Effectiveness of wheeled mobility skill interventions in children and young people with cerebral palsy: A systematic review.

AIM: To systematically review the effectiveness of wheeled mobility interventions in children and young people with cerebral palsy (CP). METHOD: A systematic literature search was performed in MEDLINE, Embase, Cochrane Central Register of Controlled Trials, EBSCO, PEDro, and Web of Science using database-specific concepts such as 'child' and 'wheelchair'. Studies focusing on wheeled mobility skill interventions with participants aged 6 to 21 years with CP were included. RESULTS: Twenty studies with 203 participants were included. The impact of wheeled mobility skill interventions was assessed on mobility skills (n = 18), activity and participation (n = 10), and quality of life (n = 3). No studies reported effects on stress, fatigue, and motivational aspects. Interventions included power wheelchair skill training (n = 12), computer-based training (n = 5), smart wheelchair training (n = 2), and manual wheelchair training (n = 1), showing positive wheeled mobility intervention effects. Study quality based on the Methodological Index for Non-Randomized Studies scale was 9 out of 16 and 14 out of 24 for non-comparative and comparative studies respectively. Risk of bias was serious-to-critical on the Risk of Bias in Non-Randomized Studies of Interventions. INTERPRETATION: Wheeled mobility interventions showed promising beneficial effects on wheeled mobility, activity and participation, and quality of life for children and young people with CP. Future studies with structured and standardized training programmes and assessment tools are warranted to further accelerate the wheeled mobility skill acquisition process in this population. WHAT THIS PAPER ADDS: Wheeled mobility interventions improve wheeled mobility skills in individuals with cerebral palsy. The mobility skill acquisition process benefits from structured and standardized training. Knowledge of the intervention effects on participation and quality of life is limited. Most studies had moderate quality of evidence because of methodological limitations.

Reliability and Discriminative Validity of Wearable Sensors for the Quantification of Upper Limb Movement Disorders in Individuals with Dyskinetic Cerebral Palsy.

Background-Movement patterns in dyskinetic cerebral palsy (DCP) are characterized by abnormal postures and involuntary movements. Current evaluation tools in DCP are subjective and time-consuming. Sensors could yield objective information on pathological patterns in DCP, but their reliability has not yet been evaluated. The objectives of this study were to evaluate (i) reliability and (ii) discriminative ability of sensor parameters. Methods-Inertial measurement units were placed on the arm, forearm, and hand of individuals with and without DCP while performing reach-forward, reach-and-grasp-vertical, and reach-sideways tasks. Intra-class correlation coefficients (ICC) were calculated for reliability, and Mann-Whitney U-tests for between-group differences. Results-Twenty-two extremities of individuals with DCP (mean age 16.7 y) and twenty individuals without DCP (mean age 17.2 y) were evaluated. ICC values for all sensor parameters except jerk and sample entropy ranged from 0.50 to 0.98 during reach forwards/sideways and from 0.40 to 0.95 during reach-and-grasp vertical. Jerk and maximal acceleration/angular velocity were significantly higher for the DCP group in comparison with peers. Conclusions-This study was the first to assess the reliability of sensor parameters in individuals with DCP, reporting high between- and within-session reliability for the majority of the sensor parameters. These findings suggest that pathological movements of individuals with DCP can be reliably captured using a selection of sensor parameters.

Towards functional improvement of motor disorders associated with cerebral palsy.

Cerebral palsy is a lifelong neurodevelopmental condition arising from non-progressive disorders occurring in the fetal or infant brain. Cerebral palsy has long been categorised into discrete motor types based on the predominance of spasticity, dyskinesia, or ataxia. However, these motor disorders, muscle weakness, hypotonia, and impaired selective movements should also be discriminated across the range of presentations and along the lifespan. Although cerebral palsy is permanent, function changes across the lifespan, indicating the importance of interventions to improve outcomes in motor disorders associated with the condition. Mounting evidence exists for the inclusion of several interventions, including active surveillance, adapted physical activity, and nutrition, to prevent secondary and tertiary complications. Avenues for future research include the development of evidence-based recommendations, low-cost and high-quality alternatives to existing therapies to ensure universal access, standardised cerebral palsy registers to harmonise epidemiological and clinical information, improved adult screening and check-up programmes to facilitate positive lived experiences, and phase 3 trials for new interventions.

Teleintervention for users of augmentative and alternative communication devices: A systematic review.

AIM: To synthesize existing evidence on the effectiveness of speech-language teleinterventions delivered via videoconferencing to users of augmentative and alternative communication (AAC) devices. METHOD: A systematic literature search was conducted in 10 electronic databases, from inception until August 2021. Included were speech-language teleinterventions delivered by researchers and/or clinicians via videoconferencing to users of AAC devices, without restrictions on chronological age and clinical diagnosis. The quality of the studies included in the review was appraised using the Downs and Black checklist and the Single-Case Experimental Design Scale; risk of bias was assessed using the Risk Of Bias In Non-Randomized Studies - of Interventions and the single-case design risk of bias tools. RESULTS: Six teleinterventions including 25 participants with a variety of conditions, such as Down syndrome, autism, Rett syndrome, and amyotrophic lateral sclerosis met the inclusion criteria. Five studies used a single-case experimental design and one was a cohort study. Teleinterventions included active consultation (n = 2), functional communication training (n = 2), brain-computer interface (n = 1), and both teleintervention and in-person intervention (n = 1). All teleinterventions reported an increase in participants' independent use of AAC devices during the training sessions compared to baseline, as well as an overall high satisfaction and treatment acceptability. INTERPRETATION: Speech-language teleinterventions for users of AAC devices show great potential for a successful method of service delivery. Future telehealth studies with larger sample sizes and more robust methodology are strongly encouraged to allow the generalization of results across different populations. WHAT THIS PAPER ADDS: Individuals can learn to use augmentative and alternative communication (AAC) devices independently during tele-AAC interventions. Service providers and recipients reported an overall high satisfaction and acceptability for AAC services delivered via teleinterventions. Speech-language teleinterventions may be an effective method of providing AAC intervention services.

Psychometric properties of upper limb kinematics during functional tasks in children and adolescents with dyskinetic cerebral palsy.

Dyskinetic cerebral palsy (DCP) is characterised by involuntary movements, and the movement patterns of children with DCP have not been extensively studied during upper limb tasks. The aim of this study is to evaluate psychometric properties of upper limb kinematics in participants with DCP and typically developing (TD) participants. In current repeatability and validity study, forty individuals with typical development (n = 20) and DCP (n = 20) performed a reach forward/sideways and a reach and grasp task during motion analysis on two occasions. Joint angles at point of task achievement (PTA) and spatio-temporal parameters were evaluated within-and between-sessions using intra-class correlation coefficients (ICC) and standard error of measurement (SEM). Independent t-tests/Mann-Whitney-U tests were used to compare parameters between groups. Within-session ICC values ranged from 0.45 to 1.0 for all parameters for both groups. Within-session SEM values ranged from 1.1° to 11.7° for TD participants and from 1.9° to 13.0° for participants with DCP. Eight within-session repetitions resulted in the smallest change in ICC and SEM values for both groups. Within-session variability was higher for participants with DCP in comparison with the TD group for the majority of the joint angles and spatio-temporal parameters. Intrinsic variability over time was small for all angles and spatio-temporal parameters, whereas extrinsic variability was higher for elbow and scapula angles. Between-group differences revealed lower shoulder adduction and higher elbow flexion, pronation and wrist flexion, as well as higher trajectory deviation and a lower maximal velocity for participants with DCP. This is the first study to assess the psychometric properties of upper limb kinematics in children and adolescents with DCP, showing that children with DCP show higher variability during task execution, requiring a minimum of eight repetitions. However, their variable movement pattern can be reliably captured within-and between-sessions, confirming the potential of three-dimensional motion analysis for assessment of rehabilitation interventions in DCP.

Eye movements and stress during eye-tracking gaming performance in children with dyskinetic cerebral palsy.

AIM: This study aimed to explore eye movements and stress during eye-tracking gaming performance in children with dyskinetic cerebral palsy (CP) compared with typically developing children, and associations between eye-tracking performance, eye movements, stress, and participants' characteristics. METHOD: This cohort study included 12 children with dyskinetic CP aged 5 to 12 years (mean age 8 years 7 months, standard deviation [SD] 2 years 3 months) and 23 typically developing children aged 5 to 13 years (mean age 9 years 0 months, SD 2 years 7 months). Participants played 10 eye-tracking games. Tobii X3-120 and Tobii Pro Lab were used to record and analyse eye movements. Stress was assessed through heart rate variability (HRV), recorded during rest, and eye-tracking performance using the Bittium Faros360° ECG Holter device. Eye-tracking performance was measured using gaming completion time. Fixation and saccade variables were used to quantify eye movements, and time- and frequency-domain variables to quantify HRV. Non-parametric statistics were used. RESULTS: Gaming completion time was significantly different (p < 0.001) between groups, and it was negatively correlated with experience (rs  = -0.63, p = 0.029). No significant differences were found between groups in fixation and saccade variables. HRV significantly changed from rest to eye-tracking performance only in typically developing children and not in children with dyskinetic CP. INTERPRETATION: Children with dyskinetic CP took longer to perform the 10 games, especially the inexperienced users, indicating the importance of the early provision of eye-tracking training opportunities. It seems that eye-tracking tasks are not a source of increased stress and effort in children with dyskinetic CP. WHAT THIS PAPER ADDS: Participants with dyskinetic cerebral palsy (CP) took twice as long to perform 10 eye-tracking games than typically developing peers. Participants with dyskinetic CP with previous eye-tracking experience performed the games faster. Fixation and saccade variables were not significantly different between children with and without dyskinetic CP. Heart rate variability showed no differences between rest and performance in participants with dyskinetic CP. Gross Motor Function Classification System, Manual Ability Classification System, and Viking Speech Scale levels were not correlated to the eye movements or stress variables.

Exercise load and physical activity intensity in relation to dystonia and choreoathetosis during powered wheelchair mobility in children and youth with dyskinetic cerebral palsy.

PURPOSE: To explore the relation between exercise load, physical activity intensity, and movement disorders during powered wheelchair (PW) mobility in people with severe dyskinetic cerebral palsy (DCP). METHODS: Ten participants with DCP, 6-21 years old, users of a head/foot steering system were included. Dystonia and choreoathetosis were assessed using the Dyskinesia Impairment Mobility Scale (DIMS), heart rate (HR) was used to assess the exercise load of the tasks on the participants, and the accelerometry-based activity index (AI) to measure the physical activity intensity and energy expenditure during mobility task performance. RESULTS: Neck- and distal arm dystonia showed significant correlations with HR (0.64 < rs < 0.77; 0.009 < p < 0.048), whereas neck- and proximal arm choreoathetosis with AI (0.64 < rs < 0.76, 0.011 < p < 0.044). Total-body AI was strongly correlated to the AI of the arms (0.66 < rs < 0.90, < 0.001 < p < 0.038), but not to the AI of the head. CONCLUSIONS: During PW mobility tasks, dystonia is associated to exercise load and choreoathetosis to physical activity intensity and energy expenditure. Findings highlight the difficulties in measuring exercise load and activity intensity in PW users with DCP due to the involuntary hypertonic and/or hyperkinetic hallmark of the movement disorders. Nevertheless, a relaxed surrounding with minimal distractions during PW training may increase learning efficiency. Future studies with a bigger sample size are highly recommended to fully establish the relationship between the variables and to allow generalizability of results.Implications for rehabilitationDystonia is positively related to heart rate during powered mobility, which may be explained by the hypertonic hallmark of dystonia causing an increase in exercise load.Choreoathetosis is positively related to the physical activity index during powered mobility where the hyperkinetic hallmark of choreoathetosis may lead to an increase in physical activity intensity and energy expenditure.Arm overflow movements are the component which contribute the most to total-body activity index, thus, minimizing these movements may lower the overall energy expenditure during powered mobility.Mobility training in a relaxed surrounding with minimal distractions and minimized arm overflow movements may lead to a less-demanding powered wheelchair mobility experience and increased learning efficiency.

Stakeholder consensus for decision making in eye-gaze control technology for children, adolescents and adults with cerebral palsy service provision: findings from a Delphi study.

BACKGROUND: Limited research exists to guide clinical decisions about trialling, selecting, implementing and evaluating eye-gaze control technology. This paper reports on the outcomes of a Delphi study that was conducted to build international stakeholder consensus to inform decision making about trialling and implementing eye-gaze control technology with people with cerebral palsy. METHODS: A three-round online Delphi survey was conducted. In Round 1, 126 stakeholders responded to questions identified through an international stakeholder Advisory Panel and systematic reviews. In Round 2, 63 respondents rated the importance of 200 statements generated by in Round 1. In Round 3, 41 respondents rated the importance of the 105 highest ranked statements retained from Round 2. RESULTS: Stakeholders achieved consensus on 94 of the original 200 statements. These statements related to person factors, support networks, the environment, and technical aspects to consider during assessment, trial, implementation and follow-up. Findings reinforced the importance of an individualised approach and that information gathered from the user, their support network and professionals are central when measuring outcomes. Information required to support an application for funding was obtained. CONCLUSION: This Delphi study has identified issues which are unique to eye-gaze control technology and will enhance its implementation with people with cerebral palsy.

Clinical Presentation of Spasticity and Passive Range of Motion Deviations in Dyskinetic Cerebral Palsy in Relation to Dystonia, Choreoathetosis, and Functional Classification Systems.

Objectives: To map the presence, severity, and distribution of spasticity and passive range of motion (pROM) deviations in dyskinetic cerebral palsy (DCP), and to explore their relation with dystonia, choreoathetosis, and functional abilities.Methods: This cross-sectional study included 53 participants with DCP. Spasticity was assessed with the Modified Ashworth Scale, limited- and increased pROM (hypermobility) with a goniometer, dystonia and choreoathetosis with the Dyskinesia Impairment Scale, gross motor and manual abilities with corresponding functional classification systems.Results: Spasticity and limited pROM were correlated with dystonia of the upper limbs (0.41< rs<0.47, <0.001 < p < .002) and lower limbs (0.31< rs<0.41, 0.002 < p < .025), and both functional systems of gross motor (0.32< rs<0.51, <0.001 < p < .018) and fine manual abilities (0.34< rs<0.44, 0.001 < p < .014). Hypermobility is correlated only with choreoathetosis of the lower limbs (0.44, p = .001).Conclusions: Coexisting spasticity and pROM deviations in DCP are functionally limiting and should be addressed accordingly. Hypermobility may lead to an increased luxation risk.

Dystonia and choreoathetosis presence and severity in relation to powered wheelchair mobility performance in children and youth with dyskinetic cerebral palsy.

INTRODUCTION: Power wheelchairs (PW) with head/foot steering systems are used as an alternative to joysticks in children with severe dyskinetic cerebral palsy (DCP). Mobility training programs are unstandardized to date, and insight on dystonia, choreoathetosis, and mobility performance may lead to greater independent mobility. OBJECTIVE: To map the presence and severity of dystonia and choreoathetosis during PW mobility in DCP and their relation with mobility performance. METHODS: Ten participants with DCP performed four PW mobility tasks using a head/foot steering system. Dystonia and choreoathetosis in the neck and arm regions were evaluated using the Dyskinesia Impairment Mobility Scale (DIMS). PW mobility performance was assessed using time-on-task and the number of errors during performance. The Wilcoxon-signed rank test and the Spearman's correlation coefficients were used to explore differences and correlations. RESULTS: Median levels of dystonia (83.6%) were significantly higher (p < 0.01) than median levels of choreoathetosis (34.4%). Positive significant correlations were found between the Arm Proximal DIMS and the PW mobility experience (rs=-0.92, p < 0.001), and between the Arm Distal DIMS and the number of errors (rs = 0.66, p = 0.039) during mobility performance. CONCLUSIONS: Dystonia is more present and severe during PW mobility than choreoathetosis. The hypertonic hallmark of dystonia may mask the hyperkinetic hallmark of choreoathetosis, resulting in lower median levels. Results may suggest that with an increase in driving experience, children with DCP adopt deliberate strategies to minimize the negative impact of arm overflow movements on mobility performance, however, future research with bigger sample size and additional outcome measures is strongly encouraged.

Eye Gaze Gaming Intervention in Children with Dyskinetic Cerebral Palsy: A Pilot Study of Task Performance and Its Relation with Dystonia and Choreoathetosis.

OBJECTIVES: To investigate the operational competences screen navigation and dwell function underlying eye gaze performance, and the relation of dystonia and choreoathetosis with eye gaze performance in children with dyskinetic cerebral palsy (DCP). METHODS: During a 5-week intervention, ten participants with DCP played eye gaze video games daily for 30 minutes. Six games were used to assess task performance, fixation count, and eye movement accuracy during four measurements. Dystonia and choreoathetosis were evaluated using the Dyskinesia Impairment Scale. RESULTS: Eye gaze performance improved over time (p = .013). Moderate to strong within-subject correlations were found between eye movement accuracy and task performance, and between eye movement accuracy and fixation count. No significant correlations were found with the movement disorders. CONCLUSIONS: Eye gaze technology shows great potential to be a successful computer interface for children with severe DCP, thereby potentially improving their communication skills, participation levels, and quality of life.

Presence and severity of dystonia and choreoathetosis overflow movements in participants with dyskinetic cerebral palsy and their relation with functional classification scales.

Background: This cross-sectional study aims to investigate the presence and severity of overflow movements of dystonia and choreoathetosis in dyskinetic cerebral palsy (CP) and to assess the relationship of overflow movements with functional classification scales.Methods: Fifty-two subjects with dyskinetic CP were included. Presence and severity of dystonia and choreoathetosis overflow movements were assessed with the Dyskinesia Impairment Scale. Functional abilities were classified with the Gross Motor Function Classification System, Manual Ability Classification System, Viking Speech Scale, Communication Function Classification System, and Eating and Drinking Ability Classification System.Results: Dystonia and choreoathetosis overflow movements were simultaneously present. Median scores of dystonia overflow movements were significantly higher than choreoathetosis overflow movements. Dystonia and choreoathetosis overflow movements were significantly higher in extremities than in the central body. Correlations between dystonia and choreoathetosis overflow movements were fair. Moderate to good correlations were found between dystonia overflow score and Gross Motor Function Classification System, Manual Ability Classification System, and Eating and Drinking Ability Classification System.Conclusions: This is the first study to assess overflow movements in dyskinetic CP. All participants presented with dystonia and choreoathetosis overflow movements, with higher values for dystonia overflow movements. Dystonia overflow movements seem to have a larger impact on functional abilities.Implications for rehabilitationDystonia and choreoathetosis overflow movements are both present in children with dyskinetic cerebral palsy, with dystonia overflow movements being more severe than choreoathetosis overflow movements.Overflow movements impact heavily on daily functional ability and the execution of voluntary activities.Dystonia overflow movements show good correlations with functional classification scales.The measurable characteristics of overflow movements can be used as a guideline for targeted treatment with, e.g., botulinum-toxin-A injections.

Development of a Data Logger for Capturing Human-Machine Interaction in Wheelchair Head-Foot Steering Sensor System in Dyskinetic Cerebral Palsy.

The use of data logging systems for capturing wheelchair and user behavior has increased rapidly over the past few years. Wheelchairs ensure more independent mobility and better quality of life for people with motor disabilities. Especially, for people with complex movement disorders, such as dyskinetic cerebral palsy (DCP) who lack the ability to walk or to handle objects, wheelchairs offer a means of integration into daily life. The mobility of DCP patients is based on a head-foot wheelchair steering system. In this work, a data logging system is proposed to capture data from human-wheelchair interaction for the head-foot steering system. Additionally, the data logger provides an interface to multiple Inertial Measurement Units (IMUs) placed on the body of the wheelchair user. The system provides accurate and real-time information from head-foot navigation system pressure sensors on the wheelchair during driving. This system was used as a tool to obtain further insights into wheelchair control and steering behavior of people diagnosed with DCP in comparison with a healthy subject.