65 results on '"Monbaliu E"'
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2. The Use of Night Orthoses in Cerebral Palsy Treatment: Sleep Disturbance in Children and Parental Burden or Not?
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Mol, E. M., Monbaliu, E., Ven, M., Vergote, M., and Prinzie, P.
- Abstract
In this study, we investigated whether (1) children with cerebral palsy (CP) using night orthoses experience more sleep disturbance than those not using night orthoses, (2) parental personality is related to the experienced parental burden of night orthoses, and (3) parental sense of competence in the parenting role mediates the relation between parental personality and parental burden. Eighty-two Flemish children with CP (55 using/27 not using night orthoses) with a mean age of 9 years and 10 months (GMFCS level I-V) participated in this cross-sectional questionnaire study, using the Sleep Disturbance Scale (SDSC), Parenting Stress Index (PSI), Big Five Inventory (BFI) and a newly developed inventory to assess the parental burden of night orthoses. Multivariate analysis of covariance revealed no statistical significant differences in sleep disturbance between children using/not using night orthoses. These findings are positive as the use of night orthoses are presumed to be important in providing adequate postural care in children with CP. Extraverted and emotionally stable parents experienced less parental burden. Mediation analysis revealed that parental sense of competence partially mediated the relation between parental personality and the parental burden. These results suggest that integrating parental sense of competence in treatment programs can improve the understanding of experienced burden. Implications of such integrations for therapy are discussed. (Contains 2 figures and 4 tables.)
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- 2012
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3. Rating Scales for Dystonia in Cerebral Palsy: Reliability and Validity
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Monbaliu, E., Ortibus, E., and Roelens, F.
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Aim: This study investigated the reliability and validity of the Barry-Albright Dystonia Scale (BADS), the Burke-Fahn-Marsden Movement Scale (BFMMS), and the Unified Dystonia Rating Scale (UDRS) in patients with bilateral dystonic cerebral palsy (CP). Method: Three raters independently scored videotapes of 10 patients (five males, five females; mean age 13y 3mo, SD 5y 2mo, range 5-22y). One patient each was classified at levels I-IV in the Gross Motor Function Classification System and six patients were classified at level V. Reliability was measured by (1) intraclass correlation coefficient (ICC) for interrater reliability, (2) standard error of measurement (SEM) and smallest detectable difference (SDD), and (3) Cronbach's alpha for internal consistency. Validity was assessed by Pearson's correlations among the three scales used and by content analysis. Results: Moderate to good interrater reliability was found for total scores of the three scales (ICC: BADS = 0.87; BFMMS = 0.86; UDRS = 0.79). However, many subitems showed low reliability, in particular for the UDRS. SEM and SDD were respectively 6.36% and 17.72% for the BADS, 9.88% and 27.39% for the BFMMS, and 8.89% and 24.63% for the UDRS. High internal consistency was found. Pearson's correlations were high. Content validity showed insufficient accordance with the new CP definition and classification. Interpretation: Our results support the internal consistency and concurrent validity of the scales; however, taking into consideration the limitations in reliability, including the large SDD values and the content validity, further research on methods of assessment of dystonia is warranted.
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- 2010
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4. Relationship between dystonia and choreoathetosis, and activity, participation and quality of life in dyskinetic cerebral palsy: FP3-5
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MONBALIU, E, DE COCK, P, MAILLEUX, L, KLINGELS, K, ORTIBUS, E, and FEYS, H
- Published
- 2014
5. The reliability and validity of the Dyskinetic Measurement Scale: a new instrument to measure dystonia and choreoathetosis in cerebral palsy: G7
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MONBALIU, E, FEYS, H, ORTIBUS, E, DE CAT, J, HEYRMAN, L, PRINZIE, P, and DE COCK, P
- Published
- 2011
6. The Trunk Control Measurement Scale: a new assessment of trunk control in children with cerebral palsy: G2
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FEYS, H, HEYRMAN, L, MOLENAERS, G, DESLOOVERE, K, VERHEYDEN, G, DE CAT, J, and MONBALIU, E
- Published
- 2011
7. Behavioral problems in children with motor and intellectual disabilities: Prevalence and associations with maladaptive personality and marital relationship.
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Vrijmoeth C, Monbaliu E, Lagast E, and Prinzie P
- Published
- 2012
8. The Dyskinesia Impairment Scale: a new instrument to measure dystonia and choreoathetosis in dyskinetic cerebral palsy.
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Monbaliu E, Ortibus E, De Cat J, Dan B, Heyrman L, Prinzie P, De Cock P, Feys H, Monbaliu, Elegast, Ortibus, Els, De Cat, Jos, Dan, Bernard, Heyrman, Lieve, Prinzie, Peter, De Cock, Paul, and Feys, Hilde
- Abstract
Aim: The aim of this study was to examine the reliability and validity of the Dyskinesia Impairment Scale (DIS). The DIS consists of two subscales: dystonia and choreoathetosis. It measures both phenomena in dyskinetic cerebral palsy (CP).Method: Twenty-five participants with dyskinetic CP (17 males; eight females; age range 5–22y; mean age 13y 6mo; SD 5y 4mo), recruited from special schools for children with motor disorders, were included. Exclusion criteria were changes in muscle relaxant medication within the previous 3 months, orthopaedic or neurosurgical interventions within the previous year, and spinal fusion. Interrater reliability was verified by two independent raters. For interrater reliability, intraclass correlation coefficients were assessed. Standard error of measurement, the minimal detectable difference, and Cronbach’s alpha for internal consistency were determined. For concurrent validity of the DIS dystonia subscale, the Barry–Albright Dystonia Scale was administered.Results: The intraclass correlation coefficient for the total DIS score and the two subscales ranged between 0.91 and 0.98 for interrater reliability. The reliability of the choreoathetosis subscale was found to be higher than that of the dystonia subscale. The standard error of the measurement and minimal detectable difference values were adequate. Cronbach’s alpha values ranged from 0.89 to 0.93. Pearson’s correlation between the dystonia subscale and Barry–Albright Dystonia Scale was 0.84 (p<0.001).Interpretation: Good to excellent reliability and validity were found for the DIS. The DIS may be promising for increasing insights into the natural history of dyskinetic CP and evaluating interventions. Future research on the responsiveness of the DIS is warranted. [ABSTRACT FROM AUTHOR]- Published
- 2012
9. PP1.6 – 1680 Clinical patterns of secondary dystonia and choreoathetosis in dyskinetic CP.
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Monbaliu, E, Ortibus, E, Prinzie, P, De Cock, P, Klingels, K, Heyrman, L, and Feys, H
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- 2013
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10. Upper limb movement characteristics of children and youth with dyskinetic cerebral palsy – A sensor approach.
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Vanmechelen, I., Bekteshi, S., Konings, M., Feys, H., Desloovere, K., Aerts, J.M., and Monbaliu, E.
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DYSKINESIAS , *PEOPLE with cerebral palsy , *HUMAN mechanics , *WALKING speed , *MOTOR ability testing of children - Published
- 2020
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11. Three-dimensional upper limb movement characteristics in children and youth with dyskinetic cerebral palsy.
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Vanmechelen, I., Bekteshi, S., Konings, M., Feys, H., Desloovere, K., Aerts, J.M., and Monbaliu, E.
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DYSKINESIAS , *PEOPLE with cerebral palsy , *HUMAN kinematics , *WALKING speed , *EXTENSOR muscles , *MUSCLE strength testing - Published
- 2020
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12. An intensive eye-tracking intervention in children with dyskinetic cerebral palsy: a multiple case study.
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Puttemans F, Copermans E, Karlsson P, Ortibus E, Dan B, Monbaliu E, and Bekteshi S
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This study aimed to explore the effects of a four-week intensive eye-tracking intervention on children with dyskinetic cerebral palsy (DCP), focusing on goal attainment, communication competencies, stress levels, subjective workload, and caregivers' perception of psychosocial impact. A multiple case study design with non-concurrent, staggered multiple baselines was employed, involving three children aged 7, 12, and 13 years. The study included a randomized baseline period of two or three weeks, an intensive eye-tracking intervention, and a six-month follow-up. Two individual eye-tracking goals were identified and assessed using the Goal Attainment Scale, while communication competencies were evaluated with the Augmentative and Alternative Communication Profile: A Continuum of Learning. Stress levels were monitored through Heart Rate Variability measured by the Bittium Faros 360° ECG Holter during eye-tracking tasks. Subjective workload and psychosocial impact were assessed using pictograms and the Psychosocial Impact of Assistive Devices Scale, respectively. Descriptive statistics were applied for analysis. All participants attained and retained their eye-tracking goals, regardless of their initial functional profiles or prior experience with eye-tracking technology. Post-intervention improvements in communication competencies were maintained at the six-month follow-up. Variations in stress levels, subjective workload, and psychosocial impact were observed among participants across different phases of the study, aiding the interpretation of the results. The study concludes that a structured, tailored, four-week intensive eye-tracking intervention can yield successful results in children with DCP, irrespective of their baseline communication abilities or functional profile. Recommendations for future research, including more robust methodologies and reliable computerized tests, are provided.
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- 2024
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13. Digital technologies for pediatric rehabilitation: current access and use in the European Rehatech4child survey.
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Mensah-Gourmel J, Bekteshi S, Brochard S, Monbaliu E, Grigoriu AI, Newman CJ, Konings M, DE LA Cruz J, and Pons C
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Background: Digital technologies such as robotics and treadmill-systems (RobTS), virtual-reality and active video-gaming (VR-AVG), and telehealth and apps (T&Apps) used within pediatric motor rehabilitation may promote recovery and improve function. However, digital technology uptake may be limited in clinical practice., Aim: To explore access to and use of digital technologies for pediatric motor rehabilitation (DT4R) in Europe as a function of individual and environmental factors, as well as potential barriers to their use., Design: This observational study was based on RehaTech4child, a cross-sectional survey (2022), supported by the European Academy of Childhood Disability., Setting: Online survey available in 20 European languages., Population: The survey was disseminated through convenience and snowball sampling to pediatric motor rehabilitation professionals in Europe., Methods: The survey included items on outcomes (access, use, purposes of use and intention to use for the three categories of DT4R, i.e. RobTS, VR-AVG and T&Apps), determinants (socio-demographics, rehabilitation practice) and barriers. The association between access and use, and individual and environmental determinants was assessed using logistic regression adjusted for age, gender and profession., Results: Of the 1397 responses received, 635 were included. Respectively 67.7% and 74.3% of respondents reported using and having access to at least one of the three categories of DT4R. T&Apps and VR-AVG were used by 50.8% and 45.5% of respondents, respectively, and RobTS by 36.6% (P<0.001). Ease of access was the main determinant of use and frequency of use. Individual (e.g. age) and environmental (e.g. healthcare facility, patients' age) factors were access determinants. At least 70% of professionals intended to use a DT4R if available. Lack of financial resources and training were the most frequently reported severe barriers., Conclusions: This study found that DT4R were already used in clinical practice by around two-thirds of respondents and that they generally wished to use them even more. Access was the main determinant of use and frequency of use., Clinical Rehabilitation Impact: To facilitate access and use of DT4R, infrastructure and financial resources should be outlined, and training opportunities provided for professionals. Practice guidance should be developed and adapted for specific age groups and rehabilitation goals.
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- 2024
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14. Pharmacological and neurosurgical management of cerebral palsy and dystonia: Clinical practice guideline update.
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Fehlings D, Agnew B, Gimeno H, Harvey A, Himmelmann K, Lin JP, Mink JW, Monbaliu E, Rice J, Bohn E, and Falck-Ytter Y
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- Humans, Neurosurgical Procedures standards, Practice Guidelines as Topic standards, Cerebral Palsy surgery, Cerebral Palsy complications, Dystonia drug therapy, Dystonia surgery
- Abstract
Dystonia, typically characterized by slow repetitive involuntary movements, stiff abnormal postures, and hypertonia, is common among individuals with cerebral palsy (CP). Dystonia can interfere with activities and have considerable impact on motor function, pain/comfort, and ease of caregiving. Although pharmacological and neurosurgical approaches are used clinically in individuals with CP and dystonia that is causing interference, evidence to support these options is limited. This clinical practice guideline update comprises 10 evidence-based recommendations on the use of pharmacological and neurosurgical interventions for individuals with CP and dystonia causing interference, developed by an international expert panel following the Grading of Recommendations, Assessment, Development and Evaluations (GRADE) approach. The recommendations are intended to help inform clinicians in their use of these management options for individuals with CP and dystonia, and to guide a shared decision-making process in selecting a management approach that is aligned with the individual's and the family's values and preferences., (© 2024 The Authors. Developmental Medicine & Child Neurology published by John Wiley & Sons Ltd on behalf of Mac Keith Press.)
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- 2024
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15. Wheelchair skills training improves power mobility and participation in young people with cerebral palsy.
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Naaris M, Konings M, Ortibus E, and Monbaliu E
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- Humans, Female, Male, Adolescent, Child, Young Adult, Treatment Outcome, Cerebral Palsy rehabilitation, Cerebral Palsy physiopathology, Wheelchairs, Motor Skills physiology
- Abstract
Aim: To explore the effect of a 4-week structured power wheelchair skills training programme (WSTP) intervention on mobility skills and participation in children and young people (CYP) with cerebral palsy (CP)., Method: This was a one-group, repeated-measures study; baseline, intervention, and retention phases, each lasting 4 weeks, were used. Twelve participants (three females, nine males) with a mean age of 15 years 11 months (SD = 3 years 6 months) classified in Gross Motor Function Classification System levels IV and V participated in the study. To be included in the study, individuals had to be aged 6 to 21 years and currently using a power wheelchair. Participants received 12 WSTP training sessions of 45 minutes, 3 times per week. Power mobility skills were assessed using the Wheelchair Skills Test (WST) before baseline, before the intervention, after the intervention, and at the follow-up; mobility-related participation was assessed with the Canadian Occupational Performance Measure (COPM). Generalized mixed models with Bonferroni correction were used to assess the differences between the assessment points (p < 0.05)., Results: Statistical analysis showed a 10.4% (12.5) increase in WST total scores (p < 0.001) after the intervention compared to before the intervention, and a 1-point (0.9) increase in the COPM performance subdomain (p = 0.002)., Interpretation: Power mobility skills and mobility-related participation improved after a 4-week WSTP intervention in CYP with CP. Thus, task-based power mobility skills training based on the WSTP, and in line with individualized needs and capabilities, taking place in a natural environment, should be recommended. Power mobility skills training needs to be structured and individualized; the training interventions must consider the individual, the task, and the environment., (© 2024 Mac Keith Press.)
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- 2024
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16. Markerless motion analysis to assess reaching-sideways in individuals with dyskinetic cerebral palsy: A validity study.
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Vanmechelen I, Van Wonterghem E, Aerts JM, Hallez H, Desloovere K, Van de Walle P, Buizer AI, Monbaliu E, and Haberfehlner H
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- Humans, Male, Female, Child, Adolescent, Young Adult, Movement physiology, Child, Preschool, Biomechanical Phenomena, Adult, Range of Motion, Articular physiology, Video Recording, Shoulder physiopathology, Cerebral Palsy physiopathology
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This study aimed to evaluate clinical utility of 2D-markerless motion analysis (2DMMA) from a single camera during a reaching-sideways-task in individuals with dyskinetic cerebral palsy (DCP) by determining (1) concurrent validity by correlating 2DMMA against marker-based 3D-motion analysis (3DMA) and (2) construct validity by assessing differences in 2DMMA features between DCP and typically developing (TD) peers. 2DMMA key points were tracked from frontal videos of a single camera by DeepLabCut and accuracy was assessed against human labelling. Shoulder, elbow and wrist angles were calculated from 2DMMA and 3DMA (as gold standard) and correlated to assess concurrent validity. Additionally, execution time and variability features such as mean point-wise standard deviation of the angular trajectories (i.e. shoulder elevation, elbow and wrist flexion/extension) and wrist trajectory deviation by mean overshoot and convex hull were calculated from key points. 2DMMA features were compared between the DCP group and TD peers to assess construct validity. Fifty-one individuals (30 DCP;21 TD; age:5-24 years) participated. An accuracy of approximately 1.5 cm was reached for key point tracking. While significant correlations were found for wrist (ρ = 0.810;p < 0.001) and elbow angles (ρ = 0.483;p < 0.001), 2DMMA shoulder angles were not correlated (ρ = 0.247;p = 0.102) to 3DMA. Wrist and elbow angles, execution time and variability features all differed between groups (Effect sizes 0.35-0.81;p < 0.05). Videos of a reaching-sideways-task processed by 2DMMA to assess upper extremity movements in DCP showed promising validity. The method is especially valuable to assess movement variability., Competing Interests: Declaration of competing interest The authors declare that they have no known competing financial interests or personal relationships that could have appeared to influence the work reported in this paper., (Copyright © 2024 Elsevier Ltd. All rights reserved.)
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- 2024
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17. A Novel Video-Based Methodology for Automated Classification of Dystonia and Choreoathetosis in Dyskinetic Cerebral Palsy During a Lower Extremity Task.
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Haberfehlner H, Roth Z, Vanmechelen I, Buizer AI, Jeroen Vermeulen R, Koy A, Aerts JM, Hallez H, and Monbaliu E
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- Humans, Adolescent, Male, Female, Child, Lower Extremity physiopathology, Machine Learning, Cerebral Palsy physiopathology, Cerebral Palsy complications, Cerebral Palsy classification, Cerebral Palsy diagnosis, Dystonia physiopathology, Dystonia diagnosis, Dystonia classification, Dystonia etiology, Video Recording, Athetosis physiopathology, Athetosis diagnosis, Athetosis etiology
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Background: Movement disorders in children and adolescents with dyskinetic cerebral palsy (CP) are commonly assessed from video recordings, however scoring is time-consuming and expert knowledge is required for an appropriate assessment., Objective: To explore a machine learning approach for automated classification of amplitude and duration of distal leg dystonia and choreoathetosis within short video sequences., Methods: Available videos of a heel-toe tapping task were preprocessed to optimize key point extraction using markerless motion analysis. Postprocessed key point data were passed to a time series classification ensemble algorithm to classify dystonia and choreoathetosis duration and amplitude classes (scores 0, 1, 2, 3, and 4), respectively. As ground truth clinical scoring of dystonia and choreoathetosis by the Dyskinesia Impairment Scale was used. Multiclass performance metrics as well as metrics for summarized scores: absence (score 0) and presence (score 1-4) were determined., Results: Thirty-three participants were included: 29 with dyskinetic CP and 4 typically developing, age 14 years:6 months ± 5 years:15 months. The multiclass accuracy results for dystonia were 77% for duration and 68% for amplitude; for choreoathetosis 30% for duration and 38% for amplitude. The metrics for score 0 versus score 1 to 4 revealed an accuracy of 81% for dystonia duration, 77% for dystonia amplitude, 53% for choreoathetosis duration and amplitude., Conclusions: This methodology study yielded encouraging results in distinguishing between presence and absence of dystonia, but not for choreoathetosis. A larger dataset is required for models to accurately represent distinct classes/scores. This study presents a novel methodology of automated assessment of movement disorders solely from video data., Competing Interests: Declaration of Conflicting InterestsThe author(s) declared no potential conflicts of interest with respect to the research, authorship, and/or publication of this article.
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- 2024
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18. The relationship between manual ability, dystonia and choreoathetosis severity and upper limb movement patterns during reaching and grasping in children and young adults with dyskinetic cerebral palsy.
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Vanmechelen I, Haberfehlner H, Martens BHM, Vermeulen RJ, Buizer AI, Desloovere K, Aerts JM, Feys H, and Monbaliu E
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- Humans, Male, Female, Adolescent, Child, Young Adult, Hand Strength physiology, Athetosis physiopathology, Movement physiology, Cerebral Palsy physiopathology, Cerebral Palsy complications, Upper Extremity physiopathology, Dystonia physiopathology, Severity of Illness Index
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Introduction: Impaired upper limb movements are a key feature in dyskinetic cerebral palsy (CP). However, information on how specific movement patterns relate to manual ability, performance and underlying movement disorders is lacking. Insight in these associations may contribute to targeted upper limb management in dyskinetic CP. This study aimed to explore associations between deviant upper limb movement patterns and (1) manual ability, (2) severity of dystonia/choreoathetosis, and (3) movement time/trajectory deviation during reaching and grasping., Participants/methods: Participants underwent three-dimensional upper limb analysis during reaching forwards (RF), reaching sideways (RS) and reach-and-grasp vertical (RGV) as well as clinical assessment. Canonical correlation and regression analysis with statistical parametric mapping were used to explore associations between clinical/performance parameters and movement patterns (mean and variability)., Results: Thirty individuals with dyskinetic CP participated (mean age 16±5 y; 20 girls). Lower manual ability was related to higher variability in wrist flexion/extension during RF and RS early in the reaching cycle (p < 0.05). Higher dystonia severity was associated with higher mean wrist flexion (40-82 % of the reaching cycle; p = 0.004) and higher variability in wrist flexion/extension (31-75 %; p < 0.001) and deviation (2-14 %; p = 0.007/60-73 %; p = 0.006) during RF. Choreoathetosis severity was associated with higher elbow pro/supination variability (12-19 %; p = 0.009) during RGV. Trajectory deviation was associated with wrist and elbow movement variability (p < 0.05)., Conclusion: Current novel analysis of upper limb movement patterns and respective timings allows to detect joint angles and periods in the movement cycle wherein associations with clinical parameters occur. These associations are not present at each joint level, nor during the full movement cycle. This knowledge should be considered for individualized treatment strategies., Competing Interests: Declaration of competing interest The authors declare that they have no known competing financial interests or personal relationships that could have appeared to influence the work reported in this paper., (© 2024 Published by Elsevier Ltd on behalf of European Paediatric Neurology Society.)
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- 2024
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19. 'Composing a new symphony': Co-construction, impact creation, and innovation for individuals with childhood-onset disabilities.
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Monbaliu E
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- Child, Humans, Disabled Children
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- 2024
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20. Altered upper limb kinematics in individuals with dyskinetic cerebral palsy in comparison with typically developing peers - A statistical parametric mapping study.
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Vanmechelen I, Desloovere K, Haberfehlner H, Martens B, Vermeulen JR, Buizer AI, Aerts JM, Feys H, and Monbaliu E
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- Humans, Adolescent, Biomechanical Phenomena, Upper Extremity, Movement, Wrist Joint, Cerebral Palsy
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Background: Dyskinetic cerebral palsy (DCP) is clinically characterized by involuntary movements and abnormal postures, which can aggravate with activity. While upper limb movement variability is often detected in the clinical picture, it remains unknown how movement patterns of individuals with DCP differ from typically developing (TD) peers., Research Question: Do individuals with DCP show i) higher time-dependent standard deviations of upper limb joint angles and ii) altered upper limb kinematics in time and/or amplitude during functional upper limb tasks in comparison with TD individuals?, Methods: Three-dimensional upper limb movement patterns were cross-sectionally compared in 50 individuals with and without DCP during three functional tasks: reach forward (RF), reach and grasp vertical (RGV) and reach sideways (RS). Mean and point-wise standard deviations of angular waveform of the upper limb joint angles were compared between groups to evaluate differences in time and/or amplitude using traditional and non-linear registration statistical parametric mapping., Results: Thirty-five extremities from 30 individuals (mean age 17y4m, range 5-25 y; MACS level I(n = 2); II(n = 15); III(n = 16); IV(n = 2)) with DCP and twenty TD individuals (mean age 16y8m, range 8-25 y) were evaluated. The DCP compared to TD group showed higher point-wise standard deviations at the level of all joints, which was time-dependent and varied between tasks. Mean wrist and elbow flexion was higher for the DCP group during RF (0-83 % wrist; 57-100 % elbow), RGV (0-82 % wrist; 12-100 % elbow) and RS (0-43 % wrist; 70-100 % elbow)., Significance: This is the first study exploring the movement patterns of individuals with DCP during reaching using quantitative measures. Analyzing these individual movement patterns by statistical parametric mapping (SPM) allows us to focus on both specific joint or on specific timing during the movement cycle. The individual information that this method yields can guide individual therapy aiming to improve reaching function in different parts of the movement cycle or evaluate intervention effects on upper extremity treatment., Competing Interests: Declaration of Competing Interest The authors declare no conflict of interest., (Copyright © 2023 Elsevier B.V. All rights reserved.)
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- 2024
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21. Effectiveness of wheeled mobility skill interventions in children and young people with cerebral palsy: A systematic review.
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Naaris M, Bekteshi S, Aufheimer M, Gerling K, Hallez H, Ortibus E, Konings M, and Monbaliu E
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- Humans, Child, Adolescent, Quality of Life, Cerebral Palsy
- Abstract
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., (© 2023 Mac Keith Press.)
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- 2023
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22. Long-Term Follow-Up of Pediatric Patients with Dyskinetic Cerebral Palsy and Deep Brain Stimulation.
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Koy A, Kühn AA, Schiller P, Huebl J, Schneider GH, Eckenweiler M, Rensing-Zimmermann C, Coenen VA, Krauss JK, Saryyeva A, Hartmann H, Lorenz D, Volkmann J, Matthies C, Schnitzler A, Vesper J, Gharabaghi A, Weiss D, Bevot A, Marks W, Howser A, Monbaliu E, Mueller J, Prinz-Langenohl R, Visser-Vandewalle V, and Timmermann L
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- Humans, Child, Adolescent, Follow-Up Studies, Prospective Studies, Globus Pallidus, Treatment Outcome, Cerebral Palsy therapy, Deep Brain Stimulation, Dyskinesias etiology, Dyskinesias therapy, Movement Disorders therapy
- Abstract
Background: Deep brain stimulation (DBS) has been increasingly used in the management of dyskinetic cerebral palsy (DCP). Data on long-term effects and the safety profile are rare., Objectives: We assessed the efficacy and safety of pallidal DBS in pediatric patients with DCP., Methods: The STIM-CP trial was a prospective, single-arm, multicenter study in which patients from the parental trial agreed to be followed-up for up to 36 months. Assessments included motor and non-motor domains., Results: Of the 16 patients included initially, 14 (mean inclusion age 14 years) were assessed. There was a significant change in the (blinded) ratings of the total Dyskinesia Impairment Scale at 36 months. Twelve serious adverse events (possibly) related to treatment were documented., Conclusion: DBS significantly improved dyskinesia, but other outcome parameters did not change significantly. Investigations of larger homogeneous cohorts are needed to further ascertain the impact of DBS and guide treatment decisions in DCP. © 2023 The Authors. Movement Disorders published by Wiley Periodicals LLC on behalf of International Parkinson and Movement Disorder Society., (© 2023 The Authors. Movement Disorders published by Wiley Periodicals LLC on behalf of International Parkinson and Movement Disorder Society.)
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- 2023
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23. The Dyskinesia Impairment Scale, Second Edition: Development, construct validity, and reliability.
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Vanmechelen I, Danielsson A, Lidbeck C, Tedroff K, Monbaliu E, and Krumlinde-Sundholm L
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- Male, Humans, Adolescent, Reproducibility of Results, Severity of Illness Index, Psychometrics, Dystonia diagnosis, Dyskinesias diagnosis, Cerebral Palsy diagnosis, Dystonic Disorders
- Abstract
Aim: To create a shortened, more user-friendly Second Edition of the Dyskinesia Impairment Scale (DIS-II) to assess dystonia and choreoathetosis, and evaluate its construct validity and reliability., Method: Scale development included an online expert meeting (n = 21) and iterative discussions within the research group (n = 6). A Rasch measurement model analysis on DIS scores from individuals with dyskinetic cerebral palsy or inherited/idiopathic dystonia (n = 123, 74 males, mean age 14 years, SD 5 years) was performed to evaluate the construct validity and reliability of the DIS-II., Results: The DIS-II evaluates dystonia and choreoathetosis in action and rest in 11 body regions, with action items scored from 0 to 3 and rest items 0 to 2. The number of videos to record are reduced from 26 to 14 and the items to score are reduced from 144 to 88. Rating scale functioning, goodness-of-fit evaluation, principal component analysis, and targeting met the predefined quality criteria of the study and construct validity was therefore considered good. Furthermore, person reliability indicated that the DIS-II can separate individuals into eight distinct ability levels., Interpretation: The DIS-II provides valid and reliable measures for dystonia and choreoathetosis, and reduces the administration and scoring time compared with the DIS. The DIS-II logit scores (interval level data) enhance comparison over time and between individuals in clinical practice and research., What This Paper Adds: Compared with the Dyskinesia Impairment Scale (DIS), the shortened edition (DIS-II) requires half of the number of videos to be scored. The DIS-II has a simplified rating scale, requiring scoring of 88 instead of 144 items. The DIS-II has shown excellent reliability and good construct validity. The interval properties of the DIS-II are superior to the ordinal level outcome measures of the DIS., (© 2022 Mac Keith Press.)
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- 2023
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24. Digital technologies for motor rehabilitation in children: protocol for a cross-sectional European survey.
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Pons C, Brochard S, Grigoriu A, Newman CJ, Monbaliu E, Mensah-Gourmel J, Gaudin-Drouelle D, Toumi A, Konings M, and de la Cruz J
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- Humans, Child, Cross-Sectional Studies, Europe, Digital Technology, Technology
- Abstract
Introduction: Digital technologies can be used as part of paediatric motor rehabilitation to remediate impairment, promote recovery and improve function. However, the uptake of digital technologies in this clinical field may be limited.The aim of this study is to describe and explain digital technology use for paediatric motor rehabilitation. The specific objectives will be: (1) to describe the access to, acceptance of and use of digital technologies as a function of individual factors related to professionals practicing motor rehabilitation with children, and of environmental factors related to paediatric rehabilitation practice and (2) to explain digital technology use with a causal model based on the 'unified theory of acceptance and use of technology'., Methods and Analysis: RehaTech4child (Rehabilitation Technologies For children) is a cross-sectional study involving an online survey, that is sponsored by the European Academy of Childhood Disability (EACD). The survey protocol follows the Strengthening the Reporting of Observational Studies in Epidemiology and CHERRIES (Checklist for Reporting Results of Internet E-Surveys) guidelines. The survey includes 43 questions about (1) respondents' individual and environmental characteristics; (2) the ease of access to digital technologies, and the frequency, type and purpose of use of those technologies and (3) acceptance of technologies and barriers to their use. The survey is intended for professionals involved in paediatric motor rehabilitation. It is disseminated across Europe by the EACD network in 20 languages. Participation is anonymous and voluntary. We aim to include 500 respondents to ensure sufficient precision for the description of study outcomes and to perform stratified analyses by the main determinants., Ethics and Dissemination: Ethics approval was waived by the Brest CHRU Institutional Review Board. The study is conducted according to current French legislation (loi Jardé (n°2012-300)) and the survey is GDPR compliant. Study findings will be presented at national and international meetings and submitted for publication in a peer-reviewed journal., Trial Registration Number: NCT05176522., Competing Interests: Competing interests: None declared., (© Author(s) (or their employer(s)) 2023. Re-use permitted under CC BY-NC. No commercial re-use. See rights and permissions. Published by BMJ.)
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- 2023
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25. Towards functional improvement of motor disorders associated with cerebral palsy.
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Bekteshi S, Monbaliu E, McIntyre S, Saloojee G, Hilberink SR, Tatishvili N, and Dan B
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- Infant, Adult, Humans, Exercise, Muscle Spasticity, Cerebral Palsy complications, Cerebral Palsy therapy, Motor Disorders complications
- Abstract
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., Competing Interests: Declaration of interests We declare no competing interests., (Copyright © 2023 Elsevier Ltd. All rights reserved.)
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- 2023
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26. Quantitative assessment of trunk movements in functional reaching in children and adolescents with dyskinetic cerebral palsy.
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Van Wonterghem E, Vanmechelen I, Haberfehlner H, Decrock B, and Monbaliu E
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- Humans, Child, Adolescent, Cross-Sectional Studies, Movement, Upper Extremity, Range of Motion, Articular, Biomechanical Phenomena, Cerebral Palsy
- Abstract
Background: Trunk control and upper limb function are often disturbed in people with dyskinetic cerebral palsy. While trunk control is fundamental in upper limb activities, insights in trunk control in dyskinetic cerebral palsy are missing. This study aimed to determine trunk movement characteristics in individuals with dyskinetic cerebral palsy during reaching., Methods: Twenty individuals with dyskinetic cerebral palsy (MACS level I-III (16y6m)) and 20 typical developing peers (17y2m) were included. Participants performed three tasks: reach forward, reach sideways, and reach and grasp vertically, using a cross-sectional study design. Movements were analyzed using 3D motion capture and a sensor on the trunk. Trunk range of motion, joint angle at point of task achievement, peak and range of angular velocity and linear acceleration were compared between groups using Mann-Whitney U and independent t-tests., Findings: Participants with dyskinetic cerebral palsy showed higher trunk range of motion in all planes during reach forward and reach and grasp vertically, and in rotation and lateral flexion during reach sideways. During reach and grasp vertically, the joint angle at point of task achievement differed in the transversal plane. Ranges of angular velocity and linear acceleration were higher for all tasks and planes for participants with dyskinetic cerebral palsy, and for peak values in nearly all planes., Interpretation: Current results provide insights in trunk control at population level. This is a first step towards a better and individualized evaluation and treatment for trunk control, being an important factor in improving functional activities for individuals with dyskinetic cerebral palsy., (Copyright © 2023 Elsevier Ltd. All rights reserved.)
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- 2023
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27. Teleintervention for users of augmentative and alternative communication devices: A systematic review.
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Bekteshi S, Konings M, Karlsson P, Criekinge TV, Dan B, and Monbaliu E
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- Humans, Cohort Studies, Language Therapy methods, Communication, Communication Disorders etiology, Communication Disorders therapy, Autistic Disorder
- Abstract
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., (© 2022 Mac Keith Press.)
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- 2023
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28. Reliability and Discriminative Validity of Wearable Sensors for the Quantification of Upper Limb Movement Disorders in Individuals with Dyskinetic Cerebral Palsy.
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Vanmechelen I, Bekteshi S, Haberfehlner H, Feys H, Desloovere K, Aerts JM, and Monbaliu E
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- Humans, Adolescent, Reproducibility of Results, Upper Extremity, Movement, Cerebral Palsy diagnosis, Movement Disorders, Dyskinesias, Wearable Electronic Devices
- Abstract
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.
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- 2023
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29. Assessment of movement disorders using wearable sensors during upper limb tasks: A scoping review.
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Vanmechelen I, Haberfehlner H, De Vleeschhauwer J, Van Wonterghem E, Feys H, Desloovere K, Aerts JM, and Monbaliu E
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Background: Studies aiming to objectively quantify movement disorders during upper limb tasks using wearable sensors have recently increased, but there is a wide variety in described measurement and analyzing methods, hampering standardization of methods in research and clinics. Therefore, the primary objective of this review was to provide an overview of sensor set-up and type, included tasks, sensor features and methods used to quantify movement disorders during upper limb tasks in multiple pathological populations. The secondary objective was to identify the most sensitive sensor features for the detection and quantification of movement disorders on the one hand and to describe the clinical application of the proposed methods on the other hand. Methods: A literature search using Scopus, Web of Science, and PubMed was performed. Articles needed to meet following criteria: 1) participants were adults/children with a neurological disease, 2) (at least) one sensor was placed on the upper limb for evaluation of movement disorders during upper limb tasks, 3) comparisons between: groups with/without movement disorders, sensor features before/after intervention, or sensor features with a clinical scale for assessment of the movement disorder. 4) Outcome measures included sensor features from acceleration/angular velocity signals. Results: A total of 101 articles were included, of which 56 researched Parkinson's Disease. Wrist(s), hand(s) and index finger(s) were the most popular sensor locations. Most frequent tasks were: finger tapping, wrist pro/supination, keeping the arms extended in front of the body and finger-to-nose. Most frequently calculated sensor features were mean, standard deviation, root-mean-square, ranges, skewness, kurtosis/entropy of acceleration and/or angular velocity, in combination with dominant frequencies/power of acceleration signals. Examples of clinical applications were automatization of a clinical scale or discrimination between a patient/control group or different patient groups. Conclusion: Current overview can support clinicians and researchers in selecting the most sensitive pathology-dependent sensor features and methodologies for detection and quantification of upper limb movement disorders and objective evaluations of treatment effects. Insights from Parkinson's Disease studies can accelerate the development of wearable sensors protocols in the remaining pathologies, provided that there is sufficient attention for the standardisation of protocols, tasks, feasibility and data analysis methods., Competing Interests: The authors declare that the research was conducted in the absence of any commercial or financial relationships that could be construed as a potential conflict of interest., (Copyright © 2023 Vanmechelen, Haberfehlner, De Vleeschhauwer, Van Wonterghem, Feys, Desloovere, Aerts and Monbaliu.)
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- 2023
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30. Eye movements and stress during eye-tracking gaming performance in children with dyskinetic cerebral palsy.
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Bekteshi S, Karlsson P, De Reyck L, Vermeerbergen K, Konings M, Hellin P, Aerts JM, Hallez H, Dan B, and Monbaliu E
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- Child, Cohort Studies, Eye Movements, Eye-Tracking Technology, Humans, Cerebral Palsy, Video Games
- Abstract
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 (r
s = -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., (© 2022 Mac Keith Press.)- Published
- 2022
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31. Psychometric properties of upper limb kinematics during functional tasks in children and adolescents with dyskinetic cerebral palsy.
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Vanmechelen I, Bekteshi S, Konings M, Feys H, Desloovere K, Aerts JM, and Monbaliu E
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- Adolescent, Biomechanical Phenomena, Child, Humans, Movement, Psychometrics, Upper Extremity, Cerebral Palsy
- Abstract
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., Competing Interests: The authors have declared that no competing interests exist.
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- 2022
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32. Exercise load and physical activity intensity in relation to dystonia and choreoathetosis during powered wheelchair mobility in children and youth with dyskinetic cerebral palsy.
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Bekteshi S, Nica IG, Gakopoulos S, Konings M, Maes R, Cuyvers B, Aerts JM, Hallez H, and Monbaliu E
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- Adolescent, Adult, Child, Exercise, Humans, Young Adult, Cerebral Palsy, Dystonia etiology, Movement Disorders complications, Wheelchairs
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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 < r
s < 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.- Published
- 2022
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33. Quality of Life After Deep Brain Stimulation of Pediatric Patients with Dyskinetic Cerebral Palsy: A Prospective, Single-Arm, Multicenter Study with a Subsequent Randomized Double-Blind Crossover (STIM-CP).
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Koy A, Kühn AA, Huebl J, Schneider GH, van Riesen AK, Eckenweiler M, Rensing-Zimmermann C, Coenen VA, Krauss JK, Saryyeva A, Hartmann H, Haeussler M, Volkmann J, Matthies C, Horn A, Schnitzler A, Vesper J, Gharabaghi A, Weiss D, Bevot A, Marks W, Pomykal A, Monbaliu E, Borck G, Mueller J, Prinz-Langenohl R, Dembek T, Visser-Vandewalle V, Wirths J, Schiller P, Hellmich M, and Timmermann L
- Subjects
- Adolescent, Canada, Child, Globus Pallidus, Humans, Prospective Studies, Quality of Life, Treatment Outcome, Cerebral Palsy therapy, Deep Brain Stimulation methods, Dystonia, Dystonic Disorders
- Abstract
Background: Patients with dyskinetic cerebral palsy are often severely impaired with limited treatment options. The effects of deep brain stimulation (DBS) are less pronounced than those in inherited dystonia but can be associated with favorable quality of life outcomes even in patients without changes in dystonia severity., Objective: The aim is to assess DBS effects in pediatric patients with pharmacorefractory dyskinetic cerebral palsy with focus on quality of life., Methods: The method used is a prospective, single-arm, multicenter study. The primary endpoint is improvement in quality of life (CPCHILD [Caregiver Priorities & Child Health Index of Life with Disabilities]) from baseline to 12 months under therapeutic stimulation. The main key secondary outcomes are changes in Burke-Fahn-Marsden Dystonia Rating Scale, Dyskinesia Impairment Scale, Gross Motor Function Measure-66, Canadian Occupational Performance Measure (COPM), and Short-Form (SF)-36. After 12 months, patients were randomly assigned to a blinded crossover to receive active or sham stimulation for 24 hours each. Severity of dystonia and chorea were blindly rated. Safety was assessed throughout. The trial was registered at ClinicalTrials.gov, number NCT02097693., Results: Sixteen patients (age: 13.4 ± 2.9 years) were recruited by seven clinical sites. Primary outcome at 12-month follow-up is as follows: mean CPCHILD increased by 4.2 ± 10.4 points (95% CI [confidence interval] -1.3 to 9.7; P = 0.125); among secondary outcomes: improvement in COPM performance measure of 1.1 ± 1.5 points (95% CI 0.2 to 1.9; P = 0.02) and in the SF-36 physical health component by 5.1 ± 6.2 points (95% CI 0.7 to 9.6; P = 0.028). Otherwise, there are no significant changes., Conclusion: Evidence to recommend DBS as routine treatment to improve quality of life in pediatric patients with dyskinetic cerebral palsy is not yet sufficient. Extended follow-up in larger cohorts will determine the impact of DBS further to guide treatment decisions in these often severely disabled patients. © 2021 The Authors. Movement Disorders published by Wiley Periodicals LLC on behalf of International Parkinson and Movement Disorder Society., (© 2021 The Authors. Movement Disorders published by Wiley Periodicals LLC on behalf of International Parkinson and Movement Disorder Society.)
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- 2022
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34. Clinical Presentation of Spasticity and Passive Range of Motion Deviations in Dyskinetic Cerebral Palsy in Relation to Dystonia, Choreoathetosis, and Functional Classification Systems.
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Bekteshi S, Vanmechelen I, Konings M, Ortibus E, Feys H, and Monbaliu E
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- Adult, Cerebral Palsy classification, Child, Dyskinesias classification, Dystonia classification, Female, Humans, Male, Muscle Spasticity classification, Muscle Spasticity diagnosis, Upper Extremity physiopathology, Cerebral Palsy diagnosis, Dyskinesias diagnosis, Dystonia diagnosis, Range of Motion, Articular
- Abstract
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< r
s <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.- Published
- 2021
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35. Biomechanical maturation of foot joints in typically developing boys: Novel insight in mechanics and energetics from a cross-sectional study.
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Deschamps K, Eerdekens M, Monbaliu E, Gijon G, and Staes F
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- Adolescent, Age Factors, Ankle Joint growth & development, Ankle Joint physiology, Biomechanical Phenomena physiology, Child, Child, Preschool, Cross-Sectional Studies, Foot physiology, Humans, Male, Tarsal Joints growth & development, Tarsal Joints physiology, Walking physiology, Young Adult, Foot Joints growth & development, Foot Joints physiology
- Abstract
Background: A growing body of quantitative evidence has been provided regarding age-related differences in plantar foot loading, multi-segment foot kinematics and muscle activity. Fundamental insight into the joint mechanics and energetics of the maturing foot has yet to be provided., Research Question/hypothesis: It was hypothesized that so-called 'biomechancial maturation' joint kinetics would be observed in children underneath the age of eight and that older age-groups would not differ from each other in these parameters., Methods: Fourty-three typically developing boys were recruited and allocated to three different age groups: 1) an early childhood group, 2) a middle childhood group, and 3) an early and late adolescence group. Multi-segment joint kinematics and kinetics of the Ankle-, Chopart-, Lisfranc- and Hallux joint were collected during barefoot walking. One-way Analysis of Covariance was conducted to examine differences among the outcome measures with group as a fixed factor and walking cadence as covariate., Results: The youngest group differed significantly from the other two age groups with respect to their ankle and chopart joint peak plantarflexion moment (p < 0.05). Ankle and chopart joint peak power generation as well as the lisfranc peak plantarflexion moment was found to be significantly lower in the youngest age group compared to the oldest group (p < 0.05). At the lisfranc joint, the youngest age group demonstrated a significantly higher peak plantarflexion velocity compared to the two older age groups (p < 0.05)., Significance: This study provides novel insight into the biomechanical maturation of the developing foot which may guide clinical interventions in paediatric cohorts., (Copyright © 2021 Elsevier B.V. All rights reserved.)
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- 2021
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36. 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.
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Karlsson P, Griffiths T, Clarke MT, Monbaliu E, Himmelmann K, Bekteshi S, Allsop A, Pereksles R, Galea C, and Wallen M
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- Adolescent, Adult, Child, Consensus, Delphi Technique, Female, Humans, Male, Surveys and Questionnaires, Cerebral Palsy, Clinical Decision-Making, Fixation, Ocular, Technology instrumentation, User-Computer Interface
- Abstract
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.
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- 2021
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37. Eye Gaze Gaming Intervention in Children with Dyskinetic Cerebral Palsy: A Pilot Study of Task Performance and Its Relation with Dystonia and Choreoathetosis.
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Bekteshi S, Konings M, Vanmechelen I, Deklerck J, Ortibus E, Aerts JM, Hallez H, Karlsson P, Dan B, and Monbaliu E
- Subjects
- Adolescent, Athetosis etiology, Cerebral Palsy complications, Child, Child, Preschool, Dyskinesias etiology, Dystonia etiology, Eye Movements, Female, Humans, Male, Pilot Projects, Psychomotor Performance, Task Performance and Analysis, Video Games, Athetosis rehabilitation, Cerebral Palsy rehabilitation, Dyskinesias rehabilitation, Dystonia rehabilitation, Fixation, Ocular
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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.
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- 2020
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38. Dystonia and choreoathetosis presence and severity in relation to powered wheelchair mobility performance in children and youth with dyskinetic cerebral palsy.
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Bekteshi S, Konings M, Nica IG, Gakopoulos S, Aerts JM, Hallez H, and Monbaliu E
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- Adolescent, Child, Child, Preschool, Female, Humans, Male, Young Adult, Basal Ganglia Diseases, Cerebral Palsy complications, Dystonia, Wheelchairs
- Abstract
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 (r
s= -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., Competing Interests: Declaration of competing interest The authors declare no conflict of interest. The funders had no role in the design of the study; in the collection, analyses, or interpretation of data; in the writing of the manuscript, or in the decision to publish the results., (Copyright © 2020 European Paediatric Neurology Society. Published by Elsevier Ltd. All rights reserved.)- Published
- 2020
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39. Reliability and Validity of the Dyskinesia Impairment Scale in Children and Young Adults with Inherited or Idiopathic Dystonia.
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Danielsson A, Vanmechelen I, Lidbeck C, Krumlinde-Sundholm L, Ortibus E, Monbaliu E, and Tedroff K
- Abstract
Background: The Dyskinesia Impairment Scale (DIS) is a new assessment scale for dystonia and choreoathetosis in children and youth with dyskinetic cerebral palsy. Today, the Burke-Fahn-Marsden Dystonia Rating Scale (BFM) is mostly used to assess dystonia in children with inherited dystonia. The aim of this study was to assess reliability and validity of the DIS in children and youth with inherited or idiopathic dystonia., Methods: Reliability was measured by (1) the intraclass correlation coefficients (ICCs) for inter-rater and test-retest reliability, as well as (2) standard error of measurement (SEM) and minimal detectable difference (MDD). For concurrent validity of the DIS-dystonia subscale, the BFM was administered., Results: In total, 11 males and 9 females (median age 16 years and 7 months, range 6 to 24 years) were included. For inter-rater reliability, the ICCs for the DIS total score and the dystonia and choreoathetosis subscale scores were 0.83, 0.87, and 0.71, respectively. For test-retest reliability, the ICCs for the DIS total score and the dystonia and choreoathetosis subscale scores were 0.95, 0.88, and 0.93, respectively. The SEM and MDD for the total DIS were 3.98% and 11.04%, respectively. The Spearman correlation coefficient between the dystonia subscale and the BFM was 0.88 ( p < 0.01)., Conclusions: Good to excellent inter-rater, test-retest reliability, and validity were found for the total DIS and the dystonia subscale. The choreoathetosis subscale showed moderate inter-rater reliability and excellent test-retest reliability. The DIS may be a promising tool to assess dystonia and choreoathetosis in children and young adults with inherited or idiopathic dystonia.
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- 2020
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40. Presence and severity of dystonia and choreoathetosis overflow movements in participants with dyskinetic cerebral palsy and their relation with functional classification scales.
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Vanmechelen I, Bekteshi S, Bossier K, Feys H, Deklerck J, and Monbaliu E
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- Child, Cross-Sectional Studies, Drinking, Humans, Severity of Illness Index, Cerebral Palsy, Dyskinesias, Dystonia
- Abstract
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.
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- 2020
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41. Use of the Dyskinesia Impairment Scale in non-ambulatory dyskinetic cerebral palsy.
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Haberfehlner H, Bonouvrié LA, Boeschoten K, Fleuren S, Monbaliu E, Becher JG, Vermeulen RJ, and Buizer AI
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- Adolescent, Baclofen administration & dosage, Baclofen therapeutic use, Cerebral Palsy drug therapy, Cerebral Palsy physiopathology, Child, Disability Evaluation, Dyskinesias drug therapy, Dyskinesias physiopathology, Female, Humans, Injections, Spinal, Male, Muscle Relaxants, Central administration & dosage, Muscle Relaxants, Central therapeutic use, Severity of Illness Index, Treatment Outcome, Young Adult, Cerebral Palsy diagnosis, Dyskinesias diagnosis
- Abstract
Aim: To assess the responsiveness, concurrent validity, and feasibility of the Dyskinesia Impairment Scale (DIS) in non-ambulatory patients with dyskinetic cerebral palsy (CP)., Method: The study is a secondary analysis of data collected in the IDYS trial, a randomized controlled trial on the effects of intrathecal baclofen (ITB). The DIS and Barry-Albright Dystonia Scale (BADS) were conducted at baseline and after 3 months of ITB or placebo treatment. Responsiveness was assessed by comparing the effect sizes and correlation of change after treatment between the DIS and BADS. Concurrent validity was evaluated by assessing the correlations between scales. Feasibility was evaluated for each DIS item by the number of participants who successfully accomplished the item., Results: Thirty-three non-ambulatory patients (9 females, 24 males) with dyskinetic CP (ITB-treated: n=17, mean [SD] age: 14y 1mo [4y 1mo]; placebo-treated: n=16, mean [SD] age: 14y 7mo [4y]) were included in the study. The effect sizes for BADS and DIS were similar in The ITB-treated group (-0.29 and -0.22 respectively). Changes after treatment on the DIS dystonia subscale correlated with changes on the BADS (r=0.64; p<0.001). The DIS dystonia subscale and BADS correlated at baseline and follow-up (r=0.78; p<0.001 and r=0.79; p<0.001). Not all DIS activity items could be performed in this sample of patients., Interpretation: For non-ambulatory patients with dyskinetic CP, the responsiveness of the DIS equalled the responsiveness of BADS. Concurrent validity was adequate. Feasibility for activity items was restricted in patients with severe dyskinetic CP., What This Paper Adds: The Dyskinesia Impairment Scale (DIS) and Barry-Albright Dystonia Scale showed similar responsiveness in non-ambulatory patients with dyskinetic cerebral palsy (CP). No floor or ceiling effect was observed for DIS in non-ambulatory participants. The concurrent validity of DIS was adequate in non-ambulatory participants. Patients with dyskinetic CP in Gross Motor Function Classification System levels IV and V could not perform all DIS activity items., (© 2019 The Authors. Developmental Medicine & Child Neurology published by John Wiley & Sons Ltd on behalf of Mac Keith Press.)
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- 2020
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42. Test-retest reliability of the Dyskinesia Impairment Scale: measuring dystonia and choreoathetosis in dyskinetic cerebral palsy.
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Vanmechelen I, Dan B, Feys H, and Monbaliu E
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- Adolescent, Cerebral Palsy physiopathology, Child, Child, Preschool, Dyskinesias physiopathology, Dystonia physiopathology, Female, Humans, Male, Reproducibility of Results, Severity of Illness Index, Young Adult, Cerebral Palsy diagnosis, Dyskinesias diagnosis, Dystonia diagnosis
- Abstract
Aim: To assess test-retest reliability of the Dyskinesia Impairment Scale (DIS) in children and young adults with dyskinetic cerebral palsy (CP)., Method: Dystonia and choreoathetosis were assessed in 15 participants with dyskinetic CP (13 males, 2 females; age range 5-22y, mean 14y, SD 4y) using the DIS in two separate sessions over 7 days. Exclusion criteria were changes in muscle relaxant medication within the previous 3 months, orthopaedic or neurosurgical interventions within the previous year, and spinal fusion. Intraclass correlation coefficient, confidence intervals (CI), standard error of measurement, and the minimal detectable difference (MDD) were determined for test-retest reliability., Result: Intraclass correlation coefficients of the DIS, the dystonia subscale of the DIS, and the choreoathetosis subscale of the DIS were 0.98 (95% CI 0.94-0.99), 0.97 (95% CI 0.92-0.99), and 0.96 (95% CI 0.90-0.99). The standard error of measurement and MDD were 2.6% and 7.2%., Interpretation: The DIS is a reliable tool to assess dystonia and choreoathetosis; it remains stable over time in children and young adults with dyskinetic CP. These results add to the current evidence for good clinimetric properties of the DIS., What This Paper Adds: The Dyskinesia Impairment Scale (DIS) shows stability in scoring dystonia and choreoathetosis. The total DIS score and dystonia and choreoathetosis subscales are clinically useful., (© 2019 Mac Keith Press.)
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- 2020
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43. Development of a Data Logger for Capturing Human-Machine Interaction in Wheelchair Head-Foot Steering Sensor System in Dyskinetic Cerebral Palsy.
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Gakopoulos S, Nica IG, Bekteshi S, Aerts JM, Monbaliu E, and Hallez H
- Subjects
- Cerebral Palsy psychology, Disabled Persons, Equipment Design, Humans, Movement, Robotics instrumentation, Signal Processing, Computer-Assisted, Wheelchairs, Wireless Technology, Brain-Computer Interfaces, Cerebral Palsy physiopathology, Robotics methods
- Abstract
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., Competing Interests: Abbreviations
- Published
- 2019
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44. Eyes on communication: trialling eye-gaze control technology in young children with dyskinetic cerebral palsy.
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Karlsson P, Bech A, Stone H, Vale C, Griffin S, Monbaliu E, and Wallen M
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- Cerebral Palsy physiopathology, Child, Child, Preschool, Female, Humans, Male, Cerebral Palsy rehabilitation, Communication Aids for Disabled standards, Fixation, Ocular, Nonverbal Communication
- Abstract
Purpose: This study aims to identify eye-gaze control technology outcomes, parent perception of the technology and support received, and gauge the feasibility of available measures., Methods: Five children with dyskinetic cerebral palsy, mean age 4 years, 4 months (1 year, 0 months); n = 4 males; trialled two eye-gaze control technology systems, each for six weeks. Parents completed pre- and post-questionnaires., Results: Parents found the 6-week home-based trial period to be the right length. Written guidelines and instructions about set-up, calibration, and play and learning activities were perceived as important. Children demonstrated improvements in goal achievement and performance. Parents found questionnaires on quality of life, participation, behaviours involved in mastering a skill and communication outcomes challenging to complete resulting in substantial missing data., Conclusion: Eye-gaze control technology warrants further investigation for young children with dyskinetic cerebral palsy in a large international study.
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- 2019
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45. Time Course of Upper Limb Function in Children with Unilateral Cerebral Palsy: A Five-Year Follow-Up Study.
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Klingels K, Meyer S, Mailleux L, Simon-Martinez C, Hoskens J, Monbaliu E, Verheyden G, Verbeke G, Molenaers G, Ortibus E, and Feys H
- Subjects
- Adolescent, Cerebral Palsy diagnosis, Child, Child, Preschool, Cohort Studies, Female, Follow-Up Studies, Humans, Longitudinal Studies, Male, Time Factors, Cerebral Palsy physiopathology, Hand Strength physiology, Upper Extremity physiology
- Abstract
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.
- Published
- 2018
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46. Pharmacological and neurosurgical interventions for managing dystonia in cerebral palsy: a systematic review.
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Fehlings D, Brown L, Harvey A, Himmelmann K, Lin JP, Macintosh A, Mink JW, Monbaliu E, Rice J, Silver J, Switzer L, and Walters I
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- Cerebral Palsy complications, Dystonia etiology, Humans, Baclofen therapeutic use, Deep Brain Stimulation methods, Dystonia therapy, Muscle Relaxants, Central therapeutic use, Neurosurgical Procedures methods
- Abstract
Aim: To systematically review evidence for pharmacological/neurosurgical interventions for managing dystonia in individuals with cerebral palsy (CP) to inform a care pathway., Method: Searches included studies with a minimum of five participants with dystonia in CP receiving oral baclofen, benzodiazepines (clonazepam, diazepam, lorazepam), clonidine, gabapentin, levodopa, trihexyphenidyl, botulinum toxin, intrathecal baclofen (ITB), or deep brain stimulation (DBS). Evidence was classified according to American Academy of Neurology guidelines., Results: Twenty-eight articles underwent data extraction: one levodopa, five trihexyphenidyl, three botulinum toxin, six ITB, and 13 DBS studies. No articles for oral baclofen, benzodiazepines, clonidine, or gabapentin met the inclusion criteria. Evidence for reducing dystonia was level C (possibly effective) for ITB and DBS; level C (possibly ineffective) for trihexyphenidyl; and level U (inadequate data) for botulinum toxin., Interpretation: For dystonia reduction, ITB and DBS are possibly effective, whereas trihexyphenidyl was possibly ineffective. There is insufficient evidence to support oral medications or botulinum toxin to reduce dystonia. There is insufficient evidence for pharmacological and neurosurgical interventions to improve motor function, decrease pain, and ease caregiving. The majority of the pharmacological and neurosurgical management of dystonia in CP is based on clinical expert opinion., What This Paper Adds: Intrathecal baclofen and deep brain stimulation are possibly effective in reducing dystonia. Current evidence does not support effectiveness of oral medications or botulinum toxin to reduce dystonia. Evidence is inadequate for pharmacological/neurosurgical interventions impact on improving motor function, pain/comfort, and easing caregiving. The majority of the care pathway rests on expert opinion., (© 2018 Mac Keith Press.)
- Published
- 2018
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47. Clinical presentation and management of dyskinetic cerebral palsy.
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Monbaliu E, Himmelmann K, Lin JP, Ortibus E, Bonouvrié L, Feys H, Vermeulen RJ, and Dan B
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- Cerebral Palsy therapy, Dyskinesias therapy, Humans, Cerebral Palsy physiopathology, Dyskinesias physiopathology
- Abstract
Cerebral palsy is the most frequent cause of severe physical disability in childhood. Dyskinetic cerebral palsy (DCP) is the second most common type of cerebral palsy after spastic forms. DCP is typically caused by non-progressive lesions to the basal ganglia or thalamus, or both, and is characterised by abnormal postures or movements associated with impaired tone regulation or movement coordination. In DCP, two major movement disorders, dystonia and choreoathetosis, are present together most of the time. Dystonia is often more pronounced and severe than choreoathetosis, with a major effect on daily activity, quality of life, and societal participation. The pathophysiology of both movement disorders is largely unknown. Some emerging hypotheses are an imbalance between indirect and direct basal ganglia pathways, disturbed sensory processing, and impaired plasticity in the basal ganglia. Rehabilitation strategies are typically multidisciplinary. Use of oral drugs to provide symptomatic relief of the movement disorders is limited by adverse effects and the scarcity of evidence that the drugs are effective. Neuromodulation interventions, such as intrathecal baclofen and deep brain stimulation, are promising options., (Copyright © 2017 Elsevier Ltd. All rights reserved.)
- Published
- 2017
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48. Functional outcomes in children and young people with dyskinetic cerebral palsy.
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Monbaliu E, De La Peña MG, Ortibus E, Molenaers G, Deklerck J, and Feys H
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- Adolescent, Age Factors, Child, Child, Preschool, Communication, Disability Evaluation, Disease Progression, Drinking, Eating, Female, Hand physiopathology, Humans, Male, Motor Activity, Netherlands, Severity of Illness Index, Speech, Young Adult, Cerebral Palsy classification, Cerebral Palsy physiopathology, Dyskinesias classification, Dyskinesias physiopathology
- Abstract
Aim: This cross-sectional study aimed to map the functional profile of individuals with dyskinetic cerebral palsy (CP), to determine interrelationships between the functional classification systems, and to investigate the relationship of functional abilities with dystonia and choreoathetosis severity., Methods: Fifty-five children (<15y) and young people (15-22y) (30 males, 25 females; mean age 14y 6mo, standard deviation 4y 1mo) with dyskinetic CP were assessed using the Gross Motor Function Classification System (GMFCS), Manual Ability Classification System (MACS), Communication Function Classification System (CFCS), Eating and Drinking Ability Classification System (EDACS), and Viking Speech Scale (VSS), as well as the Dyskinesia Impairment Scale., Results: Over 50 per cent of the participants exhibited the highest limitation levels in GMFCS, MACS, and VSS. Better functional abilities were seen in EDACS and CFCS. Moderate to excellent interrelationship was found among the classification scales. All scales had significant correlation (r
s =0.65 - 0.81) with dystonia severity except for CFCS in the young people group. Finally, only MACS (rs =0.40) and EDACS (rs =0.55) in the young people group demonstrated significant correlation with choreoathetosis severity., Interpretation: The need for inclusion of speech, eating, and drinking in the functional assessment of dyskinetic CP is highlighted. The study further supports the strategy of managing dystonia in particular at a younger age followed by choreoathetosis in a later stage., (© 2017 Mac Keith Press.)- Published
- 2017
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49. The relationship of dystonia and choreoathetosis with activity, participation and quality of life in children and youth with dyskinetic cerebral palsy.
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Monbaliu E, De Cock P, Mailleux L, Dan B, and Feys H
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- Adolescent, Athetosis physiopathology, Athetosis psychology, Cerebral Palsy complications, Child, Chorea physiopathology, Chorea psychology, Cross-Sectional Studies, Dystonia physiopathology, Dystonia psychology, Exercise physiology, Female, Humans, Male, Severity of Illness Index, Social Participation psychology, Surveys and Questionnaires, Young Adult, Athetosis complications, Cerebral Palsy physiopathology, Cerebral Palsy psychology, Chorea complications, Dystonia complications, Quality of Life
- Abstract
Aim: To relate dystonia and choreoathetosis with activity, participation and quality of life (QOL) in children and youth with dyskinetic Cerebral Palsy (CP)., Methods: Fifty-four participants with dyskinetic CP (mean age 14y6m, SD 4y2m, range 6-22y) were included. The Dyskinesia Impairment Scale (DIS) was used to evaluate dystonia and choreoathetosis. Activity, participation and quality of life (QOL) were assessed with the Gross Motor Function Measure (GMFM), the Functional Mobility Scale (FMS), the Jebsen-Taylor Hand Function Test (JTT), the ABILHAND-Kids Questionnaire (ABIL-K), the Life Habits Kids (LIFE-H) and the Quality of Life Questionnaire for children with CP (CP-QOL). Spearman's rank correlation coefficient (r
s ) was used to assess the relationship between the movement disorders and activity, participation and QOL measures., Results: Significant negative correlations were found between dystonia and the activity scales with Spearman's rank correlation coefficient (rs ) varying between -0.65 (95% CI = -0.78 to -0.46) and -0.71 (95% CI = -0,82 to -0.55). Correlations were also found with the LIFE-H (rs = -0.43; 95%CI = -0.64 to -0.17) and the CP-QOL (rs = -0.32; 95%CI = -0.56 to -0.03). As far as choreoathetosis is concerned, no or only weak relationships were found with the activity, participation and quality of life scales., Interpretation: This cross-sectional study is the first to examine the relationship of dystonia and choreoathetosis in dyskinetic CP with the level of activity, participation and QOL. The results revealed dystonia has a higher impact on activity, participation and quality of life than choreoathetosis. These findings seem to suggest it is necessary to first focus on dystonia reducing intervention strategies and secondly on choreoathetosis., (Copyright © 2016 European Paediatric Neurology Society. Published by Elsevier Ltd. All rights reserved.)- Published
- 2017
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50. Clinical patterns of dystonia and choreoathetosis in participants with dyskinetic cerebral palsy.
- Author
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Monbaliu E, de Cock P, Ortibus E, Heyrman L, Klingels K, and Feys H
- Subjects
- Adolescent, Adult, Athetosis epidemiology, Athetosis etiology, Basal Ganglia Diseases pathology, Cerebral Palsy complications, Cerebral Palsy epidemiology, Child, Chorea epidemiology, Chorea etiology, Cross-Sectional Studies, Dystonia epidemiology, Dystonia etiology, Female, Humans, Male, Severity of Illness Index, Thalamus pathology, Young Adult, Athetosis physiopathology, Cerebral Palsy physiopathology, Chorea physiopathology, Dystonia physiopathology
- Abstract
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., (© 2015 Mac Keith Press.)
- Published
- 2016
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