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.
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GEOZS, IJS, IMTLJ, KILJ, KISLJ, NUK, OILJ, PNG, SAZU, SBCE, SBJE, UL, UM, UPCLJ, UPUK, ZRSKP
Aim The aim of this article was to clarify the impact and consequences of very preterm birth (born <32wks of gestation) and/or very low birthweight (VLBW, weighing <1500g) on brain volume ...development throughout childhood and adolescence.
Method The computerized databases PubMed, Web of Knowledge, and EMBASE were searched for studies that reported volumetric outcomes during childhood or adolescence using magnetic resonance imaging and included a term‐born comparison group. Fifteen studies were identified, encompassing 818 very preterm/VLBW children and 450 term‐born peers. Average reductions in the total brain volume, white matter volume, grey matter volume, and in the size of the cerebellum, hippocampus, and corpus callosum were investigated using meta‐analytic methods.
Results Very preterm/VLBW children were found to have a significantly smaller total brain volume than the comparison group (d=−0.58; 95% confidence interval CI −0.43 to −0.73; p<0.001), smaller white matter volume (d=−0.53; CI −0.40 to −0.67; p<0.001), smaller grey matter volume (d=−0.62; CI −0.48 to −0.76; p<0.001), smaller cerebellum (d=−0.74; CI −0.56 to −0.92; p<0.001), smaller hippocampus (d=−0.47; CI −0.26 to −0.69; p<0.001), and smaller corpus callosum (d=−0.71; CI −0.34 to −1.07; p<0.001). Reductions have been associated with decreased general cognitive functioning, and no relations with age at assessment were found.
Interpretation Very preterm/VLBW birth is associated with an overall reduction in brain volume, which becomes evident in equally sized reductions in white and grey matter volumes, as well as in volumes of diverse brain structures throughout childhood and adolescence.
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BFBNIB, FZAB, GIS, IJS, KILJ, NLZOH, NUK, OILJ, SAZU, SBCE, SBMB, UL, UM, UPUK
Late-onset sepsis is frequently seen in preterm infants and is associated with poor neurodevelopmental outcome. White matter damage is proposed as substrate of poor outcome, with contributing factors ...as regional hypoxia and effects of cytokines on oligodendrocytes. We investigated the relation between cerebral oxygenation during (suspected) late-onset sepsis and neurodevelopmental outcome. Prospective cohort study, including preterm infants (gestational age <32 weeks and/or birthweight <1500 grams) with (suspected) late-onset sepsis underwent NIRS registration during the first 72 hours of suspected late-onset sepsis. At two years corrected age neurodevelopment was scored using the Bayley Scales of Infant Development-II. Thirty-two infants were included. Twenty-seven infants were identified with proven late-onset sepsis and five infants had clinical sepsis without positive blood culture. In this study, late-onset sepsis was predominantly caused by coagulase negative staphylococci (CoNS) (72%). All NIRS values were within normal limits. No association was found between NIRS and impaired neurodevelopmental outcome (n = 4) at corrected age two years: composite cognitive score 105 (80-115), composite motor score 103 (82-118) (median and range). In this pilot study, late-onset sepsis (predominantly caused by CoNS with a relatively mild clinical course), was not associated with aberrant NIRS values, nor with impaired neurodevelopmental outcome. Further research might establish our findings and elucidate effects of other micro-organisms on cerebral perfusion.
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DOBA, IZUM, KILJ, NUK, PILJ, PNG, SAZU, SIK, UILJ, UKNU, UL, UM, UPUK
Early brain lesions which produce cerebral palsy (CP) may affect the development of walking. It is unclear whether or how neuromuscular control, as evaluated by muscle synergy analysis, differs in ...young children with CP compared to typically developing (TD) children with the same walking ability, before and after the onset of independent walking. Here we grouped twenty children with (high risk of) CP and twenty TD children (age 6.5-52.4 months) based on their walking ability, supported or independent walking. Muscle synergies were extracted from electromyography data of bilateral leg muscles using non-negative matrix factorization. Number, synergies' structure and variability accounted for when extracting one (VAF
) or two (VAF
) synergies were compared between CP and TD. Children in the CP group recruited fewer synergies with higher VAF
and VAF
compared to TD children in the supported and independent walking group. The most affected side in children with asymmetric CP walking independently recruited fewer synergies with higher VAF
compared to the least affected side. Our findings suggest that early brain lesions result in early alterations of neuromuscular control, specific for the most affected side in asymmetric CP.
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IZUM, KILJ, NUK, PILJ, PNG, SAZU, UL, UM, UPUK
Despite the increased prevalence of comorbid attention deficit hyperactivity disorder (ADHD) in children with myotonic dystrophy type 1, the effects of methylphenidate treatment on associated ...cognitive deficits in this population is not yet investigated.
We describe a case study of an eleven-year-old male patient with myotonic dystrophy type 1 and comorbid ADHD that was treated with methylphenidate in a twice daily regime (0.60 mg/kg/day). Positive effects on learning and cognition were reported by the parents and teachers. No negative side effects were reported. Sequential neuropsychological assessments before and 45 minutes after methylphenidate intake were conducted to quantify the cognitive effects of methylphenidate treatment. Significant improvements in regulation of attention were behaviorally observed and were quantified using eye tracking technology.
We conclude that methylphenidate may be an effective treatment for ADHD-related cognitive deficits and learning difficulties in children with myotonic dystrophy type 1 which merits further research.
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GEOZS, IJS, IMTLJ, KILJ, KISLJ, NLZOH, NUK, OILJ, PNG, SAZU, SBCE, SBJE, UILJ, UL, UM, UPCLJ, UPUK, ZAGLJ, ZRSKP
► This systematic review examines the of current knowledge of neural correlates and executive function in children with cerebral palsy (CP). ► To our knowledge this is the first systematic review on ...brain structure in CP cerebral palsy and executive function. ► Executive function was poorer in all domains in children with CP than typically developing children (TDC) and a range of associated neural structures were identified. ► Poorer executive function was associated with a range of neural structures.
This systematic review aimed to establish the current knowledge about brain structure and executive function (EF) in children with cerebral palsy (CP). Five databases were searched (up till July 2012). Six articles met the inclusion criteria, all included structural brain imaging though no functional brain imaging. Study quality was assessed using the STROBE checklist. All articles scored between 58.7% and 70.5% for quality (100% is the maximum score). The included studies all reported poorer performance on EF tasks for children with CP compared to children without CP. For the selected EF measures non-significant effect sizes were found for the CP group compared to a semi-control group (children without cognitive deficits but not included in a control group). This could be due to the small sample sizes, group heterogeneity and lack of comparison of the CP group to typically developing children. The included studies did not consider specific brain areas associated with EF performance. To conclude, there is a paucity of brain imaging studies focused on EF in children with CP, especially of studies that include functional brain imaging. Outcomes of the present studies are difficult to compare as each study included different EF measures and cortical abnormality measures.
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GEOZS, IJS, IMTLJ, KILJ, KISLJ, NUK, OILJ, PNG, SAZU, SBCE, SBJE, UL, UM, UPCLJ, UPUK
We investigated the impact of pediatric traumatic brain injury (TBI) on attention, a prerequisite for behavioral and neurocognitive functioning.
Children aged 6 to 13 years who were diagnosed with ...TBI (n = 113; mean 1.7 years postinjury) were compared with children with a trauma control injury (not involving the head) (n = 53). TBI severity was defined as mild TBI with or without risk factors for complicated TBI (mild(RF+) TBI, n = 52; mild(RF-) TBI, n = 24) or moderate/severe TBI (n = 37). Behavioral functioning was assessed by using parent and teacher questionnaires, and the Attention Network Test assessed alerting, orienting, and executive attention. Ex-Gaussian modeling determined the contribution of extremely slow responses (lapses of attention) to mean reaction time (MRT).
The TBI group showed higher parent and teacher ratings of attention and internalizing problems, higher parent ratings of externalizing problems, and lower intelligence than the control group (P < .05, d ≥ 0.34). No effect of TBI on alerting, orienting, and executive attention was observed (P ≥ .55). MRT was slower in the TBI group (P = .008, d = 0.45), traced back to increased lapses of attention (P = .002, d = 0.52). The mild(RF-) TBI group was unaffected, whereas the mild(RF+) TBI and moderate/severe TBI groups showed elevated parent ratings of behavior problems, lower intelligence, and increased lapses of attention (P ≤ .03, d ≥ 0.48). Lapses of attention fully explained the negative relation between intelligence and parent-rated attention problems in the TBI group (P = .02).
Lapses of attention represent a core attention deficit in children with mild(RF+) TBI (even in the absence of intracranial pathology) or moderate/severe TBI, and relate to daily life problems after pediatric TBI.
In this systematic review we investigate which instrumented measurements are available to assess motor impairments, related activity limitations and participation restrictions in children and young ...adults with dyskinetic cerebral palsy. We aim to classify these instrumented measurements using the categories of the international classification of functioning, disability and health for children and youth (ICF-CY) and provide an overview of the outcome parameters.
A systematic literature search was performed in November 2019. We electronically searched Pubmed, Embase and Scopus databases. Search blocks included (a) cerebral palsy, (b) athetosis, dystonia and/or dyskinesia, (c) age 2-24 years and (d) instrumented measurements (using keywords such as biomechanics, sensors, smartphone, and robot).
Our search yielded 4537 articles. After inspection of titles and abstracts, a full text of 245 of those articles were included and assessed for further eligibility. A total of 49 articles met our inclusion criteria. A broad spectrum of instruments and technologies are used to assess motor function in dyskinetic cerebral palsy, with the majority using 3D motion capture and surface electromyography. Only for a small number of instruments methodological quality was assessed, with only one study showing an adequate assessment of test-retest reliability. The majority of studies was at ICF-CY function and structure level and assessed control of voluntary movement (29 of 49) mainly in the upper extremity, followed by assessment of involuntary movements (15 of 49), muscle tone/motor reflex (6 of 49), gait pattern (5 of 49) and muscle power (2 of 49). At ICF-CY level of activities and participation hand and arm use (9 of 49), fine hand use (5 of 49), lifting and carrying objects (3 of 49), maintaining a body position (2 of 49), walking (1 of 49) and moving around using equipment (1 of 49) was assessed. Only a few methods are potentially suitable outside the clinical environment (e.g. inertial sensors, accelerometers).
Although the current review shows the potential of several instrumented methods to be used as objective outcome measures in dyskinetic cerebral palsy, their methodological quality is still unknown. Future development should focus on evaluating clinimetrics, including validating against clinical meaningfulness. New technological developments should aim for measurements that can be applied outside the laboratory.
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IZUM, KILJ, NUK, PILJ, PNG, SAZU, UL, UM, UPUK
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.
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?
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.
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).
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.
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GEOZS, IJS, IMTLJ, KILJ, KISLJ, NLZOH, NUK, OILJ, PNG, SAZU, SBCE, SBJE, UILJ, UL, UM, UPCLJ, UPUK, ZAGLJ, ZRSKP
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