Checks and balances: a meta-analysis on the known-groups validity of functional postural control tests in children.

INTRODUCTION: Pediatric physical therapists commonly treat children with postural control deficits. Ideally, pediatric functional postural control tests should therefore be able to identify postural control deficits in children with various disorders. Despite a plethora of available tests, evidence for their validity - especially known-groups - remains scarce. This review aims to determine the known-group validity of available functional postural control tests to differentiate various pediatric pathological groups of different ages from their typically developing (TD) peers. EVIDENCE ACQUISITION: PubMed, Web of Science and Scopus were systematically searched (last update: February 2023; PROSPERO: CRD42023408982). Forty case-control studies with a pathological pediatric sample (N.=1331) and TD peers (N.=1889) were included and selected for data-extraction and -analysis. Risk of bias was assessed using the SIGN checklist and level of evidence was scored using GRADE. Random-effect meta-analyses were performed to estimate pooled standardized mean differences (SMD) for the various test types and subclassified based on pathology and/or age. EVIDENCE SYNTHESIS: When compared with TD peers, children with underlying pathologies performed significantly worse on pediatric functional postural control test batteries (SMD=-2.21), the Timed Up and Go Test and variants (SMD=2.30), the One Leg Stance test and variants (SMD=-2.14), while the Reach tests showed a smaller difference (SMD=-1.19). Subclassification within the meta-analyses showed that pathology was an influencing factor for the test batteries and the one leg stance test and variants. Age was an influencing factor for the reach tests. None of the included functional postural control tests exceeded a low level of evidence. CONCLUSIONS: Pediatric functional postural control tests that assess multiple aspects of postural control (such as test batteries) seem to offer higher known-groups validity than single-task tests (e.g. reach tests). The underlying pathology has a larger impact on the validity of these tests than age. There remains an overall low level of evidence for the known-groups validity of pediatric functional postural control tests indicating the need for research with more homogenous groups and norm reference data.

Social responsiveness in children with developmental coordination disorder.

BACKGROUND: Developmental coordination disorder (DCD) is a neurodevelopmental disorder characterized by deficits in performing motor tasks. Research suggests social skills are also altered. OBJECTIVE: To investigate (1) whether the presence of DCD affects social responsiveness, (2) whether the co-occurrence of autism spectrum disorder (ASD) affects social responsiveness in children with DCD, and (3) whether there is an association between motor performance and social responsiveness in children with DCD. METHODS: Based on parental reports, children aged 5 to 15.5 years were assigned to one of three groups: DCD only (noASD, n = 67), DCD and suspected ASD (sASD, n = 13), and DCD and confirmed ASD (cASD, n = 22). Parental answers to the Social Responsiveness Scale (SRS-2) and the DCD-Questionnaire (DCD-Q) were compared to norm values using one sample t-tests, and between groups using ANOVA and MANOVA. Pearson correlation coefficients explored the relationship between the SRS-2 and DCD-Q in the total group and per group. RESULTS: Compared to norm values, difficulties in all areas of social responsiveness were reported in children with DCD, regardless of group (p<0.001). Compared to the noASD group, more unfavorable SRS-2 total T-scores and poorer DCD-Q scores were observed in sASD and cASD groups. Only in the total group, motor performance showed significant weak to moderate associations with the SRS-2 total T-score and all subscales except for 'social motivation' (r=-0.306 to -0.405; p ≤ 0.02). CONCLUSION: Social responsiveness difficulties are more common in children with DCD and are more severe in the ASD groups. Motor performance and social responsiveness are weak to moderately associated. CLINICAL TRIAL REGISTRATION NUMBER: NCT05092893 (https://clinicaltrials.gov/study/NCT05092893).

Muscle morphology and architecture of the medial gastrocnemius between typically developing children with different ancestral background.

Muscle ultrasonography is frequently used to improve the understanding of musculoskeletal impairments in children with spastic cerebral palsy (SCP). So far, most studies on muscle morphology and architecture have included typically developing children and children with SCP with similar ancestry, being mainly Caucasian. Less is known about differences in muscle morphology between children with different ancestral backgrounds. Therefore, the aim of this study was to compare muscle morphology and architecture of the medial gastrocnemius in typically developing children with African, South Asian and Southeast Asian descent from Suriname. This explorative cohort study identified children as Maroon (Ghana, African descent), Hindustani (India, South Asian) or Javanese (Indonesia, Southeast Asian), aged 5-10 years. Using 3D freehand ultrasound with the subject prone, the following medial gastrocnemius parameters were defined: muscle tendon unit (MTU) length, muscle belly length, tendon length, muscle volume, muscle thickness, anatomical cross-sectional area (ACSA), fascicle length, pennation angle, and physiological cross-sectional area (PCSA). In addition, differences between ancestral groups were assessed for the length of the MTU, muscle, tendon and fascicles in two passive stretch conditions corresponding to an externally applied joint torque of 1Nm and 4Nm. One-way ANOVA with post hoc t-tests were used to investigate differences between the ancestral groups. In total, 100 Hindustani (n = 34), Javanese (n = 34) and Maroon (n = 32) children were included. For statistical analyses, we matched the children by age, which resulted in groups of 25 children per ancestral group (n = 75). There were no differences found in MTU length, muscle belly length, ACSA, PCSA and muscle volume. Tendon length, fascicle length and pennation angle were different between ancestral groups. Compared to Javanese children, tendon length was longer (p = 0.001) and pennation angle (p = 0.001) was larger in Maroon children and fascicle length was shorter in both Maroon and Hindustani children (p < 0.001). While there was a difference found in MTU length at different conditions of passive stretch between ancestries, no differences were found in the muscle, tendon and fascicles. This is the first study that investigated macroscopic morphological and architectural parameters for the medial gastrocnemius in one extended cohort of typically developing children, stratified in three ancestral subgroups. The current results imply that ancestry-specific reference data for children are needed, especially for tendon length, fascicle length and pennation angle when investigating altered muscle morphology in neurological or neuromuscular pathologies, such as SCP. Future studies should report the ancestral background when describing muscle morphology and architecture of children and ancestral specifications should be included in normative databases.

Electromyographic biofeedback-driven gaming to alter calf muscle activation during gait in children with spastic cerebral palsy.

BACKGROUND: Children with cerebral palsy often show deviating calf muscle activation patterns during gait, with excess activation during early stance and insufficient activation during push-off. RESEARCH QUESTION: Can children with cerebral palsy improve their calf muscle activation patterns during gait using one session of biofeedback-driven gaming? METHODS: Eighteen children (6-17 y) with spastic cerebral palsy received implicit game-based biofeedback on electromyographic activity of the calf muscle (soleus or gastrocnemius medialis) while walking on a treadmill during one session. Biofeedback alternately aimed to reduce early stance activity, increase push-off activity, and both combined. Early stance and push-off activity and the double-bump-index (early stance divided by push-off activity) were determined during baseline and walking with feedback. Changes were assessed at group level using repeated measures ANOVA with simple contrast or Friedman test with post-hoc Wilcoxon signed rank test, as well as individually using independent t-tests or Wilcoxon rank sum tests. Perceived competence and interest-enjoyment were assessed through a questionnaire. RESULTS: Children successfully decreased their electromyographic activity during early stance feedback trials (relative decrease of 6.8 ± 12.2 %, P = 0.025), with a trend during the combined feedback trials (6.5 ± 13.9 %, P = 0.055), and increased their electromyographic activity during push-off feedback trials (8.1 ± 15.8 %, P = 0.038). Individual improvements were seen in twelve of eighteen participants. All children experienced high levels of interest-enjoyment (8.4/10) and perceived competence (8.1/10). SIGNIFICANCE: This exploratory study suggests that children with cerebral palsy can achieve small within-session improvements of their calf muscle activation pattern when provided with implicit biofeedback-driven gaming in an enjoyable manner. Follow-up gait training studies can incorporate this method to assess retention and long-term functional benefits of electromyographic biofeedback-driven gaming.

Reliability of 3D freehand ultrasound to assess lower limb muscles in children with spastic cerebral palsy and typical development.

This study investigated the reliability of 3-dimensional freehand ultrasound (3DfUS) to quantify the size (muscle volume [MV] and anatomical cross-sectional area [aCSA]), length (muscle length [ML], tendon length [TL], and muscle tendon unit length [MTUL]), and echo-intensity (EI, whole muscle and 50% aCSA), of lower limb muscles in children with spastic cerebral palsy (SCP) and typical development (TD). In total, 13 children with SCP (median age 14.3 (7.3) years) and 13 TD children (median age 11.1 (1.7) years) participated. 3DfUS scans of rectus femoris, semitendinosus, medial gastrocnemius, and tibialis anterior were performed by two raters in two sessions. The intra- and inter-rater and intra- and inter-session reliability were defined with relative and absolute reliability measures, that is, intra-class correlation coefficients (ICCs) and absolute and relative standard error of measurement (SEM and SEM%), respectively. Over all conditions, ICCs for muscle size measures ranged from 0.818 to 0.999 with SEM%s of 12.6%-1.6%. For EI measures, ICCs varied from 0.233 to 0.967 with SEM%s of 15.6%-1.7%. Length measure ICCs ranged from 0.642 to 0.999 with SEM%s of 16.0%-0.5%. In general, reliability did not differ between the TD and SCP cohort but the influence of different muscles, raters, and sessions was not constant for all 3DfUS parameters. Muscle length and muscle tendon unit length were the most reliable length parameters in all conditions. MV and aCSA showed comparable SEM%s over all muscles, where tibialis anterior MV was most reliable. EI had low-relative reliability, but absolute reliability was better, with better reliability for the distal muscles in comparison to the proximal muscles. Combining these results with earlier studies describing muscle morphology assessed in children with SCP, 3DfUS seems sufficiently reliable to determine differences between cohorts and functional levels. The applicability on an individual level, for longitudinal follow-up and after interventions is dependent on the investigated muscle and parameter. Moreover, the semitendinosus, the acquisition, and processing of multiple sweeps, and the definition of EI and TL require further investigation. In general, it is recommended, especially for longitudinal follow-up studies, to keep the rater the same, while standardizing acquisition settings and positioning of the subject.

The influence of wearing an ultrasound device on gait in children with cerebral palsy and typically developing children.

BACKGROUND: Ultrasonography with motion analysis enables dynamic imaging of medial gastrocnemius (MG) muscles and tendons during gait. This revealed pathological muscle-tendon dynamics in children with spastic cerebral palsy (CP) compared to typically developing (TD) children. However, wearing an ultrasound probe on the lower leg could interfere with gait and bias muscle length changes observed with ultrasound. RESEARCH QUESTION: Does wearing an ultrasound probe on the MG influence gait in children with CP and TD children? METHODS: Eighteen children with spastic CP and 16 age-matched TD children walked at comfortable walking speed on an instrumented treadmill. One baseline gait condition (BASE) and two conditions with an ultrasound probe and custom-made probe holder were measured: on the mid-muscle fascicles (FAS) and on the muscle-tendon junction (MTJ). The effect of condition and group on spatiotemporal parameters, hip, knee and ankle kinematics, ankle moment, ankle power, and modeled MG muscle-tendon unit (MTU) length was assessed using two-way repeated measures ANOVA's. Statistical non-parametric mapping was applied for time-series. Post-hoc paired-samples t-tests were conducted, and the root mean square difference was calculated for significant parts. RESULTS: Children took wider steps during FAS (CP, TD) and MTJ (TD) compared to BASE, and during FAS compared to MTJ (CP). Hip extension was lower (2.7°) during terminal stance for MTJ compared to FAS for TD only. There was less swing knee flexion (FAS 4.9°; MTJ 4.0°) and ankle plantarflexion around toe-off (FAS 3.0°; MTJ 2.4°) for both ultrasound placements, with no group effect. Power absorption during loading response was slightly increased for both ultrasound placements (0.12 W/kg), with no group effect. MTU shortened less in swing for both ultrasound placements (FAS 3.6 mm; MTJ 3.7 mm), with no group effect. SIGNIFICANCE: Wearing an ultrasound probe causes minimal lower-limb gait alterations and MTU length changes that are mostly similar in CP and TD.

Associations between muscle morphology and spasticity in children with spastic cerebral palsy.

INTRODUCTION: Due to the heterogeneous clinical presentation of spastic cerebral palsy (SCP), which makes spasticity treatment challenging, more insight into the complex interaction between spasticity and altered muscle morphology is warranted. AIMS: We studied associations between spasticity and muscle morphology and compared muscle morphology between commonly observed spasticity patterns (i.e. different muscle activation patterns during passive stretches). METHODS: Spasticity and muscle morphology of the medial gastrocnemius (MG) and semitendinosus (ST) were defined in 74 children with SCP (median age 8 years 2 months, GMFCS I/II/III: 31/25/18, bilateral/unilateral: 46/27). Using an instrumented assessment, spasticity was quantified as the difference in muscle activation recorded during passive stretches at low and high velocities and was classified in mixed length-/velocity-dependent or pure velocity-dependent activation patterns. Three-dimensional freehand ultrasound was used to assess muscle morphology (volume and length) and echogenicity intensity (as a proxy for muscle quality). Spearman correlations and Mann-Whitney-U tests defined associations and group differences, respectively. RESULTS: A moderate negative association (r = -0.624, p < 0.001) was found between spasticity and MG muscle volume, while other significant associations between spasticity and muscle morphology parameters were weak. Smaller normalized muscle volume (MG p = 0.004, ST p=<0.001) and reduced muscle belly length (ST p = 0.015) were found in muscles with mixed length-/velocity-dependent patterns compared to muscles with pure velocity-dependent patterns. DISCUSSION: Higher spasticity levels were associated with smaller MG and ST volumes and shorter MG muscles. These muscle morphology alterations were more pronounced in muscles that activated during low-velocity stretches compared to muscles that only activated during high-velocity stretches.

Personalisation of Plantarflexor Musculotendon Model Parameters in Children with Cerebral Palsy.

Neuromusculoskeletal models can be used to evaluate aberrant muscle function in cerebral palsy (CP), for example by estimating muscle and joint contact forces during gait. However, to be accurate, models should include representative musculotendon parameters. We aimed to estimate personalised parameters that capture the mechanical behaviour of the plantarflexors in children with CP and typically developing (TD) children. Ankle angle (using motion capture), torque (using a load-cell), and medial gastrocnemius fascicle lengths (using ultrasound) were measured during slow passive ankle dorsiflexion rotation for thirteen children with spastic CP and thirteen TD children. Per subject, the measured rotation was input to a scaled OpenSim model to simulate the torque and fascicle length output. Musculotendon model parameters were personalised by the best match between simulated and experimental torque-angle and fascicle length-angle curves according to a least-squares fit. Personalised tendon slack lengths were significantly longer and optimal fibre lengths significantly shorter in CP than model defaults and than in TD. Personalised tendon compliance was substantially higher in both groups compared to the model default. The presented method to personalise musculotendon parameters will likely yield more accurate simulations of subject-specific muscle mechanics, to help us understand the effects of altered musculotendon properties in CP.

Revising the stretch reflex threshold method to measure stretch hyperreflexia in cerebral palsy.

Hyper-resistance is an increased resistance to passive muscle stretch, a common feature in neurological disorders. Stretch hyperreflexia, an exaggerated stretch reflex response, is the neural velocity-dependent component of hyper-resistance, and has been quantitatively measured using the stretch reflex threshold (i.e., joint angle at the stretch reflex electromyographic onset). In this study, we introduce a correction in how the stretch reflex threshold is calculated, by accounting for the stretch reflex latency (i.e., time between the stretch reflex onset at the muscle spindles and its appearance in the electromyographic signal). Furthermore, we evaluated how this correction affects the stretch reflex threshold in children and young adults with spastic cerebral palsy. A motor-driven ankle dynamometer induced passive ankle dorsiflexions at four incremental velocities in 13 children with cerebral palsy (mean age: 13.5 years, eight males). The stretch reflex threshold for soleus and medial gastrocnemius muscles was calculated as 1) the joint angle corresponding to the stretch reflex electromyographic onset (i.e., original method); and as 2) the joint angle corresponding to the electromyographic onset minus the individual Hoffmann-reflex latency (i.e., latency corrected method). The group linear regression slopes between stretch velocity and stretch reflex threshold differed in both muscles between methods (p < 0.05). While the original stretch reflex threshold was velocity dependent in both muscles (p < 0.05), the latency correction rendered it velocity independent. Thus, the effects of latency correction on the stretch reflex threshold are substantial, especially at higher stretch velocities, and should be considered in future studies.

Developmental Coordination Disorder before the Age of Three: A Longitudinal Retrospective Study in a Belgian Center for Developmental Disabilities.

This study aimed to explore the association between developmental coordination disorder (DCD) diagnosed after the age of three and both a standardized motor test­the Alberta Infant Motor Scale (AIMS)­and non-standardized observation of movement quality carried out before the age of three. Children at risk or with developmental concerns were studied retrospectively. Children were excluded in case of a diagnosis, excluding DCD, e.g., cerebral palsy, or IQ < 70. Of the 503 included children, 246 were diagnosed with (at-risk) DCD. Multivariate binary logistic regression revealed a significant association between DCD diagnosis after the age of three and male gender and with different aspects of poor movement quality in different age groups before the age three. Univariate analyses revealed an association between DCD diagnosis and the number of poor movement-quality descriptions at 0−6 months, 6−12 months, and 18 months−3 years but not with the AIMS scores. The MABC-2 scores after the age of three were significantly correlated with the number of poor movement-quality descriptions in age groups 0−6 months and 18 months−3 years and with the AIMS scores in age groups 6−12 months and 12−18 months. The results suggest that DCD can be associated with poor movement quality before the age of three.

The reliability of measuring medial gastrocnemius muscle-tendon unit lengths during gait.

BACKGROUND: Ultrasound imaging combined with 3D motion analysis allows for in-vivo assessment of muscle-tendon unit lengths during gait. The clinical relevance of analysing the medial gastrocnemius (MG) and Achilles muscle-tendon junction (MTJ), MG mid-muscle belly fascicles (FAS) and muscle thickness was shown. However, their reliability error estimations over the gait cycle is unknown. RESEARCH QUESTION: What are the intra- and inter-session errors associated with extracting MG belly, thickness, FAS and tendon lengths using ultrasound during gait in healthy participants? METHODS: 3D gait analysis was carried out in ten healthy adults as they walked on an instrumented treadmill at a comfortable walking speed. An ultrasound probe was secured on the leg and tracked by 3D motion analysis. Images were collected during two walking trials with the probe on the MG muscle-belly to estimate FAS lengths and muscle thickness, and during two trials with the probe on the MTJ to estimate muscle-belly and tendon lengths. A second session was performed after 5 ± 4 days where a different operator placed the ultrasound probe. The standard deviation (SD) of absolute and relative lengths changes during the gait cycle over different trials were calculated per participant. SD values averaged over participants represented intra- and inter-session errors. RESULTS: For all assessed variables, the intra-session errors were <2.2 mm, except for the FAS lengths (3.1 mm). The inter-session errors were larger than the intra-session, with the highest values found for the absolute muscle-tendon unit lengths (5.6 mm). Relative length errors were smaller than absolute length errors. SIGNIFICANCE: Intra-session errors, which may reflect natural variability and data processing errors, seem more critical when extracting absolute FAS than muscle-tendon lengths. Standardized probe positioning on the MTJ between sessions may improve the inter-session reliability. Expressing the lengths relative to their lengths as the beginning of the gait cycle reduces the inter-session errors.

The Contribution of Decreased Muscle Size to Muscle Weakness in Children With Spastic Cerebral Palsy.

Muscle weakness is a common clinical symptom in children with spastic cerebral palsy (SCP). It is caused by impaired neural ability and altered intrinsic capacity of the muscles. To define the contribution of decreased muscle size to muscle weakness, two cohorts were recruited in this cross-sectional investigation: 53 children with SCP [median age, 8.2 (IQR, 4.1) years, 19/34 uni/bilateral] and 31 children with a typical development (TD) [median age, 9.7 (IQR, 2.9) years]. Muscle volume (MV) and muscle belly length for m. rectus femoris, semitendinosus, gastrocnemius medialis, and tibialis anterior were defined from three-dimensional freehand ultrasound acquisitions. A fixed dynamometer was used to assess maximal voluntary isometric contractions for knee extension, knee flexion, plantar flexion, and dorsiflexion from which maximal joint torque (MJT) was calculated. Selective motor control (SMC) was assessed on a 5-point scale for the children with SCP. First, the anthropometrics, strength, and muscle size parameters were compared between the cohorts. Significant differences for all muscle size and strength parameters were found (p ≤ 0.003), except for joint torque per MV for the plantar flexors. Secondly, the associations of anthropometrics, muscle size, gross motor function classification system (GMFCS) level, and SMC with MJT were investigated using univariate and stepwise multiple linear regressions. The associations of MJT with growth-related parameters like age, weight, and height appeared strongest in the TD cohort, whereas for the SCP cohort, these associations were accompanied by associations with SMC and GMFCS. The stepwise regression models resulted in ranges of explained variance in MJT from 29.3 to 66.3% in the TD cohort and from 16.8 to 60.1% in the SCP cohort. Finally, the MJT deficit observed in the SCP cohort was further investigated using the TD regression equations to estimate norm MJT based on height and potential MJT based on MV. From the total MJT deficit, 22.6-57.3% could be explained by deficits in MV. This investigation confirmed the disproportional decrease in muscle size and muscle strength around the knee and ankle joint in children with SCP, but also highlighted the large variability in the contribution of muscle size to muscle weakness.

Reliability of Processing 3-D Freehand Ultrasound Data to Define Muscle Volume and Echo-intensity in Pediatric Lower Limb Muscles with Typical Development or with Spasticity.

This investigation assessed the processer reliability of estimating muscle volume and echo-intensity of the rectus femoris, tibialis anterior and semitendinosus. The muscles of 10 typically developing children (8.15 [1.40] y) and 15 children with spastic cerebral palsy (7.67 [3.80] y; Gross Motor Function Classification System I = 5, II = 5, III = 5) were scanned with 3-D freehand ultrasonography. For the intra-processer analysis, the intra-class correlations coefficients (ICCs) for muscle volume ranged from 0.943-0.997, with relative standard errors of measurement (SEM%) ranging from 1.24%-8.97%. For the inter-processer analysis, these values were 0.853 to 0.988 and 3.47% to 14.02%, respectively. Echo-intensity had ICCs >0.947 and relative SEMs <4% for both analyses. Muscle volume and echo-intensity can be reliably extracted for the rectus femoris, semitendinosus and tibialis anterior in typically developing children and children with cerebral palsy. The need for a single processer to analyze all data is dependent on the size of the expected changes or differences.

Muscle Characteristics in Pediatric Hereditary Spastic Paraplegia vs. Bilateral Spastic Cerebral Palsy: An Exploratory Study.

Hereditary spastic paraplegia (HSP) is a neurological, genetic disorder that predominantly presents with lower limb spasticity and muscle weakness. Pediatric pure HSP types with infancy or childhood symptom onset resemble in clinical presentation to children with bilateral spastic cerebral palsy (SCP). Hence, treatment approaches in these patient groups are analogous. Altered muscle characteristics, including reduced medial gastrocnemius (MG) muscle growth and hyperreflexia have been quantified in children with SCP, using 3D-freehand ultrasound (3DfUS) and instrumented assessments of hyperreflexia, respectively. However, these muscle data have not yet been studied in children with HSP. Therefore, we aimed to explore these MG muscle characteristics in HSP and to test the hypothesis that these data differ from those of children with SCP and typically developing (TD) children. A total of 41 children were retrospectively enrolled including (1) nine children with HSP (ages of 9-17 years with gross motor function levels I and II), (2) 17 age-and severity-matched SCP children, and (3) 15 age-matched typically developing children (TD). Clinically, children with HSP showed significantly increased presence and severity of ankle clonus compared with SCP (p = 0.009). Compared with TD, both HSP and SCP had significantly smaller MG muscle volume normalized to body mass (p ≤ 0.001). Hyperreflexia did not significantly differ between the HSP and SCP group. In addition to the observed pathological muscle activity for both the low-velocity and the change in high-velocity and low-velocity stretches in the two groups, children with HSP tended to present higher muscle activity in response to increased stretch velocity compared with those with SCP. This exploratory study is the first to reveal MG muscle volume deficits in children with HSP. Moreover, high-velocity-dependent hyperreflexia and ankle clonus is observed in children with HSP. Instrumented impairment assessments suggested similar altered MG muscle characteristics in pure HSP type with pediatric onset compared to bilateral SCP. This finding needs to be confirmed in larger sample sizes. Hence, the study results might indicate analogous treatment approaches in these two patient groups.

Interventions and lower-limb macroscopic muscle morphology in children with spastic cerebral palsy: a scoping review.

AIM: To identify and map studies that have assessed the effect of interventions on lower-limb macroscopic muscle-tendon morphology in children with spastic cerebral palsy (CP). METHOD: We conducted a literature search of studies that included pre- and post-treatment measurements of lower-limb macroscopic muscle-tendon morphology in children with spastic CP. Study quality was evaluated and significant intervention effects and effect sizes were extracted. RESULTS: Twenty-eight articles were identified. They covered seven different interventions including stretching, botulinum neurotoxin A (BoNT-A), strengthening, electrical stimulation, whole-body vibration, balance training, and orthopaedic surgery. Study quality ranged from poor (14 out of 28 studies) to good (2 out of 28). Study samples were small (n=4-32) and studies were variable regarding which muscles and macroscopic morphological parameters were assessed. Inconsistent effects after intervention (thickness and cross-sectional area for strengthening, volume for BoNT-A), no effect (belly length for stretching), and small effect sizes were reported. INTERPRETATION: Intervention studies reporting macroscopic muscle-tendon remodelling after interventions are limited and heterogeneous, making it difficult to generalize results. Studies that include control groups and standardized assessment protocols are needed to improve study quality and data synthesis. Lack or inconclusive effects at the macroscopic level could indicate that the effects of interventions should also be evaluated at the microscopic level. WHAT THIS PAPER ADDS: Muscle-targeted interventions to remodel muscle morphology are not well understood. Studies reporting macroscopic muscle remodelling following interventions are limited and heterogeneous. Passive stretching may preserve but does not increase muscle length. The effects of isolated botulinum neurotoxin A injections on muscle volume are inconsistent. Isolated strengthening shows no consistent increase in muscle volume or thickness.

Gastrocnemius Medialis Muscle Geometry and Extensibility in Typically Developing Children and Children With Spastic Paresis Aged 6-13 Years.

Gait of children with spastic paresis (SP) is frequently characterized by a reduced ankle range of motion, presumably due to reduced extensibility of the triceps surae (TS) muscle. Little is known about how morphological muscle characteristics in SP children are affected. The aim of this study was to compare gastrocnemius medialis (GM) muscle geometry and extensibility in children with SP with those of typically developing (TD) children and assess how GM morphology is related to its extensibility. Thirteen children with SP, of which 10 with a diagnosis of spastic cerebral palsy and three with SP of unknown etiology (mean age 9.7 ± 2.1 years; GMFCS: I-III), and 14 TD children (mean age 9.3 ± 1.7 years) took part in this study. GM geometry was assessed using 3D ultrasound imaging at 0 and 4 Nm externally imposed dorsal flexion ankle moments. GM extensibility was defined as its absolute length change between the externally applied 0 and 4 Nm moments. Anthropometric variables and GM extensibility did not differ between the SP and TD groups. While in both groups, GM muscle volume correlated with body mass, the slope of the regression line in TD was substantially higher than that in SP (TD = 3.3 ml/kg; SP = 1.3 ml/kg, p < 0.01). In TD, GM fascicle length increased with age, lower leg length and body mass, whereas in SP children, fascicle length did not correlate with any of these variables. However, the increase in GM physiological cross-sectional area as a function of body mass did not differ between SP and TD children. Increases in lengths of tendinous structures in children with SP exceeded those observed in TD children (TD = 0.85 cm/cm; SP = 1.16 cm/cm, p < 0.01) and even exceeded lower-leg length increases. In addition, only for children with SP, body mass (r = -0.61), height (r = -0.66), muscle volume (r = - 0.66), physiological cross-sectional area (r = - 0.59), and tendon length (r = -0.68) showed a negative association with GM extensibility. Such negative associations were not found for TD children. In conclusion, physiological cross-sectional area and length of the tendinous structures are positively associated with age and negatively associated with extensibility in children with SP.

A Survey on the Use and Barriers of Surface Electromyography in Neurorehabilitation.

Historical, educational, and technical barriers have been reported to limit the use of surface electromyography (sEMG) in clinical neurorehabilitation settings. In an attempt to identify, review, rank, and interpret potential factors that may play a role in this scenario, we gathered information on (1) current use of sEMG and its clinical potential; (2) professional figures primarily dealing with sEMG; (3) educational aspects, and (4) possible barriers and reasons for its apparently limited use in neurorehabilitation. To this aim, an online 30-question survey was sent to 52 experts on sEMG from diverse standpoints, backgrounds, and countries. Participants were asked to respond to each question on a 5-point Likert scale or by ranking items. A cut-off of 75% agreement was chosen as the consensus threshold. Thirty-five invitees (67%) completed the electronic survey. Consensus was reached for 77% of the proposed questions encompassing current trends in sEMG use in neurorehabilitation, educational, technical, and methodological features as well as its translational utility for clinicians and patients. Data evidenced the clinical utility of sEMG for patient assessment, to define the intervention plan, and to complement/optimize other methods used to quantify muscle and physical function. The aggregate opinion of the interviewed experts confirmed that sEMG is more frequently employed in technical/methodological than clinical research. Moreover, the slow dissemination of research findings and the lack of education on sEMG seem to prevent prompt transfer into practice. The findings of the present survey may contribute to the ongoing debate on the appropriateness and value of sEMG for neurorehabilitation professionals and its potential translation into clinical settings.

Treatment Response to Botulinum Neurotoxin-A in Children With Cerebral Palsy Categorized by the Type of Stretch Reflex Muscle Activation.

While Botulinum NeuroToxin-A (BoNT-A) injections are frequently used to reduce the effects of hyperactive stretch reflexes in children with cerebral palsy (CP), the effects of this treatment vary strongly. Previous research, combining electromyography (EMG) with motion analysis, defined different patterns of stretch reflex muscle activation in muscles, those that reacted more to a change in velocity (velocity dependent -VD), and those that reacted more to a change in length (length dependent -LD). The aim of this study was to investigate the relation between the types of stretch reflex muscle activation in the semitendinosus with post-BoNT-A outcome as assessed passively and with 3D gait analysis in children with spastic CP. Eighteen children with spastic CP (10 bilaterally involved) between the ages of 12 and 18 years were assessed before and on average, 8 weeks post-treatment. EMG and motion analysis were used to assess the degree and type of muscle activation dependency in the semitendinosus during passive knee extensions performed at different joint angular velocities. Three-dimensional gait analysis was used to assess knee gait kinematics as a measure of functional outcome. Pre-treatment, 9 muscles were classified as VD and 9 as LD, but no differences between the groups were evident in the baseline knee gait kinematics. Post-treatment, stretch reflex muscle activation decreased significantly in both groups but the reduction was more pronounced in those muscles classified pre-treatment as VD (-72% vs. -50%, p = 0.005). In the VD group, these changes were accompanied by greater knee extension at initial contact and during the swing phase of gait. In the LD group, there was significantly increased post-treatment knee hyperextension in late stance. Although results vary between patients, the reduction of stretch reflex muscle activation in the semitendinosus generally translated to an improved functional outcome, as assessed with 3D gait analysis. However, results were less positive for those muscles with pre-treatment length-dependent type of stretch reflex muscle activation. The study demonstrates the relevance of categorizing the type of stretch reflex muscle activation as a possible predictor of treatment response.

Joint and Muscle Assessments of the Separate Effects of Botulinum NeuroToxin-A and Lower-Leg Casting in Children With Cerebral Palsy.

Botulinum NeuroToxin-A (BoNT-A) injections to the medial gastrocnemius (MG) and lower-leg casts are commonly combined to treat ankle equinus in children with spastic cerebral palsy (CP). However, the decomposed treatment effects on muscle or tendon structure, stretch reflexes, and joint are unknown. In this study, BoNT-A injections to the MG and casting of the lower legs were applied separately to gain insight into the working mechanisms of the isolated treatments on joint, muscle, and tendon levels. Thirty-one children with spastic CP (GMFCS I-III, age 7.4 ± 2.6 years) received either two weeks of lower-leg casts or MG BoNT-A injections. During full range of motion slow and fast passive ankle rotations, joint resistance and MG stretch reflexes were measured. MG muscle and tendon lengths were assessed at resting and at maximum dorsiflexion ankle angles using 3D-freehand ultrasound. Treatment effects were compared using non-parametric statistics. Associations between the effects on joint and muscle or tendon levels were performed using Spearman correlation coefficients (p < 0.05). Increased joint resistance, measured during slow ankle rotations, was not significantly reduced after either treatment. Additional joint resistance assessed during fast rotations only reduced in the BoNT-A group (-37.6%, p = 0.013, effect size = 0.47), accompanied by a reduction in MG stretch reflexes (-70.7%, p = 0.003, effect size = 0.56). BoNT-A increased the muscle length measured at the resting ankle angle (6.9%, p = 0.013, effect size = 0.53). Joint angles shifted toward greater dorsiflexion after casting (32.4%, p = 0.004, effect size = 0.56), accompanied by increases in tendon length (5.7%, p = 0.039, effect size = 0.57; r = 0.40). No associations between the changes in muscle or tendon lengths and the changes in the stretch reflexes were found. We conclude that intramuscular BoNT-A injections reduced stretch reflexes in the MG accompanied by an increase in resting muscle belly length, whereas casting resulted in increased dorsiflexion without any changes to the muscle length. This supports the need for further investigation on the effect of the combined treatments and the development of treatments that more effectively lengthen the muscle.

Physics-Based Simulations to Predict the Differential Effects of Motor Control and Musculoskeletal Deficits on Gait Dysfunction in Cerebral Palsy: A Retrospective Case Study.

Physics-based simulations of walking have the theoretical potential to support clinical decision-making by predicting the functional outcome of treatments in terms of walking performance. Yet before using such simulations in clinical practice, their ability to identify the main treatment targets in specific patients needs to be demonstrated. In this study, we generated predictive simulations of walking with a medical imaging based neuro-musculoskeletal model of a child with cerebral palsy presenting crouch gait. We explored the influence of altered muscle-tendon properties, reduced neuromuscular control complexity, and spasticity on gait dysfunction in terms of joint kinematics, kinetics, muscle activity, and metabolic cost of transport. We modeled altered muscle-tendon properties by personalizing Hill-type muscle-tendon parameters based on data collected during functional movements, simpler neuromuscular control by reducing the number of independent muscle synergies, and spasticity through delayed muscle activity feedback from muscle force and force rate. Our simulations revealed that, in the presence of aberrant musculoskeletal geometries, altered muscle-tendon properties rather than reduced neuromuscular control complexity and spasticity were the primary cause of the crouch gait pattern observed for this child, which is in agreement with the clinical examination. These results suggest that muscle-tendon properties should be the primary target of interventions aiming to restore an upright gait pattern for this child. This suggestion is in line with the gait analysis following muscle-tendon property and bone deformity corrections. Future work should extend this single case analysis to more patients in order to validate the ability of our physics-based simulations to capture the gait patterns of individual patients pre- and post-treatment. Such validation would open the door for identifying targeted treatment strategies with the aim of designing optimized interventions for neuro-musculoskeletal disorders.