Literature DB >> 31861380

Machine Learning of Infant Spontaneous Movements for the Early Prediction of Cerebral Palsy: A Multi-Site Cohort Study.

Espen A F Ihlen1, Ragnhild Støen2,3, Lynn Boswell4, Raye-Ann de Regnier4,5, Toril Fjørtoft3,6, Deborah Gaebler-Spira5,7, Cathrine Labori8, Marianne C Loennecken9, Michael E Msall10,11, Unn I Möinichen9, Colleen Peyton5,12, Michael D Schreiber10, Inger E Silberg9, Nils T Songstad13, Randi T Vågen6, Gunn K Øberg8,14, Lars Adde3,6.   

Abstract

BACKGROUND: Early identification of cerebral palsy (CP) during infancy will provide opportunities for early therapies and treatments. The aim of the present study was to present a novel machine-learning model, the Computer-based Infant Movement Assessment (CIMA) model, for clinically feasible early CP prediction based on infant video recordings.
METHODS: The CIMA model was designed to assess the proportion (%) of CP risk-related movements using a time-frequency decomposition of the movement trajectories of the infant's body parts. The CIMA model was developed and tested on video recordings from a cohort of 377 high-risk infants at 9-15 weeks corrected age to predict CP status and motor function (ambulatory vs. non-ambulatory) at mean 3.7 years age. The performance of the model was compared with results of the general movement assessment (GMA) and neonatal imaging.
RESULTS: The CIMA model had sensitivity (92.7%) and specificity (81.6%), which was comparable to observational GMA or neonatal cerebral imaging for the prediction of CP. Infants later found to have non-ambulatory CP had significantly more CP risk-related movements (median: 92.8%, p = 0.02) compared with those with ambulatory CP (median: 72.7%).
CONCLUSION: The CIMA model may be a clinically feasible alternative to observational GMA.

Entities:  

Keywords:  cerebral palsy; general movement assessment; machine learning; premature infants

Year:  2019        PMID: 31861380      PMCID: PMC7019773          DOI: 10.3390/jcm9010005

Source DB:  PubMed          Journal:  J Clin Med        ISSN: 2077-0383            Impact factor:   4.241


1. Introduction

Cerebral palsy (CP) encompasses a heterogeneous group of motor impairments in childhood that affect the development of movement and posture, causing activity limitation [1]. The prevalence of CP is 2.1 cases per 1000 in high-income countries and occurs in up to 10% of infants at highest risk [2]. CP is a diagnosis based on clinical and neurological signs and is typically determined between age 12 and 24 months [3]. Earlier identification of infants with CP would improve access to community services [4], improve well-being for parents [5] and provide social and economic support for those infants and families in need of care [6]. Early identification would also facilitate earlier onset of therapies and treatments in the period when plasticity of the infant brain is at its highest [7]. Today, the most accurate risk assessments of CP in infants before 5 months of age are the observational general movement assessment (GMA) and cerebral imaging [3]. However, these risk assessments are either based on qualitative perception, requiring considerable training and clinical expertise (GMA), or demand highly expensive equipment (cerebral imaging) [8]. Thus, research on low-cost alternatives for early risk assessment of CP based on automatic and objective detection of infant spontaneous movements has rapidly increased the last two decades [9,10]. Automatic detection of infant spontaneous movements is based on several types of technology including 3D motion capture, inertial sensors, and video recordings [9]. The most clinically feasible technology is a video recording, which is non-intrusive, not dependent on body worn reflective markers or inertial sensors, and available in most clinical and home-based settings using commercially available video and smartphone cameras [10]. Because of the clinical use of observational GMA, large databases of video recordings and CP outcomes are becoming available. These serve as rich sources of data that are important for the generation of robust prediction models based on machine learning. Furthermore, novel methods within machine learning and computer vision have improved possibilities for automated infant motion tracking and facilitated the further development of a computer-based assessment of infant movement kinematics [11]. Several studies have predicted CP based on an automatic movement assessment from infant video recordings with performance comparable to observational GMA [12,13,14,15,16]. A summary of the results of these studies, the methods used, and sample sizes are shown in Table 1. Kanemura et al. [17] also found that infants developing CP had higher average velocity and jerky movements of the legs. However, this study did not report the sensitivity and specificity of the method predicting CP.
Table 1

Summary of results in previous studies for the prediction of cerebral palsy (CP) with video-based automated infant movement analysis.

StudySample Size 1Sens. (%)Spec. (%)Acc (%)Features
Adde [12]30 (13)858888 *CSD, QoM
Rahmati [13]78 (14)509587FFT features
Rahmati [14]78 (14)869291FFT features
Stahl [15]82 (15)859594Wavelet features
Orlandi [16]127 (16)449992FFT/time features

1 Sample size and number of infants with later CP diagnosis in parenthesis (..). * Value is area under receiver operating characteristic (ROC) curve. FFT = fast Fourier transformation (i.e., amplitude and frequency of infant movements); CSD = standard deviation of the center of motion; QoM = quantity-of-motion.

Despite promising results, previous studies using automatic assessment of spontaneous infant movements have several fundamental shortcomings: First, all studies, except the study of Orlandi et al. [16], are based on convenience samples that do not reflect typical clinical cohorts. Second, study samples are small (N = 13–16) in terms of number of children with CP, and it is uncertain whether the prediction models in these studies have external validity for application in a representative population of high-risk infants. Third, the construct validity of the movement features included in previous prediction models is questionable. Observational GMA defines that infant spontaneous movements have complexity denoted by a flow of changes in the movement direction of the participating body parts and variation across time where the infant explores the movement possibilities that the body offers. These spatial and temporal changes in movements are tightly intertwined [18]. The spatial and temporal changes in these movement features are difficult to represent as a single feature across the entire video recording, as was carried out in a previous study using the standard deviation of the center of motion [12]. Our hypothesis is that complex and variable spontaneous movements could be characterized by multiple features of temporal modulation in movement frequencies and covariation that will outperform single features, and that spatial and temporal changes in infant movement can be assessed by dividing the video recording into smaller movement periods to obtain a percentage (i.e., proportion) of periods with CP risk-related movements. Furthermore, previous studies have not investigated the relationship between CP prediction models and gross motor function in children with CP. These are important elements to ensure the construct validity and, consequently, the feasibility of the CP prediction model for clinical decision support. The aim of the present study was to present a novel machine-learning model, the Computer-based Infant Movement Assessment (CIMA) model, for clinically feasible early CP prediction and for the prediction of ambulatory (gross motor function classification scale (GMFCS I–III) versus non-ambulatory function (GMFCS IV–V) in children with CP.

2. Experimental Section

2.1. Study Participants

This study is part of a multi-center, observational study on early CP prediction in high-risk infants. Four hundred and fifty infants admitted to one of five participating level III–IV Neonatal Infant Care Units (NICU) in Norway or the United States were enrolled at discharge from the NICU based on extreme prematurity, neonatal neurologic abnormalities, cardiac surgery or medical complexity. Video recordings during the fidgety movements period were taken according to Prechtl’s methodology for observation of general movements, and the GMA results for early CP prediction are presented in a different paper [19]. In the study arm presented here, the aim was to develop a novel machine-learning algorithm for early CP prediction based on the same videos. In total, 377 infants constituted the study sample after exclusion of video recordings (see Figure 1). The median length of the included video recordings was 5 min (range: 1–5 min) and the mean age of the infants at recording was 12 (9–15) weeks corrected age (CA). For details on the clinical characteristics and neonatal risk factors of the 377 included infants, see Appendix A.
Figure 1

Flow-chart of exclusion of video recordings for the development and testing of the Computer-based Infant Movement Assessment (CIMA) model.

2.2. The Computer-Based Infant Movement Assessment (CIMA) Model

The goal for the development of the CIMA model was to improve the early risk assessment of CP in high-risk infants before 5 months post-term age. Figure 2 summarizes the steps of the CIMA model.
Figure 2

Steps of the CIMA model. First, infant movements are detected by motion tracking of six body parts (head, trunk, arms, and legs) in the video. Second, features for the movement frequencies, amplitude, and covariation of the different body parts are extracted from the body part movement trajectories and used in the CP prediction model. The CP prediction model identifies 5 second periods with CP risk-related movements. Finally, the proportion (%) of periods with CP risk-related movements typically found in infants with CP is summarized and communicated as a CP risk indicator.

2.2.1. Infant Motion Detection in Video Recording

Infants were video recorded during active wakefulness when in a comfortable state at 9 to 15 weeks CA using a standardized set-up, and in cases of more than one available video, the one closest to 12 weeks CA was selected. A commercially available digital video camera (Sanyo VPC-HD2000, SANYO Electric Co, Ltd., Osaka, Japan) was used. The processing of the video recording contained five steps: video screening, preprocessing, pixel tracking using large displacement optical flow, segmentation of six body parts, and extraction of vertical and horizontal coordinates of body part’s movements. All videos were cropped so that only the infant was visible in the video. Large displacement optical flow was used to track pixel movements and a manual annotation was performed on each 500 frames to identify the pixel center of six parts of the infant’s body—arms, legs, head, and torso. Two research assistants without any expertise in infant spontaneous movements performed the manual annotation. Technical details of the infant motion tracker method are described elsewhere [20].

2.2.2. Movement Feature Extraction

To quantify the temporal variation in body part movement frequencies, amplitude, and covariations, the horizontal (x) and vertical (y) coordinates of the pixel center of the six body parts was decomposed into the time–frequency domain by multivariate empirical mode decomposition (MEMD) and Hilbert–Huang transformation [21,22]. In contrast to the fast Fourier transformation and wavelet methods chosen in previous studies [13,14,15], the movement components defined by MEMD have the potential to reflect intrinsic properties of the infant movement dynamics. Technical details of the procedure are described in Appendix B. The body parts’ mean movement frequency, amplitude, and covariation was computed for 5 second non-overlapping time periods and finally resulted in a set of 990 features describing all the infant’s movement repertoire in each 5 second period.

2.2.3. CP Prediction Model and Validity of the Model

Each 5 second period in the video was labeled as CP or non-CP according to the child’s CP status diagnosed according to the decision tree published by the Surveillance of cerebral palsy in Europe (SCPE, [23]). In total, 1898 periods were available from videos of children with CP and 18,321 periods from videos of children without CP. A partial least square (PLS) regression with a backward feature selection was performed to select features that predicted CP from the large set of 990 movement features without overfitting the model [24]. The selected movement features in each 5 second period were clustered into 5 composite scores which were used in a linear discriminative analysis (LDA) to classify movements typically found in children with or without CP (Appendix C) [25]. To avoid overfitting of the CIMA model, the dataset was divided into training, validation, and test sets in a double cross-validation procedure (Appendix D). The final CIMA model classified each 5 second period with either 0 or 1 according to the absence or presence of CP risk-related movements. The final risk classification was averaged across each video recording defining the proportion (%) of periods with CP risk-related movements. A decision threshold of 50% was set to decide whether the video represented an infant with overall absence or presence of CP risk-related movements.

2.3. Observational GMA, Cerebral Imaging, Cerebral Palsy and Gross Motor Function

Observational GMA was carried out on the same video recordings as the CIMA model according to the Prechtl approach [26]. Two experienced and certified GMA observers (LA and TF) who were blinded to the clinical history of the infants performed all assessments. In case of disagreement, the observers re-assessed the video together and reached consensus. Fidgety movements (FM) were classified as absent (FM−; n = 57/15%), sporadic (FM−/+; n = 29/8%), intermittent (FM+; n = 235/62%), continual (FM++; n = 49/13%) according to their presence and length of interspersed pauses [27], or as exaggerated (FMa, n = 7/1.9%) if excessive in amplitude and speed. Cerebral imaging (cerebral ultrasound (cUS) and magnetic resonance imaging (MRI)) was carried out for clinical purposes following each hospital’s guidelines, and a central classification of the results into normal/mildly abnormal or moderately/severely abnormal was carried out based on the local written reports. Lesions known to be associated with later CP were classified as abnormal, and milder abnormalities not associated with later CP were classified as normal (for details about imaging results, see [19]). Assessment of cerebral palsy and ambulatory motor function: CP was diagnosed according to the decision tree published by the Surveillance of cerebral palsy in Europe (SCPE, [23]). The CP diagnosis was performed by pediatricians who were unaware of the outcome of GMA classification and CIMA model. Gross motor function was classified using the Gross Motor Function Classification System (GMFCS) [28,29]. Forty-one (11%) of 377 included infants had CP with corresponding GMFCS status at follow-up at mean age 3.7 years (SD 0.95; range 1.2–6 years). The prevalence of CP subtypes and GMFCS levels for the 41 infants developing CP are shown in Table 2 below.
Table 2

CP subtype and gross motor function in children with CP.

CP StatusN (%)
CP subtype *
Unilateral spastic8 (20)
Bilateral spastic25 (61)
Dyskinetic5 (12)
Ataxic1 (2)
Gross motor function (GMFCS)
GMFCS I11 (27)
GMFCS II3 (7)
GMFCS III6 (15)
GMFCS IV10 (24)
GMFCS V11 (27)

* CP subtype was available in all but two of the 41 children with CP. GMFCS = Gross Motor Function Classification System.

2.4. Statistics of the Outcome of the CIMA Model

The ability of the CIMA model to predict CP was assessed by sensitivity, specificity, positive and negative predictive values (PPV and NPV) and area under receive operating characteristic curve (AUC). The performance of the CIMA model was compared with the performance of the observational GMA and cerebral imaging (cUS and MRI) [19] in addition to the automated method previously presented by our group using the variation of the spatial center of motion (CSD) [12,30]. Kruskal–Wallis’s test with the post hoc Wilcoxon rank-sum test including Bonferroni correction assessed the significance of the difference in the proportion of CP risk-related movements between the different FM categories assessed by the observational GMA. The Wilcoxon rank-sum test was also used to assess the significance of the difference in the proportion of CP risk-related movements between infants developing CP with GMFCS I, II, or III (i.e., ambulatory CP) and those developing CP with GMFCS IV or V (i.e., non-ambulatory CP). All analyses and statistics were performed in Matlab 2018a and p-values below 0.05 were considered statistically significant.

3. Results

Proportion of Periods with CP Risk-Related Movements, CP Status and Gross Motor Function

Figure 3 shows the proportion of periods with CP risk-related movements identified in each of the video recordings of the 377 infants. Three (7.3%) of 41 children with a confirmed CP diagnosis had a proportion of periods with CP risk-related movements below 50% (false negative; red bars below the horizontal line in Figure 3). Sixty-two (18.5%) of 336 infants who did not develop CP had a proportion of CP risk-related movements above 50% (false positive; blue bars above the horizontal line in Figure 3). Table 3 shows the predictive values for the current CIMA model, observational GMA [19] and neuroimaging results [19] and the variation of the spatial center of motion (CSD). The CIMA model had the best sensitivity, NPV and AUC. However, the specificity and PPV were slightly lower than for the GMA and neuroimaging results. The statistics in Table 3 are dependent on a decision threshold of 50%. The ROC curve and alternative thresholds are provided in Appendix E and cross-tables and mean square contingency coefficients are provided in Appendix F.
Figure 3

Each bar represents the proportion (%) of periods with CP risk-related movements represented in the video recordings of each of the 377 infants. The bars are centered around the decision threshold of 50% (horizontal line) for increased risk of CP. The red bars are from infants with confirmed CP diagnosis, whereas the blue bars represent the infants with a confirmed non-CP diagnosis.

Table 3

The sensitivity, specificity, positive and negative predictive values and area under the curve (AUC) with 95% confidence intervals in brackets for the prediction of CP.

MethodSens. (%)Spec. (%)PPV (%)NPV (%)AUC *
CIMA92.7 [80.1, 98.5]81.6 [77.0, 85.5]38.0 [32.5, 43.8]98.9 [96.8, 99.6]0.87 [0.81, 0.91]
GMA [19]76.2 [60.6, 88.0]82.4 [78.1, 86.2]33.3 [27.4, 39.8]96.8 [94.6, 98.1]0.82 [0.78, 0.85]
Imaging [19] 81.0 [65.9, 91.4]85.3 [81.2, 88.8]39.1 [32.5, 46.1]97.5 [95.4, 98.6]0.85 [0.81, 0.88]
CSD56.1 [39.8, 71.5]58.6 [53.2, 64.0]14.2 [10.9, 18.6]91.6 [88.5, 94.0]0.56 [0.48, 0.64]

* Values for GMA and Imaging is accuracy reported in Støen et al. [19]. PPV = positive predictive value; NPV = negative predictive value; GMA = General Movement Assessment.

The proportion of periods with CP risk-related movements showed a significant relationship with the FM classification by the observational GMA (p < 0.01, Figure 4). In the group of infants with absent FMs, a significantly higher proportion of CP risk-related movements (median: 92%, p < 0.00001) were seen in the infants later diagnosed with CP when compared with those without CP (median: 18.6%) (see boxplots in Figure 4). Within the group of infants later diagnosed with CP, the infants with intermittent fidgety movements (FM+) had a significantly lower proportion of risk-related movements (median: 63.5%, p = 0.009) compared with the infants with absent fidgety movements (FM−). The infants with continual fidgety movements (FM++) had a low proportion of periods of risk-related movements (median: 8.5%), with little intra-group variation, compared with infants with absent, sporadic or intermittent FMs (Figure 4).
Figure 4

Boxplot of the proportion of periods with CP risk-related movements assessed by the CIMA model (y-axis) and temporal organization of FMs assessed by observational GMA (x-axis) according to CP outcome. The red line indicates the median and blue box the interquartile range. The whiskers in dashed lines are 1.5 times the interquartile range and cover 99.3% of the data if normally distributed. Outliers are marked as red crosses. The horizontal dashed line represents a decision threshold of 50% for the CIMA model. FM− = absent FM; FM−/+ = sporadic FM; FM+ = intermittent FM; FM++ = continual FM; FMa = FM with exaggerated speed and amplitude.

Among the 41 children with CP, the proportion of periods with CP risk-related movements was significantly higher in those with GMFCS IV–V (non-ambulatory function) compared to those with GMFCS I–III (ambulatory function) (median: 92.8%, IQR: [75.0%, 97.2%] vs. median: 72.7%, IQR: [60.6%, 83.3%]; p = 0.02).

4. Discussion

This study presents a novel machine-learning model, the CIMA model, for the early prediction of CP with an accuracy comparable to the General Movement Assessment (GMA) and neonatal cerebral imaging. Furthermore, the CIMA model differentiated children with ambulatory CP from those with non-ambulatory CP. These findings motivate the further development of a clinical decision support system based on video recordings and machine-learning assessment that can easily be applied for screening of high-risk infants. In the present study, the CIMA model was developed based on CP outcome. This is in contrast to others who have presented machine-learning models for automated CP prediction based on the identification of abnormal general movements and absence of FMs [9]. The video recordings in the present study were performed during the fidgety movements period, making it likely that the CIMA model captures some of the features which are typical for FM. Hence, the selected features in the CIMA model (i.e., movement covariation, frequencies and amplitudes) have the potential to reflect complexity and variability of the infant spontaneous movements which is typical for FM [27]. Both CIMA and GMA deliver a high number of false positives, but the assessments are weakly correlated with r = 0.24 to 0.30 (see Appendix F for details). The false positive cases of the CIMA method are mainly in the intermittent FM category (FM+), whereas the false positive of the GMA is, by definition, in the absent and the sporadic FM category (FM−/+ and FM+). The CIMA model identified children without CP who were classified with absence of FMs (i.e., FM−) with a low proportion of CP risk-related movements. These results suggest that the CIMA model and GMA identifies different false positive cases and may identify different features of infant spontaneous movements. Thus, machine-learning approaches, like the CIMA model, could be used to detect false positive cases within the group of infants with absence of FMs. Further research could relate the movements features used in the CIMA model to the different motor phenotypes recently suggested for infants developing CP in order to gain a deeper knowledge of the appearance of false positive cases in the CIMA model [31]. The ability of the CIMA model to predict ambulatory versus non-ambulatory function in children with CP suggests a continuum in the proportion (%) of periods with CP risk-related movements, which is related to later motor function. However, the present CIMA model cannot reveal how the chosen movement features change according to later motor function. For the time being, we can, therefore, only speculate that reduced covariation between body parts and reduced variation in movement frequencies and amplitudes are typical for infants who develop CP and that the same movement features are related to the severity of CP. The proportion (%) of time periods with abnormal movements identified by the CIMA model was shown to outperform the CP prediction ability of the standard deviation of the center of motion (CSD) used in several previous computer-based studies by our group [12,30]. The previously developed CSD was based on a frame differencing method which may be susceptible to differences in contrasts, light, and infant clothing, which may vary more in this larger multi-site cohort of infants. Furthermore, as the sample size and heterogeneity of children with CP increase, it becomes more challenging for a single predefined feature, such as CSD, to contain information of various characteristics of the infant movement repertoire relevant for a clinical outcome such as CP. Thus, we argue that it is likely that the predictive performance of other suggested single features such as relative movement frequency [15] and mean and minimum velocity [30] will potentially decay in larger multi-site populations of high-risk infants. The performance of the presented CIMA model suggests that overall variables, such as the proportion (%) of periods with CP risk-related movements, should be based on a cluster of movement features rather than single “key” features. The CIMA model has several clinical and methodological limitations. First, the large distance optical flow method is not fully automatic and requires manual annotation. Thus, even though the CIMA model could be a clinically feasible alternative to observational GMA, additional resources for manual annotation are necessary at this point. Furthermore, the horizontal and vertical coordinates of the pixel centers of the six labeled infant body parts are not directly related to biomechanical features such as the joint center position or the body part’s center of mass. Consequently, the present infant movement tracker based on large distance optical flow does not provide accurate biomechanical descriptors of the infant movements. Thus, the selected features and PLS regression components of the CIMA model will be dependent on the specific motion tracker system used. Further studies should emphasize on developing fully automated movement tracker technology able to identify joint center position and the body segment’s center of mass (i.e., full biomechanical 2D model), which will have the potential to identify specific and definable biomarkers of later motor impairments. Advancement in computer vision and the development of deep convolutional neural networks make it possible to identify joint centers and body segment position with high precision [11,32]. Such a movement tracker will generate universal pools of biomechanical descriptors for the CIMA model that are not dependent on manual annotations and the choice of movement assessment technology (e.g., 3D motion capture and inertial body worn sensors). Secondly, the CIMA model is trained on video recordings from a standardized camera setup with a static mounted camera [12]. To improve clinical feasibility, the CIMA model should be trained on video recordings from hand-held smartphone cameras. Thus, a fully automated movement tracker system suggested above could include filters and post-processing procedures to remove motion artifacts of hand-held smartphone recordings [11]. This will integrate the CIMA model into future app-based platforms for clinical decision support. Thirdly, the present model was created from 5 second non-overlapping time periods. It is highly unlikely that all 5 second periods within a video recording contain infant movements related to risk of CP outcome. As an example, infants with or without CP may be quiet with little movements within a 5 second time period. These time periods containing only short movement sequences will get different CIMA model labels according to the CP outcome. Thus, the labeling of the short periods may contribute to noise and, consequently, this may affect the estimated percentage (%) of periods with CP risk-related movements. A remedy for these limitations is to use the classification score provided by linear discriminative analysis to weight the influence of each time interval. This solution was implemented in our study but did not change the reported overall performance of the CIMA model. Fourth, the present study did not provide test–retest reliability of the proportion of CP risk-related movements. The intra-session test-reliability of observational GMA is reported to be high [33]. Further test–retest reliability studies are necessary before concluding that the CIMA model is a clinically feasible alternative to observational GMA. Finally, the present study should be replicated on new samples of high-risk infants to assess the external validity of the CIMA model. The present multi-center study comprised a heterogenous selection of infants (shown in Appendix A, Table A1). The predictive values of a specific method will differ based on the prevalence of the outcome, and this should be taken into consideration in the interpretation of the results. The international community of infant movement assessment should collaborate on generating larger databases of infant video data working as a foundation for the development of more robust machine-learning algorithms for the classification of infant motor repertoire and the prediction of later motor impairments. The investigation of neurophysiological correlates (functional magnetic resonance imaging (fMRI), ultrasound, electroencephalography (EEG) and magnetic resonance imaging (MRI)) to the outcome of the CIMA model is also an important direction of future research to improve the model’s construct validity and establish new biomarkers of later motor impairments.
Table A1

Demographic variables and primary reason for referral to follow-up.

Risk GroupN (%)
GA < 28 weeks and/or BW ≤ 1000 g167 (44.3)
- Boys90 (53.9)
- GA (weeks), mean (SD)26.3 (1.7)
- BW (g), mean (SD)833 (178)
GA 28–36 weeks and BW > 1001 g59 (15.6)
Neonatal arterial ischemic stroke15 (4.0)
Neonatal encephalopathy50 (13.3)
CHD w/surgery before 4 weeks39 (10.3)
Other a47 (12.5)

BW = birth weight; GA = gestational age; CHD = Cardiac heart disease. Other a: Infants who were referred to neurodevelopmental follow-up at discharge from the neonatal intensive care unit due to significant abnormalities on cerebral imaging (intraventricular hemorrhages III–IV, other intracranial hemorrhages with or without seizures, cystic periventricular leukomalacia, ventriculomegaly, venous sinus thrombosis), central nervous system infection, medically complex infants (syndromes/chromosomal abnormalities, multiple congenital anomalies, hydrops fetalis, severe lung hypoplasia, protracted hypoglycemia, seizures with unknown etiology) and severe intrauterine growth restriction. One second twin came to follow-up due to referral of the first twin.

5. Conclusions

This study presents a novel machine-learning model, called the CIMA model, which predicts CP in a large cohort of high-risk infants with an accuracy comparable to the observational General Movement Assessment (GMA) and neonatal cerebral imaging. The model also differentiated between ambulatory and non-ambulatory CP. Movement features assessing covariation between body parts and temporal modulation in movement frequencies and amplitudes were used in the CIMA model. The present adds to developing a clinical decision support system based on video recordings and machine-learning models that can easily be applied for screening of high-risk infants.
Table A2

Performance of CP prediction with different decision thresholds (%) for the proportion of periods with CP risk-related movements. The sensitivity, specificity, positive and negative predictive values and area under the curve (AUC) with 95% confidence intervals in brackets for the prediction of CP.

Threshold (%)Sens. (%)Spec. (%)PPV (%)NPV (%)
5092.7 [80.1, 98.5]81.6 [77.0, 85.5]38.0 [32.5, 43.8]98.9 [96.8, 99.6]
5592.7 [80.1, 98.5]83.9 [79.6, 87.7]41.3 [35.2, 47.7]99.0 [96.9, 99.6]
6085.4 [70.8, 94.4]86.6 [82.5, 90.1]43.6 [36.6, 51.2]98.0 [95.9, 99.0]
6578.1 [62.4, 89.4]89.3 [85.5, 92.4]47.0 [38.5, 55.8]97.1 [94.9, 98.4]
7068.3 [51.9, 81.9]92.3 [88.9, 94.9]51.9 [41.3, 62.2]96.0 [93.8, 97.4]
Table A3

A weak correlation between imaging and CIMA.

Imaging/CIMACP Risk-Related Move > 50%CP Risk-Related Move < 50%
Abnormal MR 4242
Normal MR 57234

Mean square contingency coefficient: r = 0.29.

Table A4

A weak correlation between GMA and CIMA.

GMA/CIMACP Risk-Related Move > 50%CP Risk-Related Move < 50%
Abnormal FMs 4442
Normal FMs 56235

Mean square contingency coefficient: r = 0.30.

Table A5

A weak correlation between imaging and CIMA.

Imaging/GMAAbnormal FMsNormal FMs
Abnormal MR 3549
Normal MR 50241

Mean square contingency coefficient: r = 0.24.

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8.  Frequency Analysis and Feature Reduction Method for Prediction of Cerebral Palsy in Young Infants.

Authors:  Hodjat Rahmati; Harald Martens; Ole Morten Aamo; Oyvind Stavdahl; Ragnhild Stoen; Lars Adde
Journal:  IEEE Trans Neural Syst Rehabil Eng       Date:  2016-03-08       Impact factor: 3.802

Review 9.  Fidgety movements - tiny in appearance, but huge in impact.

Authors:  Christa Einspieler; Robert Peharz; Peter B Marschik
Journal:  J Pediatr (Rio J)       Date:  2016-03-17       Impact factor: 2.197

10.  Cerebral Palsy: Early Markers of Clinical Phenotype and Functional Outcome.

Authors:  Christa Einspieler; Arend F Bos; Magdalena Krieber-Tomantschger; Elsa Alvarado; Vanessa M Barbosa; Natascia Bertoncelli; Marlette Burger; Olena Chorna; Sabrina Del Secco; Raye-Ann DeRegnier; Britta Hüning; Jooyeon Ko; Laura Lucaccioni; Tomoki Maeda; Viviana Marchi; Erika Martín; Catherine Morgan; Akmer Mutlu; Alice Nogolová; Jasmin Pansy; Colleen Peyton; Florian B Pokorny; Lucia R Prinsloo; Eileen Ricci; Lokesh Saini; Anna Scheuchenegger; Cinthia R D Silva; Marina Soloveichick; Alicia J Spittle; Moreno Toldo; Fabiana Utsch; Jeanetta van Zyl; Carlos Viñals; Jun Wang; Hong Yang; Bilge N Yardımcı-Lokmanoğlu; Giovanni Cioni; Fabrizio Ferrari; Andrea Guzzetta; Peter B Marschik
Journal:  J Clin Med       Date:  2019-10-04       Impact factor: 4.241

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  16 in total

1.  Development and Validation of a Deep Learning Method to Predict Cerebral Palsy From Spontaneous Movements in Infants at High Risk.

Authors:  Daniel Groos; Lars Adde; Sindre Aubert; Lynn Boswell; Raye-Ann de Regnier; Toril Fjørtoft; Deborah Gaebler-Spira; Andreas Haukeland; Marianne Loennecken; Michael Msall; Unn Inger Möinichen; Aurelie Pascal; Colleen Peyton; Heri Ramampiaro; Michael D Schreiber; Inger Elisabeth Silberg; Nils Thomas Songstad; Niranjan Thomas; Christine Van den Broeck; Gunn Kristin Øberg; Espen A F Ihlen; Ragnhild Støen
Journal:  JAMA Netw Open       Date:  2022-07-01

2.  Temporal and spatial localisation of general movement complexity and variation-Why Gestalt assessment requires experience.

Authors:  Ying-Chin Wu; Ilse M van Rijssen; Maria T Buurman; Linze-Jaap Dijkstra; Elisa G Hamer; Mijna Hadders-Algra
Journal:  Acta Paediatr       Date:  2020-06-22       Impact factor: 2.299

3.  In-Motion-App for remote General Movement Assessment: a multi-site observational study.

Authors:  Lars Adde; Annemette Brown; Christine van den Broeck; Kris DeCoen; Beate Horsberg Eriksen; Toril Fjørtoft; Daniel Groos; Espen Alexander F Ihlen; Siril Osland; Aurelie Pascal; Henriette Paulsen; Ole Morten Skog; Wiebke Sivertsen; Ragnhild Støen
Journal:  BMJ Open       Date:  2021-03-04       Impact factor: 2.692

4.  3D Motion Capture May Detect Spatiotemporal Changes in Pre-Reaching Upper Extremity Movements with and without a Real-Time Constraint Condition in Infants with Perinatal Stroke and Cerebral Palsy: A Longitudinal Case Series.

Authors:  Julia Mazzarella; Mike McNally; Daniel Richie; Ajit M W Chaudhari; John A Buford; Xueliang Pan; Jill C Heathcock
Journal:  Sensors (Basel)       Date:  2020-12-19       Impact factor: 3.576

Review 5.  The future of General Movement Assessment: The role of computer vision and machine learning - A scoping review.

Authors:  Nelson Silva; Dajie Zhang; Tomas Kulvicius; Alexander Gail; Carla Barreiros; Stefanie Lindstaedt; Marc Kraft; Sven Bölte; Luise Poustka; Karin Nielsen-Saines; Florentin Wörgötter; Christa Einspieler; Peter B Marschik
Journal:  Res Dev Disabil       Date:  2021-02-08

6.  A Setup for Camera-Based Detection of Simulated Pathological States Using a Neonatal Phantom.

Authors:  Florian Voss; Simon Lyra; Daniel Blase; Steffen Leonhardt; Markus Lüken
Journal:  Sensors (Basel)       Date:  2022-01-26       Impact factor: 3.576

Review 7.  Applications of Pose Estimation in Human Health and Performance across the Lifespan.

Authors:  Jan Stenum; Kendra M Cherry-Allen; Connor O Pyles; Rachel D Reetzke; Michael F Vignos; Ryan T Roemmich
Journal:  Sensors (Basel)       Date:  2021-11-03       Impact factor: 3.576

8.  Estimation of Gross Motor Functions in Children with Cerebral Palsy Using Zebris FDM-T Treadmill.

Authors:  Mariusz Bedla; Paweł Pięta; Daniel Kaczmarski; Stanisław Deniziak
Journal:  J Clin Med       Date:  2022-02-12       Impact factor: 4.241

9.  Writhing Movement Detection in Newborns on the Second and Third Day of Life Using Pose-Based Feature Machine Learning Classification.

Authors:  Iwona Doroniewicz; Daniel J Ledwoń; Alicja Affanasowicz; Katarzyna Kieszczyńska; Dominika Latos; Małgorzata Matyja; Andrzej W Mitas; Andrzej Myśliwiec
Journal:  Sensors (Basel)       Date:  2020-10-22       Impact factor: 3.576

Review 10.  AI Approaches Towards Prechtl's Assessment of General Movements: A Systematic Literature Review.

Authors:  Muhammad Tausif Irshad; Muhammad Adeel Nisar; Philip Gouverneur; Marion Rapp; Marcin Grzegorzek
Journal:  Sensors (Basel)       Date:  2020-09-17       Impact factor: 3.576

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