Reference curves of motor function outcomes in young steroid-naïve males with Duchenne muscular dystrophy.

AIM: To investigate functional motor performance in a large cohort of young steroid-naïve males with Duchenne muscular dystrophy (DMD) and typically developing males, and to develop specific reference curves for both groups. Also, to describe associations between anthropometric values and functional motor outcomes. METHOD: Cross-sectional data of 196 steroid-naïve males with DMD aged 4 to 8 years and 497 typically developing males aged 2 years 6 months to 8 years were included. Both groups were evaluated with the time to rise from the floor test, 10-metre walk/run test, 6-minute walk test, and North Star Ambulatory Assessment. Reference curves with centiles 5%, 10%, 25%, 50%, 75%, 90%, and 95% were estimated using quantile regression. RESULTS: Males with DMD scored significantly worse on all functional motor outcomes than age-matched typically developing males (p < 0.001): 89% to 95% of the males with DMD scored below the 5th centile of the typically developing males. No or weak correlations exist between anthropometric values and functional motor outcomes. INTERPRETATION: The estimated reference curves can support consultation with families of young males with DMD and can support the evaluation of treatment for reaching motor skills and functional motor outcomes compared with typically developing males.

Experts' perspectives on how to promote implicit and explicit motor learning in children: A mixed-methods study.

BACKGROUND: Little is known about how motor learning strategies (MLSs) can promote implicit and explicit motor learning processes. This study aimed to explore experts' perspectives on therapists' use of MLSs to promote specific learning processes in children with and without developmental coordination disorder (DCD). METHODS: In this mixed-methods study, two consecutive digital questionnaires were used to ascertain the opinions of international experts. Questionnaire 2 explored the findings of Questionnaire 1 in greater depth. In order to reach a certain level of agreement about the classification of MLSs as promoting either (more) implicit or (more) explicit motor learning, 5-point Likert scales were used in addition to open-ended questions. The open-ended questions were analysed with a conventional analysis approach. Open coding was performed by two reviewers independently. Categories and themes were discussed within the research team, taking both questionnaires as one dataset. RESULTS: Twenty-nine experts from nine different countries with different backgrounds in research, education and/or clinical care completed the questionnaires. The results of the Likert scales showed large variation. Two themes emerged from the qualitative analyses: (1) Experts found it difficult to classify MLSs as promoting either implicit or explicit motor learning, and (2) experts stressed the need for clinical decisionmaking when choosing MLSs. CONCLUSIONS: Insufficient insight was gained into how MLSs could promote (more) implicit or (more) explicit motor learning in children in general and in children with DCD specifically. But this study demonstrated the importance of clinical decisionmaking to model and adapt MLSs to child, task and environment, with therapists' knowledge of MLSs being an important prerequisite. Research is needed to better understand the various learning mechanisms of children and how MLSs can be used to manipulate these mechanisms.

Exploring structural connectomes in children with unilateral cerebral palsy using graph theory.

We explored structural brain connectomes in children with spastic unilateral cerebral palsy (uCP) and its relation to sensory-motor function using graph theory. In 46 children with uCP (mean age = 10 years 7 months ± 2 years 9 months; Manual Ability Classification System I = 15, II = 16, III = 15) we assessed upper limb somatosensory and motor function. We collected multi-shell diffusion-weighted, T1-weighted and T2-FLAIR MRI and identified the corticospinal tract (CST) wiring pattern using transcranial magnetic stimulation. Structural connectomes were constructed using Virtual Brain Grafting-modified FreeSurfer parcellations and multi-shell multi-tissue constrained spherical deconvolution-based anatomically-constrained tractography. Graph metrics (characteristic path length, global/local efficiency and clustering coefficient) of the whole brain, the ipsilesional/contralesional hemisphere, and the full/ipsilesional/contralesional sensory-motor network were compared between lesion types (periventricular white matter (PWM) = 28, cortical and deep gray matter (CDGM) = 18) and CST-wiring patterns (ipsilateral = 14, bilateral = 14, contralateral = 12, unknown = 6) using ANCOVA with age as covariate. Using elastic-net regularized regression we investigated how graph metrics, lesion volume, lesion type, CST-wiring pattern and age predicted sensory-motor function. In both the whole brain and subnetworks, we observed a hyperconnectivity pattern in children with CDGM-lesions compared with PWM-lesions, with higher clustering coefficient (p = [<.001-.047], η p 2 =[0.09-0.27]), characteristic path length (p = .003, η p 2 =0.19) and local efficiency (p = [.001-.02], η p 2 =[0.11-0.21]), and a lower global efficiency with age (p = [.01-.04], η p 2 =[0.09-0.15]). No differences were found between CST-wiring groups. Overall, good predictions of sensory-motor function were obtained with elastic-net regression (R2  = .40-.87). CST-wiring pattern was the strongest predictor for motor function. For somatosensory function, all independent variables contributed equally to the model. In conclusion, we demonstrated the potential of structural connectomics in understanding disease severity and brain development in children with uCP.

In-depth quantification of bimanual coordination using the Kinarm exoskeleton robot in children with unilateral cerebral palsy.

BACKGROUND: Robots have been proposed as tools to measure bimanual coordination in children with unilateral cerebral palsy (uCP). However, previous research only examined one task and clinical interpretation remains challenging due to the large amount of generated data. This cross-sectional study aims to examine bimanual coordination by using multiple bimanual robotics tasks in children with uCP, and their relation to task execution and unimanual performance. METHODS: The Kinarm exoskeleton robot was used in 50 children with uCP (mean age: 11 years 11 months ± 2 years 10 months, Manual Ability Classification system (MACS-levels: l = 27, ll = 16, lll = 7)) and 50 individually matched typically developing children (TDC). All participants performed three tasks: object-hit (hit falling balls), ball-on-bar (balance a ball on a bar while moving to a target) and circuit task (move a cursor along a circuit by making horizontal and vertical motions with their right and left hand, respectively). Bimanual parameters provided information about bimanual coupling and interlimb differences. Differences between groups and MACS-levels were investigated using ANCOVA with age as covariate (α < 0.05, [Formula: see text]). Correlation analysis (r) linked bimanual coordination to task execution and unimanual parameters. RESULTS: Children with uCP exhibited worse bimanual coordination compared to TDC in all tasks (p ≤ 0.05, [Formula: see text] = 0.05-0.34). The ball-on-bar task displayed high effect size differences between groups in both bimanual coupling and interlimb differences (p < 0.001, [Formula: see text] = 0.18-0.36), while the object-hit task exhibited variations in interlimb differences (p < 0.001, [Formula: see text] = 0.22-0.34) and the circuit task in bimanual coupling (p < 0.001, [Formula: see text] = 0.31). Mainly the performance of the ball-on-bar task (p < 0.05, [Formula: see text] = 0.18-0.51) was modulated by MACS-levels, showing that children with MACS-level lll had worse bimanual coordination compared to children with MACS-level l and/or II. Ball-on-bar outcomes were highly related to task execution (r = - 0.75-0.70), whereas more interlimb differences of the object-hit task were moderately associated with a worse performance of the non-dominant hand (r = - 0.69-(- 0.53)). CONCLUSION: This study gained first insight in important robotic tasks and outcome measures to quantify bimanual coordination deficits in children with uCP. The ball-on-bar task showed the most discriminative ability for both bimanual coupling and interlimb differences, while the object-hit and circuit tasks are unique to interlimb differences and bimanual coupling, respectively.

Therapists' Use of Instructions and Feedback in Motor Learning Interventions in Children with Developmental Coordination Disorder: A Video Observation Study.

AIM: This qualitative study explored therapists' use of instructions and feedback when teaching motor tasks to children with developmental coordination disorder (DCD) as a first step in developing practical recommendations. METHODS: A conventional content analysis approach was used to analyze videotaped treatment sessions of physical therapists using a newly developed analysis plan. Inductive coding was used to code purposively selected video segments. The codes were sorted into categories to identify key themes. Analyses were performed independently by two researchers until data saturation was reached. RESULTS: Ten video-taped sessions were analyzed and 61 segments were coded. Three key themes were identified: (1) therapists' intention with the instructions and feedback was to motivate or to provide information; (2) the preferred therapists' teaching style was either direct or indirect; and (3) parameters to shape specific instructions and feedback were the focus of attention, modality, information content, timing and frequency. CONCLUSION: Therapists used numerous instructions and feedback with different information content, often shaped by multiple focuses and/or modalities to motivate children or to provide specific information about task performance. Although therapists adapted instructions and feedback to child and task, future research should explore how characteristics of child and task can guide therapists' clinical decision-making.

Tyneside Pegboard Test for unimanual and bimanual dexterity in unilateral cerebral palsy: association with sensorimotor impairment.

AIM: We explored the psychometric properties of the recently developed Tyneside Pegboard Test (TPT) for unimanual and bimanual dexterity in children with unilateral cerebral palsy (CP) and investigated the impact of sensorimotor impairments on manual dexterity. METHOD: In this cross-sectional study, the TPT was assessed in 49 children with unilateral CP (mean age 9y 8mo, SD 1y 11mo, range 6-15y; 30 males, 19 females; 23 with right unilateral CP). All participants additionally underwent a standardized upper limb evaluation at body function and activity level. We investigated: (1) known-group, concurrent, and construct validity and (2) impact of sensorimotor impairments including spasticity, grip force, stereognosis, and mirror movements using analysis of covariance, Spearman's rank correlation (r), and multiple linear regression (R2 ) respectively. RESULTS: TPT outcomes significantly differed according to the Manual Ability Classification System (p<0.001, known-group validity). Relationships were found between the unimanual TPT tasks and the Jebsen-Taylor Hand Function Test (r=0.86-0.88, concurrent validity). Bimanual TPT tasks were negatively correlated with the Assisting Hand Assessment, ABILHAND-Kids, and Children's Hand-use Experience Questionnaire (r=-0.38 to -0.78, construct validity). Stereognosis was the main determinant influencing all tasks (p<0.001, R2 =37-50%). Unimanual dexterity was additionally determined by grip strength (p<0.05, R2 =8-9%) and mirror movements in the more impaired hand (p<0.05, R2 =4-8%). Bimanual dexterity was also explained by mirror movements in the more impaired hand (p<0.01, R2 =10-16%) and spasticity (p=0.04, R2 =5%). INTERPRETATION: The TPT is a valid test to measure unimanual and bimanual dexterity in unilateral CP. The results further emphasize the importance of somatosensory impairments in children with unilateral CP. What this paper adds The Tyneside Pegboard Test is valid for measuring unimanual and bimanual dexterity in unilateral cerebral palsy. Children with poorer manual ability show worse unimanual and bimanual dexterity. Stereognosis is the main predictor of both unimanual and bimanual dexterity. Stronger mirror movements in the more impaired hand result in worse bimanual dexterity.

Assessment of mirror movements in children and adolescents with unilateral cerebral palsy: reliability of the Woods and Teuber scale.

AIM: To investigate the inter- and intrarater reliability of the Woods and Teuber scale to detect mirror movements in children and adolescents with unilateral cerebral palsy (CP). METHOD: A convenience sample of children and adolescents with unilateral CP (n=68; 31 males, 37 females; mean age 12y 2mo, SD 3y 6mo) in Manual Ability Classification levels I to III was recruited from Norway, Australia, and Belgium. Three therapists scored mirror movements according to the Woods and Teuber scale from three video-recorded tasks at two separate time points. A two-way, mixed model regression was used to calculate intraclass correlation coefficients (ICCs) reflecting overall inter- and intrarater reliability. In addition, ICCs for each hand and task were calculated separately. RESULTS: The overall interrater reliability ICC was 0.90 and the corresponding intrarater reliability ICC was 0.92. The ICCs for each hand ranged from 0.86 to 0.92 and for each task from 0.63 to 0.89. INTERPRETATION: The Woods and Teuber scale shows excellent reliability for scoring mirror movements in children and adolescents with unilateral CP. The assessment is easy to administer with no need for specific equipment and scoring can be determined from short video recordings, making it a feasible instrument in research and clinical practice.

Test-retest reliability of static and dynamic motor fatigability protocols using grip and pinch strength in typically developing children.

Upper limb motor fatigability is an important debilitating factor for activities of daily living in clinical pediatric populations. However, the reliability of fatigability protocols in these populations is currently unknown. Therefore, the current study investigates test-retest reliability of a static and dynamic motor fatigability protocol for grip and pinch strength in typically developing children (TDC). Eighty-nine TDC (35 boys, 54 girls; mean age 10 years 11 months) used a grip and pinch dynamometer for static (sustained) and dynamic (repeated) contractions during 30s. For static motor fatigability (SFI), mean (Fmean) and SD (Fvar) of force were calculated, and for dynamic motor fatigability, F mean and number of peaks (Npeaks) were calculated. Intraclass correlation coefficients (ICC) were calculated. ICCs of F mean in static and dynamic motor fatigability were high (ICC: 0.94-0.96 and 0.91-0.98). ICCs were moderate to high for F var (ICC: 0.67-0.85). The SFI showed moderate ICCs (ICC: 0.69-0.77). ICCs were moderate to high for N peaks (ICC: 0.78-0.91).Conclusion: The results suggest that static and dynamic motor fatigability in for grip and pinch can be used reliably in TD children aged 6-18 years. What is Known: •Psychometric properties of motor fatigability protocols using grip and pinch in children are lacking. •Motor fatigability in grip and pinch is an important debilitating symptom in multiple neurologic populations. What is New: •Static fatigability can be investigated using a 30-s maximum sustained grip strength protocol in children. •Dynamic fatigability can be investigated using a 30-s maximum repeated grip strength protocol in children.

Performance of Upper Limb module for Duchenne muscular dystrophy.

AIM: To report the differences between Performance of Upper Limb (PUL) versions 1.2 and 2.0, compare the measurement ability of the two versions, and compare their longitudinal performance in Duchenne muscular dystrophy. METHOD: Rasch analysis was performed on the dual data from three centres to confirm whether the two scales measure the same construct. Change scores in natural history for the different domains were compared for the two versions. RESULTS: Rasch analysis demonstrated that both versions measure the same construct and that the PUL 2.0 was a better fit to the construct of motor performance and better able to detect change at 12 months in all levels of ability than the PUL 1.2. This was also true when change scores were reviewed over 2 years. INTERPRETATION: Our results confirm that the PUL 1.2 and 2.0 versions detect change in all domains over 2 years. They also demonstrate that simplifying the original scoring of the PUL 1.2 for the revised PUL 2.0 maintains the validity of the construct and enhances the scale measurement qualities. WHAT THIS PAPER ADDS: The original and revised Performance of Upper Limb (PUL) scales measure the same construct. Both scales detected change in all domains over 2 years. The PUL 2.0 enhances the measurement qualities of the scale.

Reliability and concurrent validity of a modified timed up and go test for healthy preschoolers.

The purpose of this study was to assess the interrater and test-retest reliability and the concurrent validity of the modified timed up and go test for preschoolers. As such, we aim to determine the most suited outcome of the modified timed up and go test: the best or the average performance. Thirty-two children (age 3-5) performed three timed up and go test trials as fast as possible on two separate occasions. During the first session, two researchers recorded the time to perform the task simultaneously. For reliability analyses, intra-class correlation coefficients (ICCs) and the minimal detectable change were determined. A Pearson correlation coefficient was calculated to determine concurrent validity between the timed up and go test and the balance subscale of the Movement Assessment Battery for Children, 2nd edition. Interrater (ICC > 0.97) and test-retest (ICC > 0.75) reliability were good both for the average and the best timed up and go test performance. A minimal detectable change of 1.86 s was found for the best performance, and 2.30 s for the average performance. Only the best timed up and go test performance correlated significantly with the balance subscale of the Movement Assessment Battery for Children, 2nd edition, though fair (r = -0.347, p = 0.007).Conclusion: The modified timed up and go test for preschoolers using the best performance is reliable and recommended to reduce standard and measurement error. What is Known: • A large variety of timed up and go test protocols is available for children • The protocols differ in the instructions on walking speed (self-selected/fastest), the use of an extra motivation (e.g., touch a star on the wall) and the applied outcome (average/best performance) What is New: • The best timed up and go test performance induces more consistent test results between raters and sessions and also less standard and measurement error in 3- to 5-year-old children • The best timed up and go test performance should be preferred over the average performance to achieve both reliable and valid test results in 3- to 5-year-old children.

Measuring Motor Fatigability in the Upper Limbs in Individuals With Neurologic Disorders: A Systematic Review.

OBJECTIVE: To summarize the literature on definitions, assessment protocols, and outcome measures for motor fatigability in patients with neurologic problems and investigates the known clinimetric properties according to the COnsensus-based Standards for the selection of health Measurement INstruments (COSMIN) criteria. DATA SOURCES: Two databases were consulted for studies published between January 2003 and November 2018 using the terms "motor fatigability," "nervous system disease," and "upper limb." STUDY SELECTION: Studies were included if they were (1) not older than 15 years; (2) written in English, German, or Dutch; (3) involved upper limbs of patients with neurologic disease; and (4) adequately described protocols using maximum voluntary contractions. DATA EXTRACTION: Thirty-three studies were included, describing 14 definitions, 37 assessment protocols, and 9 outcome measures. The following data were obtained: (1) author and publication year; (2) aim; (3) fatigability definition; (4) sample characteristics; (5) fatigability protocol; (8) measurement system; and (9) outcome measure. DATA SYNTHESIS: Protocols relating to body function level of the International Classification of Functioning (ICF) were most often performed in patients with multiple sclerosis (MS) including maximal or submaximal, isometric or concentric, and eccentric contractions of variable duration. For ICF activities level, most protocols included wheelchair-related tasks. Clinimetric properties were known in 2 included protocols. Test-retest reliability in patients with MS were moderate to excellent for the static fatigue index and moderate for the dynamic fatigue index. CONCLUSIONS: Based on physiology, recommendations are made for protocols and outcome measures for motor fatigability at the ICF body function level. For the ICF activities level, too little is known to make sound statements on the use of protocols in populations with neurologic disease. Clinimetric properties should be further investigated for populations with neurologic problems.

Action observation training for rehabilitation in brain injuries: a systematic review and meta-analysis.

BACKGROUND: To systematically review and analyse the effects of Action Observation Training on adults and children with brain damage. METHODS: Seven electronic databases (Cochrane, EBSCO, Embase, Eric, PubMed, Scopus and Web of Science) were searched up to 16 September 2018 to select Randomized Controlled Trials focused on adults and children with brain damage that included AOT training on upper and/or lower limb carried out for at least 1 week. Identification of studies and data extraction was conducted with two reviewers working independently. Oxford Centre for Evidence-based Medicine (March2009) - Levels of Evidence and Physiotherapy Evidence Database scale were used to grade studies. The data collected from the articles were analysed using software R, version 3.4.3. Hedge's g values were calculated and effect size estimates were pooled across studies. Separate meta-analyses were carried out for each ICF domain (i.e. body function and activity) for upper and lower limb. RESULTS: Out of the 210 records identified after removing duplicates, 22 were selected for systematic review and 19 were included in the meta-analysis. Thirteen studies included in the meta-analysis focused on upper limb rehabilitation (4 in children and 9 in adults) and 6 on lower limb rehabilitation (only studies in adults). A total of 626 patients were included in the meta-analysis. An overall statistically significant effect size was found for upper limb body function (0.44, 95% CI: [0.24, 0.64], p < 0.001) and upper limb activity domain (0.47, 95% CI: [0.30, 0.64], p < 0.001). For lower limb, only the activity domain was analysed, revealing a statistically significant overall effect size (0.56, 95% CI: [0.28, 0.84], p < 0.001). CONCLUSIONS: Action Observation Training (AOT) is an innovative rehabilitation tool for individuals with brain damage, which shows promising results in improving the activity domain for upper and lower limbs, and also the body function domain for the upper limb. However, the examined studies lack uniformity and further well-designed, larger controlled trials are necessary to determine the most suitable type of AOT particularly in children. SYSTEMATIC REVIEW REGISTRATION: CRD42019119600.

The Timed Up and Go Test in Children: Does Protocol Choice Matter? A Systematic Review.

PURPOSE: Results on reliability and normative data for the Timed Up and Go test (TUG) in children who are developing typically are systematically reviewed. SUMMARY OF KEY POINTS: Six different TUG protocols are presented for which normative data are available for ages 3 to 18 years. TUG time is consistent within and between raters and sessions and is influenced by age. The choice of protocol, self-selected versus fastest walking speed, and use of a motivational aspect and of the outcome calculation affect TUG time as well as its consistency within and between sessions. CONCLUSIONS: A standard protocol for the TUG is lacking and should be developed with attention to reliability. RECOMMENDATIONS FOR CLINICAL PRACTICE: If the TUG is to be used as a screening tool for dynamic balance control, clinicians need to apply protocols that include fastest walking speed motivation.

Windmill-task as a New Quantitative and Objective Assessment for Mirror Movements in Unilateral Cerebral Palsy: A Pilot Study.

OBJECTIVE: To introduce the Windmill-task, a new objective assessment tool to quantify the presence of mirror movements (MMs) in children with unilateral cerebral palsy (UCP), which are typically assessed with the observation-based Woods and Teuber scale (W&T). DESIGN: Prospective, observational, cohort pilot study. SETTING: Children's hospital. PARTICIPANTS: Prospective cohort of children (N=23) with UCP (age range, 6-15y, mean age, 10.5±2.7y). INTERVENTIONS: Not applicable. MAIN OUTCOME MEASURES: The concurrent validity of the Windmill-task is assessed, and the sensitivity and specificity for MM detection are compared between both assessments. To assess the concurrent validity, Windmill-task data are compared with W&T data using Spearman rank correlations (ρ) for 2 conditions: affected hand moving vs less affected hand moving. Sensitivity and specificity are compared by measuring the mean percentage of children being assessed inconsistently across both assessments. RESULTS: Outcomes of both assessments correlated significantly (affected hand moving: ρ=.520; P=.005; less affected hand moving: ρ=.488; P=.009). However, many children displayed MMs on the Windmill-task, but not on the W&T (sensitivity: affected hand moving: 27.5%; less affected hand moving: 40.6%). Only 2 children displayed MMs on the W&T, but not on the Windmill-task (specificity: affected hand moving: 2.9%; less affected hand moving: 1.4%). CONCLUSIONS: The Windmill-task seems to be a valid tool to assess MMs in children with UCP and has an additional advantage of sensitivity to detect MMs.

Combining constraint-induced movement therapy and action-observation training in children with unilateral cerebral palsy: a randomized controlled trial.

BACKGROUND: Upper limb (UL) deficits in children with unilateral cerebral palsy (uCP) have traditionally been targeted with motor execution treatment models, such as modified Constraint-Induced Movement Therapy (mCIMT). However, new approaches based on a neurophysiological model such as Action-Observation Training (AOT) may provide new opportunities for enhanced motor learning. The aim of this study is to describe a randomised controlled trial (RCT) protocol investigating the effects of an intensive treatment model, combining mCIMT and AOT compared to mCIMT alone on UL function in children with uCP. Additionally, the role of neurological factors as potential biomarkers of treatment response will be analysed. METHODS: An evaluator-blinded RCT will be conducted in 42 children aged between 6 and 12 years. Before randomization, children will be stratified according to their House Functional Classification Scale, age and type of corticospinal tract wiring. A 2-week day-camp will be set up in which children receive intensive mCIMT therapy for 6 hours a day on 9 out of 11 consecutive days (54 h) including AOT or control condition (15 h). During AOT, these children watch video sequences showing goal-directed actions and subsequently execute the observed actions with the more impaired UL. The control group performs the same actions after watching computer games without human motion. The primary outcome measure will be the Assisting Hand Assessment. Secondary outcomes comprise clinical assessments across body function, activity and participation level of the International Classification of Function, Disability and Health. Furthermore, to quantitatively evaluate UL movement patterns, a three-dimensional motion analysis will be conducted. UL function will be assessed at baseline, immediately before and after intervention and at 6 months follow up. Brain imaging comprising structural and functional connectivity measures as well as Transcranial Magnetic Stimulation (TMS) to evaluate corticospinal tract wiring will be acquired before the intervention. DISCUSSION: This paper describes the methodology of an RCT with two main objectives: (1) to evaluate the added value of AOT to mCIMT on UL outcome in children with uCP and (2) to investigate the role of neurological factors as potential biomarkers of treatment response. TRIAL REGISTRATION: NCT03256357 registered on 21st August 2017 (retrospectively registered).

Corticospinal Tract Wiring and Brain Lesion Characteristics in Unilateral Cerebral Palsy: Determinants of Upper Limb Motor and Sensory Function.

Brain lesion characteristics (timing, location, and extent) and the type of corticospinal tract (CST) wiring have been proposed as determinants of upper limb (UL) motor function in unilateral cerebral palsy (uCP), yet an investigation of the relative combined impact of these factors on both motor and sensory functions is still lacking. Here, we first investigated whether structural brain lesion characteristics could predict the underlying CST wiring and we explored the role of CST wiring and brain lesion characteristics to predict UL motor and sensory functions in uCP. Fifty-two participants with uCP (mean age (SD): 11 y and 3 m (3 y and 10 m)) underwent a single-pulse Transcranial Magnetic Stimulation session to determine CST wiring between the motor cortex and the more affected hand (n = 17 contralateral, n = 19 ipsilateral, and n = 16 bilateral) and an MRI to determine lesion timing (n = 34 periventricular (PV) lesion, n = 18 corticosubcortical (CSC) lesion), location, and extent. Lesion location and extent were evaluated with a semiquantitative scale. A standardized protocol included UL motor (grip strength, unimanual capacity, and bimanual performance) and sensory measures. A combination of lesion locations (damage to the PLIC and frontal lobe) significantly contributed to differentiate between the CST wiring groups, reclassifying the participants in their original group with 57% of accuracy. Motor and sensory functions were influenced by each of the investigated neurological factors. However, multiple regression analyses showed that motor function was predicted by the CST wiring (more preserved in individuals with contralateral CST (p < 0.01)), lesion extent, and damage to the basal ganglia and thalamus. Sensory function was predicted by the combination of a large and later lesion and an ipsilateral or bilateral CST wiring, which led to increased sensory deficits (p < 0.05). These novel insights contribute to a better understanding of the underlying pathophysiology of UL function and may be useful to delineate individualized treatment strategies.

Time Course of Upper Limb Function in Children with Unilateral Cerebral Palsy: A Five-Year Follow-Up Study.

Knowledge on long-term evolution of upper limb function in children with unilateral cerebral palsy (CP) is scarce. The objective was to report the five-year evolution in upper limb function and identify factors influencing time trends. Eighty-one children (mean age 9 y and 11 mo, SD 3 y and 3 mo) were assessed at baseline with follow-up after 6 months, 1, and 5 years. Passive range of motion (PROM), tone, muscle, and grip strength were assessed. Activity measurements included Melbourne Assessment, Jebsen-Taylor test, Assisting Hand Assessment (AHA), and ABILHAND-Kids. At 5-year follow-up, PROM (p < 0.001) and AHA scores (p < 0.001) decreased, whereas an improvement was seen for grip strength (p < 0.001), Melbourne Assessment (p = 0.003), Jebsen-Taylor test (p < 0.001), and ABILHAND-Kids (p < 0.001). Age influenced the evolution of AHA scores (p = 0.003), with younger children being stable over time, but from 9 years onward, children experienced a decrease in bimanual performance. Manual Ability Classification System (MACS) levels also affected the evolution of AHA scores (p = 0.02), with stable scores in MACS I and deterioration in MACS II and III. In conclusion, over 5 years, children with unilateral CP develop more limitations in PROM, and although capacity measures improve, the spontaneous use of the impaired limb in bimanual tasks becomes less effective after the age of 9 years.

Clinical patterns of dystonia and choreoathetosis in participants with dyskinetic cerebral palsy.

AIM: The aim of the study was to map clinical patterns of dystonia and choreoathetosis and to assess the relation between functional classifications and basal ganglia and thalamus lesions in participants with dyskinetic cerebral palsy (CP). METHODS: In this cross-sectional study, 55 participants with dyskinetic CP (mean age 14y 6mo, SD 4y 1mo; range 6-22y) were assessed with the Dyskinesia Impairment Scale and classified with the Gross Motor Function Classification System (GMFCS), Manual Ability Classification System (MACS), and Communication Function Classification System (CFCS). RESULTS: Dystonia and choreoathetosis are simultaneously present. Median levels of dystonia (70.2%) were significantly higher than levels of choreoathetosis (26.7%) and both were significantly higher during activity than at rest (both p<0.01). High correlations were found between dystonia levels and GMFCS level (Spearman's rank correlation coefficient, rS =0.70; 95% confidence interval [CI] 0.53-0.81; p<0.01) and MACS (rS =0.65; 95% CI 0.47-0.81; p<0.01), and fair correlation with CFCS (rs =0.36; 95% CI=0.11-0.57; p<0.05). No significant correlation was found between choreoathetosis levels and motor classifications. Finally, higher choreoathetosis levels were found in participants with pure thalamus and basal ganglia lesions (p=0.03) than mixed lesions, but not for dystonia (p=0.41). INTERPRETATION: Dystonia and choreoathetosis increase during activity. However, dystonia predominates and seems to have a larger impact on functional abilities. Our findings further suggest that choreoathetosis seems to be more linked to pure thalamus and basal ganglia lesions than dystonia.

Do mirror movements relate to hand function and timing of the brain lesion in children with unilateral cerebral palsy?

AIM: This study aimed to systematically map the severity of mirror movements in both hands in a prospective cohort of children with unilateral cerebral palsy, and to explore the relationship with hand function and brain lesion type. METHOD: Seventy-eight children were included (41 males, 37 females; age 9y 4mo, SD 3y 1mo, range 5-15y). Mirror movements were scored during three repetitive tasks following Woods and Teuber criteria. Strength, tone, Melbourne Assessment, Jebsen-Taylor test, and Assisting Hand Assessment were evaluated. Lesions were classified into malformations (n=5), periventricular (n=43), cortico-subcortical (n=22), and postnatally acquired lesions (n=8). RESULTS: Significantly more mirror movements were observed in the non-paretic versus the paretic hand (p≤0.003). Higher mirror movement scores in the non-paretic hand significantly correlated with lower distal strength and lower scores on unimanual and bimanual assessments (r=0.29-0.41). In the paretic hand, significant differences were found between lesion types (p=0.03). INTERPRETATION: The occurrence of mirror movements in the non-paretic hand seems related to hand function while mirror movements in the paretic hand seem more related to the lesion timing, whereby children with earlier lesions present with more mirror movements.

Guidelines for future research in constraint-induced movement therapy for children with unilateral cerebral palsy: an expert consensus.

AIM: The aim of this study was to provide an overview of what is known about constraintinduced movement therapy (CIMT) in children with unilateral cerebral palsy (CP), to identify current knowledge gaps, and to provide suggestions for future research. METHOD: Nine experts participated in a consensus meeting. A comprehensive literature search was conducted and data were summarized before the meeting. The core model produced by the European network for Health Technology Assessment was used as a framework for discussion and to identify critical issues for future research. RESULTS: All models of CIMT have demonstrated improvements in the upper limb abilities of children with unilateral CP. A consensus was reached on 11 important questions to be further explored in future studies. The areas of highest priority included the effect of dosage, the effect of repeated CIMT, and the impact of predictive factors, such as age, on the response to CIMT. Consensus suggestions for future study designs and the use of validated outcome measures were also provided. INTERPRETATION: The CIMT construct is complex, and much remains unknown. It is unclear whether a specific model of CIMT demonstrates superiority over others and whether dosage of training matters. Future research should build upon existing knowledge and aim to provide information that will help implement CIMT in various countries with different healthcare resources and organizational structures.