Locomotion Efficiency in Children With Cerebral Palsy Experiencing Limited Gross Motor Function: Walking Versus Cycling.

PURPOSE: This study compares cycling and walking efficiency, and energy expenditure in children with bilateral spastic cerebral palsy (CP). In children with CP, locomotion with body weight support aids such as a tricycle is a potential alternative for less exhausting movements. METHODS: Nine children with CP traveled at comfortable speed for 6 minutes by cycling and walking. The energy expenditure index (EEI) and the percentage of the reserve heart rate (%HRR) were calculated. RESULTS: The EEI was lower while cycling than walking, the traveled distance was higher while cycling than walking, and %HRR remained similar between cycling and walking. CONCLUSION: Cycling appears an efficient alternative to walking for children with CP for adapted school environments and in the community.

Static Postural Control in Youth With Osteogenesis Imperfecta Type I.

OBJECTIVES: To assess static postural control in eyes-open and eyes-closed conditions in individuals with osteogenesis imperfecta (OI) type I as compared with typically developing (TD) individuals and to explore the relation between postural control and lower limb muscle function. DESIGN: Cross-sectional study. SETTING: Outpatient department of a pediatric orthopedic hospital. PARTICIPANTS: A convenience sample (N=38) of individuals with OI type I (n=22; mean age, 13.1y; range, 6-21y) and TD individuals (n=16; mean age, 13.1y; range, 6-20y) was selected. Participants were eligible if they were between 6 and 21 years and if they did not have any fracture or surgery in the lower limb in the 12 months before testing. INTERVENTIONS: Not applicable. MAIN OUTCOMES MEASURES: Postural control was assessed through static balance tests and muscle function through mechanographic tests on a force platform. Selected postural parameters were path length, velocity, 90% confidence ellipse area, and the ellipse's length of the mediolateral and anteroposterior axes. Mechanographic parameters were peak force and peak power as measured using the multiple two-legged hopping and the single two-legged jump test, respectively. RESULTS: Individuals with OI type I had poorer postural control than did TD individuals as indicated by longer and faster displacements and a larger ellipse area. Muscle function was unrelated to postural control in the OI group. Removing visual information resulted in a larger increase in postural control parameters in the OI group than in the TD group. CONCLUSIONS: A proprioceptive deficit could explain poorer postural control in individuals with OI type I.

Gross Motor Function Measure Evolution Ratio: Use as a Control for Natural Progression in Cerebral Palsy.

OBJECTIVE: To develop a new way to interpret Gross Motor Function Measure (GMFM-66) score improvement in studies conducted without control groups in children with cerebral palsy (CP). DESIGN: The curves, which describe the pattern of motor development according to the children's Gross Motor Function Classification System level, were used as historical control to define the GMFM-66 expected natural evolution in children with CP. These curves have been modeled and generalized to fit the curve to particular children characteristics. SETTING: Research center. PARTICIPANTS: Not applicable. INTERVENTIONS: Not applicable. MEAN OUTCOME MEASURES: Not applicable. RESULTS: Assuming that the GMFM-66 score evolution followed the shape of the Rosenbaum curves, by taking into account the age and GMFM-66 score of children, the expected natural evolution of the GMFM-66 score was predicted for any group of children with CP who were <8 years old. Because the expected natural evolution could be predicted for a specific group of children with CP, the efficacy of a treatment could be determined by comparing the GMFM-66 score evolution measured before and after treatment with the expected natural evolution for the same period. A new index, the Gross Motor Function Measure Evolution Ratio, was defined as follows: Gross Motor Function Measure Evolution Ratio=measured GMFM-66 score change/expected natural evolution. CONCLUSIONS: For practical or ethical reasons, it is almost impossible to use control groups in studies evaluating effectiveness of many therapeutic modalities. The Gross Motor Function Measure Evolution Ratio gives the opportunity to take into account the expected natural evolution of the gross motor function of children with CP, which is essential to accurately interpret the therapy effect on the GMFM-66.

Effect of power training on locomotion capacities in children with cerebral palsy with GMFCS level III-IV.

PURPOSE: Power training (PT) is a promising training modality to improve functional abilities in children with cerebral palsy (CP). This study aimed to implement PT in an adapted school and to assess its effect on locomotion capacities in children with Gross Motor Function Classification System (GMFCS) level III-IV. MATERIALS AND METHODS: Nine children with CP (GMFCS level III-IV) were trained three times/week for 12 weeks. The training sessions took place during the 50-minute physical activity classes and included high intensity exercise. The outcome measures were the performance on the 10-meter comfortable and fast walking tests, the 50-meter sprint test (50M-ST), and the energy expenditure index (EEI). RESULTS: Participants spent 12 ± 7 and 7 ± 9 min per session at an intensity superior to 40% and 60% of the heart rate reserve, respectively. Performance in the 10-meter walking test (13.5 ± 7.8 to 9.9 ± 4.6 s, p < 0.05), the 10-meter fast walking test (8.8 ± 3.1 to 7.0 ± 3.2 s, p < 0.05), the 6-minute walking exercise (199.0 ± 48.6 to 316.6 ± 107.2 m, p < 0.05), and in 50M-ST (53.8 ± 29.5 to 42.3 ± 16.2 s, p < 0.05) increased after training. The EEI was reduced after training (p = 0.01), resulting in a more efficient gait. CONCLUSIONS: PT was successfully implemented in children with CP with GMFCS level III-IV. Results suggest that PT increases walking capacities.Implications for rehabilitationPower training (PT) is feasible with children with bilateral cerebral palsy with GMFCS level III and IV.Physical education course in specialist school is a valuable environment to implement PT in children with poor gross motor function.Power training results in locomotion capacities improvement.

How many observations in the reference dataset are required to compute a consistent Gait Deviation Index & Gait Profile Score?

BACKGROUND: The Gait Deviation Index (GDI) and the Gait Profile Score (GPS) are the most used scores to sum up gait deviations and are used as primary outcomes in many clinical studies. They are considered as equivalent scores. The computation of these scores is based on a reference dataset but often no description is provided. Among other characteristics, the number of observations needed and its possible influence on the computation of the scores remains unknown. RESEARCH QUESTION: Define the number of observations needed in the reference dataset to compute consistent and reliable GDI and GPS. METHODS: Fifty individuals with cerebral palsy (CP) were randomly selected from our laboratory database. Both scores were computed based on the reference dataset of Schwartz et al. (2008). A bootstrap analysis was performed, for every individual, to assess the effect of the number of observations on both scores. N number of observations were randomly selected, with replacement, from the reference dataset. This procedure was repeated 2000 times for every individual and every N and performed from N = 5 to N = 165 with an increment of 5. The 95 % of the absolute error distribution was considered for every individual and every N. The smallest detectable change (SDC) for both scores was considered as a threshold (GDI: 10.8; GPS:1.3°) to determine the minimum N required. RESULTS AND SIGNIFICANCE: A minimum of 90 and 20 observations are required to compute consistent GDI and GPS, respectively. The number of observations has a higher impact on the GDI than the GPS, mainly because the GPS calculation does not rely on the standard deviation (SD). Furthermore, the GDI absolute error seems to be higher in individuals with greater gait deviations, i.e. lower GDI value. This effect was not observed on the GPS. In the case of a small reference dataset, the GPS should therefore be preferred.

Static Postural Control Deficits in Adults with Myotonic Dystrophy Type 1, Steinert Disease.

BACKGROUND: Myotonic dystrophy type 1 (DM1) is characterized by progressive and predominantly distal muscle atrophy and myotonia. Gait and balance impairments, resulting in falls, are frequently reported in this population. However, the extent to which individuals with DM1 rely more on a specific sensory system for balance than asymptomatic individuals (AI) is unknown. OBJECTIVE: Evaluate postural control performance in individuals with DM1 and its dependence on vision compared to AI. METHODS: 20 participants with DM1, divided into two groups based on their diagnosis, i.e. adult and congenital phenotype, and 12 AI participants were recruited. Quiet standing postural control was assessed in two visual conditions: eyes-open and eyes-closed. The outcomes measures were: center of pressure (CoP), mean velocity, CoP range of displacement in anteroposterior and mediolateral axis, and the 95% confidence ellipse's surface. Friedman and Kruskal-Wallis analysis of variance were used to compare outcomes between conditions and groups, respectively. RESULTS: Significant group effect and condition effect were observed on postural control performance. No significant difference was observed between the two DM1 groups. The significant differences observed between the AI group and the two DM1 groups in the eyes-open condition were also observed in the eyes-closed condition. CONCLUSIONS: The result revealed poorer postural control performance in people with DM1 compared to AI. The DM1 group also showed similar decrease in performance than AI in eyes-closed condition, suggesting no excessive visual dependency.

Running capacity in children with bilateral cerebral palsy: What are the biomechanical and neuromotor differences between runners and walkers?

BACKGROUND: Running is a high-level locomotor activity requiring more from joints, muscles and a more complex interaction of the neuromuscular system than walking. High-level locomotor activity has the potential to shed light on motor function deficits that lower-level activity does not reveal. Therefore, the study aimed to compare biomechanical and neuromotor function between a group of children with bilateral cerebral palsy who are able and unable to run. METHODS: Retrospectively, children with bilateral cerebral palsy aged between 6 and 18 years who completed a clinical gait analysis between 2006 and 2019 were included. Participants were categorized as walkers or runners based on the presence of a double floating phase. Spasticity, selectivity, muscle weakness, and passive range of motion of the lower limbs were measured and dichotomized as «normal¼ or «abnormal¼ based on reference values. Functional tasks reflecting balance (standing on one leg) and power (single leg and two-legged jumps) were realized and evaluated as failure or success. FINDINGS: 75 children with bilateral cerebral palsy (53 runners/22 walkers) were included. Children classified as runners were stronger (hip flexors, p = 0.006; hip abductors, p = 0.022; knee flexors, p = 0.001; dorsiflexors, p = 0.014), had greater selectivity (hip flexors, p = 0.011; dorsiflexors, p = 0.001; plantiflexors, p = 0.043) and lower spasticity at the knee extensors (p = 0.045). No differences were observed in the passive range of motion between the two groups. Children classified as runners performed better at all tasks of balance and power (p < 0.05). INTERPRETATION: Flexors muscles strength and selectivity and knee extensor spasticity are key points for running ability in children with bilateral cerebral palsy.

Relationship between lower limb strength and walking capacities in children with spastic bilateral cerebral palsy.

PURPOSE: Evaluate the relationship between different walking capacities and muscle strength in children with bilateral cerebral palsy (BCP) and assess these relationships in stronger and weaker children. MATERIALS AND METHODS: Thirty-two children with spastic BCP were included. All participants walked under three speed conditions: comfortable, fast, and for a longer period (6 min). Walking speeds, Energy Expenditure Index (EEI), and lower limb muscle strength were measured. A global strength index (GSI) was computed as the sum of each muscle group strength. Pearson's coefficient and regression models were computed between walking capacities and the GSI. RESULTS: GSI was correlated with the EEI and all walking speeds. Logarithmic regressions models explained between 24 and 34% of the variance of walking capacities. Then, the group was divided in two subgroups (weaker and stronger children). GSI was correlated with comfortable and endurance waking speed in weaker children, but not in stronger children. CONCLUSION: This study reports logarithmic relationship between muscle strength and walking capacities in children with BCP. The subgroup analysis implies that muscle strength has an impact on walking capacities solely in weaker children, suggesting that muscle strength must be preserved and reinforced in interventions targeting motor function in weaker children with BCP.Implications for rehabilitationIn a sample of children with spastic bilateral cerebral palsy, this study shows that global muscle strength is associated with walking capacities and the relationship seems more complex than linear.Based on the results, interventions should focus on maintaining or improving muscle strength in weaker children as no association was observed between muscle strength and walking capacities in stronger children.In stronger children, intervention should focus on factors other than muscle strength as it does not influence walking capacities.Based on this study, a more accurate screening of children who could benefit from strength training could be completed by initial global muscle strength.

Are Clinical Impairments Related to Kinematic Gait Variability in Children and Young Adults With Cerebral Palsy?

Intrinsic gait variability (GV), i.e., fluctuations in the regularity of gait patterns between repetitive cycles, is inherent to the sensorimotor system and influenced by factors such as age and pathology. Increased GV is associated with gait impairments in individuals with cerebral palsy (CP) and has been mainly studied based on spatiotemporal parameters. The present study aimed to describe kinematic GV in young people with CP and its associations with clinical impairments [i.e., passive range of motion (pROM), muscle weakness, reduced selective motor control (selectivity), and spasticity]. This retrospective study included 177 participants with CP (age range 5-25 years; Gross Motor Function Classification System I-III) representing 289 clinical gait analyses [n = 172 for unilateral CP (uCP) vs. 117 for bilateral CP (bCP)]. As variability metrics, Root Mean Square Deviation (RMSD) for nine lower-limb kinematic parameters and Gait Standard Deviation (GaitSD) - as composite score of the kinematic parameters - were computed for the affected (unilateral = uCP) and most affected side (bilateral = bCP), respectively, as defined by clinical scores. GaitSD was then computed for the non/less-affected side for between leg comparisons. Uni- and multivariate linear regressions were subsequently performed on GaitSD of the affected/most affected side with all clinical impairments (composite scores) as independent variables. Highest RMSD were found in the transverse plane (hip, pelvis), for distal joints in the sagittal plane (knee, ankle) and for foot progression. GaitSD was not different between uCP and bCP (affected/most affected side) but higher in the non-affected vs. affected side in uCP. GaitSD was associated with age (p < 0.001), gait deviation index (GDI) (p < 0.05), muscle weakness (p < 0.001), selectivity (p < 0.05), and pROM (p < 0.001). After adjustment for age and GDI, GaitSD remained associated with muscle weakness (uCP: p = 0.003, bCP: p < 0.001) and selectivity (bCP: p = 0.024). Kinematic GV can be expressed as global indicator of variability (GaitSD) in young people with CP given the strong correlation of RMSD for lower-limb kinematic parameters. In terms of asymmetry, increased variability of the non-affected vs. affected side may indicate contralateral compensation mechanisms in uCP. Notably muscle weakness (uCP, bCP) and selectivity (bCP) - but not spasticity - were associated with GaitSD. Further studies need to explore the clinical relevance of kinematic GV in CP to support the interpretation of clinical gait analyses and therapeutic decision-making.

What can we learn from the relationship between gait deviations and clinical impairments when comparing two databases?

BACKGROUND: Several previous studies have tried to determine the relationship between gait and clinical impairments in children with Cerebral Palsy (CP). The heterogeneity of the population and the methodology used could explain the discrepancy within the results. Recently, Papageorgiou et al. (2019) used a Statistical Parametric Mapping (SPM) analysis to investigate this relationship, allowing to test across the kinematic waveforms parameters. RESEARCH QUESTION: Are we able to replicate the results of Papageorgiou et al. (2019) on a population of children with CP from another center? METHODS: Retrospectively, youth with spastic unilateral (uCP) or bilateral (bCP) CP (3-18 years of age) who underwent a clinical gait analysis at the Geneva University Hospitals (HUG) were screened. Following Papageorgiou et al. 2019, the same inclusion and exclusion criteria as well as the same methodology were applied. Mann-Whitney-U test was used to compare the impairments score between the two centers. A Student T-Test using SPM was applied to compare the kinematic waveforms from the two centers. A canonical correlation analysis using SPM was realized to assess the relationship between clinical impairments and the combined sagittal motion of the pelvis, hip, knee and ankle. RESULTS AND SIGNIFICANCE: A total of 211 patients were included with 131 uCP (10 [8-14] years old) and 80 bCP (11 [7-14] years old). The distribution of the Gross Motor Function Classification System levels and the proportion of previous treatment differs between centers. In both CP groups, significant differences were observed in the composite score and lower limb kinematics, reflecting less impaired patients with CP at HUG compared to Papagergiou et al. (2019). While similar associations between spasticity and kinematic were observed in both centers, the association with muscle weakness, selectivity, and range of motion differed.

Short Walking Exercise Leads to Gait Changes and Muscle Fatigue in Children With Cerebral Palsy Who Walk With Jump Gait.

OBJECTIVE: The aim of this study was to evaluate kinematic changes and muscle fatigue in jump gait during a walking exercise and the relationship between kinematic changes and muscle fatigue and strength. DESIGN: This preliminary study included 10 children with cerebral palsy who walk with jump gait. Hip and knee maximal isometric muscle strength were measured using a dynamometer. Then, lower-limb kinematics and electromyography were collected while children walked continuously for 6 min at their self-selected speed. Electromyography median frequency and lower-limb joint angles were compared between the first and the sixth minutes of the walking exercise using t test and Wilcoxon rank test. The relationship between kinematic changes and muscle strength and changes in electromyography median frequency were assessed using correlation analyses. RESULTS: During stance, maximal knee flexion significantly increased at the sixth minute (P = 0.01) and was associated with knee extensor muscle weakness (ρ = -0.504, P = 0.03). Muscle fatigue was observed only in the gluteus medius muscle (P = 0.01). CONCLUSIONS: Children with cerebral palsy who walked with jump gait and who had knee extensor weakness were more prone to an increase in knee flexion during a continuous walk. The fatigue in the gluteus medius muscle suggests that physical intervention should target the endurance of this muscle to improve jump gait.

Muscle fatigue during a short walking exercise in children with cerebral palsy who walk in a crouch gait.

BACKGROUND: A deterioration of crouch gait was found in a group of children with cerebral palsy (CP) after a short walking exercise. The increased knee flexion reported after a continuous walk could be related with muscle fatigue and muscle strength. AIM: Does muscle fatigue appears at the end of a walking exercise in children with CP who walk in a crouch gait? METHODS: Eleven children with cerebral palsy (GMFCS I to III) who walk in a crouch gait were included. Isometric muscle strength was assessed using a handheld dynamometer. Children were asked to walk for 6 min at comfortable speed. Spatio-temporal, kinematic and electromyographic (EMG) measurements were recorded at the first and the last minute of the 6-minute walking exercise. Muscle fatigue was evaluated using the shift of EMG signals median frequency. RESULTS: There was no significant difference in walking speed, cadence, and step length at the end of the 6mwe. Maximal and mean anterior pelvic tilt decreased and knee flexion increased (p < 0.05). Rectus femoris EMG median frequency decreased (p < 0.05). The median frequency in other muscles did not decrease significantly. Greater hip extensor strength was associated with lesser knee flexion at the end of the 6-minute walking exercise (p < 0.05). SIGNIFICANCE: The increase in knee flexion at the end of the 6-minute walking exercise can be explained by muscle fatigue found in rectus femoris. Hip extensor strength can limit the deterioration of crouch gait after a 6-minute walking exercise representative of daily activities.

Lower limb extension is improved in fast walking condition in children who walk in crouch gait.

Background and purpose: The strategies for walking fast have never been reported in children with cerebral palsy who walk in crouch gait. This study aimed to assess to what extent children who walk in crouch gait are able to increase their gait speed and to report the corresponding three-dimensional kinematic adaptations.Methods: Eleven children and adolescents (aged between 7 and 17 years) with bilateral cerebral palsy, who walk in crouch gait, were asked to walk at their self-selected comfortable speed and then as fast as possible without running. The spatio-temporal and kinematic parameters, as well as the center of mass displacements were compared between walking conditions.Results: Children were able to walk 30% faster than their comfortable speed (+0.30 m/s, p = 0.000) by increasing both cadence (+21 step/min, p = 0.000) and step length (+0.05 m, p = 0.001). During the stance phase, pelvis anteversion (+3 Deg, p = 0.010), hip flexion-extension range of motion (+4 Deg, p = 0.002), and knee extension (+5 Deg, p = 0.000) were increased in fast walking. During fast walking, the center of mass showed larger range of vertical displacements (p < 0.05).Conclusions: Children with cerebral palsy who walk in crouch gait increased their walking speed by adopting a less crouched posture. Compared to comfortable walking speed condition, fast walking could be beneficial in rehabilitation to solicit higher lower limbs range of motion.Implications for rehabilitation:Children who walk in crouch gait can walk 30% fasterFast walking required higher hip and knee extensions during stance phaseFast walking could be an interesting training modality to improve the lower limb range of motion of children who walk in crouch gait.

Impact of a short walking exercise on gait kinematics in children with cerebral palsy who walk in a crouch gait.

BACKGROUND: Crouch gait results in an increase of the joint stress due to an excessive knee flexion. Daily walking exercises, even when performed at a self-selected speed, may result in a decrease of the extensor muscle strength which could lead to a more severe crouch gait pattern. The aim of this study was to assess the impact of a short walking exercise on gait kinematics in children with cerebral palsy who walk with a crouch gait. METHODS: Seven children with cerebral palsy who walk with a crouch gait were asked to walk for 6min at a self-selected speed. The spatio-temporal and kinematic measures, as well as the center of mass position were compared before and after the exercise. FINDINGS: There was no significant difference between walking speed before and after the walking exercise. Knee flexion and the maximal ankle dorsiflexion increased after the walking exercise. The vertical position of the center of mass decreased. No significant difference was found at the hip. INTERPRETATION: Children with cerebral palsy who walk with a crouch gait were more crouched after a 6-min walking exercise performed at their self-selected speed. These gait modifications could be due to fatigue of the extensor muscle groups. This study highlighted that a short walking exercise, corresponding to daily mobility, results in gait pattern modifications. Since therapies in children with cerebral palsy aim to improve motor function in everyday life situations, it could be relevant to evaluate gait adaptation after a few minutes of walking exercise.

The functional muscle-bone unit in patients with osteogenesis imperfecta type I.

CONTEXT: Osteogenesis imperfecta (OI) type I is a heritable bone fragility disorder that is caused by mutations affecting collagen type I. We recently showed that patients with OI type I frequently have muscle weakness. As muscle force and bone mass are usually closely related, we hypothesized that muscle weakness in OI type I could contribute to increase bone mass deficit in the lower extremities. OBJECTIVE: To assess the muscle-bone relationship in the lower extremities of children and adolescents with OI type I. SETTING: The study was carried out in the outpatients department of a pediatric orthopedic hospital. Patients and other participants Thirty children and adolescents with OI type I (20 females; mean age [SD]: 11.2 years [3.9]) were compared with 30 healthy age- and sex-matched controls (mean age [SD]: 11.1 years [4.5]). MAIN OUTCOME MEASURES: Tibia bone mineral content (BMC; mg/mm) was measured by peripheral quantitative computed tomography to estimate bone strength at the 4% and 14% sites. Lower extremity peak force (kN) was measured by mechanography using the multiple two-legged hopping test. RESULTS: Compared with age- and sex-matched controls, patients with OI type I had 17% lower peak force (1.3 kN vs. 1.7 kN; p=0.002) as well as a 22% lower BMC (128 mg/mm vs. 165 mg/mm; p<0.001). Stepwise regression analysis showed that muscle force and tibia length were positively related to bone strength (r(2)=0.90, p<0.001) whereas there was no effect of the disease status (OI vs. control). CONCLUSIONS: These results suggest that the muscle-bone relationship is similar between children and adolescents with OI type I and healthy age and sex-matched controls. It also suggests that muscle weakness may contribute to decreased bone strength in individuals with OI type I.

Muscle anatomy and dynamic muscle function in osteogenesis imperfecta type I.

CONTEXT: Results of previous studies suggested that children and adolescents with osteogenesis imperfecta (OI) type I have a muscle force deficit. However, muscle function has only been assessed by static isometric force tests and not in more natural conditions such as dynamic force and power tests. OBJECTIVE: The purpose of this study was to assess lower extremity dynamic muscle function and muscle anatomy in OI type I. SETTING: The study was performed in the outpatient department of a pediatric orthopedic hospital. PATIENTS AND OTHER PARTICIPANTS: A total of 54 individuals with OI type I (6-21 years; 20 male) and 54 age- and sex-matched controls took part in this study. MAIN OUTCOME MEASURES: Calf muscle cross-sectional area and density were measured by peripheral quantitative computed tomography. Lower extremity muscle function (peak force per body weight and peak power per body mass) was measured by jumping mechanography through 5 tests: multiple two-legged hopping, multiple one-legged hopping, single two-legged jump, chair-rise test, and heel-rise test. RESULTS: Compared with age- and sex-matched controls, patients with OI type I had smaller muscle size (P = .04) but normal muscle density (P = .21). They also had lower average peak force and lower specific force (peak force/muscle cross-sectional area; all P < .008). Average peak power was lower in patients with OI type I but not significantly so (all P > .054). CONCLUSIONS: Children and adolescents with OI type I have, on average, a significant force deficit in the lower limb as measured by dynamic force tests. Nonetheless, these data also show that OI type I is compatible with normal muscle performance in some individuals.