies utilising a cross-sectional design (vs. longitudinal), convenience sampling (vs. population screening) and a majority male sample. CONCLUSIONS: The findings demonstrate that individuals with DCD experience greater levels of internalising symptoms than their peers. This highlights the importance of routine screening for emotional difficulties in DCD, raising awareness of the condition in mental health services and developing psychosocial interventions that extend beyond a focus on motor impairments. However, there is a need for higher quality, longitudinal studies to better understand the causal relationship between DCD and internalising symptoms.

A comparison of viral microneutralization and haemagglutination inhibition assays as measures of seasonal inactivated influenza vaccine immunogenicity in the first year after reduced intensity conditioning, lymphocyte depleted allogeneic haematopoietic stem cell transplant.

Traditionally, immune response to influenza vaccines has been measured using the haemagglutination inhibition (HAI) assay. A broader repertoire of techniques including the sensitive viral microneutralization (VMN) assay is now recommended by the European Medicines Agency (EMA). Comparing HAI and VMN, we determined immune response to a trivalent 2015-2016 seasonal inactivated influenza vaccine (SIIV) administered to 28 recipients of allogeneic haematopoietic stem cell transplant (HSCT). Vaccination was within the first-year post-transplant at a median of 78.5 (24-363) days. The proportion of patients with baseline and post-vaccination HAI titres ≥ 1:40 were 28.6% and 25% for A(H1N1)pdm09, 14.3% at both timepoints for A(H3N2), and 32.1% and 25% for B(Phuket). Pre and Post-vaccination geometric mean titres(GMT) were higher by VMN than HAI for A(H1N1)pdm09 and A(H3N2), but lower for B(Phuket)(p=<0.05). Geometric mean ratios(GMR) of baseline and post-vaccination titres were similar by HAI and VMN(p > 0.05) for all components. A single seroconversion to A(H1N1) was detected by ELISA-VMN. None of patient age, lymphocyte count, days from transplant to vaccination, donor type, or graft-versus-host disease (GVHD) or immunosuppressive therapy (IST) at vaccination correlated with baseline or post-vaccination titres by either assay. This absence of seroresponse to SIIV in the first-year post HSCT highlights the need for novel immunogenic vaccination formulations and schedules in this high-risk population.

Sociodemographic and psychological determinants of influenza vaccine intention among recipients of autologous and allogeneic haematopoietic stem cell transplant: a cross-sectional survey of UK transplant recipients using a modified health belief model.

OBJECTIVES: Studies exploring vaccination rates among haematopoietic stem cell transplant (HSCT) recipients have focused on physician factors that limit uptake. Understanding the patient factors that determine vaccination intention is crucial to delivering a successful vaccination programme. Using a modified health belief model (mHBM), we conducted a cross-sectional survey with the objective of exploring the sociodemographic and psychological factors that determined autologous and allogeneic HSCT recipients' intention to receive the seasonal inactivated influenza vaccine (SIIV) during the 2015-2016 influenza season. SETTING: The setting of our study was three tertiary level, UK National Health Service (NHS) autologous and allogeneic HSCT centres. PARTICIPANTS: Eligible patients were aged 16 years or over and recipients of autologous or allogeneic HSCT for any disease indication, with no absolute contraindication to receiving the SIIV during the next influenza season, and having not received the SIIV since transplant. 93 participants from 3 UK NHS HSCT centres completed an anonymous study-specific questionnaire. 78.5% were recipients of allogeneic and 21.5% autologous HSCT. RESULTS: 23.7% of participants expressed low intent to receive the SIIV. Patients aged over 65 (OR 0.02, 95% CI 0.01 to 0.57, p=0.02) and those who had not received the SIIV prior to HSCT (OR 0.04, 95% CI 0.02 to 0.56, p=0.02) were less likely to have high intent. A multivariate logistic regression model incorporating constructs of the mHBM was statistically significant (p<0.001) and explained 74.7% of variation in SIIV intention. More patients felt that a recommendation from their HSCT team than their general practitioner would prompt them to receive the SIIV, and this was most pronounced in those who had low intent. CONCLUSIONS: The mHBM may provide a useful structure for addressing low vaccine intent among HSCT recipients and further interventional studies are warranted. We would encourage HSCT and general practitioners to discuss SIIV intention as a routine part of care.

An Exploration of the Factor Structure of Executive Functioning in Children.

There has been considerable debate and interest in the factor structure of executive functioning (EF). For children and young people, there is evidence of a progression from a single factor to a more differentiated structure, although the precise nature of these factors differs between investigations. The purpose of the current study was to look at this issue again with another sample, and try to understand possible reasons for previous differences between investigations. In addition, we examined the relationship between less central EF tasks, such as fluency and planning, to the more common tasks of updating/executive working memory (EWM), inhibition, and switching/shifting. A final aim was to carry out analyses which are relevant to the debate about whether EF is influenced by language ability, or language ability is influenced by EF. We reasoned that if language ability affects EF, a factor analysis of verbal and non-verbal EF tasks might result in the identification of a factor which predominantly contains verbal tasks and a factor that predominately contains non-verbal tasks. Our investigation involved 128 typically developing participants (mean age 10:4) who were given EF assessments that included verbal and non-verbal versions of each task: EWM; switching; inhibition; fluency; and planning. Exploratory factor analyses on EWM, switching, and inhibition produced a structure consisting of inhibition in one factor and the remaining tasks in another. It was decided to exclude verbal planning from the next analyses of all the ten tasks because of statistical considerations. Analysis of the remaining nine EF tasks produced two factors, one factor containing the two inhibition tasks, and another factor that contained all the other tasks (switching, EWM, fluency, and non-verbal planning). There was little evidence that the verbal or non-verbal elements in these tasks affected the factor structure. Both these issues are considered in the discussion, where there is a general evaluation of findings about the factor structure of EF.

Comparing Attention to Socially-Relevant Stimuli in Autism Spectrum Disorder and Developmental Coordination Disorder.

Difficulties with social interaction have been reported in both children with an autism spectrum disorder (ASD) and children with developmental coordination disorder (DCD), although these disorders have very different diagnostic characteristics. To date, assessment of social skills in a DCD population has been limited to paper-based assessment or parent report. The present study employed eye tracking methodology to examine how children attend to socially-relevant stimuli, comparing 28 children with DCD, 28 children with ASD and 26 typically-developing (TD) age-matched controls (aged 7-10). Eye movements were recorded while children viewed 30 images, half of which were classed as 'Individual' (one person in the scene, direct gaze) and the other half were 'Social' (more naturalistic scenes showing an interaction). Children with ASD spent significantly less time looking at the face/eye regions in the images than TD children, but children with DCD performed between the ASD and TD groups in this respect. Children with DCD demonstrated a reduced tendency to follow gaze, in comparison to the ASD group. Our findings confirm that social atypicalities are present in both ASD and to a lesser extent DCD, but follow a different pattern. Future research would benefit from considering the developmental nature of the observed findings and their implications for support.

Executive functions in children with developmental coordination disorder: a 2-year follow-up study.

AIM: Executive function impairments have been identified in children with poor motor skills, with and without a diagnosis of developmental coordination disorder (DCD). However, most studies are cross-sectional. This study investigates the development of executive function in children with poor motor skills over 2 years. METHOD: Children aged 7 to 11 years (n=51) were assessed twice, 2 years apart, on verbal and nonverbal measures of executive functions: executive-loaded working memory (ELWM); fluency; response inhibition; planning; and cognitive flexibility. Typically developing children (n=17) were compared with those with a clinical diagnosis of DCD (n=17) and those with identified motor difficulties (n=17) but no formal diagnosis of DCD. RESULTS: Developmental gains in executive function were similar between groups, although a gap between children with poor motor skills and typically developing children on nonverbal executive functions persisted. Specifically, children with DCD performed significantly more poorly than typically developing children on all nonverbal executive function tasks and verbal fluency tasks at both time points; and children with motor difficulties but no diagnosis of DCD showed persistent executive function problems in nonverbal tasks of ELWM and fluency. INTERPRETATION: Children with DCD and motor difficulties demonstrated executive function difficulties over 2 years, which may affect activities of daily living and academic achievement, in addition to their motor deficit. WHAT THIS PAPER ADDS: Executive function difficulties in children with poor motor skills persist throughout middle childhood. Children with motor difficulties, without a developmental coordination disorder (DCD) diagnosis, demonstrate less pervasive executive function difficulties than those with DCD. Executive function problems in the groups with motor difficulties and DCD affect mostly nonverbal domains. All groups showed similar developmental gains in executive function.

The role of older siblings in infant motor development.

Previous research has suggested that infant motor skills may be affected by older siblings but has not considered whether this is due to specific characteristics of the older sibling or of the quality of the sibling relationship. The current study used a longitudinal diary method to record infant motor milestones from 23 infants with older siblings along with parent reports and standardized assessments of motor skills. Parent reports of the older siblings' motor skills and the sibling relationship were also collected until the infants were 18months old. The motor skills, age, and sex of the older siblings were not significantly related to any measure of infant motor development. A significant correlation was revealed between perceived agonism between siblings and infant fine motor skills at 18months, suggesting the importance of considering reciprocal effects of motor development on sibling relationships. Overall, the suggestion that older siblings may provide a good model of motor skills for infants is not supported by the current data. In the future, it will be important to assess the dynamic interactions between different factors in predicting infant motor development, allowing early identification of motor difficulties, which could affect other areas of cognitive development and health.

Coordination difficulty and internalizing symptoms in adults: A twin/sibling study.

Increased anxiety and depression symptoms have been reported in individuals with neurodevelopmental disorders, and have been found to be associated with motor coordination difficulties, but little is known about the etiology of these associations. This study aimed to assess genetic, shared (making twins/siblings alike) and non-shared (individual-specific) environmental influences on the association between poor coordination and symptoms of anxiety and depressed mood using a sample of adult twin and sibling pairs. Participants were asked about their coordination skill and anxiety and depression symptoms. About half of the variance in coordination difficulty was explained by familial (combined genetic and shared environmental) influences, with the remaining variance explained by non-shared environmental influences. Phenotypic associations between coordination and anxiety (r=.46) and depression symptoms (r=.44) were largely underpinned by shared familial liability for the three traits. Non-shared environment accounted for about a third of the phenotypic association. Results suggest that both familial and non-shared environmental influences play a role in the etiology of coordination difficulty and its association with internalizing symptoms. The current study highlights that both biological and environmental pathways shared between these symptoms should be examined in future research to inform prevention and treatment approaches in clinical settings.

Overlapping Phenotypes in Autism Spectrum Disorder and Developmental Coordination Disorder: A Cross-Syndrome Comparison of Motor and Social Skills.

Motor and social difficulties are often found in children with an autism spectrum disorder (ASD) and with developmental coordination disorder (DCD), to varying degrees. This study investigated the extent of overlap of these problems in children aged 7-10 years who had a diagnosis of either ASD or DCD, compared to typically-developing controls. Children completed motor and face processing assessments. Parents completed questionnaires concerning their child's early motor and current motor and social skills. There was considerable overlap between the ASD and DCD groups on the motor and social assessments, with both groups more impaired than controls. Furthermore, motor skill predicted social functioning for both groups. Future research should consider the relationships between core symptoms and their consequences in other domains.