OBJECTIVE:The objective of this study was to investigate the influences on motor development in infants who are at low risk from Belgium, India, Norway, and the United States using the General Movement Assessment at 10-16 weeks postterm age. METHODS:This was a cross-sectional study of prospectively enrolled full-term infants at low risk (n = 186). Certified General Movement Assessment observers rated the fidgety movements, quality of the movement patterns, age-adequacy of the movement repertoire, postural patterns, movement character, and overall Motor Optimality Score-Revised (MOS-R). Scores were evaluated for associations with sex, birth weight category, gestational age, postterm age at video, and country. RESULTS:The majority of infants had normal fidgety movements (179/186, 96.2%). This did not vary by sex, birth weight, gestational age, postterm age at video, or country. All infants showed normal>atypical movement patterns. Variability was seen for age adequacy (optimal: 137/183, 74.9%), postural patterns (normal>atypical: 164/183, 89.6%), and smooth/ fluent movement character (138/183, 75.4%). Gestational age and postterm age at video were associated with atypical postural patterns, but in multivariable regression, only younger postterm age retained significance (OR = 2.94, 95% CI = 1.05-8.24). Lack of age adequacy was associated with postterm age (OR = 13.15, 95% CI = 4.36-39.72) and country (compared with Norway; Belgium OR = 3.38 95% CI = 12.4-9.22; India OR = 3.16, 95% CI = 1.01-9.87; United States not significant). Infants from India also showed lower rates of an optimal MOS-R (25-28) than infants from Norway. CONCLUSION:The normality and temporal organization of fidgety movements did not differ by sex, birth weight, postterm age, or country, suggesting that the fidgety movements are free of cultural and environmental influences. The majority of full-term infants who were healthy in this cohort showed normal scores for all aspects of motor development tested using the MOS-R. Differences in age adequacy and MOS-R by country warrant investigation with larger cohorts and longitudinal follow-up. IMPACT:Understanding variations in typical motor development is essential to interpreting patterns of movement and posture in infants at risk for atypical development. Using the framework of Prechtl's General Movement Assessment, this study showed that the development of movement and posture in healthy infants was affected by age and country of birth, but the development of the fidgety movements appeared to be free of these influences. Local norms may be needed to interpret the Motor Optimality Score-Revised in all populations, but further research on this topic is needed.
Objective To survey the incidence of intraventricular hemorrhage (IVH) and periventricular leukomalacia (PVL) by gestational age and to report the impact on mortality and neurodevelopmental outcome in very preterm/very low birthweight infants. Study design This was a population-based cohort study of 1927 very preterm/very low birthweight infants born in 2014-2016 and admitted to Flemish neonatal intensive care units. Infants underwent standard follow-up assessment until 2 years corrected age with the Bayley Scales of Infant and Toddler Development and neurological assessments. Results No brain lesion was present in 31% of infants born at <26 weeks of gestation and 75.8% in infants born at 29-32 weeks of gestation. The prevalence of low-grade IVH/PVL (grades I and II) was 16.8% and 12.7%, respectively. Low-grade IVH/PVL was not related significantly to an increased likelihood ofmortality, motor delay, or cognitive delay, except for PVL grade II, which was associated with a 4-fold increase in developing cerebral palsy (OR, 4.1; 95% CI, 1.2-14.6). High-grade lesions (III-IV) were present in 22.0% of the infants born at <26 weeks of gestational and 3.1% at 29-32 weeks of gestation, and the odds of death were (3)14.0 (IVH: OR, 14.0; 95% CI, 9.0-21.9; PVL: OR, 14.1; 95% CI, 6.6-29.9). PVL grades III-IV showed an increased odds of 17.2 for motor delay and 12.3 for cerebral palsy, but were not found to be associated significantly with cognitive delay (OR, 2.9; 95% CI, 0.5-17.5; P =.24). Conclusions Both the prevalence and severity of IVH/PVL decreased significantly with advancing gestational age. More than 75% of all infants with low grades of IVH/PVL showed normal motor and cognitive outcome at 2 years corrected age. High-grade PVL/IVH has become less common and is associated with adverse outcomes.
Introduction Urinary incontinence is the most frequently observed lower urinary tract symptom in children with cerebral palsy (CP). Being continent can positively influence quality of life of the child and the social environment. Objective To investigate the effectiveness of incontinence training with urotherapy in children with CP. Study design A population-based case-control study was conducted including 21 children with CP and 24 typically developing children between 5 and 12 years old, both with daytime incontinence or combined daytime incontinence and enuresis. Children received treatment for one year with three-monthly examination by means of uroflowmetry, a structured questionnaire and bladder diaries. Children started with three months of standard urotherapy. After three, six and nine months of training, specific urotherapy interventions (pelvic floor muscle training with biofeedback, alarm treatment or neuromodulation) and/or pharmacotherapy could be added to the initial treatment. Therapy was individualized to probable underlying conditions. Effectiveness was controlled for spontaneous improvement due to maturation and analysed by means of longitudinal linear models, generalized estimating equations and multilevel cumulative odds models. Comparison with typically developing children was assessed by means of Kaplan-Meier survival analysis. Results Results suggest effectivity rate of incontinence training is lower and changes occur more slowly in time in children with CP compared to typically developing children (Figure). Within the group of children with CP, significant changes during one year of training were found for daytime incontinence (p < 0.001), frequency of daytime incontinence (p = 0.002), frequency of enuresis (p = 0.048), storage symptoms (p = 0.011), correct toilet posture (p = 0.034) and fecal incontinence (p = 0.026). Discussion Maximum voided volume and fluid intake at the start of training were significantly lower in children with CP and could explain a delayed effectiveness of urotherapy. Treatment of constipation demonstrated a positive effect on maximum voided volume and should be initiated together with standard urotherapy when constipation is still present after implementation of a correct fluid intake schedule. Future research with a larger sample size is recommended. Conclusions Incontinence training with urotherapy can be an effective treatment for urinary incontinence in children with cerebral palsy. In the current cohort, effectivity rate of incontinence training was lower and changes occurred more slowly in children with cerebral palsy compared to typically developing children. [GRAPHICS] .
IMPORTANCE Early identification of cerebral palsy (CP) is important for early intervention, yet expert-based assessments do not permit widespread use, and conventional machine learning alternatives lack validity. OBJECTIVE To develop and assess the external validity of a novel deep learning-based method to predict CP based on videos of infants' spontaneous movements at 9 to 18 weeks' corrected age. DESIGN, SETTING, AND PARTICIPANTS This prognostic study of a deep learning-based method to predict CP at a corrected age of 12 to 89 months involved 557 infants with a high risk of perinatal brain injury who were enrolled in previous studies conducted at 13 hospitals in Belgium, India, Norway, and the US between September 10, 2001, and October 25, 2018. Analysis was performed between February 11, 2020, and September 23, 2021. Included infants had available video recorded during the fidgety movement period from 9 to 18 weeks' corrected age, available classifications of fidgety movements ascertained by the general movement assessment (GMA) tool, and available data on CP status at 12 months' corrected age or older. A total of 418 infants (75.0%) were randomly assigned to the model development (training and internal validation) sample, and 139 (25.0%) were randomly assigned to the external validation sample (1 test set). EXPOSURE Video recording of spontaneous movements. MAIN OUTCOMES AND MEASURES The primary outcome was prediction of CP. Deep learning-based prediction of CP was performed automatically from a single video. Secondary outcomes included prediction of associated functional level and CP subtype. Sensitivity, specificity, positive and negative predictive values, and accuracy were assessed. RESULTS Among 557 infants (310 [55.7%] male), the median (IQR) corrected age was 12 (11-13) weeks at assessment, and 84 infants (15.1%) were diagnosed with CP at a mean (SD) age of 3.4 (1.7) years. Data on race and ethnicity were not reported because previous studies (from which the infant samples were derived) used different study protocols with inconsistent collection of these data. On external validation, the deep learning-based CP prediction method had sensitivity of 71.4% (95% CI, 47.8%-88.7%), specificity of 94.1%(95% CI, 88.2%-97.6%), positive predictive value of 68.2% (95% CI, 45.1%-86.1%), and negative predictive value of 94.9% (95% CI, 89.2%-98.1%). In comparison, the GMA tool had sensitivity of 70.0% (95% CI, 45.7%-88.1%), specificity of 88.7% (95% CI, 81.5%-93.8%), positive predictive value of 51.9% (95% CI, 32.0%-71.3%), and negative predictive value of 94.4% (95% CI, 88.3%-97.9%). The deep learning method achieved higher accuracy than the conventional machine learning method (90.6% [95% CI, 84.5%-94.9%] vs 72.7% [95% CI, 64.5%-79.9%]; P < .001), but no significant improvement in accuracy was observed compared with the GMA tool (85.9%; 95% CI, 78.9%-91.3%; P = .11). The deep learning prediction model had higher sensitivity among infants with nonambulatory CP (100%; 95% CI, 63.1%-100%) vs ambulatory CP (58.3%; 95% CI, 27.7%-84.8%; P = .02) and spastic bilateral CP (92.3%; 95% CI, 64.0%-99.8%) vs spastic unilateral CP (42.9%; 95% CI, 9.9%-81.6%; P < .001). CONCLUSIONS AND RELEVANCE In this prognostic study, a deep learning-based method for predicting CP at 9 to 18 weeks' corrected age had predictive accuracy on external validation, which suggests possible avenues for using deep learning-based software to provide objective early detection of CP in clinical settings.
Objective: To describe the inter-observer reliability of the General Movement Assessment (GMA) among a sample of infants at high-risk of cerebral palsy (CP) among raters with various levels of experience. Methods: Video assessments of 150 high-risk infants at 10-15 weeks corrected age were rated by three Prechtl GMA-certified observers with varied experience using the assessment. Videos were scored based on temporal organization of fidgety movements (FMs), presence of abnormal FMs, or absence of FMs. Inter-observer agreements were analyzed with Gwet's AC1 statistic. Results: We found fair to moderate agreement when subcategories of normal FMs (continuous and intermittent) were included (AC1 = 0.32-0.57) and moderate to near perfect agreement when normal categories of FMs were combined (AC1 = 0.60-0.95). Reliability was higher among observers with more experience using the GMA (AC1 = 0.57-0.98) than the observer with less experience (AC1 = 0.32-0.61). Conclusions: Caution may be warranted when the GMA is used to differentiate "continuous and intermittent" FMs temporal organization. The GMA is highly reliable among experienced raters when comparing normal FMs to other FMs categorizations.
OBJECTIVES:To determine whether videos taken by parents of their infants' spontaneous movements were in accordance with required standards in the In-Motion-App, and whether the videos could be remotely scored by a trained General Movement Assessment (GMA) observer. Additionally, to assess the feasibility of using home-based video recordings for automated tracking of spontaneous movements, and to examine parents' perceptions and experiences of taking videos in their homes.DESIGN:The study was a multi-centre prospective observational study.SETTING:Parents/families of high-risk infants in tertiary care follow-up programmes in Norway, Denmark and Belgium.METHODS:Parents/families were asked to video record their baby in accordance with the In-Motion standards which were based on published GMA criteria and criteria covering lighting and stability of smartphone. Videos were evaluated as GMA 'scorable' or 'non-scorable' based on predefined criteria. The accuracy of a 7-point body tracker software was compared with manually annotated body key points. Parents were surveyed about the In-Motion-App information and clarity.PARTICIPANTS:The sample comprised 86 parents/families of high-risk infants.RESULTS:The 86 parent/families returned 130 videos, and 121 (96%) of them were in accordance with the requirements for GMA assessment. The 7-point body tracker software detected more than 80% of body key point positions correctly. Most families found the instructions for filming their baby easy to follow, and more than 90% reported that they did not become more worried about their child's development through using the instructions.CONCLUSIONS:This study reveals that a short instructional video enabled parents to video record their infant's spontaneous movements in compliance with the standards required for remote GMA. Further, an accurate automated body point software detecting infant body landmarks in smartphone videos will facilitate clinical and research use soon. Home-based video recordings could be performed without worrying parents about their child's development.TRIALS REGISTRATION NUMBER:NCT03409978.
Background: With constant changes in neonatal care practices, recent information is valuable for healthcare providers and for parental counselling. The aim of the study was to describe the neuro-developmental outcome in a cohort of very preterm (VPT)/very-low-birthweight (VLBW) infants at 2 years corrected age (CA). Material and methods: This is a population-based cohort study of all infants born with a GA <31 weeks and/or BW <1500 g between 2014 and 2016 admitted to the Flemish (Belgium) neonatal intensive care units. Infants had routine clinical follow-up around 2 years CA. The diagnosis of cerebral palsy (CP), visual and hearing impairments were recorded. Motor, cognitive and language outcomes were assessed using the Bayley-III. Neurodevelopmental impairment (NDI) was classified as mild (<1 standard deviation [SD]) or moderate-severe (<2SD) based on the defined categories of motor, cognitive, hearing, and vision impairments. Results: Of the 1941 admissions, 92% survived to discharge and follow-up data were available for 1089 infants (61.1%). Overall, 19.3%, 18.9% and 41.8% of infants had a motor, cognitive and language delay, respectively. CP was diagnosed in 4.3% of the infants. Mild and moderate-to-severe NDI was observed in 25.2% and 10.9% of the infants, respectively. The number of infants with a normal outcome increased from nearly 40% in the category of GA<26 weeks to 70% for infants in the category of 30-31 weeks GA. Conclusion: At 2 years CA, 64% were free from NDI and 90% were free from moderate-to-severe NDI. However, a lower GA and BW are associated with higher rates of adverse neurodevelopmental outcomes at 2 years CA. (c) 2020 European Paediatric Neurology Society. Published by Elsevier Ltd. All rights reserved.
BACKGROUND:Unilateral spastic cerebral palsy (USCP) occurs in 30%-68% of infants with perinatal stroke. Early detection of USCP is essential for referring infants to early intervention. The aims of this study were to report motor outcomes after perinatal stroke, and to determine the predictive value of the General Movements Assessment (GMA) and Hand Assessment for Infants (HAI) for detection of USCP. MATERIALS AND METHODS:This was a prospective observational study involving infants with perinatal stroke. GMA was conducted between 10 and 15 weeks post term-age (PTA). The HAI was performed between 3 and 5 months PTA. Motor outcome was collected between 12 and 36 months PTA. RESULTS:The sample consisted of 46 infants. Fifteen children (32.6%) were diagnosed with CP, two children with bilateral CP and 13 with USCP. Abnormal GMA had a sensitivity of 85% (95% confidence interval [CI] 55-98%) and a specificity of 52% (95% CI 33-71%) to predict USCP. When asymmetrically presented FMs were also considered as abnormal, sensitivity increased to 100%, hence the specificity declined to 43%. A HAI asymmetry index cut-off of 23, had both a sensitivity and a specificity of 100% to detect USCP. CONCLUSION:Using GMA and HAI can enable prediction of USCP before the age of 5 months in infants with perinatal stroke. Nevertheless, GMA must be interpreted with caution in this particular population. The HAI was found to be a very accurate screening tool for early detection of asymmetry and prediction of USCP.
AimThe purpose of this systematic review was to provide an up‐to‐date global overview of the separate prevalences of motor and cognitive delays and cerebral palsy (CP) in very preterm (VPT) and very‐low‐birthweight (VLBW) infants.MethodA comprehensive search was conducted across four databases. Cohort studies reporting the prevalence ofCPand motor or cognitive outcome from 18 months corrected age until 6 years ofVPTorVLBWinfants born after 2006 were included. Pooled prevalences were calculated with random‐effects models.ResultsThirty studies were retained, which included a total of 10 293 infants. The pooled prevalence of cognitive and motor delays, evaluated with developmental tests, was estimated at 16.9% (95% confidence interval [CI] 10.4–26.3) and 20.6% (95%CI13.9–29.4%) respectively. Mild delays were more frequent than moderate‐to‐severe delays. Pooled prevalence ofCPwas estimated to be 6.8% (95%CI5.5–8.4). Decreasing gestational age and birthweight resulted in higher prevalences. Lower pooled prevalences were found with the Third Edition of the Bayley Scales of Infant Development than with the Second Edition.InterpretationEven though neonatal intensive care has improved over recent decades, there is still a wide range of neurodevelopmental disabilities resulting fromVPTandVLBWbirths. However, pooled prevalences ofCPhave diminished over the years.What this paper addsThe Bayley Scales of Infant and Toddler Development, Third Edition reported lower pooled prevalences of motor and cognitive delays than the Second Edition.The pooled prevalence of cerebral palsy in infants born extremely preterm was reduced compared with previous meta‐analyses.
Aims To investigate if the standard protocol for uroflowmetry, recommended by the International Children's Continence Society, remains accurate when integrating EMG measurement by means of superficial electrodes. Methods A cross-sectional study was conducted including healthy children. Group A performed two direct repetitions of uroflowmetry in combination with electromyography (uroflow/EMG). Group B performed a preceding measurement of isolated uroflowmetry, followed by two randomized measurements of uroflowmetry with and without EMG. Interpretation of uroflow curve was assessor blinded by a pediatric urologist and secondly performed using the flow index methodology. Statistical analysis compared different voids within each group and between group A and B. Results Eighty-three children were included and 206 uroflow measurements were obtained. In both groups statistical findings confirmed the hypothesis that it is preferable to perform an additional measurement before the use of uroflow/EMG. Although both groups showed improvement between voids, the group with initial uroflow measurement followed by uroflow/EMG measurement showed more improvement in concern of curve pattern. An initially better first void in group A, but no statistical difference between the second void in group A and uroflow/EMG testing in group B further demonstrates a higher improvement in group B. This suggests the use of a precedent uroflowmetry without EMG is preferable to immediate testing with EMG. Conclusions It should be mandatory to perform one measurement in advance to ensure the reliability of the results. It is suggested to initiate the procedure with a single uroflowmetry measurement followed by one measurement of uroflow with EMG testing.