Parental Beliefs About the Motor Development of Dutch Infants Born Very Preterm: A Cohort Study.

PURPOSE: To explore the influence of preterm birth on parental beliefs about gross motor development and parents' supportive role in infants' motor development. METHODS: Prospective cohort study: Parents of infants born very preterm (VPT) (gestation ≤32 weeks, birth weight <1500 g, without perinatal complications) and parents of healthy infants born full-term (FT) completed the Parental Beliefs on Motor Development questionnaire. RESULTS: Questionnaires from 37 parents of infants born VPT, aged 3.5 to 7.5 months (corrected), and 110 parents of infants born FT, aged 3.5 months, were analyzed. Parents of infants born VPT believed stimulating motor development to be more important than parents of infants born FT (F = 5.22; P = .024; ηp2 = 0.035). Most parents of infants born VPT (82.4%) and FT (85.2%) acknowledged their role in supporting motor development. More parents of infants born VPT (41.2% vs 12.0%) believed they should follow their infant's natural developmental pace. CONCLUSION: Knowledge of parental beliefs and parents' supporting role in motor development is relevant for tailoring pediatric physiotherapists' interventions with families.

Surgical treatment options for spasticity in children and adolescents with hereditary spastic paraplegia.

PURPOSE: To provide an overview of outcome and complications of selective dorsal rhizotomy (SDR) and intrathecal baclofen pump implantation (ITB) for spasticity treatment in children with hereditary spastic paraplegia (HSP). METHODS: Retrospective study including children with HSP and SDR or ITB. Gross motor function measure (GMFM-66) scores and level of spasticity were assessed. RESULTS: Ten patients were included (most had mutations in ATL1 (n = 4) or SPAST (n = 3) genes). Four walked without and two with walking aids, four were non-walking children. Six patients underwent SDR, three patients ITB, and one both. Mean age at surgery was 8.9 ± 4.5 years with a mean follow-up of 3.4 ± 2.2 years. Five of the SDR patients were walking. Postoperatively spasticity in the legs was reduced in all patients. The change in GMFM-66 score was + 8.0 (0-19.7 min-max). The three ITB patients treated (SPAST (n = 2) and PNPLA6 (n = 1) gene mutation) were children with a progressive disease course. No complications of surgery occurred. CONCLUSIONS: SDR is a feasible treatment option in carefully selected children with HSP, especially in walking patients. The majority of patients benefit with respect to gross motor function, complication risk is low. ITB was used in children with severe and progressive disease.

Physiological Response to the 6-Minute Frame Running Test in Children and Adults With Cerebral Palsy.

PURPOSE: To determine the physiological response and association to peak oxygen uptake of the 6-minute Frame Running test (6-MFRT) in persons with cerebral palsy (CP). METHODS: Twenty-four participants with CP, Gross Motor Function Classification System II/III/IV, performed the 6-MFRT. Distance, peak heart rate (HR peak ), peak respiratory exchange ratio (RER peak ), and peak oxygen uptake ( O 2peak ) were measured. RESULTS: HR peak ranged from 146 to 201 beats per minute, RER peak from 0.94 to 1.49, 6-MFRT distance from 179 to 1220 m and O 2peak from 0.62 to 2.18 L/min. HR peak was achieved in 63%, RER peak in 71%. A strong correlation was observed between 6-MFRT and O 2peak . CONCLUSIONS: The 6-MFRT represented a (near) maximum effort for 75% of the participants and the 6-MFRT can be used to estimate oxygen consumption on an individual basis.

The Impact of Frame Running on Quality of Life in Young Athletes With Mobility Limitations.

Purpose: The para-athletic sport Frame Running is developed for persons with neurological impairments causing severe limitations of walking ability. Participating in sports can contribute to a better quality of life (QoL). It is unknown if participation in Frame Running contributes to QoL in children with mobility limitations. This study aims to explore the changes in QoL in children and youth who started Frame Running. Materials and Methods: We conducted a cross-sectional study amongst young Frame Running athletes with mobility limitations due to various underlying causes, aged 6-19 years, who are members of one of the Frame Running groups in the Netherlands. For 62 athletes, parents completed the Psychosocial Impact of Assistive Devices Scale (PIADS) questionnaire (subscales: competence, adaptability, and self-esteem). For six athletes, parents were interviewed to obtain more in-depth insight in the perceived changes in the QoL of their children. Results: Parents (of 58% boys, mean age 12 years 4 months; SD 3 years 3 months; 52% supported walkers) reported a significant positive change on all three subscales of the PIADS questionnaire since their children started Frame Running. Most change was experienced in the items performance, the ability to participate, happiness and self-confidence. Quotes of the parents who were interviewed elucidated these changes. Conclusion: Frame Running increased QoL in young athletes with a mobility limitation. Frame Running may therefore be advised for these children and youth to improve QoL.

Athlete-Perceived Impact of Frame Running on Physical Fitness, Functional Mobility and Psychosocial Outcomes.

OBJECTIVE: Frame Running (RaceRunning) allows people with moderate-to-severe mobility impairments to participate in physical activity using a 3-wheeled frame with a saddle and handlebars. The aim of this study was to investigate athlete-perceived impact of Frame Running on aspects of physical fitness, functional mobility and psychosocial outcomes. DESIGN: Survey. PARTICIPANTS: Frame Running athletes aged 5 years and over. METHODS: A survey was distributed to athletes through their club or sports organization. RESULTS: The survey was completed by 115 athletes (53 females). Median age was 17 years (range 5-62 years) and 64 (57%) used a wheelchair or walker for distances over 50 m. Many felt that Frame Running stretched their muscles (n = 93, 87%) and increased their self-confidence (n = 63, 93%). Four (4%) reported extreme fatigue or sore muscles after training (n = 17, 15%). Of the 110 athletes who had been participating in Frame Running for over 3 months, 46 (47%) reported being less out of breath during mobility tasks and 66 (66%) felt they had improved their functional mobility. However, 7 (7%) reported increased muscle tightness and 4 (4%) reported a Frame Running-related injury lasting more than 4 weeks. CONCLUSION: Frame Running is a safe physical activity with athlete-perceived benefits on physical fitness, functional mobility and psychosocial outcomes.

Neurodevelopment of children born very preterm and/or with a very low birth weight: 8-Year follow-up of a nutritional RCT.

BACKGROUND: Children born very preterm are at risk for cognitive deficits and motor impairment. Enhanced protein intake immediately after very preterm birth has been associated with favorable growth and improved neurodevelopment. It is unknown whether increased protein intake after discharge from the hospital affects long-term neurodevelopment. OBJECTIVE: The primary objective was to assess neurodevelopment from infancy to 8 years in preterm-born children who received either protein-enriched formula (PDF), standard term formula (TF), or human milk (HM) after discharge. The secondary objective was to assess the correlation between outcomes obtained at 24 months corrected age (CA) and at 8 years. METHODS: This RCT included 152 children born very preterm (gestational age ≤32 weeks) and/or with a very low birth weight (≤1500 g) of whom 102 were randomly assigned to receive PDF (n = 54) or TF (n = 48) from term age to 6 months CA. A control group of infants fed HM (n = 50) was also included. Neurodevelopmental outcomes were assessed at 24 months CA (cognitive and motor functioning; n = 123) and at 8 years (estimated Full Scale Intelligence Quotient, visual-motor skills, verbal memory, attention, and motor functioning; n = 76). RESULTS: The PDF and TF groups were not significantly different in neurodevelopmental outcomes. The HM group had a better cognitive score compared with the PDF group: at 24 months CA 92.9 ± 12.5 vs. 105.2 ± 18.6, P < 0.001 and at 8 years 98.1 ± 11.3 vs. 105.8 ± 9.1, P = 0.017 (P = 0.002 and P = 0.080, respectively, after adjustment for parental educational level). Correlations between outcomes at 24 months CA and 8 years were weak: r = 0.35 and r = 0.37 for cognitive and motor outcomes, respectively. CONCLUSIONS: PDF did not improve long-term neurodevelopmental outcomes as compared with TF. However, these results should be interpreted with caution considering the substantial attrition at follow-up. Furthermore, the correlation between outcomes at different ages was weak, emphasizing the need for long-term follow-up of nutritional intervention studies in preterm-born children.

Factors Associated With Long-Term Improvement of Gait After Selective Dorsal Rhizotomy.

OBJECTIVE: To identify factors associated with long-term improvement in gait in children after selective dorsal rhizotomy (SDR). DESIGN: Retrospective cohort study. SETTING: University medical center. PARTICIPANTS: Children (N=36) (age 4-13y) with spastic diplegia of Gross Motor Function Classification System (GMFCS) level I (n=14), II (n=15), and III (n=7) were included retrospectively from the database of our hospital. Children underwent SDR between January 1999 and May 2011. Patients were included if they received clinical gait analysis before and 5 years post-SDR, age >4 years at time of SDR and if brain magnetic resonance imaging (MRI) scan was available. INTERVENTION: Selective dorsal rhizotomy. MAIN OUTCOME MEASURES: Overall gait quality was assessed with Edinburgh visual gait score (EVGS), before and 5 years after SDR. In addition, knee and ankle angles at initial contact and midstance were evaluated. To identify predictors for gait improvement, several factors were evaluated including functional mobility level GMFCS, presence of white matter abnormalities on brain MRI, and selective motor control during gait (synergy analysis). RESULTS: Overall gait quality improved after SDR, with a large variation between patients. Multiple linear regression analysis revealed that worse score on EVGS and better GMFCS were independently related to gait improvement. Gait improved more in children with GMFCS I and II compared to III. No differences were observed between children with or without white matter abnormalities on brain MRI. Selective motor control during gait was predictive for improvement of knee angle at initial contact and midstance, but not for EVGS. CONCLUSION: Functional mobility level and baseline gait quality are both important factors to predict gait outcomes after SDR. If candidates are well selected, SDR can be a successful intervention to improve gait both in children with brain MRI abnormalities as well as other causes of spastic diplegia.

Early-life growth of preterm infants and its impact on neurodevelopment.

BACKGROUND: Increasing numbers of preterm-born children survive nowadays, and improving long-term health and neurodevelopment is becoming more important. Early-life growth has been linked to neurodevelopmental outcomes. We aimed to study whether this association has changed with time. METHODS: We studied two cohorts of preterm-born children (gestational age ≤32 weeks and/or birth weight ≤1500 g) from 1983 (n = 708) and 2003-2006 (n = 138), respectively. We distinguished four early-life growth patterns at 3 months corrected age: appropriate for gestational age (AGA) with or without growth restriction (AGA GR+/AGA GR-), and small for gestational age (SGA) with or without catch-up growth (SGA CUG+/SGA CUG-). Intelligence quotient (IQ), neuromotor function, and behavior were assessed at ages 19 and 8 years, respectively, for the cohorts. RESULTS: In the 2003-2006 cohort, less children had early-life GR. In both cohorts, SGA CUG- subjects had unfavorable growth trajectories and neurodevelopmental outcomes (IQ ß -6.5, 95% confidence interval (CI) -9.8; -3.2, P < 0.001; neuromotor score ß -1.9%, 95% CI -3.2; -0.6, P = 0.005), while SGA CUG+ subjects were comparable to adequately grown subjects. CONCLUSION: Although the incidence of adverse growth patterns decreased between the cohorts, possibly indicating improvements in care over time, the impact of these growth patterns on neurodevelopmental outcomes was not significantly different. Achieving adequate early-life growth may be crucial for improving neurodevelopmental outcomes, especially for preterms born SGA.

Evolution of gait in adolescents and young adults with spastic diplegia after selective dorsal rhizotomy in childhood: A 10 year follow-up study.

BACKGROUND: Short-term benefit on gait of selective dorsal rhizotomy (SDR) surgery, which relieves spasticity of the lower extremities has been demonstrated in children with cerebral palsy (CP). However very little is known of the evolution of gait when patients become adolescents and young adults. RESEARCH QUESTION: How does the gait pattern evolve in adolescents and young adults who underwent SDR during childhood? METHODS: A longitudinal study was performed including 19 ambulant patients with spastic diplegia due to CP or other causes (mean age at SDR: 6.6 ±â€¯1.6 years) who were assessed four times: pre-SDR, 2 years post- SDR, 5 years post-SDR and at least 10 years post-SDR. From 2D video recordings, Edinburgh Visual Gait Score and lower limb joint kinematic parameters were calculated. RESULTS: Our data show that the improvement in the gait pattern obtained short-term after SDR continues during into adolescence and adulthood. Ten years after SDR all patients improved compared to baseline. Considering the lower limb joint kinematics, most notable improvements were found at knee and ankle joints. Compared to the evaluation before SDR, the range of motion of the knee increased: the knee was more extended at initial contact and knee flexion in midswing improved. Excessive ankle plantar flexion was reduced during the entire gait cycle. Only minor changes were found at hip and pelvis. Eight patients underwent additional orthopaedic surgery in the years after SDR, and the present findings should be considered as a combination of SDR, development and additional treatment. SIGNIFICANCE: We demonstrate lasting improvement of gait quality in ambulant patients with spastic diplegia who underwent SDR during childhood when they become adolescents and young adults.

Risk Factors for Dystonia after Selective Dorsal Rhizotomy in Nonwalking Children and Adolescents with Bilateral Spasticity.

We recently showed a beneficial effect of selective dorsal rhizotomy (SDR) on daily care and comfort in nonwalking children with severe bilateral spasticity. However, despite careful selection, some patients showed dystonia after the intervention, in which cases caregivers tended to be less satisfied with the result.The aim of this study is to identify risk factors for dystonia after SDR in children and adolescents with severe bilateral spasticity (GMFCS levels IV/V).Clinical and MRI risk factors for dystonia after SDR were studied in our cohort of 24 patients. Patients with clinical evidence of dystonia and brain MRI showing basal ganglia abnormalities were excluded for SDR.Nine of 24 patients (38%) showed some degree of dystonia after SDR. There was a significant association between the cause of spasticity and dystonia after SDR; in six (67%) patients with a congenital disorder, dystonia was present versus three (20%) with an acquired disorder (Chi-squared test: C(1) = 5.23, p = 0.02).This study allows more optimal selection of patients that may benefit from SDR. Patients with an acquired cause of spasticity, when selected carefully on clinical examination and MRI, rarely show dystonia after SDR. However, patients with an underlying congenital disorder have a considerable risk of dystonia after SDR.

Sudden falls as a persistent complication of selective dorsal rhizotomy surgery in children with bilateral spasticity: report of 3 cases.

Selective dorsal rhizotomy (SDR) surgery is a well-established treatment for ambulatory children with bilateral spastic paresis and is performed to eliminate spasticity and improve walking. The objective of this case report is to describe sudden falls as a persistent complication of SDR. The authors report on 3 patients with bilateral spastic paresis, aged 12, 6, and 7 years at the time of surgery. The percentage of transected dorsal rootlets was around 40% at the L2-S1 levels. Sudden falls were reported with a frequency of several a day, continuing for years after SDR. The falls were often triggered by performing dual tasks as well as occurring in the transition from sitting to standing, during running, after strenuous exercise, or following a fright. Patients also had residual hyperesthesia and dysesthesia of the foot sole. The authors hypothesize that the sudden falls are caused by a muscle inhibition reflex of the muscles in the legs, as an abnormal reaction to a sensory stimulus that is perceived with increased intensity by a patient with hyperesthesia. A favorable effect of gabapentin medication supports this hypothesis.

Effect of (minor or major) maternal trauma on fetal motility: A prospective study.

BACKGROUND: 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. AIM: To study motility in fetuses after maternal trauma. STUDY DESIGN: Prospective study; 1-h sonographic observations at 2-8h (T1), 24-72h (T2) and >72h (T3) after trauma. SUBJECTS: Fetuses exposed to trauma after 20weeks gestational age. OUTCOME MEASURES: 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. RESULTS: 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. CONCLUSIONS: 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.

Long-term motor and behavioral outcome after perinatal hypoxic-ischemic encephalopathy.

BACKGROUND: A cohort of children born with perinatal hypoxic-ischemic encephalopathy (HIE) was followed prospectively until school age. AIMS: 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. METHODS: 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-8 y 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. RESULTS: 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. CONCLUSIONS: 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.

Developmental trajectories of receptive and expressive communication in children and young adults with cerebral palsy.

AIM: The aim of this study was to determine the developmental trajectories of expressive (speech) and receptive (spoken and written language) communication by type of motor disorder and intellectual disability in individuals with cerebral palsy (CP). METHOD: The development of 418 participants (261 males, 157 females; mean age 9y 6mo [SD 6y 2mo], range 1-24y; Gross Motor Function Classification System (GMFCS) level I [n=206], II [n=57], III [n=59], IV [n=54], V [n=42]) was followed for 2 to 4 years in a longitudinal study. Communication performance was measured using the Vineland Adaptive Behavior Scales. The type of motor disorder was differentiated by type of CP as unilateral spastic (USCP, n=161), bilateral spastic (BSCP, n=202), and non-spastic (NSCP, n=55), while intellectual disability was determined by IQ or school type (regular or special). A multilevel analysis was then used to model the developmental trajectories. RESULTS: The most favourable development of expressive communication was seen in USCP (vs BSCP ß [SE]-2.74 [1.06], NSCP ß [SE]-2.67 [1.44]). The difference between the development trajectory levels of children with and without intellectual disability was smaller for children with USCP than for those with BSCP and NSCP. For receptive communication, the most favourable development was found for all children with USCP and for BSCP or NSCP without intellectual disability (vs intellectual disability ß [SE]-4.00 [1.16]). Development of written language was most favourable for children without intellectual disability (vs intellectual disability ß [SE]-23.11 [2.85]). INTERPRETATION: The development of expressive communication was found to be most closely related to type of motor disorder, whereas the development of receptive communication was found to be most closely related to intellectual disability.

Neurodevelopmental outcome during the first year of life in preterm infants after supplementation of a prebiotic mixture in the neonatal period: a follow-up study.

AIM: The study aimed to determine the effect of enteral supplementation of a prebiotic mixture (neutral and acidic oligosaccharides) in the neonatal period on neurodevelopmental outcome in preterm infants with a gestational age of less than 32 weeks and/or birth weight of less than 1,500 g in the first year of life. METHODS: Neurodevelopmental outcome consisted of tone and motility according to Touwen, motor ability according to the Alberta Infant Motor Scale, as well as vision and hearing were evaluated at 0, 3, 6, and 12 months corrected age (CA). To adjust for potential confounders, data were analyzed by multinomial or logistic regression. RESULTS: In total, 93 of 101 infants (92%) participated in the study (prebiotic mixture group [n = 48] and placebo group [n = 45]). Incidence of suspect and abnormal neurodevelopmental outcome and delayed motor ability was not different in the prebiotic mixture and placebo group at 0, 3, 6, and 12 months (CA). Infants with 1 or more neonatal infections or more had a higher incidence of abnormal neurodevelopmental outcome (OR: 20.73; 95% CI: 1.31-328.46; p = 0.03) at 6 months CA. CONCLUSION: Short-term enteral supplementation of a prebiotic mixture in the neonatal period had no effect on neurodevelopmental outcome in preterm infants in the first year of life. However, serious neonatal infections in preterm infants were associated with adverse neurodevelopmental outcome at 6 months, but not at 12 months.

Development of social functioning and communication in school-aged (5-9 years) children with cerebral palsy.

The aim of this study was to examine determinants of the course and level of social functioning and communication in school-aged children with cerebral palsy (CP) over a 2-year period. A clinic-based sample of 5 and 7 years old children with CP (n=108; 72 males; mean age 6 y 3 mo, SD 12 mo; Gross Motor Function Classification System (GFMCS) level I-V) was followed longitudinally in three yearly assessments. Social functioning and communication were measured with the Vineland Adaptive Behavior Scales (VABS). Data were analyzed with generalized estimated equations. The results showed that social function followed a course of progressive restrictions over time in non-ambulatory children with CP aged 5-9 compared to children who could walk with or without walking aids. Overall lower levels of social functioning were found in children with GMFCS V, epilepsy, speech problems, lower intellectual capacity and older age at baseline. For communication more restrictions over time were found in children with lower intellectual capacity. Children with GMFCS V, speech problems and older age at baseline had overall greater restrictions in communication. It was concluded that motor functioning and intellectual ability can be used to identify children at risk for progressive restrictions in social functioning and communication. For children with CP and social and communicative restrictions, multidisciplinary assessment and treatment may be indicated to counteract an unfavorable development.

Long-term effect of selective dorsal rhizotomy on gross motor function in ambulant children with spastic bilateral cerebral palsy, compared with reference centiles.

AIM: The aim of this study was to evaluate the long-term effect of selective dorsal rhizotomy (SDR) on the gross motor function of ambulant children with spastic bilateral cerebral palsy (CP), compared with reference centiles. METHOD: The study used a prospective cohort design and participants comprised 29 children classified using the Gross Motor Function Classification System (GMFCS) in level I (n=7), II (n=4), or III (n=18; 18 males, 11 females; median age at time of surgery 6 y 4 mo; range 2 y 10 mo-12 y 1 mo), who were examined 5 years and 10 years after SDR. We used individual centiles based on Gross Motor Function Measure (GMFM-66) scores and age, corresponding to the GMFCS levels. Individual improvement or deterioration was defined as a change of more than 20 centiles. Side effects experienced and additional treatment received after SDR were also recorded. RESULTS: Five years after SDR, 10 out of 28 children showed improvement, and 10 years after SDR 6 out of 20 children had improved. Spinal side effects were noted in two children and hip subluxation in three. Additional treatments included subtalar arthrodesis (n=13), endorotational osteotomy of the tibia (n=5), and botulinum toxin treatment (n=13). INTERPRETATION: None of the children showed deterioration of gross motor function based on centile ranking. Five and 10 years after SDR, gross motor function in some children had improved more than would have been expected according to the reference centiles. This suggests, taking the limitations of this study into account, that the applied criteria for selection were adequate. However, the children still required additional treatment after SDR.

Intrathecal baclofen for progressive neurological disease in childhood: a systematic review of literature.

BACKGROUND: Intrathecal baclofen (ITB) treatment is frequently used for individuals with severe, but non-progressive, spasticity refractory to oral treatment. However, experiences with ITB in patients with progressive neurological disorders of childhood causing spasticity are limited. AIM: To investigate whether ITB is an option in patients with progressive neurological disorders causing spasticity in childhood. DESIGN: A systematic literature search in Embase, Pubmed and the Cochrane Library was performed. RESULTS: We identified six eligible studies considering patients with progressive neurological disease in childhood and receiving ITB treatment. The studies included a total of seven paediatric patients and four adult patients. Improvement was reported in spasticity, spasms, pain, gait, activities of daily life and providing care. High satisfaction is described. CONCLUSIONS: ITB has beneficial effects in paediatric patients with progressive neurological disease. However, the level of evidence is limited due to the small number of available studies and due to the poor quality of these studies.

Effects of intrathecal baclofen on daily care in children with secondary generalized dystonia: a pilot study.

AIM: Treatment options for dystonic cerebral palsy (CP) are limited. Our aims were to determine whether intrathecal baclofen (ITB) improves daily care, decreases dystonia and decreases pain in patients with dystonic CP. METHODS: Patients received randomized blinded treatment with ITB or placebo. Scores on problems of daily care were recorded and dystonia, pain and comfort were assessed. RESULTS: Four patients (three males, average age 12 years 6 months) were included (all Gross Motor Function Classification System level V). During the trial period problem scores and dystonia scores decreased in all four patients. CONCLUSION: In this pilot study we report positive functional effects of ITB trial treatment in four patients with dystonic CP. A randomized trial with a larger cohort is needed to verify these results.