Three-Dimensional Upper Limb Movement Analysis in Children and Adolescents With Brachial Plexus Birth Injury: A Systematic Review.

BACKGROUND: To synthesize the current evidence on clinical use of three-dimensional upper limb movement analysis (3D-ULMA) in children and adolescents with brachial plexus birth injury (BPBI). METHODS: MEDLINE, Embase, and Web of Science were searched for relevant studies up to April 2022. An automatic e-mail alert was installed to ensure no eligible article was missed. Articles evaluating 3D-ULMA in children and adolescents with BPBI were included. Covidence web-based platform was used for blind screening of eligible articles. Twenty-one observational studies with a final sample size of 609, encompassing 493 BPBI cases, met the inclusion criteria. Data were extracted using a custom form to support standardized extraction conforming to the Cochrane Checklist of items. Risk of bias was assessed using the Newcastle-Ottawa Scale, the Strengthening the Reporting of Observational Studies in Epidemiology checklist, and a specifically established quality assessment form for kinematic analysis studies. RESULTS: Study setups differed, including six different types of kinematic devices. Twelve studies used the (modified) Mallet positions for their 3D-ULMA. Throughout the studies, 3D-ULMA was used for various purposes. The Newcastle-Ottawa Scale scored 16 articles with five stars or more, indicating fair to moderate quality. CONCLUSIONS: This systematic review summarizes the different 3D-ULMA kinematic devices, test protocols, and their clinical use for BPBI. The use of 3D-ULMA provides valuable, objective, and quantified data to clinicians with regard to movement strategies; it complements existing clinical scales and can be implemented to evaluate effectiveness of therapy interventions. Implications for future research and clinical practice are discussed.

Patient reported outcomes in brachial plexus birth injury: results from the iPLUTO world-wide consensus survey.

PURPOSE: Brachial plexus birth injuries (BPBI) can have lifelong effects on the development and functional use of the upper extremity. Currently there is no agreement with regards to what patient-reported outcome (PRO) measures should be used. Therefore, the ability to compare the effects of treatment between individuals and institutions is challenging. This study aimed to achieve consensus among clinicians on the use of PRO measures within this patient group to allow for improved comparison of treatments and outcomes in the future. MATERIALS AND METHODS: Online, a 3 round Delphi survey was completed by 35 international multi-disciplinary specialist centers. RESULTS: All respondents (100%) agreed that PRO measures are useful for clinical evaluation and patient treatment. None of the outcome measures scored >75% agreement for ability to assess responsiveness and current state in children with BPBI as most outcome measures were judged as not specific for BPBI. Additionally, participant centers were asked their perspective on the best available PRO option for each of the 3 categories: functional use of the upper limb, quality of life and pain. This resulted in endorsement by the participant centers of the Brachial Plexus Outcome Measure - Self-Evaluation, the Pediatric Quality of Life Inventory, and Visual Analogue Scale/Brief Pain Inventory respectively. CONCLUSION: International specialists in BPBI agree that PRO measures are important to use both clinically and in research in children aged 5 years and above.

Patient-reported outcome measures were judged as useful both in clinic and in research for brachial plexus birth injury (BPBI), according to a panel of specialized centers.Currently available outcome measures were judged as not specific for BPBI.The panel endorsed the following measures as best available: the Brachial Plexus Outcome Measure ­ Self-Evaluation scale for functional evaluation, the Pediatric Quality of Life Inventory for disease-related quality of life and the Faces Pain Scale - Revised/Visual Analogue Scale/Brief Pain Inventory for pain.

Defining characteristics of independent walking persons after stroke presenting with different arm swing coordination patterns.

BACKGROUND: Persons after stroke present with an altered arm swing during walking. Given the known influence of the arm swing on gait, it is important to identify the characteristics of persons with stroke with different arm-to-leg coordination patterns during walking. METHODS: Twenty-five persons after stroke walked on a self-paced treadmill at comfortable walking speed. The frequency of shoulder movements per stride was detected by Fast Fourier transform analysis on the kinematic data for hemiplegic shoulder movements in the sagittal plane. An independent-sample t-test or Mann-Whitney U test was used to compare clinical and biomechanical parameters between identified subgroups. RESULTS: Two earlier described subgroups based on the number of shoulder flexion-extension movements during one stride could be confirmed. Participants in the 1:1 ratio subgroup (one arm swing during one stride, N = 15) presented with a less upper limb impairment and less spasticity of the elbow extensors (p = 0.012) than the participants in the 2:1 ratio subgroup (two arm swings during one stride, N = 9). Although not significant, the participants in the 1:1 subgroup also seemed to have less spasticity of the shoulder internal rotators (p = 0.06) and a less walking variability based on the standard deviation of the step width. Further research on a greater sample should confirm these findings. CONCLUSION: Fast Fourier transform analysis was used to identify subgroups based on sagittal shoulder kinematics during walking. The clinical and gait related differences between the identified subgroups can be taken into account in future research investigating post-stroke gait interventions aiming to improve the arm swing.

Physical activity and physical fitness in children with heritable connective tissue disorders.

Objectives: Health problems in patients with heritable connective tissue disorders (HCTD) are diverse and complex and might lead to lower physical activity (PA) and physical fitness (PF). This study aimed to investigate the PA and PF of children with heritable connective tissue disorders (HCTD). Methods: PA was assessed using an accelerometer-based activity monitor (ActivPAL) and the mobility subscale of the Pediatric Evaluation of Disability Inventory Computer Adaptive Test (PEDI-CAT). PF was measured in terms of cardiovascular endurance using the Fitkids Treadmill Test (FTT); maximal hand grip strength, using hand grip dynamometry (HGD) as an indicator of muscle strength; and motor proficiency, using the Bruininks-Oseretsky Test of Motor Proficiency-2 (BOTMP-2). Results: A total of 56 children, with a median age of 11.6 (interquartile range [IQR], 8.8-15.8) years, diagnosed with Marfan syndrome (MFS), n = 37, Loeys-Dietz syndrome (LDS), n = 6, and genetically confirmed Ehlers-Danlos (EDS) syndromes, n = 13 (including classical EDS n = 10, vascular EDS n = 1, dermatosparaxis EDS n = 1, arthrochalasia EDS n = 1), participated. Regarding PA, children with HCTD were active for 4.5 (IQR 3.5-5.2) hours/day, spent 9.2 (IQR 7.6-10.4) hours/day sedentary, slept 11.2 (IQR 9.5-11.5) hours/day, and performed 8,351.7 (IQR 6,456.9-1,0484.6) steps/day. They scored below average (mean (standard deviation [SD]) z-score -1.4 (1.6)) on the PEDI-CAT mobility subscale. Regarding PF, children with HCTD scored well below average on the FFT (mean (SD) z-score -3.3 (3.2)) and below average on the HGD (mean (SD) z-score -1.1 (1.2)) compared to normative data. Contradictory, the BOTMP-2 score was classified as average (mean (SD) z-score.02 (.98)). Moderate positive correlations were found between PA and PF (r(39) = .378, p < .001). Moderately sized negative correlations were found between pain intensity and fatigue and time spent actively (r(35) = .408, p < .001 and r(24) = .395 p < .001, respectively). Conclusion: This study is the first to demonstrate reduced PA and PF in children with HCTD. PF was moderately positively correlated with PA and negatively correlated with pain intensity and fatigue. Reduced cardiovascular endurance, muscle strength, and deconditioning, combined with disorder-specific cardiovascular and musculoskeletal features, are hypothesized to be causal. Identifying the limitations in PA and PF provides a starting point for tailor-made interventions.

Comparing spatiotemporal gait parameters between overground walking and self-paced treadmill walking in persons after stroke.

PURPOSE: Since self-paced treadmills enable more natural gait patterns compared to fixed-speed treadmills we examined the use of a self-paced treadmill as a alternative for overground gait analysis in persons after stroke. MATERIAL AND METHODS: Twenty-five persons after stroke (10 males/15 females; 53 ± 12.05 years; 40.72 ± 42.94 months post-stroke) walked at self-selected speed overground (GAITRite, CIR Systems) and on a self-paced treadmill (GRAIL, Motek) in randomized order. Spatiotemporal parameters, variability and symmetry measures were compared using paired-sample t-tests or Wilcoxon Signed Rank tests. Concurrent validity was assessed using intraclass correlation coefficients and Bland-Altman plots. A regression model determined the contribution of the walking velocity to the changes in spatiotemporal parameters. RESULTS: The velocity on the treadmill was significant lower compared to overground (p < 0.001). This difference predicted the significant changes in other spatiotemporal parameters to varying degrees (27.7%-83.8%). Bland-Altman plots showed large percentage of bias and limits of agreement. Variability and symmetry measures were similar between conditions. CONCLUSIONS: When considering gait analysis in persons after stroke a self-paced treadmill may be a valuable alternative for overground analysis. Although a slower walking velocity, and accompanying changes in other spatiotemporal parameters, should be taken into account compared to overground walking.Implications for rehabilitationConsidering the advantages regarding space and time, instrumented treadmills provide opportunities for gait assessment and training in a stroke population.When using self-paced treadmills for clinical gait analysis in persons after stroke, the slower walking velocity and accompanying changes in other spatiotemporal parameters need to be taken into account.Stroke patients seem to preserve their walking pattern on a self-paced treadmill.

Efficacy of interdisciplinary rehabilitation in child cancer survivors: Impact on physical fitness, fatigue and body composition after 1-year follow-up.

OBJECTIVE: Childhood cancer survivors experience reduced physical activity level, participation as well as health-related quality of life. This prospective, pre-/post-intervention and follow-up cohort study aims to determine the efficacy of an interdisciplinary rehabilitation on improving physical fitness, fatigue and body composition. METHODS: A total of 24 childhood cancer survivors (mean age: 12.15 years ± 3.2; 14 females; 10 males) were recruited 6 months after medical treatment and received a 4-month interdisciplinary intervention. Cardiorespiratory fitness (PredVO2peak and PredLoadmax ), body composition (dry lean weight) and quality of life (general fatigue) were assessed at baseline, post-intervention and 1-year follow-up. Linear mixed models were used to analyse data. RESULTS: Linear mixed modelling revealed a significant main effect of time on predicted maximal load (F = 13.189, df = 36.179, p < 0.001), dry lean weight (F = 64.813, df = 37.019, p < 0.001) but also significant improvement of general fatigue score (-9.039 ± 4.300, 95% CI -17.741 to -0.336, p = 0.042), indicating a decline in general fatigue. CONCLUSION: With emerging evidence that physical activity is safe and feasible, together with increasing numbers of childhood cancer survivors at risk for long-term chronic co-morbidities, this study advocates for better access to interdisciplinary rehabilitation programmes in order to improve their physical condition and their body composition and reduce fatigue.

Motor Functioning and Intelligence Quotient in Paediatric Survivors of a Fossa Posterior Tumor Following a Multidisciplinary Rehabilitation Program.

BACKGROUND: Short- and long-term consequences after treatment for childhood fossa posterior tumors are extensively reported in the literature; however, papers highlighting physical function throughout rehabilitation and its correlation with Intelligence Quotient (IQ) are sparse. This study aims to describe the physical functioning and IQ of these survivors, their progression during rehabilitation, and the association with histopathological tumor classification. Additionally, the correlation between gross motor functioning and cognitive functioning was investigated. METHODS: This retrospective single-center cohort study included 56 children (35 (62.5%) males and 21 (37.5%) females, with an average age of 6.51 years (SD 4.13)) who followed a multidisciplinary program at the Child Rehabilitation Centre, Ghent University Hospital in the period from 2005 to 2020. Descriptive statistical analysis was performed with the use of non-parametric tests and linear regression to determine the relationship between gross motor functioning and IQ. RESULTS: This report shows impaired motor and intelligence performance in children with a fossa posterior tumor. Although multidisciplinary rehabilitation is beneficial, it is not able to counteract the further decline of several motor skills and intelligence during oncological treatment, more specifically in children with a medulloblastoma. A correlation between gross motor function and total IQ was found. CONCLUSION: Pediatric survivors of a fossa posterior tumor experience impaired physical and intellectual functions, with more decline during oncological treatment despite simultaneous multidisciplinary rehabilitation.

Neonatal brachial plexus palsy and hand representation in children and young adults.

AIM: To assess the impact of neonatal brachial plexus palsy (NBPP) on higher-order hand representation. METHOD: Eighty-two left-handed children and adolescents with and without right-sided NBPP were recruited. Thirty-one participants with NBPP (mean age [SD] 11y 4mo [4y 4mo]; age range 6y 2mo-21y 0mo; 15 females; C5-6, n=4, C5-7, n=12, C5-T1, n=11, C5-T1 with Horner sign, n=4) were assessed along with 30 controls (mean age 11y 5mo [4y 4mo]; age range 6y 7mo-21y 7mo; 14 females). Participants' estimated hand size and shape on measure of implicit and explicit hand representation was assessed. A linear mixed model (LMM) was used to investigate the effect of condition, sensorimotor impairment, and age. RESULTS: Individuals with NBPP showed a significant difference in implicit hand representation between affected and non-affected hands. LMM confirmed a significant influence of the severity of sensorimotor injury. Only the estimated implicit hand representation was associated with age, with a significant difference between 6- to 8-year-olds and 9- to 10-year-olds. INTERPRETATION: The effect of sensorimotor impairment on central hand representation in individuals with NBPP is specific due to its implicit component and is characterized by finger length underestimation in the affected hand compared to the characteristic underestimation in the unaffected hand. Neither NBPP nor age impacted the explicit hand estimate. This study confirms the importance of sensorimotor contribution to the development of implicit hand representation.

Hand size representation in healthy children and young adults.

Whereas we experience our body as a coherent volumetric object, the brain appears to maintain highly fragmented representations of individual body parts. Little is known about how body representations of hand size and shape are built and evolve during infancy and young adulthood. This study aimed to investigate the effect of hand side, handedness, and age on the development of central hand size representation. The observational study with comparison groups was conducted with 90 typically developing Belgian school children and young adults (48 male and 42 female; age range = 5.0-23.0 years; 49 left-handed and 41 right-handed). Participants estimated their hand size and shape using two different tasks. In the localization task, participants were verbally cued to judge the locations of 10 anatomical landmarks of an occluded hand. An implicit hand size map was constructed and compared with actual hand dimensions. In the template selection task, the explicit hand shape was measured with a depictive method. Hand shape indexes were calculated and compared for the actual, implicit, and explicit conditions. Participants were divided into four age groups (5-8 years, 9-10 years, 11-16 years, and 17-23 years). Implicit hand maps featured underestimation of finger length and overestimation of hand width, which is already present in the youngest children. Linear mixed modeling revealed no influence of hand side on finger length underestimation; nonetheless, a significant main effect of age (p = .001) was exposed. Sinistrals aged 11 to 16 years showed significantly less underestimation (p = .03) than dextrals of the same age. As for the hand shape, the implicit condition differed significantly with the actual and explicit conditions (p < .001). Again, the implicit shape index was subjected to handedness and age effects, with significant differences being found between sinistrals and dextrals in the age groups of 9 and 10 years (p = .029) and 11 to 16 years (p < .001). In conclusion, the implicit metric component of the hand representation in children and young adults is misperceived, featuring shortened fingers and broadened hands since a very young age. Crucially, the finger length underestimation increases with age and shows a different developmental trajectory for sinistrals and dextrals. In contrast, the explicit hand shape is approximately veridical and seems immune from age and handedness effects. This study confirms the dual character of somatoperception and establishes a point of reference for children and young adults.

Risk factors for neonatal brachial plexus palsy: a systematic review and meta-analysis.

AIM: To provide a comprehensive update on the most prevalent, significant risk factors for neonatal brachial plexus palsy (NBPP). METHOD: Cochrane CENTRAL, MEDLINE, Web of Science, Embase, and ClinicalTrials.gov were searched for relevant publications up to March 2019. Studies assessing risk factors of NBPP in relation to typically developing comparison individuals were included. Meta-analysis was performed for the five most significant risk factors, on the basis of the PRISMA statement and MOOSE guidelines. Pooled odds ratios (ORs), 95% confidence intervals (CIs), and across-study heterogeneity (I2 ) were reported. Reporting bias and quality of evidence was rated. In addition, we assessed the incidence of NBPP. RESULTS: Twenty-two observational studies with a total sample size of 29 419 037 live births were selected. Significant risk factors included shoulder dystocia (OR 115.27; 95% CI 81.35-163.35; I2 =92%), macrosomia (OR 9.75; 95% CI 8.29-11.46; I2 =70%), (gestational) diabetes (OR 5.33; 95% CI 3.77-7.55; I2 =59%), instrumental delivery (OR 3.8; 95% CI 2.77-5.23; I2 =77%), and breech delivery (OR 2.49; 95% CI 1.67-3.7; I2 =70%). Caesarean section appeared as a protective factor (OR 0.13; 95% CI 0.11-0.16; I2 =41%). The pooled overall incidence of NBPP was 1.74 per 1000 live births. It has decreased in recent years. INTERPRETATION: The incidence of NBPP is decreasing. Shoulder dystocia, macrosomia, maternal diabetes, instrumental delivery, and breech delivery are risk factors for NBPP. Caesarean section appears as a protective factor. WHAT THIS PAPER ADDS: The overall incidence of neonatal brachial plexus palsy is 1.74 per 1000 live births. The incidence has declined significantly. Shoulder dystocia, macrosomia, maternal diabetes, instrumental delivery, and breech delivery are the main risk factors. Prevention is difficult owing to unpredictability and often labour-related risk.

Early electrodiagnosis in the management of neonatal brachial plexus palsy: A systematic review.

Neonatal brachial plexus palsy (NBPP) is a prominent form of newborn morbidity with a potentially disabling persistence. Neurosurgical intervention is indicated in select NBPP patients. Early prognostic assessment would facilitate rational selection of those infants for surgery. We conducted a systematic literature review to determine the prognostic value of early electrodiagnosis (EDx) in NBPP. We included 16 observational studies with a total sample size of 747 children. Risk of bias and quality of evidence were rated. Wide variation was found in EDx techniques, outcome algorithms, and decisionmaking. Nevertheless, the most methodologically sound studies support the use of EDx, at standardized time-frames, as a key prognostic modality for complementing clinical judgment and neuroimaging. An accurate knowledge of the underlying anatomy of the nerve injury helps to counsel families and to guide reconstructive strategy.