Muscle synergy structure and gait patterns in children with spastic cerebral palsy.

AIM: To determine if muscle synergy structure (activations and weights) differs between gait patterns in children with spastic cerebral palsy (CP). METHOD: In this cross-sectional study, we classified 188 children with unilateral (n=82) or bilateral (n=106) spastic CP (mean age: 9y 5mo, SD: 4y 3mo, range: 3y 9mo-17y 7mo; 75 females; Gross Motor Function Classification System [GMFCS] level I: 106, GMFCS level II: 55, GMFCS level III: 27) into a minor deviations (n=34), drop foot (n=16), genu recurvatum (n=26), apparent equinus (n=53), crouch (n=39), and jump gait pattern (n=20). Surface electromyography recordings from eight lower limb muscles of the most affected side were used to calculate synergies with weighted non-negative matrix factorization. We compared synergy activations and weights between the patterns. RESULTS: Synergy structure was similar between gait patterns, although weights differed in the more impaired children (crouch and jump gait) when compared to the other patterns. Variability in synergy structure between participants was high. INTERPRETATION: The similarity in synergy structure between gait patterns suggests a generic motor control strategy to compensate for the brain lesion. However, the differences in weights and high variability between participants indicate that this generic motor control strategy might be individualized and dependent on impairment level.

Instrumented assessment of motor function in dyskinetic cerebral palsy: a systematic review.

BACKGROUND: In this systematic review we investigate which instrumented measurements are available to assess motor impairments, related activity limitations and participation restrictions in children and young adults with dyskinetic cerebral palsy. We aim to classify these instrumented measurements using the categories of the international classification of functioning, disability and health for children and youth (ICF-CY) and provide an overview of the outcome parameters. METHODS: A systematic literature search was performed in November 2019. We electronically searched Pubmed, Embase and Scopus databases. Search blocks included (a) cerebral palsy, (b) athetosis, dystonia and/or dyskinesia, (c) age 2-24 years and (d) instrumented measurements (using keywords such as biomechanics, sensors, smartphone, and robot). RESULTS: Our search yielded 4537 articles. After inspection of titles and abstracts, a full text of 245 of those articles were included and assessed for further eligibility. A total of 49 articles met our inclusion criteria. A broad spectrum of instruments and technologies are used to assess motor function in dyskinetic cerebral palsy, with the majority using 3D motion capture and surface electromyography. Only for a small number of instruments methodological quality was assessed, with only one study showing an adequate assessment of test-retest reliability. The majority of studies was at ICF-CY function and structure level and assessed control of voluntary movement (29 of 49) mainly in the upper extremity, followed by assessment of involuntary movements (15 of 49), muscle tone/motor reflex (6 of 49), gait pattern (5 of 49) and muscle power (2 of 49). At ICF-CY level of activities and participation hand and arm use (9 of 49), fine hand use (5 of 49), lifting and carrying objects (3 of 49), maintaining a body position (2 of 49), walking (1 of 49) and moving around using equipment (1 of 49) was assessed. Only a few methods are potentially suitable outside the clinical environment (e.g. inertial sensors, accelerometers). CONCLUSION: Although the current review shows the potential of several instrumented methods to be used as objective outcome measures in dyskinetic cerebral palsy, their methodological quality is still unknown. Future development should focus on evaluating clinimetrics, including validating against clinical meaningfulness. New technological developments should aim for measurements that can be applied outside the laboratory.

Muscle synergies demonstrate only minimal changes after treatment in cerebral palsy.

BACKGROUND: Children with cerebral palsy (CP) have altered synergies compared to typically-developing peers, reflecting different neuromuscular control strategies used to move. While these children receive a variety of treatments to improve gait, whether synergies change after treatment, or are associated with treatment outcomes, remains unknown. METHODS: We evaluated synergies for 147 children with CP before and after three common treatments: botulinum toxin type-A injection (n = 52), selective dorsal rhizotomy (n = 38), and multi-level orthopaedic surgery (n = 57). Changes in synergy complexity were measured by the number of synergies required to explain > 90% of the total variance in electromyography data and total variance accounted for by one synergy. Synergy weights and activations before and after treatment were compared using the cosine similarity relative to average synergies of 31 typically-developing (TD) peers. RESULTS: There were minimal changes in synergies after treatment despite changes in walking patterns. Number of synergies did not change significantly for any treatment group. Total variance accounted for by one synergy increased (i.e., moved further from TD peers) after botulinum toxin type-A injection (1.3%) and selective dorsal rhizotomy (1.9%), but the change was small. Synergy weights did not change for any treatment group (average 0.001 ± 0.10), but synergy activations after selective dorsal rhizotomy did change and were less similar to TD peers (- 0.03 ± 0.07). Only changes in synergy activations were associated with changes in gait kinematics or walking speed after treatment. Children with synergy activations more similar to TD peers after treatment had greater improvements in gait. CONCLUSIONS: While many of these children received significant surgical procedures and prolonged rehabilitation, the minimal changes in synergies after treatment highlight the challenges in altering neuromuscular control in CP. Development of treatment strategies that directly target impaired control or are optimized to an individual's unique control may be required to improve walking function.

Associations Between Muscle Synergies and Treatment Outcomes in Cerebral Palsy Are Robust Across Clinical Centers.

OBJECTIVE: To determine whether patient-specific differences in motor control quantified using muscle synergy analysis were associated with changes in gait after treatment of cerebral palsy (CP) across 2 clinical centers with different treatments and clinical protocols. DESIGN: Retrospective cohort study. SETTING: Clinical medical center. PARTICIPANTS: Center 1: children with CP (n=473) and typically developing (TD) children (n=84). Center 2: children with CP (n=163) and TD children (n=12). INTERVENTIONS: Standard clinical care at each center. MAIN OUTCOME MEASURES: The Dynamic Motor Control Index During Walking (walk-DMC) was computed from electromyographic data during gait using muscle synergy analysis. Regression analysis was used to evaluate whether pretreatment walking speed or kinematics, muscle synergies, treatment group, prior treatment, or age were associated with posttreatment changes in gait at both clinical centers. RESULTS: Walk-DMC was significantly associated with changes in speed and kinematics after treatment with similar regression models at both centers. Children with less impaired motor control were more likely to have improvements in walking speed and gait kinematics after treatment, independent of treatment group. CONCLUSIONS: Dynamic motor control evaluated with synergy analysis was associated with changes in gait after treatment at both centers, despite differences in treatments and clinical protocols. This study further supports the finding that walk-DMC provides additional information, not captured in traditional gait analysis, that may be useful for treatment planning.

Influence of musculoskeletal pain during gait on kinematics and selective motor control in individuals with spastic cerebral palsy: A pilot study.

BACKGROUND: Individuals with cerebral-palsy commonly present with altered kinematics and selective-motor-control during gait, and may also experience musculoskeletal pain. This pilot study aims to investigate if the immediate experience of musculoskeletal pain during gait influences kinematics and selective-motor-control in individuals with spastic cerebral-palsy. METHODS: Retrospective treadmill-based gait-analysis data for 145 individuals with spastic cerebral-palsy were screened. Participants were asked about experiencing lower-extremity musculoskeletal pain immediately during gait, with 26 individuals (18%) reporting this was the case (pain-group; mean 11.55 ± 3.15 years, Gross-Motor-Function-Classification-System levels I/II/III n = 5/13/8, Uni/bilateral involvement n = 11/15). Of the 77 individuals who did not report any pain, a no-pain group (n = 26) was individually matched. Kinematics were evaluated using the Gait-Profile-Score and spatiotemporal parameters (dimensionless-walking-speed, single-leg-support percentage and step-time). Selective-motor-control was assessed using the Walking-Dynamic-Motor-Control index. FINDINGS: In the pain-group, 58% reported experiencing pain in their more-involved leg, 8% in the less-involved leg and 34% in both legs. Regarding the pain location, 38% of the pain-group reported experiencing pain in multiple locations. On a more specific level, 35%, 46% and 54% reported pain around the hip/thigh, knee/calf and ankle/ft, respectively. No significant differences were observed between the pain and no-pain groups for any of the outcome measures, in each leg or bilaterally. INTERPRETATION: No significant differences in kinematics and selective-motor-control during gait were found between individuals with spastic cerebral-palsy, with and without musculoskeletal pain. This suggests that the individuals in this study may not present with obvious antalgic gait patterns, which may relate to the pre-existing altered kinematics and selective-motor-control.

Is dynamic motor control clinically important for identifying gait deviations in individuals with cerebral palsy?

INTRODUCTION: Individuals with cerebral palsy (CP) often present with altered motor control. This can be assessed selectively during sitting/lying with the Selective Control Assessment of the Lower Extremity (SCALE), or dynamically with the dynamic motor control index during walking (walk-DMC). Both approaches suggest that altered selective motor control relate to larger gait deviations. RESEARCH QUESTION: Does the walk-DMC provide valuable information in addition to the SCALE for estimating gait deviations in individuals with CP. METHODS: Retrospective, treadmill-based gait analysis data of 157 children with spastic CP (mean 11.4±3.5 years) and Gross Motor Function Classification System levels I (n=45), II (n=88) or III (n=24) were extracted. Gait kinematic deviations were evaluated using the Gait Profile Score (GPS). The SCALE, walk-DMC and GPS were extracted for the more clinically involved leg (unilateral-analysis), and for both legs together (bilateral-analysis). RESULTS: GPS moderately correlated with both SCALE and walk-DMC scores, unilaterally and bilaterally (r≥0.4; p<0.001). Multivariate linear regression analyses were conducted, taking into account potential confounding factors. In the unilateral analysis, 54% of the GPS variance was explained (p<0.001), with both walk-DMC and SCALE significantly contributing to the GPS variance (p=0.006 and p=0.008, respectively). In the bilateral analysis, 61% of the GPS variance was explained (p<0.001), with both walk-DMC and SCALE significantly contributing to the GPS variance (p=0.006 and p<0.001, respectively). Dimensionless walking speed and use of assistive devices were the only confounding factors included in each analysis. SIGNIFICANCE: Both SCALE and walk-DMC significantly contribute to GPS variance, suggesting that they likely measure different components of motor control, and both may be useful in understanding the underlying relationship between motor control and deviations in gait kinematics.

Exploring harmonic walking development in children with unilateral cerebral palsy and typically developing toddlers: Insights from walking experience.

This longitudinal study investigated the impact of the first independent steps on harmonic gait development in unilateral cerebral palsy (CP) and typically developing (TD) children. We analysed the gait ratio values (GR) by comparing the duration of stride/stance, stance/swing and swing/double support phases. Our investigation focused on identifying a potential trend towards the golden ratio value of 1.618, which has been observed in the locomotion of healthy adults as a characteristic of harmonic walking. Locomotor ability was assessed in both groups at different developmental stages: before and after the emergence of independent walking. Results revealed that an exponential fit was observed only after the first unsupported steps were taken. TD children achieved harmonic walking within a relatively short period (approximately one month) compared to children with CP, who took about seven months to develop harmonic walking. Converging values for stride/stance and stance/swing gait ratios, averaged on the two legs, closely approached the golden ratio in TD children (R2 = 0.9) with no difference in the analysis of the left vs right leg separately. In contrast, children with CP exhibited a trend for stride/stance and stance/swing (R2 = 0.7), with distinct trends observed for the most affected leg which did not reach the golden ratio value for the stride/stance ratio (GR = 1.5), while the least affected leg exceeded it (GR = 1.7). On the contrary, the opposite trend was observed for the stance/swing ratio. These findings indicate an overall harmonic walking in children with CP despite the presence of asymmetry between the two legs. These results underscore the crucial role of the first independent steps in the progressive development of harmonic gait over time.

Current practices in clinical gait analysis in Europe: A comprehensive survey-based study from the European society for movement analysis in adults and children (ESMAC) standard initiative.

BACKGROUND: Clinical gait analysis (CGA) is a systematic approach to comprehensively evaluate gait patterns, quantify impairments, plan targeted interventions, and evaluate the impact of interventions. However, international standards for CGA are currently lacking, resulting in various national initiatives. Standards are important to ensure safe and effective healthcare practices and to enable evidence-based clinical decision-making, facilitating interoperability, and reimbursement under national healthcare policies. Collaborative clinical and research work between European countries would benefit from common standards. RESEARCH OBJECTIVE: This study aimed to review the current laboratory practices for CGA in Europe. METHODS: A comprehensive survey was conducted by the European Society for Movement Analysis in Adults and Children (ESMAC), in close collaboration with the European national societies. The survey involved 97 gait laboratories across 16 countries. The survey assessed several aspects related to CGA, including equipment used, data collection, processing, and reporting methods. RESULTS: There was a consensus between laboratories concerning the data collected during CGA. The Conventional Gait Model (CGM) was the most used biomechanical model for calculating kinematics and kinetics. Respondents also reported the use of video recording, 3D motion capture systems, force plates, and surface electromyography. While there was a consensus on the reporting of CGA data, variations were reported in training, documentation, data preprocessing and equipment maintenance practices. SIGNIFICANCE: The findings of this study will serve as a foundation for the development of standardized guidelines for CGA in Europe.

A longitudinal analysis of selective motor control during gait in individuals with cerebral palsy and the relation to gait deviations.

OBJECTIVE: To investigate longitudinal changes in selective motor control during gait (SMCg) in individuals with cerebral palsy (CP), and to assess if they are related to changes in gait deviations. METHOD: Twenty-three children/adolescents with spastic CP (mean ± SD age = 9.0±2.5 years) and two 3D gait assessments (separated by 590±202 days) with no interim surgical intervention, were included. SMCg was assessed using muscle synergy analysis to determine the dynamic motor control index (walk-DMC). Gait deviation was assessed using the Gait profile score (GPS) and Gait variable scores (GVS). RESULTS: There were no mean changes in walk-DMC score, GPS or GVS between assessments. However, changes in walk-DMC scores in the more involved leg related to changes in hip flexion-extension and hip internal-external GVS (rp = -0.56; p = 0.017 and rp = 0.65; p = 0.004, respectively). CONCLUSIONS: On average, there were no significant longitudinal changes in SMCg. However, there was considerable variability between individuals, which may relate to changes in hip joint kinematics. This suggests that a combination of neural capacity and biomechanical factors influence lower limb muscle co-activation in individuals with CP, with a potential important role for the hip muscles. These findings highlight the importance of taking an individualized approach when evaluating SMCg in individuals with CP.

Influence of the number of muscles and strides on selective motor control during gait in individuals with cerebral palsy.

OBJECTIVE: To evaluate the influence of the number of muscles and strides on estimating motor control accuracy during treadmill-gait, in individuals with cerebral palsy (CP). METHODS: Bilateral lower limb electromyography data were extracted for 44 children/adolescents with CP. The number of synergy solutions required to explain 90 % of the variance (tVAF-threshold) and the total variance accounted for by one synergy (tVAF1) were calculated for a different number of strides (between 5 and 50) and muscles both unilaterally (four to seven) and bilaterally (eight to 14). The kappa and intraclass correlation coefficients were used to assess similarities in tVAF-threshold and tVAF1 between the different number of strides and muscle sets. RESULTS: In both analyses, the number of muscles influenced the tVAF-threshold. Additionally, using <30 strides led to only substantial-moderate agreement with 50 strides (k < 0.80). In both analyses, the mean tVAF1 values demonstrated high-agreement between the different number of muscles (intraclass-correlations = 0.88-0.93) and strides (intraclass-correlations = 0.96-0.99); In the group level, it may result in an error of ≤2.3 %. However, in the individual level, using different number of muscles or <40 strides may result in an error of ≥6 %. CONCLUSION: Differing numbers of muscles and strides did not influence the group mean tVAF1 value, but it influenced the tVAF-threshold value. In addition, using different number of muscles or strides can lead to a large measurement error in the individual tVAF1 value.

The Role of Walking Experience in the Emergence of Gait Harmony in Typically Developing Toddlers.

The ability to walk without support usually develops in the first year of a typically developing toddler's life and matures further in the following years. Mature walking is characterized by the correct timing of the different gait phases that make up a full gait cycle formed by stance, swing, and double support phases. The harmony of mature walking is given by a specific self-similar structure of the ratios between the durations of these phases (stride/stance, stance/swing, swing/double support), that in adults all converge to the golden ratio (phi, about 1.618). The aim of this longitudinal, prospective, experimental study was to investigate the evolution of this gait harmonic structure in the transition from supported to independent walking. In total, 27 children were recruited and recorded at various stages of locomotor development, ranging from neonatal stepping up to an independent walking experience of about six months. Differently from walking speed that progressively increased with age, the gait phase ratios started to converge to golden value only after the first independent steps, suggesting a relation to the maturation of the walking experience. The independent walking experience seems to represent a trigger for the evolution of a locomotor harmonic pattern in toddlers learning to walk.

Longitudinal Alterations in Gait Features in Growing Children With Duchenne Muscular Dystrophy.

Prolonging ambulation is an important treatment goal in children with Duchenne muscular dystrophy (DMD). Three-dimensional gait analysis (3DGA) could provide sensitive parameters to study the efficacy of clinical trials aiming to preserve ambulation. However, quantitative descriptions of the natural history of gait features in DMD are first required. The overall goal was to provide a full delineation of the progressive gait pathology in children with DMD, covering the entire period of ambulation, by performing a so-called mixed cross-sectional longitudinal study. Firstly, to make our results comparable with previous literature, we aimed to cross-sectionally compare 31 predefined gait features between children with DMD and a typically developing (TD) database (1). Secondly, we aimed to explore the longitudinal changes in the 31 predefined gait features in growing boys with DMD using follow-up 3DGA sessions (2). 3DGA-sessions (n = 124) at self-selected speed were collected in 27 boys with DMD (baseline age: 4.6-15 years). They were repeatedly measured over a varying follow-up period (range: 6 months-5 years). The TD group consisted of 27 children (age: 5.4-15.6 years). Per measurement session, the spatiotemporal parameters, and the kinematic and kinetic waveforms were averaged over the selected gait cycles. From the averaged waveforms, discrete gait features (e.g., maxima and minima) were extracted. Mann-Whitney U tests were performed to cross-sectionally analyze the differences between DMD at baseline and TD (1). Linear mixed effect models were performed to assess the changes in gait features in the same group of children with DMD from both a longitudinal (i.e., increasing time) as well as a cross-sectional perspective (i.e., increasing baseline age) (2). At baseline, the boys with DMD differed from the TD children in 17 gait features. Additionally, 21 gait features evolved longitudinally when following-up the same boys with DMD and 25 gait features presented a significant cross-sectional baseline age-effect. The current study quantitatively described the longitudinal alterations in gait features in boys with DMD, thereby providing detailed insight into how DMD gait deteriorates. Additionally, our results highlight that gait features extracted from 3DGA are promising outcome measures for future clinical trials to quantify the efficacy of novel therapeutic strategies.

Muscle weakness has a limited effect on motor control of gait in Duchenne muscular dystrophy.

AIM: Our aim was to determine if synergy weights and activations are altered in Duchenne muscular dystrophy (DMD) and if these alterations could be linked to muscle weakness. METHODS: In 22 children with DMD and 22 typical developing (TD) children of a similar age, surface electromyography (sEMG) of the gluteus medius, rectus femoris (REF), medial hamstrings, tibialis anterior, and medial gastrocnemius (GAS) were recorded during gait. Muscle weakness was assessed with maximal voluntary isometric contractions (MVIC). Synergies were calculated with non-negative matrix factorization. The number of synergies explaining ≥90% of the variance in the sEMG signals (N90), were extracted and grouped with k-means cluster analysis. We verified differences in weights with a Mann-Whitney U test. Statistical non-parametric mapping (Hotelling's T2 test and two-tailed t-test) was used to assess group differences in synergy activations. We used Spearman's rank correlation coefficients and canonical correlation analysis to assess if weakness was related to modifications in weights and activations, respectively. RESULTS: For both groups, average N90 was three. In synergy one, characterized by activity at the beginning of stance, the DMDs showed an increased REF weight (p = 0.001) and decreased GAS weight (p = 0.007). Synergy activations were similar, with only a small difference detected in mid-swing in the combined activations (p<0.001). Weakness was not associated with these differences. CONCLUSION: Despite the apparent weakness in DMD, synergy weights and activations were similar between the two groups. Our findings are in line with previous research suggesting non-neural alterations have limited influence on muscle synergies.

Muscle Synergy Constraints Do Not Improve Estimates of Muscle Activity From Static Optimization During Gait for Unimpaired Children or Children With Cerebral Palsy.

Neuromusculoskeletal simulation provides a promising platform to inform the design of assistive devices or inform rehabilitation. For these applications, a simulation must be able to accurately represent the person of interest, such as an individual with a neurologic injury. If a simulation fails to predict how an individual recruits and coordinates their muscles during movement, it will have limited utility for informing design or rehabilitation. While inverse dynamic simulations have previously been used to evaluate anticipated responses from interventions, like orthopedic surgery or orthoses, they frequently struggle to accurately estimate muscle activations, even for tasks like walking. The simulated muscle activity often fails to represent experimentally measured muscle activity from electromyographic (EMG) recordings. Research has theorized that the nervous system may simplify the range of possible activations used during dynamic tasks, by constraining activations to weighted groups of muscles, referred to as muscle synergies. Synergies are altered after neurological injury, such as stroke or cerebral palsy (CP), and may provide a method for improving subject-specific models of neuromuscular control. The aim of this study was to test whether constraining simulation to synergies could improve estimated muscle activations compared to EMG data. We evaluated modeled muscle activations during gait for six typically developing (TD) children and six children with CP. Muscle activations were estimated with: (1) static optimization (SO), minimizing muscle activations squared, and (2) synergy SO (SynSO), minimizing synergy activations squared using the weights identified from EMG data for two to five synergies. While SynSO caused changes in estimated activations compared to SO, the correlation to EMG data was not higher in SynSO than SO for either TD or CP groups. The correlations to EMG were higher in CP than TD for both SO (CP: 0.48, TD: 0.36) and SynSO (CP: 0.46, TD: 0.26 for five synergies). Constraining activations to SynSO caused the simulated muscle stress to increase compared to SO for all individuals, causing a 157% increase with two synergies. These results suggest that constraining simulated activations in inverse dynamic simulations to subject-specific synergies alone may not improve estimation of muscle activations during gait for generic musculoskeletal models.

Combining muscle morphology and neuromotor symptoms to explain abnormal gait at the ankle joint level in cerebral palsy.

BACKGROUND: Individuals with spastic cerebral palsy (CP) have neuromotor symptoms contributing towards their gait patterns. However, the role of altered muscle morphology alongside these symptoms is yet to be fully investigated. RESEARCH QUESTION: To what extent can medial gastrocnemius and tibialis anterior volume and echo-intensity, plantar/dorsiflexion strength and selective motor control, plantarflexion spasticity and passive ankle dorsiflexion explain abnormal ankle gait. METHOD: In thirty children and adolescents with spastic CP (8.6 ± 3.4 y/mo) and ten typically developing peers (9.9 ± 2.4 y/mo), normalised muscle volume and echo-intensity were estimated. Both cohorts also underwent three-dimensional gait analysis, whilst for participants with spastic CP, plantar/dorsi-flexion strength and selective motor control, plantarflexion spasticity and maximum ankle dorsiflexion were also measured. The combined contribution of these parameters towards five clinically meaningful features of gait were evaluated, using backwards multiple regression analyses. RESULTS: With respect to the typically developing cohort, the participants with spastic CP had deficits in normalised medial gastrocnemius and tibialis anterior volume of 40% and 33%, and increased echo-intensity values of 19% and 16%, respectively. The backwards multiple regression analyses revealed that the combination of reduced ankle dorsiflexion, muscle volume, plantarflexion strength and dorsiflexion selective motor control could account for 12-62% of the variance in the chosen features of gait. SIGNIFICANCE: The combination of altered muscle morphology and neuromotor symptoms partly explained abnormal gait at the ankle in children with spastic CP. Both should be considered as important measures for informed treatment decision-making, but further work is required to better unravel the complex pathophysiology.

A new strength assessment to evaluate the association between muscle weakness and gait pathology in children with cerebral palsy.

AIM: The main goal of this validation study was to evaluate whether lower limb muscle weakness and plantar flexor rate of force development (RFD) related to altered gait parameters in children with cerebral palsy (CP), when weakness was assessed with maximal voluntary isometric contractions (MVICs) in a gait related test position. As a subgoal, we analyzed intra- and intertester reliability of this new strength measurement method. METHODS: Part 1 -Intra- and intertester reliability were determined with the intra-class correlation coefficient (ICC2,1) in 10 typical developing (TD) children (age: 5-15). We collected MVICs in four lower limb muscle groups to define maximum joint torques, as well as plantar flexor RFD. Part 2 -Validity of the strength assessment was explored by analyzing the relations of lower limb joint torques and RFD to a series of kinematic- and kinetic gait features, the GDI (gait deviation index), and the GDI-kinetic in 23 children with CP (GMFCS I-II; age: 5-15) and 23 TD children (age: 5-15) with Spearman's rank correlation coefficients. RESULTS: Part 1 -The best reliability was found for the torque data (Nm), with the highest ICC2,1 (0.951) for knee extension strength (inter) and the lowest (0.693) for dorsiflexion strength (intra). For plantar flexor RFD, the most reliable window size was 300 milliseconds (ICC2,1: 0.828 (inter) and 0.692 (intra)). Part 2 -The children with CP were significantly weaker than the TD children (p <0.001). Weakness of the dorsiflexors and plantar flexors associated with delayed and decreased knee flexion angle during swing, respectively. No other significant correlations were found. CONCLUSION: While our new strength assessment was reliable, intra-joint correlations between weakness, RFD, and gait deviations were low. However, we found inter-joint associations, reflected by a strong association between plantar- and dorsiflexor weakness, and decreased and delayed knee flexion angle during swing.

The influence of maximum isometric muscle force scaling on estimated muscle forces from musculoskeletal models of children with cerebral palsy.

BACKGROUND: Musculoskeletal models do not include patient-specific muscle forces but rely on a scaled generic model, with muscle forces left unscaled in most cases. However, to use musculoskeletal simulations to inform clinical decision-making in children with cerebral palsy (CP), inclusion of subject-specific muscle forces is of utmost importance in order to represent each child's compensation mechanisms introduced through muscle weakness. RESEARCH AIM: The aims of this study were to (i) evaluate if maximum isometric muscle forces (MIMF) in musculoskeletal models of children with CP can be scaled based on strength measurements obtained with a hand-held-dynamometer (HHD), (ii) evaluate the impact of the HHD based scaling approach and previously published MIMF scaling methods on computed muscle forces during gait, and (iii) compare maximum muscle forces during gait between CP and typically developing (TD) children. METHODS: Strength and motion capture data of six CP and motion capture data of six TD children were collected. The HHD measurements to obtain hip, knee and ankle muscle strength were simulated in OpenSim and used to modify MIMF of the 2392-OpenSim model. These muscle forces were compared to the MIMF scaled on the child's body mass and a scaling approach, which included the body mass and muscle-tendon lengths. OpenSim was used to calculate peak muscle forces during gait. RESULTS: Ankle muscle strength was insufficient to reproduce joint moments during walking when MIMF were scaled based on HHD. During gait, peak hip and knee extensor muscle forces were higher and peak ankle dorsi-flexor forces were lower in CP compared to TD participants. SIGNIFICANCE: HHD measurements can be used to scale MIMF for the hip and knee muscle groups but underestimate the force capacity of the ankle muscle groups during walking. Muscle-tendon-length and mass based scaling methods affected muscle activations but had little influence on peak muscle forces during gait.

Non-neural Muscle Weakness Has Limited Influence on Complexity of Motor Control during Gait.

Cerebral palsy (CP) and Duchenne muscular dystrophy (DMD) are neuromuscular disorders characterized by muscle weakness. Weakness in CP has neural and non-neural components, whereas in DMD, weakness can be considered as a predominantly non-neural problem. Despite the different underlying causes, weakness is a constraint for the central nervous system when controlling gait. CP demonstrates decreased complexity of motor control during gait from muscle synergy analysis, which is reflected by a higher total variance accounted for by one synergy (tVAF1). However, it remains unclear if weakness directly contributes to higher tVAF1 in CP, or whether altered tVAF1 reflects mainly neural impairments. If muscle weakness directly contributes to higher tVAF1, then tVAF1 should also be increased in DMD. To examine the etiology of increased tVAF1, muscle activity data of gluteus medius, rectus femoris, medial hamstrings, medial gastrocnemius, and tibialis anterior were measured at self-selected walking speed, and strength data from knee extensors, knee flexors, dorsiflexors and plantar flexors, were analyzed in 15 children with CP [median (IQR) age: 8.9 (2.2)], 15 boys with DMD [8.7 (3.1)], and 15 typical developing (TD) children [8.6 (2.7)]. We computed tVAF1 from 10 concatenated steps with non-negative matrix factorization, and compared tVAF1 between the three groups with a Mann-Whiney U-test. Spearman's rank correlation coefficients were used to determine if weakness in specific muscle groups contributed to altered tVAF1. No significant differences in tVAF1 were found between DMD [tVAF1: 0.60 (0.07)] and TD children [0.65 (0.07)], while tVAF1 was significantly higher in CP [(0.74 (0.09)] than in the other groups (both p < 0.005). In CP, weakness in the plantar flexors was related to higher tVAF1 (r = -0.72). In DMD, knee extensor weakness related to increased tVAF1 (r = -0.50). These results suggest that the non-neural weakness in DMD had limited influence on complexity of motor control during gait and that the higher tVAF1 in children with CP is mainly related to neural impairments caused by the brain lesion.

Gait deviations in Duchenne muscular dystrophy-Part 1. A systematic review.

BACKGROUND: Although prolonged ambulation is considered important in children with Duchenne muscular dystrophy (DMD), articles describing gait deviations in DMD are scarce. RESEARCH QUESTION: Therefore, our research questions were the following: 1) what are the most consistently reported spatiotemporal-, kinematic-, kinetic-, and muscle activity deviations in children with DMD in literature, 2) what is the quality of the studies describing these deviations, and 3) is there need for further research? METHODS: We conducted a systematic literature search for studies published before the end of June 2017 in six online databases. We created a data extraction form to define information on materials and methods and on the analyzed gait parameters for each paper included in the review. If enough information was available, we calculated standardized mean differences (SMDs). RESULTS: The search yielded nine articles, but generalizability was poor. Seventy-nine parameters were analyzed by seven research groups, but they only agreed on a decrease in walking speed (minimal SMD: 1.26), stride length (1.83), step length (1.80), dorsiflexion during swing (1.43), maximal power generation at the hip (0.92), maximal knee extension torque (0.99), maximal dorsiflexion torque (-1.30), and maximal power generation at the ankle (0.92), and an increased knee range of motion (-0.82) in DMD. SIGNIFICANCE: In order to keep children with DMD ambulant as long as possible, a clear understanding of their pathological gait pattern is necessary. However, gait deviations in DMD appear not well defined. Previous studies appear to be of an exploratory nature while using predefined gait parameters to assess an undirected null hypothesis. This made them prone to regional focus bias, thereby increasing the chance of a type I error. Therefore, further research is required to define the altered gait pattern in children with DMD.

Gait deviations in Duchenne muscular dystrophy-Part 2. Statistical non-parametric mapping to analyze gait deviations in children with Duchenne muscular dystrophy.

BACKGROUND: Prolonged ambulation is considered important in children with Duchenne muscular dystrophy (DMD). However, previous studies analyzing DMD gait were sensitive to false positive outcomes, caused by uncorrected multiple comparisons, regional focus bias, and inter-component covariance bias. Also, while muscle weakness is often suggested to be the main cause for the altered gait pattern in DMD, this was never verified. RESEARCH QUESTION: Our research question was twofold: 1) are we able to confirm the sagittal kinematic and kinetic gait alterations described in a previous review with statistical non-parametric mapping (SnPM)? And 2) are these gait deviations related to lower limb weakness? METHODS: We compared gait kinematics and kinetics of 15 children with DMD and 15 typical developing (TD) children (5-17 years), with a two sample Hotelling's T2 test and post-hoc two-tailed, two-sample t-test. We used canonical correlation analyses to study the relationship between weakness and altered gait parameters. For all analyses, α-level was corrected for multiple comparisons, resulting in α = 0.005. RESULTS: We only found one of the previously reported kinematic deviations: the children with DMD had an increased knee flexion angle during swing (p = 0.0006). Observed gait deviations that were not reported in the review were an increased hip flexion angle during stance (p = 0.0009) and swing (p = 0.0001), altered combined knee and ankle torques (p = 0.0002), and decreased power absorption during stance (p = 0.0001). No relationships between weakness and these gait deviations were found. SIGNIFICANCE: We were not able to replicate the gait deviations in DMD previously reported in literature, thus DMD gait remains undefined. Further, weakness does not seem to be linearly related to altered gait features. The progressive nature of the disease requires larger study populations and longitudinal analyses to gain more insight into DMD gait and its underlying causes.