R Jeroen Vermeulen

Maastricht Universitair Medisch Centrum, Maestricht, Limburg, Netherlands

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Publications (49)132.6 Total impact

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    ABSTRACT: Purpose: PCDH19 mutations cause epilepsy and mental retardation limited to females (EFMR) or Dravet-like syndromes. Especially in the first years of life, epilepsy is known to be highly pharmacoresistant. The aim of our study was to evaluate the effectiveness of antiepileptic therapy in patients with PCDH19 mutations. Methods: We report a retrospective multicenter study of antiepileptic therapy in 58 female patients with PCDH19 mutations and epilepsy aged 2-27 years (mean age 10.6 years). Results: The most effective drugs after 3 months were clobazam and bromide, with a responder rate of 68% and 67%, respectively, where response was defined as seizure reduction of at least 50%. Defining long-term response as the proportion of responders after 12 months of treatment with a given drug in relation to the number of patients treated for at least 3 months, the most effective drugs after 12 months were again bromide and clobazam, with a long-term response of 50% and 43%, respectively. Seventy-four percent of the patients became seizure-free for at least 3 months, 47% for at least one year. Significance: The most effective drugs in patients with PCDH19 mutations were bromide and clobazam. Although epilepsy in PCDH19 mutations is often pharmacoresistant, three quarters of the patients became seizure-free for at least for 3 months and half of them for at least one year. However, assessing the effectiveness of the drugs is difficult because a possible age-dependent spontaneous seizure remission must be considered.
    No preview · Article · Jan 2016 · Seizure
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    ABSTRACT: Background: Axonal injury after traumatic brain injury (TBI) may cause impaired sensory integration. We aim to determine the effects of childhood TBI on visual integration in relation to general neurocognitive functioning. Methods: We compared children aged 6-13 diagnosed with TBI (n = 103; M = 1.7 years post-injury) to children with traumatic control (TC) injury (n = 44). Three TBI severity groups were distinguished: mild TBI without risk factors for complicated TBI (mildRF- TBI, n = 22), mild TBI with ≥1 risk factor (mildRF+ TBI, n = 46) or moderate/severe TBI (n = 35). An experimental paradigm measured speed and accuracy of goal-directed behavior depending on: (1) visual identification; (2) visual localization; or (3) both, measuring visual integration. Group-differences on reaction time (RT) or accuracy were tracked down to task strategy, visual processing efficiency and extra-decisional processes (e.g. response execution) using diffusion model analysis. General neurocognitive functioning was measured by a Wechsler Intelligence Scale short form. Results: The TBI group had poorer accuracy of visual identification and visual integration than the TC group (Ps ≤ .03; ds ≤ -0.40). Analyses differentiating TBI severity revealed that visual identification accuracy was impaired in the moderate/severe TBI group (P = .05, d = -0.50) and that visual integration accuracy was impaired in the mildRF+ TBI group and moderate/severe TBI group (Ps < .02, ds ≤ -0.56). Diffusion model analyses tracked impaired visual integration accuracy down to lower visual integration efficiency in the mildRF+ TBI group and moderate/severe TBI group (Ps < .001, ds ≤ -0.73). Importantly, intelligence impairments observed in the TBI group (P = .009, d = -0.48) were statistically explained by visual integration efficiency (P = .002). Conclusions: Children with mildRF+ TBI or moderate/severe TBI have impaired visual integration efficiency, which may contribute to poorer general neurocognitive functioning.
    Full-text · Article · Dec 2015 · PLoS ONE
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    ABSTRACT: Objective: To study the effect of vaccination-associated seizure onset on disease course and estimate the risk of subsequent seizures after infant pertussis combination and measles, mumps, and rubella (MMR) vaccinations in Dravet syndrome (DS). Methods: We retrospectively analyzed data from hospital medical files, child health clinics, and the vaccination register for children with DS and pathogenic SCN1A mutations. Seizures within 24 hours after infant whole-cell, acellular, or nonpertussis combination vaccination or within 5 to 12 days after MMR vaccination were defined as "vaccination-associated." Risks of vaccination-associated seizures for the different vaccines were analyzed in univariable and in multivariable logistic regression for pertussis combination vaccines and by a self-controlled case series analysis using parental seizure registries for MMR vaccines. Disease courses of children with and without vaccination-associated seizure onset were compared. Results: Children who had DS (n = 77) with and without vaccination-associated seizure onset (21% and 79%, respectively) differed in age at first seizure (median 3.7 vs 6.1 months, p < 0.001) but not in age at first nonvaccination-associated seizure, age at first report of developmental delay, or cognitive outcome. The risk of subsequent vaccination-associated seizures was significantly lower for acellular pertussis (9%; odds ratio 0.18, 95% confidence interval [CI] 0.05-0.71) and nonpertussis (8%; odds ratio 0.11, 95% CI 0.02-0.59) than whole-cell pertussis (37%; reference) vaccines. Self-controlled case series analysis showed an increased incidence rate ratio of seizures of 2.3 (95% CI 1.5-3.4) within the risk period of 5 to 12 days following MMR vaccination. Conclusions: Our results suggest that vaccination-associated earlier seizure onset does not alter disease course in DS, while the risk of subsequent vaccination-associated seizures is probably vaccine-specific.
    Full-text · Article · Aug 2015 · Neurology
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    ABSTRACT: 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. Copyright © 2015 by the American Academy of Pediatrics.
    No preview · Article · Aug 2015 · PEDIATRICS
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    ABSTRACT: The aims of this study were to determine the intra- and interrater reliability of the Dutch-language version of the Communication Function Classification System (CFCS-NL) and to investigate the association between the CFCS level and (1) spoken language comprehension and (2) preferred method of communication in children with cerebral palsy (CP). Participants were 93 children with CP (50 males, 43 females; mean age 7y, SD 2y 6mo, range 2y 9mo-12y 10mo; unilateral spastic [n=22], bilateral spastic [n=51], dyskinetic [n=15], ataxic [n=3], not specified [n=2]; Gross Motor Function Classification System level I [n=16], II [n=14], III, [n=7], IV [n=24], V [n=31], unknown [n=1]), recruited from rehabilitation centres throughout the Netherlands. Because some centres only contributed to part of the study, different numbers of participants are presented for different aspects of the study. Parents and speech and language therapists (SLTs) classified the communication level using the CFCS. Kappa was used to determine the intra- and interrater reliability. Spearman's correlation coefficient was used to determine the association between CFCS level and spoken language comprehension, and Fisher's exact test was used to examine the association between the CFCS level and method of communication. Interrater reliability of the CFCS-NL between parents and SLTs was fair (r=0.54), between SLTs good (r=0.78), and the intrarater (SLT) reliability very good (r=0.85). The association between the CFCS and spoken language comprehension was strong for SLTs (r=0.63) and moderate for parents (r=0.51). There was a statistically significant difference between the CFCS level and the preferred method of communication of the child (p<0.01). Also, CFCS level classification showed a statistically significant difference between parents and SLTs (p<0.01). These data suggest that the CFCS-NL is a valid and reliable clinical tool to classify everyday communication in children with CP. Preferably, professionals should classify the child's CFCS level in collaboration with the parents to acquire the most comprehensive information about the everyday communication of the child in various situations both with familiar and with unfamiliar partners. © 2015 Mac Keith Press.
    Full-text · Article · Jul 2015 · Developmental Medicine & Child Neurology
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    ABSTRACT: Fetal motility represents the spontaneous activity of the central nervous system and as such can be used to evaluate its functional integrity. Maternal mechanical trauma in pregnancy is a risk factor for hypoxic ischemic brain injury and can potentially affect the CNS and fetal motility. To study motility in fetuses after maternal trauma. Prospective study; 1-h sonographic observations at 2-8h (T1), 24-72h (T2) and >72h (T3) after trauma. Fetuses exposed to trauma after 20weeks gestational age. Motor aspects; differentiation into specific movement patterns, quality and quantity of general movements were compared to a normal population. Obstetrical outcome; neurological outcome at term and 1year of age. Sixteen fetuses were examined between 2012 and 2014. Median gestational age at time of trauma was 25+6 (range 20-38) weeks. Most traumas were traffic accidents or falls, injuries were mainly minor. Motility assessment showed abnormal differentiation in 2/16; 2/14 and 0/16; abnormal quality in 2/16; 3/14 and 6/16; and abnormal quantity in 6/16, 9/14 and 9/16 at T1, T2 and T3 respectively. Preterm delivery occurred once. Neurological development was normal in 13/14 infants at term and 14/14 at one year. This study shows that maternal trauma affected fetal motility in the majority of the fetuses. The changes in motility support the concern that even minor mechanical trauma may have influence on the functional integrity of the central nervous system, although no neurological sequelae were present at 1year. Copyright © 2015 Elsevier Ireland Ltd. All rights reserved.
    Full-text · Article · Jun 2015 · Early human development
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    ABSTRACT: To identify relations between brain abnormalities and spoken language comprehension, MRI characteristics of 80 nonspeaking children with severe CP were examined. MRI scans were analysed for patterns of brain abnormalities and scored for specific MRI measures: white matter (WM) areas; size of lateral ventricles, WM abnormality/reduction, cysts, subarachnoid space, corpus callosum thinning and grey matter (GM) areas; cortical GM abnormalities, thalamus, putamen, globus pallidus and nucleus caudatus and cerebellar abnormalities. Language comprehension was assessed with a new validated instrument (C-BiLLT). MRI scans of 35 children were classified as a basal ganglia necrosis (BGN) pattern, with damage to central GM areas; in 60% of these children damage to WM areas was also found. MRI scans of 13 children were classified as periventricular leukomalacia (PVL) with little concomitant damage to central GM areas, 13 as malformations and 19 as miscellaneous. Language comprehension was best in children with BGN, followed by malformations and miscellaneous, and was poorest in PVL. Linear regression modelling per pattern group (malformations excluded), with MRI measures as independent variables, revealed that corpus callosum thinning in BGN and parieto-occipital WM reduction in PVL were the most important explanatory factors for poor language comprehension. No MRI measures explained outcomes in language comprehension in the miscellaneous group. Comprehension of spoken language differs between MRI patterns of severe CP. In children with BGN and PVL differences in language comprehension performance is attributed to damage in the WM areas. Language comprehension was most affected in children with WM lesions in the subcortical and then periventricular areas, most characteristic for children with PVL. Copyright © 2015 European Paediatric Neurology Society. Published by Elsevier Ltd. All rights reserved.
    Full-text · Article · Jun 2015 · European journal of paediatric neurology: EJPN: official journal of the European Paediatric Neurology Society
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    ABSTRACT: Background Children with severe cerebral palsy (CP) (i.e. ‘non-speaking children with severely limited mobility’) are restricted in many domains that are important to the acquisition of language.AimsTo investigate comprehension of spoken language on sentence type level in non-speaking children with severe CP.Methods & ProceduresFrom an original sample of 87 non-speaking children with severe CP, 68 passed the pre-test (i.e. they matched at least five spoken words to the corresponding objects) of a specifically developed computer-based instrument for low motor language testing (C-BiLLT), admitting them to the actual C-BiLLT computer test. As a result, the present study included 68 children with severe CP (35 boys, 33 girls; mean age 6;11 years, SD 3;0 years; age range 1;9–11;11 years) who were investigated with the C-BiLLT for comprehension of different sentence types: phrases, simple active sentences (with one or two arguments) and compound sentences. The C-BiLLT provides norm data of typically developing (TD) children (1;6–6;6 years). Binomial logistic regression analyses were used to compare the percentage correct of each sentence type in children with severe CP with that in TD children (subdivided into age groups) and to compare percentage correct within the CP subtypes.Outcomes & ResultsSentence comprehension in non-speaking children with severe CP followed the developmental trajectory of TD children, but at a much slower rate; nevertheless, they were still developing up to at least age 12 years. Delays in sentence type comprehension increased with sentence complexity and showed a large variability between individual children and between subtypes of CP. Comprehension of simple and syntactically more complex sentences were significantly better in children with dyskinetic CP than in children with spastic CP. Of the children with dyskinetic CP, 10–13% showed comprehension of simple and compound sentences within the percentage correct of TD children, as opposed to none of the children with spastic CP.Conclusion & ImplicationsIn non-speaking children with severe CP sentence comprehension is delayed rather than deviant. Results indicate the importance of following comprehension skills across all age groups, even beyond age 12 years. Moreover, the subtype of CP should be considered when establishing an educational programme for sentence comprehension, and augmentative and alternative communication support. In addition, educational programmes for children with severe CP should take into account the linguistic hierarchy of sentence comprehension when focusing on the input and understanding of spoken language comprehension.
    Full-text · Article · Feb 2015 · International Journal of Language & Communication Disorders
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    ABSTRACT: A cohort of children born with perinatal hypoxic-ischemic encephalopathy (HIE) was followed prospectively until school age. To describe motor outcome and behavioral functioning of school-age children after perinatal HIE and the relationship with neonatal MRI and outcome at age 2. Twenty-five children (19 males), born at term with perinatal HIE, were assessed at a mean age of 7 y 6 m (range 6 y 4 m-8y 2 m). Motor ability was assessed with the Movement Assessment Battery for Children (2nd version) and behavioral functioning was assessed with the Child Behavior Checklist. Neonatal MRI was scored according to Barkovich classification. Of the 25 included children, eight had cerebral palsy (CP). Of the 17 children without CP, nine had impaired motor ability (of which 3 scored definitely abnormal), and four had behavioral problems. There was a significant difference in motor performance (p = 0.008) between children with normal and children with abnormal neonatal MRI. Two (of four) children with normal motor ability and seven (of 14) children with normal neurological examination at age 2 showed impaired motor ability at school age. Half of the children without CP had impaired motor ability at school age. A normal outcome after HIE at young age does not necessarily imply a good outcome at school age, even when neonatal MRI does not show any abnormalities. More research is needed on the behavioral and cognitive consequences of HIE at school age and on the consequences for quality of life for children with and without CP. Copyright © 2015 European Paediatric Neurology Society. Published by Elsevier Ltd. All rights reserved.
    No preview · Article · Feb 2015 · European Journal of Paediatric Neurology
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    R Jeroen Vermeulen · Jules G Becher
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    ABSTRACT: This commentary is on the original article by Tedroff et al. on pages 484-490 of this issue.
    Full-text · Article · Jan 2015 · Developmental Medicine & Child Neurology
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    ABSTRACT: Neonatal convulsions are clinical manifestations in a heterogeneous group of disorders with different etiology and outcome. They are attributed to several genetic causes. We describe a patient with intractable neonatal seizures who died from respiratory compromise during a status epilepticus. This case report provides electroencephalogram (EEG), MRI, genetic analysis, and neuropathological data. Genetic analysis revealed a de novo heterozygous missense mutation in the KCNQ2 gene, which encodes a subunit of a voltage-gated potassium channel. KCNQ2 gene mutation is associated with intractable neonatal seizures. EEG, MRI, data as well as mutation analysis have been described in other KCNQ2 cases. Post-mortem neuropathological investigation revealed mild malformation of cortical development with increased heterotopic neurons in the deep white matter compared to an age-matched control subject. The new finding of this study is the combination of a KCNQ2 mutation and the cortical abnormalities. KCNQ2 mutations should be considered in neonates with refractory epilepsy of unknown cause. The mild cortical malformation is an important new finding, though it remains unknown whether these cortical abnormalities are due to the KCNQ2 mutation or are secondary to the refractory seizures.
    Full-text · Article · Dec 2014 · Frontiers in Pediatrics
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    ABSTRACT: Lymphomatoid granulomatosis (LG) is a B-cell type lymphoproliferative disease. It mainly affects the lungs but may have extrapulmonary manifestations, especially in the central nervous system. The purpose of this study was to review the pediatric cases in the literature and add 2 new cases to the existing literature. A review of the literature was performed on children (0 to 18 years of age at diagnosis) with pathologically proven LG. We found 47 case reports, which, together with 2 new cases, were systematically analyzed. The median age was 12 years. The main symptoms were general, pulmonary, and neurological. Approximately one third of the patients were immunocompromised. High mortality rate was observed. Pediatric LG is a rare disease, which appears to be more frequently seen in immunocompromised patients, especially patients with leukemia. The disease has a high mortality rate; therefore, aggressive therapy according to a high-grade B-cell lymphoma protocol is justified.
    No preview · Article · Oct 2014 · Journal of Pediatric Hematology/Oncology
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    Joke JM Geytenbeek · R Jeroen Vermeulen · Jules G Becher · Kim J Oostrom
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    ABSTRACT: AimTo assess spoken language comprehension in non-speaking children with severe cerebral palsy (CP) and to explore possible associations with motor type and disability.Method Eighty-seven non-speaking children (44 males, 43 females, mean age 6y 8mo, SD 2y 1mo) with spastic (54%) or dyskinetic (46%) CP (Gross Motor Function Classification System [GMFCS] levels IV [39%] and V [61%]) underwent spoken language comprehension assessment with the computer-based instrument for low motor language testing (C-BiLLT), a new and validated diagnostic instrument. A multiple linear regression model was used to investigate which variables explained the variation in C-BiLLT scores. Associations between spoken language comprehension abilities (expressed in z-score or age-equivalent score) and motor type of CP, GMFCS and Manual Ability Classification System (MACS) levels, gestational age, and epilepsy were analysed with Fisher's exact test. A p-value <0.05 was considered statistically significant.ResultsChronological age, motor type, and GMFCS classification explained 33% (R=0.577, R2=0.33) of the variance in spoken language comprehension. Of the children aged younger than 6 years 6 months, 52.4% of the children with dyskinetic CP attained comprehension scores within the average range (z-score ≥−1.6) as opposed to none of the children with spastic CP. Of the children aged older than 6 years 6 months, 32% of the children with dyskinetic CP reached the highest achievable age-equivalent score compared to 4% of the children with spastic CP. No significant difference in disability was found between CP-related variables (MACS levels, gestational age, epilepsy), with the exception of GMFCS which showed a significant difference in children aged younger than 6 years 6 months (p=0.043).InterpretationDespite communication disabilities in children with severe CP, particularly in dyskinetic CP, spoken language comprehension may show no or only moderate delay. These findings emphasize the importance of introducing alternative and/or augmentative communication devices from early childhood.
    Full-text · Article · Oct 2014 · Developmental Medicine & Child Neurology
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    ABSTRACT: Background and Aims The amino acid glutamine has been shown to reduce the number of serious neonatal infections in very preterm children (< 32 weeks of gestation), which may benefit long term brain development. The aim of this study was to elucidate potential effects of glutamine supplementation on brain development in very preterm children at school-age. Methods First, we investigated growth trajectories of head circumference, weight, and length in the first year for 65 very preterm children that originally participated in a randomized controlled trial on enteral glutamine supplementation between day 3 and 30 of life. Second, we measured brain structure volumes and white matter integrity for 52 very preterm children at school-age, using magnetic resonance imaging (MRI) and Diffusion Tensor Imaging (DTI), respectively. Furthermore, differences in functional outcomes were explored. Group differences were tested using ANOVA statistics. Results Glutamine supplementation was associated with improved growth trajectories of head circumference in the first year of life (d=0.66, p=0.03). Furthermore, glutamine supplementation increased white matter (d=0.54, p=0.03), hippocampus (d=0.47, p=0.02), and brain stem (d=0.54, p=0.04) volumes at school-age. All differences were strongly related with the number of serious neonatal infections (all p<0.02). Glutamine supplementation did not influende measures of motor, cognitive, and behavioral functioning at school-age. Conclusions We found evidence that reduction of serious infections by neonatal glutamine supplementation improves head growth in the first year of life, as well as brain structure volumes at school-age. This suggests an early programming effect of nutritional intervention with enteral glutamine.
    Full-text · Article · Jul 2014 · PharmaNutrition
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    ABSTRACT: In clinical practice, a variety of diagnostic tests are available to assess a child's comprehension of spoken language. However, none of these tests have been designed specifically for use with children who have severe motor impairments and who experience severe difficulty when using speech to communicate. This article describes the process of investigating the reliability and validity of the Computer-Based Instrument for Low Motor Language Testing (C-BiLLT), which was specifically developed to assess spoken Dutch language comprehension in children with cerebral palsy and complex communication needs. The study included 806 children with typical development, and 87 nonspeaking children with cerebral palsy and complex communication needs, and was designed to provide information on the psychometric qualities of the C-BiLLT. The potential utility of the C-BiLLT as a measure of spoken Dutch language comprehension abilities for children with cerebral palsy and complex communication needs is discussed.
    Full-text · Article · Jun 2014 · Augmentative and alternative communication (Baltimore, Md.: 1985)
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    ABSTRACT: Background: Attention problems are among the most prominent behavioral deficits reported in very preterm children (below 32 wk of gestation) at school age. In this study, we aimed to elucidate the brain abnormalities underlying attention problems in very preterm children by investigating the role of abnormalities in white and gray brain matter during interference control, using functional magnetic resonance imaging (fMRI)-guided probabilistic diffusion tensor tractography. Methods: Twenty-nine very preterm children (mean (SD) age: 8.6 (0.3) y), and 47 term controls (mean (SD) age: 8.7 (0.5) y), performed a fMRI version of the Eriksen Flanker task measuring interference control. Results: Very preterm children showed slower reaction times than term controls when interfering stimuli were presented, indicating poorer interference control. Very preterm children and term controls did not differ in mean activation of the cortical regions involved in interference control. However, impaired fractional anisotropy (FA) was found in very preterm children in specifically those fiber tracts that innervate the cortical regions involved in interference control. Lower FA was related to poorer interference control in very preterm children. Conclusion: White matter alterations have a crucial role in the interference control problems of very preterm children at school age.
    Full-text · Article · Mar 2014 · Pediatric Research
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    ABSTRACT: Dystonic cerebral palsy is primarily caused by damage to the basal ganglia and central cortex. The daily care of these patients can be difficult due to dystonic movements. Intrathecal baclofen treatment is a potential treatment option for dystonia and has become common practice. Despite this widespread adoption, high quality evidence on the effects of intrathecal baclofen treatment on daily activities is lacking and prospective data are needed to judge the usefulness and indications for dystonic cerebral palsy. The primary aim of this study is to provide level one clinical evidence for the effects of intrathecal baclofen treatment on the level of activities and participation in dystonic cerebral palsy patients. Furthermore, we hope to identify clinical characteristics that will predict a beneficial effect of intrathecal baclofen in an individual patient.Methods/design: A double blind placebo-controlled multi-center randomized clinical trial will be performed in 30 children with dystonic cerebral palsy. Patients aged between 4 and 25 years old with a confirmed diagnosis of dystonic cerebral palsy, Gross Motor Functioning Classification System level IV or V, with lesions in the cerebral white matter, basal ganglia or central cortex and who are eligible for intrathecal baclofen treatment will be included. Group A will receive three months of continuous intrathecal baclofen treatment and group B will receive three months of placebo treatment, both via an implanted pump. After this three month period, all patients will receive intrathecal baclofen treatment, with a follow-up after nine months. The primary outcome measurement will be the effect on activities of and participation in daily life measured by Goal Attainment Scaling. Secondary outcome measurements on the level of body functions include dystonia, spasticity, pain, comfort and sleep-related breathing disorders. Side effects will be monitored and we will study whether patient characteristics influence outcome. The results of this study will provide data for evidence-based use of intrathecal baclofen in dystonic cerebral palsy.Trial registration: Nederlands Trial Register: NTR3642.
    Full-text · Article · Oct 2013 · BMC Pediatrics
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    ABSTRACT: Very preterm children (<32 weeks of gestation) are characterized by impaired white matter development as measured by fractional anisotropy (FA). This study investigates whether altered FA values underpin the widespread motor impairments and higher incidence of developmental coordination disorder (DCD) in very preterm children at school-age. Thirty very preterm born children (mean (SD) age of 8.6 (0.3) years) and 47 term born controls (mean [SD] age 8.7 [0.5] years) participated. Motor development was measured using the Movement Assessment Battery for Children. A score below the 15th percentile was used as a research diagnosis of DCD. FA values, as measure of white matter abnormalities, were determined for 18 major white matter tracts, obtained using probabilistic diffusion tensor tractography. Large-sized reductions in FA of the cingulum hippocampal tract right (d = 0.75, p = .003) and left (d = 0.76, p = .001), corticospinal tract right (d = 0.56, p = .02) and left (d = 0.65, p = .009), forceps major (d = 1.04, p < .001) and minor (d = 0.54, p = .02) were present in very preterms, in particular with a research diagnosis of DCD. Reduced FA values moderately to strongly related to motor impairments. A ROC curve for average FA, as calculated from tracts that significantly discriminated between very preterm children with and without a research diagnosis of DCD, showed an area under curve of 0.87 (95% CI 0.74-1.00, p = .001). This study provides clear evidence that reduced FA values are strongly underpinning motor impairment and DCD in very preterm children at school-age. In addition, outcomes demonstrate that altered white matter FA values can potentially be used to discriminate between very preterm children at risk for motor impairments, although future studies are warranted.
    Full-text · Article · Oct 2013 · European journal of paediatric neurology: EJPN: official journal of the European Paediatric Neurology Society
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    ABSTRACT: Information regarding the selection procedure for selective dorsal rhizotomy (SDR) in children with spastic cerebral palsy (CP) is scarce. Therefore, the aim of this study was to summarize the selection criteria for SDR in children with spastic CP. A systematic review was carried out using the following databases: MEDLINE, CINAHL, EMBASE, PEDro, and the Cochrane Library. Additional studies were identified in the reference lists. Search terms included 'selective dorsal rhizotomy', 'functional posterior rhizotomy', 'selective posterior rhizotomy', and 'cerebral palsy'. Studies were selected if they studied mainly children (<18y of age) with spastic CP, if they had an intervention of SDR, if they had a detailed description of the selection criteria, and if they were in English. The levels of evidence, conduct of studies, and selection criteria for SDR were scored. Fifty-two studies were included. Selection criteria were reported in 16 International Classification of Functioning, Disability and Health model domains including 'body structure and function' (details concerning spasticity [94%], other movement abnormalities [62%], and strength [54%]), 'activity' (gross motor function [27%]), and 'personal and environmental factors' (age [44%], diagnosis [50%], motivation [31%], previous surgery [21%], and follow-up therapy [31%]). Most selection criteria were not based on standardized measurements. Selection criteria for SDR vary considerably. Future studies should describe clearly the selection procedure. International meetings of experts should develop more uniform consensus guidelines, which could form the basis for selecting candidates for SDR.
    Full-text · Article · Sep 2013 · Developmental Medicine & Child Neurology
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    Jessica Warnink-Kavelaars · Roland Jeroen Vermeulen · Jules Guilhelmus Becher
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    ABSTRACT: Intramuscular injection of botulinum toxin type-A given by manual intramuscular needle placement in the lower extremity under general anaesthesia is an established treatment and standard of care in managing spasticity in children with spastic cerebral palsy. Optimal needle placement is essential. However, reports of injection and verification techniques used in previous studies have been partly incomplete and there are methodological shortcomings. This paper describes a detailed protocol for manual intramuscular needle placement checked by passive stretching and relaxing of the target muscle for each individual muscle injection location in the lower extremity during botulinum toxin type-A treatment under general anaesthesia in children with spastic cerebral palsy. It explains the design of a study to verify this protocol, which consists of an injection technique combined with a needle localizing technique, as by means of electrical stimulation to determine its precision. Setting: University Medical Centre, Department of Paediatric Rehabilitation Medicine, the Netherlands. prospective observational study.Participants: children with spastic cerebral palsy, aged 4 to 18 years, receiving regular botulinum toxin type-A treatment under general anaesthesia to improve their mobility, are recruited from the Department of Paediatric Rehabilitation Medicine at VU University Medical Centre, Amsterdam, the Netherlands. a detailed protocol for manual intramuscular needle placement checked by passive stretching and relaxing of the target muscle has been developed for each individual muscle injection location of the adductor brevis muscle, adductor longus muscle, gracilis muscle, semimembranosus muscle, semitendinosus muscle, biceps femoris muscle, rectus femoris muscle, gastrocnemius lateralis muscle, gastrocnemius medialis muscle and soleus muscle. This protocol will be verified as by means of electrical stimulation.Technical details: 25 mm or 50 mm Stimuplex-needle and a Stimuplex-HNS-12 electrical stimulator will be used. Botulinum toxin type-A injected in the intended muscle is expected to yield the greatest effect in terms of activities. Protocols for manual intramuscular needle placement should be described in detail and verified to determine its precision. Detailed and verified protocols are essential to be able to interpret the results of botulinum toxin type-A treatment studies.
    Full-text · Article · Aug 2013 · BMC Pediatrics

Publication Stats

452 Citations
132.60 Total Impact Points

Institutions

  • 2015
    • Maastricht Universitair Medisch Centrum
      Maestricht, Limburg, Netherlands
    • Maastricht University
      Maestricht, Limburg, Netherlands
  • 2008-2015
    • VU University Amsterdam
      Amsterdamo, North Holland, Netherlands
  • 2005-2015
    • VU University Medical Center
      • Rehabilitation Medicine Clinic
      Amsterdamo, North Holland, Netherlands
  • 2010
    • University of Amsterdam
      Amsterdamo, North Holland, Netherlands