Factors associated with mathematical capacity in children with Developmental Coordination Disorder.

BACKGROUND: Developmental Coordination Disorder (DCD) is a condition characterized by difficulties in motor planning and coordination and affects 5 to 6% of all school-aged children. Children with DCD frequently present with difficulties with academic activities such as handwriting. However, no study to date has comprehensively described mathematical capacity and its potential associated factors in this high-risk group. AIMS: We aimed to describe the frequency and nature of mathematical difficulties of school-aged children with DCD and to evaluate potential factors associated with mathematical performance. METHODS: A total of 55 elementary school-aged children with DCD underwent comprehensive standardized assessments of mathematical, visuoperceptual (VP), attentional, visual-motor integration (VMI), and motor skills. The contribution of each factor to mathematical capacity was established using hierarchical multivariate linear regression models. RESULTS: Children with DCD (9.1 ± 1.5 years, 44 males) had lower overall mathematical capacity compared to normative data (-0.59 SD) on the KeyMath 3rd edition, with poorer performance in basic concepts and problem-solving. Thirty-eight percent of the sample performed below the 15th percentile in overall mathematical skills. VP skills were the most important factors associated with most mathematical domains. Thirty-four percent of the variance of overall mathematical capacity was explained by VP skills, inattention, VMI and motor impairments while controlling for household income (F [5,49]=5.029, p < .0001). CONCLUSION: Children with DCD present with mathematical difficulties in basic concepts and problem-solving, which are partially explained by VP skills. Our findings stress the important of systematically assessing mathematical difficulties children with DCD to ensure they receive the necessary support that leads to academic success.

Novel KCNC2 variant associated with developmental and epileptic encephalopathy.

The KCNC2 gene encodes Kv3.2, which is a member of the voltage-gated potassium channel subfamily. It is crucial for the generation of fast-spiking properties in cortical GABAergic interneurons. Recently, KCNC2 variations were found to be associated with epileptic encephalopathy in unrelated individuals. Here, we report a Chinese patient with developmental and epileptic encephalopathy (DEE) and motor development delay. Whole-exome sequencing (WES) revealed a novel heterozygous variant in the KCNC2 gene NM_139137.4:c.1163T>C (p.Phe388Ser), and subsequent Sanger sequencing showed that it was a de novo mutation. We identified the KCNC2 likely pathogenic variant in a DEE patient by reanalysis of WES data in a Chinese family. Our study enriched the variation spectrum of the KCNC2 gene and promoted the application of WES technology and data reanalysis in the diagnosis of epilepsy.

Developmental coordination disorder subtypes in children: An unsupervised clustering.

AIM: To identify subtypes of developmental coordination disorder (DCD) in children. METHOD: Children with DCD diagnosed through comprehensive evaluation at Robert-Debré Children's University Hospital (Paris, France) were consecutively enrolled from February 2017 to March 2020. We performed an unsupervised hierarchical clustering based on principal component analysis using a large set of variables encompassing cognitive, motor, and visuospatial scores (Wechsler Intelligence Scale for Children, Fifth Edition; Developmental Neuropsychological Assessment, Second Edition; Movement Assessment Battery for Children, Second Edition). RESULTS: One hundred and sixty-four children with DCD were enrolled (median age 10 years 3 months; male:female ratio 5.56:1). We identified distinct subgroups with mixed visuospatial and gestural disorders, or with pure gestural disorders that predominantly impaired either speed or precision. Associated neurodevelopmental disorders, such as attention-deficit/hyperactivity disorder, did not influence the results of the clustering. Importantly, we identified a subgroup of children with marked visuospatial impairment with the lowest scores in almost all of the evaluated domains, and the poorest school performance. INTERPRETATION: The classification of DCD into distinct subgroups could be indicative of prognosis and provide critical information to guide patient management, taking into account the child's neuropsychological profile. Beyond this clinical interest, our findings also provide a relevant framework with homogeneous subgroups of patients for research on the pathogenesis of DCD. WHAT THIS PAPER ADDS: Unsupervised hierarchical clustering identified four subgroups of children with developmental coordination disorder. Two subgroups had combined visuospatial/gestural difficulties, and two had pure gestural disorders. Severe visuospatial impairment was associated with poor performance in most domains including school. Difficulties in the gestural-only clusters were predominantly either gestural precision or speed.

How does brain structure and function on MRI differ in children with autism spectrum disorder, developmental coordination disorder, and/or attention deficit hyperactivity disorder?

AIM: The purpose of this study was to systematically review the neural similarities and differences in brain structure and function, measured by magnetic resonance imaging (MRI), in children with neurodevelopmental disorders that commonly co-occur to understand if and how they have shared neuronal characteristics. METHOD: Using systematic review methodology, the following databases were comprehensively searched: MEDLINE, EMBASE, CINAHL, CENTRAL, PsycINFO, and ProQuest from the earliest record up to December 2021. Inclusion criteria were (1) peer-reviewed studies, case reports, or theses; (2) children under 18 years of age with at least one of the following neurodevelopmental disorders: autism spectrum disorder (ASD), attention hyperactivity deficit disorder (ADHD), developmental coordination disorder (DCD), and their co-occurrence; and (3) studies based on MRI modalities (i.e., structural MRI, diffusion tensor imaging [DTI], and resting-state fMRI). Thirty-one studies that met the inclusion criteria were included for quality assessment by two independent reviewers using the Appraisal tool for Cross-Sectional Studies (AXIS). RESULTS: Studies compared brain structure and function of children with DCD and ADHD (n = 6), DCD and ASD (n = 1), ASD and ADHD (n = 17), and various combinations of these co-occurring conditions (n = 7). Structural neuroimaging (n = 15) was the most commonly reported modality, followed by resting-state (n = 8), DTI (n = 5), and multimodalities (n = 3). INTERPRETATION: Evidence indicated that the neural correlates of the co-occurring conditions were more widespread and distinct compared to a single diagnosis. The majority of findings (77%) suggested that each neurodevelopmental disorder had more distinct neural correlates than shared neural features, suggesting that each disorder is distinct despite commonly co-occurring with each other. As the number of papers examining the co-occurrence of ASD, DCD, and/or ADHD was limited and most findings were not corrected for multiple comparisons, these results must be interpreted with caution.

Early factors associated with risk of developmental coordination disorder in very preterm children: A prospective area-based cohort study in Italy.

BACKGROUND: Developmental coordination disorder (DCD) is a motor disorder of unknown aetiology that may have long-term consequences on daily activities, and psychological and physical health. Studies investigating risk factors for DCD have so far provided inconsistent results. OBJECTIVES: To assess, using a parent-report screening tool, risk of DCD in school-age very preterm children born in Italy, and investigate the associated early biomedical and sociodemographic factors. METHODS: A prospective area-based cohort (804 children, response rate 73.4%) was assessed at 8-11 years of age in three Italian regions. Perinatal data were abstracted from medical records. DCD risk was measured using the Italian-validated version of the Developmental Coordination Disorder Questionnaire (DCDQ-IT). For this study, children with cognitive deficit (i.e. intelligence quotient <70), cerebral palsy, severe vision and hearing disabilities, and other impairments affecting movement were excluded. A total of 629 children were analysed. We used inverse probability weighting to account for loss to follow-up, and multilevel, multivariable modified Poisson models to obtain adjusted risk ratio (aRR) and 95% confidence interval (CI). Missing values in the covariates were imputed. RESULTS: 195 children (weighted proportion 31.8%, 95% CI 28.2, 35.6) scored positive on the DCDQ-IT, corresponding to the 15th centile of the reference Movement-ABC test. Factors associated with overall DCD risk were male sex (aRR 1.35, 95% CI 1.05, 1.73), intrauterine growth restriction (aRR 1.45, 95% CI 1.14, 1.85), retinopathy of prematurity (aRR 1.62, 95% CI 1.07, 2.45), and older maternal age at delivery (aRR 1.39, 95% CI 1.09, 1.77). Complete maternal milk feeding at discharge from the neonatal unit and higher parental socio-economic status were associated with decreased risk. CONCLUSIONS: Both biomedical and sociodemographic factors increase DCD risk. These findings can contribute to elucidating the origins of this disorder, and assist in the identification of children at risk for early referral and intervention.

Repercussions of the Degrees of Hearing Loss and Vestibular Dysfunction on the Static Balance of Children With Sensorineural Hearing Loss.

OBJECTIVE: The purpose of this study was to assess the static balance of children with sensorineural hearing loss (SNHL) according to the degrees of SNHL and the function of the vestibular system. METHODS: This cross-sectional study was conducted in public schools located in Caruaru, Pernambuco state, Brazil, with 130 children (65 with normal hearing and 65 with SNHL as documented by air and bone conduction audiometry) of both sexes between 7 and 11 years old. Static balance was assessed by a stabilometric analysis using a force platform consisting of the circular area of center-of-pressure displacement of the children evaluated in 3 positions: bipedal support with feet together and parallel (PF), tandem feet (TF), and 1 foot (OF), carried out under 2 sensory conditions each, with eyes open and eyes closed. After balance assessments, the children with SNHL received examinations of auditory and vestibular functions-through audiometry and computerized vectoelectronystagmography, respectively-to compose the groups according to degrees of SNHL and vestibular function. RESULTS: The children with severe and profound SNHL demonstrated more static balance instabilities than the children with normal hearing in 5 positions assessed with eyes open (PF, TF, and OF) and eyes closed (PF and TF). The same phenomenon occurred in children with SNHL and associated vestibular dysfunction in all of the positions assessed with eyes open and eyes closed (PF, TF, and OF). CONCLUSION: The larger the degree of SNHL, the greater the balance instability of the children. The children with SNHL and associated vestibular dysfunction showed the highest balance instabilities in this study. IMPACT: Children with larger degrees of SNHL and associated vestibular dysfunction might require prolonged periods to rehabilitate their balance.

Decline in gross motor skills in adult Rett syndrome; results from a Danish longitudinal study.

Longevity of individuals with neurodevelopmental diseases as Rett syndrome (RTT) has increased and many reach adulthood and old age. There is therefore a need to increase knowledge about the course of RTT in adults in order to improve medical care management and quality of life. We did a longitudinal study to address if a possible decline in motor skills in adults with RTT can be explained by the presence of common medical conditions as epilepsy, breathing disturbance, and scoliosis. Data from the Danish RTT database, medical files, and videos from visits at the national Center for Rett syndrome were reviewed. The study included 24 individuals aged 30-66 years at last visit after a follow-up period of 6-12 years. Results showed a clinically observable and significant decline in gross motor skills using the Rett syndrome Gross Motor Scale (RSGMS) with a tendency of less decline in the individuals with the best motor abilities. The frequencies of comorbidities were high. Decline in RSGMS score was associated with the presence of epilepsy and severe scoliosis that had been conservatively managed. The results emphasize that epilepsy plays a significant role in the adult RTT life and management of severe scoliosis in the younger years has impact on the motor abilities in adulthood.

The Relationship Between Developmental Coordination Disorder and Concurrent Deficits in Social Communication and Repetitive Behaviors Among Children with Autism Spectrum Disorder.

Children with Autism Spectrum Disorder (ASD), characterized by core deficits in social communication and restrictive behaviors, can exhibit concurrent motor incoordination and/or intellectual disability (ID). While pervasive delays in motor behavior are common, formal diagnosis of Development Coordination Disorder (DCD) is uncommon. It is not clear how DCD and ID impact core deficits in ASD. This study utilized the Simons Foundation SPARK cohort to describe the scope of motor incoordination among children with ASD and examine the interrelationships between DCD risk, ID, and ASD core deficits. 10,234 children with ASD, between the ages of 5 and 15 years, were included in the analysis. Parents completed online versions of the DCD Questionnaire (DCD-Q), Social Communication Questionnaire (SCQ) and Restrictive Behavior Scale (RBS-R). 85% of children with ASD had DCD-Q scores consistent with being at-risk for DCD, but only 14% reported a formal diagnosis. Children with ID exhibited significantly greater motor incoordination compared to children without ID (P < 0.001). Significantly, greater core deficits were identified in both children at-risk for DCD (P < 0.001) and with ID (P < 0.001). However, the effects of DCD risk were independent of ID and exhibited a medium effect size for SCQ (η2p = 0.063) and a small effect size for RBS-R (η2p = 0.04) scores. Collectively, study outcomes reinforce the pervasiveness of motor incoordination among children with ASD, both with and without concurrent ID, and provide further justification for the inclusion of motor behavior in the early intervention and prescription for children with ASD. LAY SUMMARY: This secondary data analysis of the Simons Foundation SPARK cohort found high rates (85%) of DCD risk among children with ASD. Deficits in motor coordination were greater among children with ASD with concurrent ID diagnoses. Meaningful differences in ASD core deficits (social communication and repetitive behaviors) were independently found in children at risk for DCD, both with and without ID. Autism Res 2021, 14: 804-816. © 2021 International Society for Autism Research and Wiley Periodicals LLC.

Differences in walking and running gait in children with and without developmental coordination disorder: A systematic review and meta-analysis.

BACKGROUND: Developmental coordination disorder (DCD) is a common motor skills disorder in children. Although gait changes are often reported clinically, affecting children's participation in play and sport, there has been no synthesis of research evidence comparing gait of children with and without DCD. Thus, the aim of this research was to determine differences in gait between children with and without DCD METHODS: A search of electronic databases (AMED, CINAHL, Cochrane Library, Embase, Medline, SPORTDiscus, & Web of Science) was conducted from inception to August 2019 for studies comparing walking and/or running gait of children with and without DCD. The McMaster Critical Appraisal Form for Quantitative Studies was used to assess risk of bias amongst included studies. Meta-analysis was completed on measures with four or more homogenous outcomes. RESULTS: 20 studies fulfilled the inclusion criteria and were eligible for review - three of which were pre-post designs, and 17 were cross-sectional designs. Data was extracted in five domains: kinetics, kinematics, physical function, electromyography, and temporospatial parameters. Meta-analysis was performed on one outcome measure - the 6 min Walk Test (6MWT), finding that children with DCD walked significantly shorter distances, indicating reduced endurance in walking gait. Other statistically significant results all favoured typically developing children, however there is little consistency between studies. CONCLUSION: Although a clear gait pattern for children with DCD is not evident, functional deficits appear to be present in endurance and cardiorespiratory fitness. The current evidence base for gait changes in DCD is currently low-level, and further high quality research is warranted. PROSPERO REGISTRATION NUMBER: CRD42018106791.

Balance control in individuals with developmental coordination disorder: A systematic review and meta-analysis.

BACKGROUND: Although it is recognized that the majority of children with developmental coordination disorder (DCD) have balance deficits, comprehensive insights into which balance domains are affected, are still lacking in literature. RESEARCH QUESTION: To what extent is balance control deficient in individuals with DCD compared to controls? METHODS: Pubmed, Scopus and Web of Science were systematically searched. Risk of bias was assessed with the Scottish Intercollegiate Guidelines Network checklist for case-control studies. Mean and standard deviations characterizing balance control were extracted to calculate standardized mean differences (SMD) and pooled, if possible, using Review Manager. RESULTS: The results of 31 studies (1152 individuals with DCD, 1103 typically developing (TD) peers, mean age 10.4 years old) were extracted of which 17 were used for meta-analysis. The mean SMD for the balance subscale of the Movement Assessment Battery for Children was 1.63 (pooled 95 %CI =[1.30;1.97]), indicating children with DCD to perform significantly poorer than their TD peers. Force plate studies also revealed that children with DCD present with a larger sway path during bipedal stance with eyes closed (pooled mean SMD = 0.55; 95 %CI=[0.32;0.78]). Children with DCD tend to have direction-specific limited stability limits and task-independent delayed onset of anticipatory postural adjustments. INTERPRETATION: Children with DCD perform poorer on different domains of balance compared to TD peers. Future research should focus on comprehensive balance assessment in these children, preferably using a longitudinal design.

Correlation between Motor Coordination Skills and Emotional and Behavioral Difficulties in Children with and without Developmental Coordination Disorder.

The purpose of this study was to compare whole factors of emotional and behavioral problems between children with and without developmental coordination disorder (DCD) and investigate the interrelationship between motor coordination skills and emotional and behavioral problems among the children. As a result of screening participants (288 children) based on DSM-5 standard, participants were classified as DCD and typically developing (TD) groups. A total of 60 children (mean age: 8.8 years ± 3.5 months; DCD group n = 30, TD group n = 30) were assessed using the Korean Behavior Assessment System for Children, Second Edition for emotional and behavioral problems. Children with DCD showed significantly poor scores in internalizing problems (p = 0.009), inattention/hyperactivity (p = 0.004), and emotional symptoms index (p = 0.001) among the criteria of emotional problems and in personal adjustment (p = 0.000) among the criteria of behavioral problems. The MABC-2 composite percentile score of participants showed a significant correlation with internalizing problem behavior (r = -0.382, p = 0.003), inattention / hyperactivity disorder (r = -0.409, p = 0.001), emotional symptoms index (r = -0.483, p = 0.000), and personal adjustment (r = 0.474, p < 0.01). Our results validated that children with DCD have more emotional and behavioral difficulties than TD children. Our results revealed that the motor coordination skills have correlated with emotional and behavioral difficulties among children.

Cognitive and motor function in developmental coordination disorder.

AIM: To analyse the development of motor skill and executive function in school-aged children with and without developmental coordination disorder (DCD). METHOD: Using a longitudinal design, 186 children (86 males, 100 females) aged 6 to 11 years at Time 1 were tested over a 2-year period, 52 of whom were diagnosed with DCD at Time 1 (27 males, 25 females; mean age 8y 5mo, SD 1y 6mo) using DSM-5 criteria. The McCarron Assessment of Neuromuscular Development assessed motor status at Time 1 and at 2-year follow-up (Time 2). Executive function was assessed using a well-validated measure, the Groton Maze Learning Test. RESULTS: The DCD cohort at Time 1 had moderate incidence of executive function deficit (41%). Most importantly, at a group level, children with persisting DCD (across Times 1 and 2) also showed significantly lower levels of executive function than children with typical motor development at both time points. At an individual level, around 26% of children in this group had persisting executive function deficits relative to normal ranges of performance. INTERPRETATION: Children with persisting DCD are at significant risk of executive function issues. The combination of motor and cognitive issues as a potential risk factor in the longer-term development of children is discussed. WHAT THIS PAPER ADDS: Around half of children initially diagnosed with developmental coordination disorder (DCD) had the same diagnosis at 2-year follow-up. 41% of children with DCD have impaired executive function. Children with persisting DCD show poorer executive function than those with typical motor development or remitting DCD.

Relationship between Mastery Motivation and Sensory Processing Difficulties in South Korean Children with Developmental Coordination Disorder.

The aim of this study was to identify the correlation between mastery motivation and sensory processing difficulties among South Korean children with developmental coordination disorder (DCD). Ninety-nine children aged 4-7 years with DCD participated. The Dimensions of Mastery Questionnaire was used to assess the mastery motivation of the children, and the Short Sensory Profile was used to assess the children's sensory processing difficulties. All subjects showed lower mastery motivation and definite differences in sensory processing. Mastery motivation was significantly correlated with sensory processing (r = -0.34, p = 0.01). Mastery motivation predicted 41.1% of the sensory processing. In particular, a negative reaction to failure in mastery situations scale (p < 0.01) and general competence compared to peers scale (p < 0.05) in mastery motivation were significant predictors. This study indicated that sensory processing difficulties and lack of mastery motivation were identified among children with DCD in South Korea. And the children with high mastery motivation show less difficulty in sensory processing. It is suggested to develop possible solution for higher mastery motivation to improve sensory processing of the children with DCD in South Korea.

Executive function in children with and without developmental coordination disorder: A comparative study.

BACKGROUND: Children with motor impairments also show poor performance in some executive functions' components. However, there is no consensus on which specific executive subdomain is more impacted. AIM: The objective of this study was to compare executive functions in children with developmental coordination disorder (DCD), at risk for DCD (r-DCD), and in typically developing (TD) children. METHODS AND PROCEDURES: A sample of 397 children was assessed using the MABC-2. Two groups of children were identified; DCD (n = 63) and at r-DCD (n = 31). A third matched group of children with TD (n = 63) was formed. The MABC-2 checklist and the WASI tests were used as screening tools. Measures of executive function including verbal and nonverbal tasks for working memory, inhibitory control, and cognitive flexibility were tested. Multivariate analysis of variance followed by analyses of variance and Bonferroni tests were used to verify group effects on executive functions. RESULTS: A significant group effects were found for Working Memory, Λ = .78, F(4, 360) = 10.12, p ≤ .001, ηp2 = .12; Inhibitory Control, Λ = .59, F(16, 294) = 5.48, p ≤ .001, ηp2 = .23; and Cognitive Flexibility and Inhibitory Control, Λ = .60, F(22, 288) = 3.74, p ≤ .001, ηp2 = .22, with moderate effect sizes. The DCD group showed lower scores compared with the TD group on the visuospatial and verbal working memory; inhibitory control and in tasks of cognitive flexibility; the r-DCD group showed lower scores compared with the TD group for visuospatial working memory and for cognitive flexibility. CONCLUSIONS AND IMPLICATIONS: Poor performance in several measures of executive functions in children with DCD emphasized the need of motor/executive task-specific interventions. Furthermore, children at r-DCD showed low scores in several executive functions; therefore, preventive services should also be provided for this subclinical group.

Impaired motor skills and atypical functional connectivity of the sensorimotor system in 40- to 65-year-old adults with autism spectrum disorders.

Impairments in fine and gross motor function, coordination, and balance in early development are common in autism spectrum disorders (ASDs). It is unclear whether these deficits persist into adulthood and whether they may be exacerbated by additional motor problems that often emerge in typical aging. We assessed motor skills and used resting-state functional magnetic resonance imaging to study intrinsic functional connectivity of the sensorimotor network in 40- to 65-year-old adults with ASDs (n = 17) and typically developing matched adults (n = 19). Adults with ASDs scored significantly lower on assessments of motor skills compared with an age-matched group of typical control adults. In addition, functional connectivity of the sensorimotor system was reduced and the pattern of connectivity was more heterogeneous in adults with ASDs. A negative correlation between functional connectivity of the motor system and motor skills, however, was only found in the typical control group. Findings suggest behavioral impairment and atypical brain organization of the motor system in middle-age adults with ASDs, accompanied by pronounced heterogeneity.

Modified-Bolus Placement as a Therapeutic Tool in the Treatment of Pediatric Feeding Disorders: Analysis From a Retrospective Chart Review.

Purpose Recognizing the need to advance the treatment of pediatric feeding disorders, an emerging area of inquiry focuses on therapeutic techniques that address expulsion and packing possibly associated with oral-motor dysfunction. In the current study, we documented the use of modified-bolus placement in the treatment of pediatric feeding disorders at an intensive, multidisciplinary day-treatment center over a 26-month period. Method The retrospective observational cohort study involved patients admitted for the assessment and treatment of chronic food refusal from August 2013 to October 2015. Results Patients (n = 23) who received modified bolus placement displayed moderate-to-severe oral-motor deficits. Use of modified-bolus placement was associated with reduced expulsion and packing of bites during meals, and treatment gains coincided with a threefold increase in grams consumed per meal. Conclusion Results provide additional support for the potential therapeutic benefits of enhancing bolus placement onto the tongue as a means to improve mealtime performance in children with pediatric feeding disorders when included as an adjunct to more traditional behavioral approaches.

Speech characteristics of childhood apraxia of speech: A survey research.

PURPOSE: There has not been any valid method for the clinical diagnosis of Childhood Apraxia of Speech (CAS) up to now, and the golden standard for diagnosis is the expert's opinion. The current research was conducted to obtain criteria used by the Iranian Speech-Language Pathologists (SLPs) to establish speech characteristics and co-occurring problems of CAS based on their knowledge and clinical experience. METHODS: This research used a questionnaire-based survey design for data collection. The questionnaire was filled out by 260 anonymous participants, both physically and online. RESULTS: The nine top speech characteristics reported with 75.6% agreement as the core symptoms of CAS included: inconsistency (86.9%), consonant sequencing problems (75%), low intelligibility (75%), groping (72.7%), slow diadochokinetic (DDK) (72.3%), articulatory configuration problems (66.2%), difficulty with multisyllabic words (62.7%), suprasegmental disturbances (56.2%) and metathesis (53.5%). The consent of the Iranian practicing SLPs was consistent with the American Speech-Language-Hearing Association (ASHA) diagnostic criteria, Strand's 10-point checklist, and Ozanne's cluster model. More than half of the respondents have been identified with fine motor deficit and language impairment as the most common co-occurring problems of CAS. CONCLUSIONS: The results of this study are in accordance with the findings of previous practicing SLPs' surveys in different languages. Carrying out researches on the speech characteristics of Persian-speaking children suspected of CAS and compare with the results of clinicians' surveys will help us to find a reliable standard for differential diagnosis of Persian-speaking children in Iran.

[Sensory-psychomotor evaluation in Autism: A new tool for functional diagnosis]. / L'évaluation sensori-psychomotrice dans l'autisme : un nouvel outil d'aide au diagnostic fonctionnel.

INTRODUCTION: Psychomotor impairments in Autism Spectrum Disorders (ASD) have frequently been described in scientific literature. Such deficits impact upon the development of social motor function and interfere with the ability to adjust to everyday life. The inclusion of sensory-motor signs in the Diagnostic and Statistical Manual of Mental Disorders (DSM-5) confirms their importance in the diagnosis of ASD. Previous literature has shown the presence precocity of these signs, sometimes before the alteration of the social communication. To our knowledge, there are no existing clinical tools to measure sensory-psychomotor deficit, specifically in ASD. The current paper presents the construction and validation of a new scale, designed to evaluate sensory-psychomotor signs in Autism: 'the Sensory-psychomotor Particularities Scale in Autism' (SPSA). METHOD: The scale is composed of 160 items describing common sensory-psychomotor signs in autism. These items are grouped into 20 variables: touch, nociception, vestibular sensitivity, proprioceptive sensitivity, vision, auditory, multimodality, tone, posture, balance, global coordination, manual dexterity, body schema, bodily self-consciousness, relational adjustment, emotional expression, use of objects, space, time and tonico-emotional regulation. For each item, the psychomotor therapist evaluated sensori-psychomotor signs according to a five-level Likert scale (0="the sign is never expressed by the person", 1="weakly expressed", 2="moderately expressed", 3="severely expressed" and 4="the sign is very characteristic of the person and very severely expressed"). This is completed by a family interview in order to assess the impact of these signs on everyday situations. The study included 111 children with autism. The presence of neurological and genetic diseases was exclusion criteria. For each child, a global developmental evaluation was carried out by an expert clinical team specializing in ASD. Standardized clinical tools were used: Autism Diagnostic Observation Schedule (ADOS), Childhood Autism Rating Scale (CARS), Behavior Summarized Evaluation scale (BSE-R), Repeated and Restricted Behavior scale (RRB), Movement Assessment Battery for Children (M-ABC), Motor Development Rating scale (MDR), Sensory Profile (SP). Developmental quotients (DQ) were evaluated using various tests depending on age and ability. RESULTS: Factor analysis produced three clinically relevant factors: F1: "sensory-emotional synchronization", F2: "multisensory integration" and F3: "motor skills": each containing a similar quantity of items. They account for roughly equal percentages of variance (18.9%, 18.0%, 16.8%, respectively). The factorial structure does not change if the 26 children with comorbid developmental coordination disorder are removed. The three factors show good internal consistency and excellent inter-rater reliability. F1 is comprised of 6 items: touch, nociception, proprioceptive sensitivity, vision, emotional expression and tonico-emotional regulation. This factor is significantly associated with items of the Sensory Profile (touch processing, poor registration, sensory seeking). F2 is comprised of 5 items: multimodality, bodily self-consciousness, relational adjustment, use of objects and space. This factor is associated with ADOS, BSE-R and RRB scores, and the item "touch processing" of the Sensory Profile. F3 is comprised of 4 items: tone, posture, global coordination, manual dexterity. This factor is associated with the M-ABC, the MDR and the item "low endurance" of the Sensory Profile. CONCLUSION: The SPSA is a relevant clinical tool to assess the severity of sensory-psychomotor clinical signs in order to describe the individual profiles of children with ASD. It represents a critical step in advancing knowledge of the complex and heterogeneous pattern of psychomotor development in autism. It could make a valuable contribution to the field, both in research and clinical practice.

Motor development in children with Dravet syndrome.

AIM: The aim of this study is to describe the course of motor development in children with Dravet syndrome. METHOD: Forty-three participants (21 males, 22 females; mean age at last assessment 53.89mo±42.50mo) met the inclusion criteria of having a confirmed diagnosis of Dravet syndrome and presence of data on motor development. All data between 1985 and 2018 were derived retrospectively from their medical records. Gross motor milestones and motor age equivalents were used to describe motor development. Standardized neurodevelopmental assessment and the Bayley Scales of Infant Development defined the overall motor development. Peabody Developmental Motor Scales, Bruininks-Oseretsky Test of Motor Proficiency, and the Beery-Buktenica Developmental Test of Visual-Motor Integration were used to describe development in specific motor domains. RESULTS: Children with Dravet syndrome showed a delay in both sitting (seven out of 14) and walking independently (11 out of 25). Overall motor age equivalents revealed a delay in 29 out of 38 assessments (age 9-115mo). All assessments of children older than 2 years (16 out of 16) showed a delay. Gross motor delay was present in seven out of seven and fine motor delay in 10 out of 13 assessments (age 19-167mo). INTERPRETATION: Motor development is delayed in the majority of children with Dravet syndrome older than 2 years and increases with age. WHAT THIS PAPER ADDS: A delay in motor development is present in most children with Dravet syndrome older than 2 years. Large diversity in early gross motor milestones confirms heterogeneity in Dravet syndrome.

Desarrollo motor en niños con el síndrome de Dravet OBJETIVO: El objetivo de este estudio es describir el curso del desarrollo motor en los niños con síndrome de Dravet. METODOLOGÍA: Cuarenta y tres participantes (21 niños, 22 niñas; con edad promedio en la última evaluación de 53,89 meses ± 42,50 meses) cumplieron los criterios de inclusión: tener un diagnóstico confirmado de síndrome de Dravet y presentar datos de desarrollo motor. Todos los datos recogidos entre 1,985 y 2,018 fueron extraídos retrospectivamente de las historias clínicas. Los hitos de motricidad gruesa y las equivalencias de edad motora se utilizaron para describir el desarrollo motor. Evaluaciones del neurodesarrollo estandarizadas y la escala de desarrollo infantil Bayley definieron el desarrollo motor global. La escala de desarrollo motor Peabody, el test de competencia motora Bruininks-Oseretsky y el test de desarrollo de la integración visomotora Beery-Buktenica se utilizaron para describir el desarrollo en los dominios motores específicos. RESULTADOS: Los niños con el síndrome de Dravet mostraron un retraso tanto en sentarse (7 de 14) como en caminar de manera independiente (11 de 25). En conjunto, las edades motoras equivalentes revelaron un retraso en 29 de las 38 evaluaciones (edad de 9-115 meses). Todas las evaluaciones de niños mayores de 2 años (16 de 16) mostraron un retraso. El retraso de la motricidad gruesa estuvo presente en 7 de 7 evaluaciones y el retraso de la motricidad fina en 10 de 13 evaluaciones (edad 19-167 meses). INTERPRETACIÓN: El desarrollo motor está retrasado en la mayoría de los niños con síndrome de Dravet mayores de dos años y aumenta con la edad.

Desenvolvimento motor em crianças com síndrome de Dravet OBJETIVO: O objetivo deste estudo é descrever o curso do desenvolvimento motor em crianças com síndrome de Dravet. MÉTODO: Quarenta e três participantes (21 do sexo masculino, 22 do sexo feminino; média de idade na última avaliação 53,89m ± 42,50m) atenderam aos critérios de inclusão de ter um diagnóstico confirmado de síndrome de Dravet e a presença de dados sobre o desenvolvimento motor. Todos os dados entre 1985 e 2018 foram derivados retrospectivamente de seus registros médicos. Marcos motores globais e idade motora equivalente foram usados para descrever o desenvolvimento motor. Avaliação padronizada do neurodesenvolvimento e a Escala Bayley de Desenvolvimento Infantil definiram o desenvolvimento global. A Escala Peabody de Desenvolvimento Motor, o Teste de Proficiência Motora de Bruininks-Oseretsky, e o Teste Desenvolvimental de Beery-Buktenica para Integração Visuo-motora foram usados para descrever o desenvolvimento em domínios motores específicos. RESULTADOS: Crianças com síndrome de Dravet mostraram atraso no sentar (sete em 14) e no andar independente (11 em 25). A idade motora global equivalente revelou atraso em 29 de 38 avaliações (idade 9-115m). Todas as avaliações de crianças com mais de 2 anos (16 de 16) mostraram atraso. O atraso motor global estava presente em sete de sete, e motor fino em 10 de 13 avaliações (idade 19-167m). INTERPRETAÇÃO: O desenvolvimento motor é atrasado na maioria das crianças com síndrome de Dravet maiores do que 2 anos, e aumenta com a idade.

Nature of motor impairments in autism spectrum disorder: A comparison with developmental coordination disorder.

INTRODUCTION: Several authors have suggested the existence of motor disorders associated with developmental coordination disorder (DCD) in individuals with autism spectrum disorder (ASD). However, there are few comparative studies of psychomotor profiles that include assessments of neurological soft signs in children with ASD or DCD. We used a neuropsychomotor assessment for children with ASD from a standardized neurodevelopmental examination to understand the nature of the difficulties these children encounter. To uncover the differences and similarities in psychomotor profiles, we compared the profiles of children with ASD with those of children with DCD and focused on two recently described DCD subgroups: visuospatial-constructional (VSC) and mixed (MX). METHODS: We compared 18 children with ASD and 58 children with DCD (33 with VSC-DCD and 25 with MX-DCD) who were assessed with a battery of French-language tests (the NP-MOT) to evaluate the neuropsychomotor functions associated with visual perception and visual-spatial-motor structuring. RESULTS: Although there were similarities between the profiles of children with ASD and those with DCD (VSC-DCD or MX-DCD), these similarities were not associated with the predictive diagnostic markers that characterized subtypes of DCD. Instead, many variables (visuospatial-motor structuration, synkinetic movements, dynamic balance, manual dexterity, coordination, praxis, bodily spatial integration, and digital perception) differed among the three groups; the best performance was observed in the children with ASD. CONCLUSION: The neuropsychomotor profiles of children with ASD and those with VSC-DCD or MX-DCD differed, and these differences are discussed. Our results highlight that impairments of ASD are specific about lateralization disturbances and support the hypothesis of proprioceptive impairment due to visual fixation problems influenced by muscular tone in relation to the subcortical and cortical structures and possible interhemispheric disorder. Thus, some neuropsychomotor functions that underpin both gestures and a set of motor skills are affected.