The development and validation of the S-scan-parental self-management support (S-scan - PS): A self-reflection tool for child healthcare professionals.

BACKGROUND: Professionals providing self-management support to parents regarding the care for their child with a chronic condition nowadays is an important aspect of child healthcare. This requires professionals to orient themselves towards partnership and collaboration with parents. The aims of the current study were the development and validation of the S-Scan-Parental self-management Support (S-scan - PS) as a tool for healthcare professionals to reflect on their attitude and practices regarding the support for parental self-management. METHODS: An existing instrument was adapted together with field experts for professionals to self-evaluate their support for self-management of parents. The resulting 36-item self-report questionnaire was filled in by healthcare professionals in the Netherlands working with children and their parents. Cognitive interviews, exploratory and confirmatory factor analysis (CFA), and test-retest reliability analysis were part of the development and validation process. RESULTS: In total, 434 professionals, including physicians, physiotherapists, occupational therapists, and nurses, from 13 rehabilitation institutes and 5 medical centres participated. The cognitive interviews with child healthcare professionals indicated adequate face and content validity. The S-scan - PS scale had acceptable internal consistency (0.71 ≤ α ≤ 0.91) for the total score as well as the domain scores. CFA showed acceptable root mean square error of approximation (RMSEA) model fit (0.066), though not on other tested goodness-of-fit indices. Test-retest reliability of the instrument was moderate with an average intraclass correlation coefficient (ICC) = 0.61. CONCLUSIONS: The S-scan - PS fulfils important psychometric criteria for use by child healthcare professionals to reflect on parental self-management support. Such self-reflection might help to improve their approach towards supporting self-management of parents in the care for their child with a chronic condition. Further research is needed into the construct validity and test-retest reliability of the instrument.

Attainment of personal goals in the first year of intrathecal baclofen treatment in dyskinetic cerebral palsy: a prospective cohort study.

PURPOSE: To assess attainment of individual treatment goals one year after intrathecal baclofen (ITB) pump implantation in individuals with dyskinetic cerebral palsy (CP). MATERIALS AND METHODS: A multi-center prospective cohort study was conducted including 34 non-walking individuals with severe dyskinetic CP, classified as Gross Motor Function Classification System (GMFCS) IV/V, aged 4-24 years, 12 months after pump implantation. The main outcome measure was Goal Attainment Scaling (GAS). Predictors of GAS results were analyzed. Complications were registered systematically. RESULTS: Seventy-one percent of individuals with dyskinetic CP fully achieved one or more treatment goals. One or more treatment goals were partially achieved in 97% of individuals. Two factors were found to be associated with attainment of goals: Dyskinesia Impairment Scale (DIS) score at baseline and the difference in pain score between baseline and follow-up. These two variables explain 30% of the variance in the outcome. CONCLUSIONS: Intrathecal baclofen is effective in achieving individual treatment goals in children and young adults with dyskinetic CP after nine to 12 months of ITB treatment. A positive outcome on treatment goals is, for a small part, associated with higher severity of dystonia at baseline and with improvement of pain during treatment. CLINICAL TRIAL REGISTRATION NUMBER: Dutch Trial Register, number NTR3642.Implications for rehabilitationIntrathecal baclofen treatment is effective in attainment of personal treatment goals, one year after pump implantation in patients with dyskinetic cerebral palsy.A positive outcome on treatment goals is, for a small part, related to higher severity of dystonia at the start and on improvement of pain during treatment.

Professionals' motivation to support parental self-management regarding children with physical disability in Dutch rehabilitation services: 'Please mind your gap'.

BACKGROUND: Professionals in child healthcare increasingly endorse the support of self-management in paediatric rehabilitation services for children with physical disability. Less understood though are their views regarding the role of the children's parents, as well as their own role in supporting parents. This study aimed to investigate the motivation of rehabilitation professionals to support self-management of parents regarding their child with physical disability, professionals' beliefs about parental self-management, and the perceptions underlying their motivation. METHODS: A mixed-methods strategy was followed using a survey among rehabilitation professionals (n = 175) and consecutive semi-structured interviews (n = 16). Associations between autonomous (intrinsic) versus controlled (extrinsic) motivation and beliefs on parental self-management were tested. For deeper understanding of their motivation, directed content analysis was used to address key themes in the qualitative data extracts. RESULTS: Professionals reported autonomous motivation for parental self-management support more often than controlled motivation (t[174] = 29.95, p < .001). Autonomous motivation was associated with the beliefs about the importance of parental self-management (r = .29, p < .001). Approximately 90% of the professionals believed that parents should have an active role, though less than 10% considered it important that parents also are independent actors and initiative takers in the rehabilitation process. Interviews revealed that individual professionals struggled with striking a balance between keeping control and 'giving away responsibility' to parents. A 'professional-like' attitude was expected of parents with 'involvement' and 'commitment' as essential preconditions. Furthermore, professionals expressed the need for additional coaching skills to support parental self-management. CONCLUSION: Professionals were predominantly autonomously motivated to support self-management of parents. However, the dilemmas in giving or taking responsibilities within the partnership with parents may limit their effectiveness in empowering parents. Reflection on the potential gaps between professionals' motivation, beliefs and actual behaviour might be crucial to support parental self-management.

Improvements in Muscle Strength Are Associated With Improvements in Walking Capacity in Young Children With Cerebral Palsy: A Secondary Analysis.

PURPOSE: To evaluate whether changes in lower-limb muscle strength explain changes in walking capacity during 14-week periods of usual care, power training and follow-up for children with spastic cerebral palsy. METHODS: Secondary analysis of a previously conducted double-baseline controlled trial of 22 children with spastic cerebral palsy. Generalized estimating equations were used to evaluate the relationships between within-subject changes in isometric muscle strength and walking capacity over 3 periods. RESULTS: Changes in hip abductor strength were associated with changes in the Muscle Power Sprint Test, changes in gastrocnemius and hip abductor strength were associated with changes in the Shuttle Run Test, and changes in gastrocnemius strength were associated with changes in the 1-minute walk test. All associations supported better walking capacity with increased strength. CONCLUSION: Walking capacity, especially sprint capacity, can be improved by increasing strength by functional power training in this population.

Gastrocnemius Medialis Muscle Geometry and Extensibility in Typically Developing Children and Children With Spastic Paresis Aged 6-13 Years.

Gait of children with spastic paresis (SP) is frequently characterized by a reduced ankle range of motion, presumably due to reduced extensibility of the triceps surae (TS) muscle. Little is known about how morphological muscle characteristics in SP children are affected. The aim of this study was to compare gastrocnemius medialis (GM) muscle geometry and extensibility in children with SP with those of typically developing (TD) children and assess how GM morphology is related to its extensibility. Thirteen children with SP, of which 10 with a diagnosis of spastic cerebral palsy and three with SP of unknown etiology (mean age 9.7 ± 2.1 years; GMFCS: I-III), and 14 TD children (mean age 9.3 ± 1.7 years) took part in this study. GM geometry was assessed using 3D ultrasound imaging at 0 and 4 Nm externally imposed dorsal flexion ankle moments. GM extensibility was defined as its absolute length change between the externally applied 0 and 4 Nm moments. Anthropometric variables and GM extensibility did not differ between the SP and TD groups. While in both groups, GM muscle volume correlated with body mass, the slope of the regression line in TD was substantially higher than that in SP (TD = 3.3 ml/kg; SP = 1.3 ml/kg, p < 0.01). In TD, GM fascicle length increased with age, lower leg length and body mass, whereas in SP children, fascicle length did not correlate with any of these variables. However, the increase in GM physiological cross-sectional area as a function of body mass did not differ between SP and TD children. Increases in lengths of tendinous structures in children with SP exceeded those observed in TD children (TD = 0.85 cm/cm; SP = 1.16 cm/cm, p < 0.01) and even exceeded lower-leg length increases. In addition, only for children with SP, body mass (r = -0.61), height (r = -0.66), muscle volume (r = - 0.66), physiological cross-sectional area (r = - 0.59), and tendon length (r = -0.68) showed a negative association with GM extensibility. Such negative associations were not found for TD children. In conclusion, physiological cross-sectional area and length of the tendinous structures are positively associated with age and negatively associated with extensibility in children with SP.

An Interdisciplinary Model of Treatment of Children with Cerebral Palsy in Poland. Recommendations of the Paediatric Rehabilitation Section of the Polish Rehabilitation Society.

Medical rehabilitation plays an important leading role in the treatment of children with cerebral palsy (CP). Collaboration between specialists in medical rehabilitation and the rehabilitation team is a prerequisite for good medical care. The quality of medical services for children with CP depends chiefly on the level of expertise of the treatment team. Through training of specialists and sharing of knowledge, we can help more patients. This idea was developed and implemented by Dr Ewa Kooyman-Piskorz, the founder and president of Wandafonds Foundation. Between 2003 and 2014, Dutch specialists working with children with CP conducted a number of training workshops in Poland under the supervision of the Polish Rehabilitation Society and Prof. Jules Becher, a world-famous expert in the rehabilitation of children. Based on these experiences, we present the recommendations of the Paediatric Rehabilitation Section of the Polish Rehabilitation Society regarding an interdisciplinary model of treatment of children with CP in Poland.

Managing oneself or managing together? Parents' perspectives on chronic condition self-management in Dutch pediatric rehabilitation services.

Purpose: With the growing attention in pediatric rehabilitation services for supporting self-management, the need increases for more shared understanding of the concept. The aim of this study was to explore parent activation, associated factors of- and underlying perceptions on parental self-management of parents of children with chronic conditions.Materials and methods: Using a mixed-methods strategy, first variations in self-management behaviors, motivation and perceived autonomy support were assessed with a cross-sectional survey among parents of children with chronic conditions (N = 239). Statistical analysis involved descriptive statistics and univariate analysis of variance. The survey was followed by 18 in-depth interviews with parents. Thematic analysis was used to recognize relevant topics in the qualitative data.Results: In the survey most parents reported being active self-managers. Nevertheless, only one third persisted in self-management when under stress. Autonomous motivation was strongly associated with parental self-management. In the interviews, parents mentioned attuning with professionals and finding balance as important aspects of self-management. To facilitate self-management, professionals were expected to have expert knowledge, be engaged and empathic.Conclusion: From the perspective of parents, self-management should be viewed as a collaborative effort in which they are supported by professionals, rather than having to manage it "by themselves".Implications for rehabilitationTo facilitate self-management, parents expect professionals to have expert knowledge and additionally show interpersonal competences as openness, engagement and empathy.Motivating parents may facilitate their level of self-management regarding the care for their child with a chronic disorder.Good communication and collaboration with professionals appear to be key aspects of parental self-management.Parents expect pediatric rehabilitation teams to tune their services to the needs, desires and expectations of parents to support them in "self-managing" the care for their child.

Foot flexibility confounds the assessment of triceps surae extensibility in children with spastic paresis during typical physical examinations.

Accurate assessment of the talo-crural (ankle) joint angle at physical examination is important for assessing extensibility of m. triceps surae (TS) in children with spastic cerebral paresis (SCP). The main aim of this study was to quantify foot flexibility during standardized measurements of TS muscle-tendon complex extensibility (i.e. based on foot-sole rotation) in SCP children, and typical developed (TD) ones. Additionally, we aim to define a method that minimizes the confounding effects of foot flexibility on estimates of talo-crural joint angles and TS extensibility. Children, aged 6-13 years, with SCP (GMFCS I-III, n = 13) and TD children (n = 14) participated in this study. Externally applied -1 Nm, 0 Nm, 1 Nm and 4 Nm dorsal flexion foot plate moments were imposed. Resulting TS origin-insertion lengths, foot sole (φFoSo) rotations, and changes in talo-crural joint angle (φTaCr) were measured. Foot flexibility was quantified as Δ(φTaCr -φFoSo) between the 0 Nm and 4 Nm dorsal flexion conditions. In both groups, φFoSo rotations of approximately 20° were observed between 0 Nm and 4 Nm dorsal flexion, of which about 6° (≈30%) was related to foot flexibility. Foot flexibility correlated to φFoSo (r = 0.69) but not to φTaCr (r = 0.11). For φFoSo no significant differences were found between groups at 4 Nm. However, for SCP children the mean estimate of φTaCr was 4.3° more towards plantar flexion compared to the TD group (p < 0.05). Normalized TS lengths show a higher coefficient of correlation with φTaCr (r2 = 0.82) than with φFoSo (r2 = 0.60), indicating that TS lengths are better estimated by talo-cural joint angles. In both SCP and TD children aged 6-13 year, estimates of TS length and extensibility based on foot sole assessments are confounded by foot flexibility. Assessments of TS extensibility at physical examination will be more accurate when based on measurements of talo-crural joint angles.

Use of the Dyskinesia Impairment Scale in non-ambulatory dyskinetic cerebral palsy.

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.

The Effect of Intrathecal Baclofen in Dyskinetic Cerebral Palsy: The IDYS Trial.

OBJECTIVE: Intrathecal baclofen treatment is used for the treatment of dystonia in patients with severe dyskinetic cerebral palsy; however, the current level of evidence for the effect is low. The primary aim of this study was to provide evidence for the effect of intrathecal baclofen treatment on individual goals in patients with severe dyskinetic cerebral palsy. METHODS: This multicenter, randomized, double-blind, placebo-controlled trial was performed at 2 university medical centers in the Netherlands. Patients with severe dyskinetic cerebral palsy (Gross Motor Functioning Classification System level IV-V) aged 4 to 24 years who were eligible for intrathecal baclofen were included. Patients were assigned by block randomization (2:2) for treatment with intrathecal baclofen or placebo for 3 months via an implanted microinfusion pump. The primary outcome was goal attainment scaling of individual treatment goals (GAS T score). A linear regression model was used for statistical analysis with study site as a covariate. Safety analyses were done for number and type of (serious) adverse events. RESULTS: Thirty-six patients were recruited from January 1, 2013, to March 31, 2018. Data for final analysis were available for 17 patients in the intrathecal baclofen group and 16 in the placebo group. Mean (standard deviation) GAS T score at 3 months was 38.9 (13.2) for intrathecal baclofen and 21.0 (4.6) for placebo (regression coefficient = 17.8, 95% confidence interval = 10.4-25.0, p < 0.001). Number and types of (serious) adverse events were similar between groups. INTERPRETATION: Intrathecal baclofen treatment is superior to placebo in achieving treatment goals in patients with severe dyskinetic cerebral palsy. ANN NEUROL 2019.

Intrathecal baclofen in metachromatic leukodystrophy.

Metachromatic leukodystrophy (MLD) is a rare progressive neurological disorder, often accompanied by motor impairments that are challenging to treat. In this case series, we report the course of treatment with intrathecal baclofen (ITB), aimed at improving daily care and comfort in children and young adults with MLD. All patients with MLD in our centre on ITB treatment for a minimum of 6 months were included (n=10; 4 males, 6 females; mean age 10y 8mo [range 6-24y]). Eight patients had MLD with a predominant spastic movement disorder (sMLD) and two were mainly dyskinetic. Patients with sMLD were compared with matched patients with spastic cerebral palsy (CP). Complication rates related to ITB treatment were similar in both groups. ITB treatment course in the first 6 months after pump implantation appears to show more dose increase in most patients MLD, compared to patients with spastic CP. This may be due to the progressive disease in MLD. ITB is a feasible therapy to improve daily care and comfort in patients with MLD and should therefore be considered early. WHAT THIS PAPER ADDS: Intrathecal baclofen (ITB) is a feasible therapy to improve comfort and daily care in children and young people with metachromatic leukodystrophy (MLD). In the first 6 months of ITB treatment, MLD seems to show more dose increase compared to spastic cerebral palsy.

Effect of continuous intrathecal baclofen therapy in children: a systematic review.

AIM: To investigate the effects of continuous intrathecal baclofen (ITB) therapy in children with cerebral palsy (CP) and other neurological conditions. METHOD: This systematic review was conducted using standardized methodology, searching four electronic databases (PubMed, Embase, CINAHL, Cochrane Library) for relevant literature published between inception and September 2017. Included studies involved continuous ITB as an intervention and outcome measures relating to all International Classification of Functioning, Disability and Health: Children and Youth (ICF-CY) components. RESULTS: Thirty-three studies were identified, of which one, including 17 children with spastic CP, produced level II evidence, and the others, mainly non-controlled cohort studies, level IV and V. Outcomes at body function level were most frequently reported. Results suggest continuous ITB may be effective in reducing spasticity and dystonia in CP, as well as other neurological conditions, and may improve the ease of care and quality of life of children with CP, but the level of evidence is low. INTERPRETATION: Despite three decades of applying ITB in children and a relatively large number of studies investigating the treatment effects, a direct link has not yet been demonstrated because of the low scientific quality of the primary studies. Further investigation into the effects of continuous ITB at all levels of the ICF-CY is warranted. Although large, controlled trials may be difficult to realize, national and international collaborations may provide opportunities. Also, multicentre prospective cohort studies with a long-term follow-up, employing harmonized outcome measures, can offer prospects to expand our knowledge of the effects of continuous ITB therapy in children. WHAT THIS PAPER ADDS: There is low-level evidence for continuous intrathecal baclofen (ITB) in children with cerebral palsy. Continuous ITB is effective in reducing spasticity and dystonia in non-controlled cohort studies. Evaluation of individual goals and systematic assessment of long-term effects in large cohort studies are required.

Intramuscular botulinum toxin prior to comprehensive rehabilitation has no added value for improving motor impairments, gait kinematics and goal attainment in walking children with spastic cerebral palsy.

OBJECTIVE: Botulinum toxin (BoNT-A) is widely used in combined treatment for spastic cerebral palsy, but its added value preceding comprehensive rehabilitation for motor impairments, gait, and goal attainment has not been studied. DESIGN: A comparative multi-centre trial, in which two groups underwent comprehensive rehabilitation (i.e. high-intensive functional physiotherapy, and indicated casting/orthoses). One group received intramuscular BoNT-A prior to rehabilitation, and the other group did not receive BoNT-A. SUBJECTS/PATIENTS: Children with spastic cerebral palsy, Gross Motor Function Classification System (GMFCS) levels I-III, age range 4-12 years, indicated for BoNT-A treatment regarding mobility problems. METHODS: Sixty-five children participated (37 boys), mean age 7.3 years (standard deviation (SD) 2.3, range 4-12 years), equally distributed across GMFCS levels. Forty-one children received BoNT-A+ comprehensive rehabilitation and 24 received comprehensive rehabilitation only. Functional leg muscle strength, passive range of motion, angle of catch, cerebral palsy-related pain, walking speed, kinematic gait parameters, goal attainment, and proxy-reported general functioning were assessed at baseline, primary end-point (12 weeks) and 24-week follow-up. Statistical analyses were performed with linear mixed models. RESULTS: At the primary end-point there were no statistically significant differences in treatment effects between the groups, except for the angle-of-catch of the rectus femoris, which was in favour of comprehensive rehabilitation without BoNT-A (12° difference, 95% confidence interval (95% CI) 2:23, p = 0.025). Results at follow-up were similar. CONCLUSION: At the group level, treating with BoNT-A prior to comprehensive rehabilitation did not add to the clinical effectiveness of rehabilitation. Thus, BoNT-A prescription and use should be critically reconsidered in this cerebral palsy age- and GMFCS-subgroup.

Improved parent-reported mobility and achievement of individual goals on activity and participation level after functional power-training in young children with cerebral palsy: a double-baseline controlled trial.

BACKGROUND: In children with cerebral palsy (CP), strength training programs to improve walking capacity and participation in activities of daily living are commonly used in clinical practice, despite lacking evidence of its effectiveness. It has been suggested that strength training with high movement velocity could be more effective than traditional resistance training to improve functional abilities such as walking. In a recently published study, we have demonstrated the positive effects of functional high-velocity resistance (power) training on muscle strength and walking capacity in young children with CP. Whether this type of training is also effective in achieving individual predefined goals in daily activities and self-reported mobility limitations, has not yet been described however. AIM: To evaluate the effect of functional power-training on parent-reported mobility and achievement of individual goals on activity and participation level in young children with CP. DESIGN: A double-baseline design was used to compare a 14-week period usual care with a 14-week period of functional power-training (3 times a week) and a follow-up period of 14 weeks. SETTING: A rehabilitation center, two special needs schools for children with physical disabilities, and a university medical center outpatient clinic. POPULATION: Twenty-two children with spastic CP (13 bilateral, GMFCS level I [N.=10] and level II [N.=12], mean age 7.5 years [SD 1.8, range 4-10 years]) and their parents participated. METHODS: Outcome measures were goal attainment scaling (GAS) of individual daily activity related treatment goals, mobility performance as measured using the Functional Mobility Scale (FMS-5 m, 50 m and 500 m), and the parent-reported Mobility Questionnaire (MobQues). RESULTS: After power-training, 86% of children achieved or exceeded their goal, compared with 14% in the usual care period (P<.001). The probability of improvement by one point or more on the FMS-500 meter after functional power-training was 10 times higher, compared with the usual care period (Relative Risk=10.0 with 95% CI 1.4 - 71.3). No changes were found in the FMS-5m and FMS-50m categories. Improvement on the MobQues was significantly greater after power-training compared with usual care (7.9% (95% CI 2.7 - 13.0, P=.005)). The improvement in performance in the activities defined in the treatment goals continued during the follow-up period. CONCLUSIONS AND CLINICAL REHABILITATION IMPACT: The results indicated that functional power-training is an effective training to achieve personalized treatment goals for activities in daily life and parent-reported mobility performance in young children with cerebral palsy.

Outcome of medial hamstring lengthening in children with spastic paresis: A biomechanical and morphological observational study.

To improve gait in children with spastic paresis due to cerebral palsy or hereditary spastic paresis, the semitendinosus muscle is frequently lengthened amongst other medial hamstring muscles by orthopaedic surgery. Side effects on gait due to weakening of the hamstring muscles and overcorrections have been reported. How these side effects relate to semitendinosus morphology is unknown. This study assessed the effects of bilateral medial hamstring lengthening as part of single-event multilevel surgery (SEMLS) on (1) knee joint mechanics (2) semitendinosus muscle morphology and (3) gait kinematics. All variables were assessed for the right side only. Six children with spastic paresis selected for surgery to counteract limited knee range of motion were measured before and about a year after surgery. After surgery, in most subjects popliteal angle decreased and knee moment-angle curves were shifted towards a more extended knee joint, semitendinosus muscle belly length was approximately 30% decreased, while at all assessed knee angles tendon length was increased by about 80%. In the majority of children muscle volume of the semitendinosus muscle decreased substantially suggesting a reduction of physiological cross-sectional area. Gait kinematics showed more knee extension during stance (mean change ± standard deviation: 34±13°), but also increased pelvic anterior tilt (mean change ± standard deviation: 23±5°). In most subjects, surgical lengthening of semitendinosus tendon contributed to more extended knee joint angle during static measurements as well as during gait, whereas extensibility of semitendinosus muscle belly was decreased. Post-surgical treatment to maintain muscle belly length and physiological cross-sectional area may improve treatment outcome of medial hamstring lengthening.

Value of botulinum toxin injections preceding a comprehensive rehabilitation period for children with spastic cerebral palsy: A cost-effectiveness study.

OBJECTIVE: Despite the widespread use of botulinum toxin in ambulatory children with spastic cerebral palsy, its value prior to intensive physiotherapy with adjunctive casting/orthoses remains unclear. DESIGN: A pragmatically designed, multi-centre trial, comparing the effectiveness of botulinum toxin + intensive physiotherapy with intensive physiotherapy alone, including economic evaluation. SUBJECTS/PATIENTS: Children with spastic cerebral palsy, age range 4-12 years, cerebral palsy-severity Gross Motor Function Classification System levels I-III, received either botulinum toxin type A + intensive physiotherapy or intensive physiotherapy alone and, if necessary, ankle-foot orthoses and/or casting. METHODS: Primary outcomes were gross motor func-tion, physical activity levels, and health-related quality-of-life, assessed at baseline, 12 (primary end-point) and 24 weeks (follow-up). Economic outcomes included healthcare and patient costs. Intention-to-treat analyses were performed with linear mixed models. RESULTS: There were 65 participants (37 males), with a mean age of 7.3 years (standard deviation 2.3 years), equally distributed across Gross Motor Function Classification System levels. Forty-one children received botulinum toxin type A plus intensive physio-therapy and 24 received intensive physiotherapy treatment only. At primary end-point, one statistically significant difference was found in favour of intensive physiotherapy alone: objectively measured percentage of sedentary behaviour (-3.42, 95% confidence interval 0.20-6.64, p=0.038). Treatment costs were significantly higher for botulinum toxin type A plus intensive physiotherapy (8,963 vs 6,182 euro, p=0.001). No statistically significant differences were found between groups at follow-up. CONCLUSION: The addition of botulinum toxin type A to intensive physiotherapy did not improve the effectiveness of rehabilitation for ambulatory children with spastic cerebral palsy and was also not cost-effective. Thus botulinum toxin is not recommended for use in improving gross motor function, activity levels or health-related quality-of-life in this cerebral palsy age- and severity-subgroup.

Measuring wearing time of knee-ankle-foot orthoses in children with cerebral palsy: comparison of parent-report and objective measurement.

Purpose state: Orthotic wearing time may be an important confounder in efficacy studies of treatment in children with spastic cerebral palsy (SCP). Most studies measure parent-reported wearing time (WTparent) with questionnaires, but it is questionable whether this yields valid results. This study aims to compare WTparent with objectively measured wearing time (WTobj) in children with SCP receiving orthotic treatment. METHOD: Eight children with SCP participated in this observational study. For one year, they received knee-ankle-foot orthosis (KAFO) treatment. WTparent was measured using questionnaires. WTobj was measured using temperature sensor-data-loggers that were attached to the KAFOs. The 2.5th and 97.5th percentiles and median of differences between methods (per participant) were used to calculate limits of agreement and systematic differences. RESULTS: There was no systematic difference between WTparent and WTobj (0.1 h per week), but high inter-individual variation of the difference was found, as reflected by large limits of agreement (lower limit/2.5th percentile: -1.7 h/week; upper limit/97.5th percentile: 11.1 h/week). CONCLUSIONS: Parent-reported wearing time (WTparent) of a KAFO differs largely from objectively measured wearing time (WTobj) using temperature sensors. Therefore, parent-reported wearing time (WTparent) of KAFOs should be interpreted with utmost care. Implications for Rehabilitation Low wearing time of orthoses may be a cause of inefficacy of orthotic treatment and incorrect reported wearing time may bias results of efficacy studies. Results of this study show that parent-reported wearing time is not in agreement with objectively measured wearing time. Parent-reported wearing time of KAFOs should be interpreted with utmost care. Objective methods are recommended for measuring orthotic wearing time.

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.

Improved Walking Capacity and Muscle Strength After Functional Power-Training in Young Children With Cerebral Palsy.

BACKGROUND: Strength training programs for children with cerebral palsy (CP) showed inconclusive evidence for improving walking, despite improvements in strength. Recent studies have suggested that strength training with high movement velocity is more effective for improving walking than traditional resistance training. OBJECTIVE: The purpose of this study was to evaluate the effect of functional high-velocity resistance training (power-training) to improve muscle strength and walking capacity of children with CP. METHOD: Twenty-two children with spastic CP participated (13 bilateral, Gross Motor Function Classification System [GMFCS] level I [n = 10] and II [n = 12], 7.5 years [SD 1.8, range 4-10 years]). Within-subjects changes in a 14-weeks usual care period were compared with changes in a 14-week functional power-training period (in groups, 3×/wk). Outcome measures were the muscle power sprint test (MPST), 1-minute walk test (1MWT), 10-m shuttle run test (SRT), gross motor function (GMFM-66), isometric strength of lower-limb muscles and dynamic ankle plantar flexor strength. RESULTS: Changes during the training period were significantly larger than changes in the usual care period for all outcome measures ( P < .05). Large improvements were found during the training period for walking capacity (ΔMPST [mean]: 27.6 W [95%CI 15.84-39.46, 83% increase], Δ1MWT: 9.4 m [95% CI 4.17-14.68, 13%], ΔSRT: 4.2 [95%CI 2.57-5.83, 56%], ΔGMFM-66: 5.5 [95% CI 3.33-7.74, 7%]) and muscle strength (18%-128%), while outcomes remained stable in the usual care period. CONCLUSIONS: The results indicate that functional power-training is an effective training for improving walking capacity in young children with cerebral palsy.