Factors for changes in self-care and mobility capabilities in young children with cerebral palsy involved in regular outpatient rehabilitation care.

BACKGROUND: Assessing prognosis of self-care and mobility capabilities in children with cerebral palsy (CP) is important for goal setting, treatment guidance and meaningful professional-caregiver conversations. AIMS: Identifying factors associated with changes in self-care and mobility capabilities in regular outpatient multidisciplinary paediatric CP rehabilitation care. METHODS AND PROCEDURES: Routinely monitored longitudinal data, assessed with the Paediatric Evaluation of Disability Inventory (PEDI-Functional-Skills-Scale, FSS 0-100) was retrospectively analysed. We determined contributions of age, gross-motor function, bimanual-arm function, intellectual function, education type, epilepsy, visual function, and psychiatric comorbidity to self-care and mobility capability changes (linear-mixed-models). OUTCOMES AND RESULTS: For 90 children (53 boys), in all Gross-Motor-Function-Classification-System (GMFCS) levels, 272 PEDI's were completed. Mean PEDI-FSS-scores at first measurement (median age: 3,2 years) for self-care and mobility were 46.3 and 42.4, and mean final FSS-scores respectively were 55.1 and 53.1 (median age: 6,5 years). Self-care capability change was significantly associated with age (2.81, p < 0.001), GMFCS levels III-V (-9.12 to -46.66, p < 0.01), and intellectual impairment (-6.39, p < 0.01). Mobility capability change was significantly associated with age (3.25, p < 0.001) and GMFCS levels II-V (-6.58 to -47.12, p < 0.01). CONCLUSIONS AND IMPLICATIONS: Most important prognostic factor for self-care and mobility capabilities is GMFCS level, plus intellectual impairment for self-care. Maximum capability levels are reached at different ages, which is important for individual goal setting and managing expectations.

The role of botulinum toxin in multimodal treatment of spasticity in ambulatory children with spastic Cerebral Palsy: extensive evaluation of a cost-effectiveness trial.

BACKGROUND: A cost-effectiveness trial (the Space Bop study) on the added value of botulinum toxin injections (BoNT-A) in the leg muscles, as part of a multimodal intervention for ambulatory children with spastic cerebral palsy in the context of a single distinct cycle of care was performed recently by our group. For a broad set of effect outcomes, we found that BoNT-A had no added value if children received comprehensive rehabilitation. However, this counterintuitive finding was met with scepticism. OBJECTIVE: Since several noteworthy facts and experiences were recorded during the course of the trial and the dissemination phase, the aim of this paper was to describe and discuss some crucial aspects of, and barriers to, the Space Bop study, related to context and perspective, design and results, as well as publication and implementation. METHODS: This paper discusses 5 issues: (i) the design, interpretation and presentation of previous research; (ii) the role of one's own clinical experience and interpretation; (iii) the aims of (BoNT-A) treatment; (iv) conflict of interest, role of industry, and the role of history; (v) optimal treatment modalities and dose-response relationships. CONCLUSION: Despite the unambiguous findings from the Space Bop study, several factors hindered acceptance of the results. Awareness of these factors is important when performing rehabilitation research and disseminating and implementing research findings.

Relationship between changes in motor capacity and objectively measured motor performance in ambulatory children with spastic cerebral palsy.

BACKGROUND: Different interventions are offered to children with cerebral palsy (CP) to improve the activity domain of the international classification of functioning (ICF). In therapy settings, the focus is mostly on motor capacity, but the ultimate goal is to improve motor performance. We therefore examined if changes in motor capacity outcomes are accompanied by changes in objectively measured motor performance after a 3-month intensive treatment period in ambulatory children with CP. METHODS: A secondary analysis on prospective clinical trial data was performed using multivariate linear regression. Sixty-five children (37 boys and 28 girls) with spastic CP, mean age 7 years and 3 months, Gross Motor Function Classification System (GMFCS) levels I-III were involved in a distinct 3-month intensive treatment period. Motor capacity (Gross Motor Function Measure [GMFM], functional muscle strength [FMS], and walking speed [WS]) and motor performance (using three Actigraph-GT3X+-derived outcome measures) were measured at baseline, 12 and 24 weeks. RESULTS: No significant associations were found for any of the change scores (∆12 ) between motor capacity and motor performance after a 12-week intensive treatment period. After 24 weeks, ∆24 FMS (p = .042) and ∆24 WS (p = .036) were significantly associated with changes in motor performance outcome measure percentage of time spent sedentary (∆24 %sedentary). In this model, 16% of variance of ∆24 %sedentary was explained by changes in motor capacity (p = .030). CONCLUSIONS: Changes in motor capacity are mostly not accompanied by changes in objectively measured motor performance after an intensive treatment period for ambulatory children with CP. These findings should be taken into account during goal setting and are important to manage expectations of both short- and longer term effects of treatment programmes.

Subgrouping patients with chronic low back pain: What are the differences in actual daily life behavior between patients classified as avoider or persister?

BACKGROUND AND OBJECTIVE: The purpose of this study is to determine whether patients, classified by their treating consultant in rehabilitation medicine as avoider or persister, show differences in a large set of detailed outcomes of actual, objectively measured daily physical behaviour. METHODS: In this explorative cross-sectional study, 16 patients were included; 9 patients were categorized as avoider and 7 patients as persister. Subjects wore the VitaMove activity monitor, a high-end accelerometry-based device that allowed automatic detection of a large set of body postures and motions. Physical behaviour was assessed in detail by total duration of body postures and motions as percentages of 24 hours, as well as by the number of sit-to-stand transfers, overall activity level, walking speed, and the distribution of bouts of physical activity and sedentary behaviour. Differences between groups were tested with the Mann Whitney U test. RESULTS: There were no significant differences between groups in any of the physical behaviour outcomes. CONCLUSIONS: Our study showed that activity-related behavioural style categorization by consultants in rehabilitation medicine is not expressed in objectively measured detailed outcomes of daily physical behaviour.

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.

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.

No recovery of cold complex regional pain syndrome after transdermal isosorbide dinitrate: a small controlled trial.

The microcirculation appears to be impaired in cold chronic complex regional pain syndrome (CRPS). This double-blind, placebo-controlled, randomized trial investigated the effect of the nitric oxide (NO) donor isosorbide dinitrate (ISDN) on the peripheral blood flow in patients with chronic CRPS. Twenty-four patients received 1% ISDN in Vaseline or a placebo ointment applied to the dorsum of the affected hand four times daily for 10 weeks. The patients participated in a physical therapy program to improve activity. The primary outcome measure was blood distribution in the affected extremity, which was determined by measuring the skin temperature using videothermography. We also measured NO and endothelin-1 concentrations in blister fluid, pain using the visual analog scale, and activity limitations using an upper limb activity monitor and the Disabilities of Arm Shoulder and Hand Questionnaire. ISDN failed to produce a significant improvement in temperature asymmetry in chronic cold CRPS patients, and it did not result in the expected reduction in pain and increase in activity compared with placebo either. There may be other central or peripheral factors contributing to the disturbed vasodynamics in cold chronic CRPS that are not influenced by NO substitution. This study does not show an improvement of the regional blood distribution by ISDN in the involved extremity of patients with cold-type CRPS.

Effect of tadalafil on blood flow, pain, and function in chronic cold complex regional pain syndrome: a randomized controlled trial.

BACKGROUND: This double-blind, randomized, controlled trial investigated the effect of the phosphodiesterase-5 inhibitor tadalafil on the microcirculation in patients with cold Complex Regional Pain Syndrome (CRPS) in one lower extremity. METHODS: Twenty-four patients received 20 mg tadalafil or placebo daily for 12 weeks. The patients also participated in a physical therapy program. The primary outcome measure was temperature difference between the CRPS side and the contralateral side, determined by measuring the skin temperature with videothermography. Secondary outcomes were: pain measured on a Visual Analogue Scale, muscle force measured with a MicroFet 2 dynamometer, and level of activity measured with an Activity Monitor (AM) and walking tests. RESULTS: At the end of the study period, the temperature asymmetry was not significantly reduced in the tadalafil group compared with the placebo group, but there was a significant and clinically relevant reduction of pain in the tadalafil group. Muscle force improved in both treatment groups and the AM revealed small, non-significant improvements in time spent standing, walking, and the number of short walking periods. CONCLUSION: Tadalafil may be a promising new treatment for patients that have chronic cold CRPS due to endothelial dysfunction, and deserves further investigation. TRIAL REGISTRATION: The registration number in the Dutch Trial Register is ISRCTN60226869.

Objective and continuous measurement of peripheral motor indicators of pain in hospitalized infants: a feasibility study.

Measurement of pain in pre-verbal infants is complex. Until now, pain behavior has mainly been assessed intermittently using observational tools. Therefore, we determined the feasibility of long-term, objective and continuous measurement of peripheral motor parameters through body-fixed sensors to discriminate between pain and no pain in hospitalized pre-verbal infants. Two pain modes were studied: for procedural pain 10 measurements were performed before, during and after routine heel lances in 9 infants (age range infants: 5-175 days), and for post-operative pain 14 infants (age range 45-400 days) were measured for prolonged periods (mean 7h) using the validated COMFORT-behavior scale as reference method. Several peripheral motor parameters were studied: three body part activity parameters derived from acceleration sensors attached to one arm and both legs, and two muscle activity parameters derived from electromyographic (EMG) sensors attached to wrist flexor and extensor muscles. Results showed that the accelerometry-based parameters legs activity and overall extremity activity (i.e. mean of arm and legs) were significantly higher during heel lance than before or after lance (p0.001), whereas arm activity accelerometry data and wrist muscle activity EMG data showed no significant change. For the post-operative pain measurements, relationships were found between accelerometry-based overall extremity activity and COMFORT-behavior (r=0.76, p<0.001), and between EMG-based wrist flexor activity and COMFORT-behavior (r=0.55, p<0.001, for a subgroup of 7 infants). We conclude that long-term, objective and continuous measurement of peripheral motor parameters is feasible, has high potential, and is promising to assess pain in pre-verbal hospitalized infants.

Impact of chronic pain on everyday physical activity.

Although patients with chronic pain are often considered to have reduced levels of everyday physical activity, data on their activity levels are scarce and inconclusive. Therefore, this study explored whether patients with chronic pain have reduced activity levels, as objectively measured with an activity monitor. The activity monitor is based on long-term ambulatory monitoring of signals from body-fixed accelerometers during everyday life, aimed at assessment of mobility-related activities. Measurements with the monitor were performed during a weekday (24 h) in 18 patients with chronic pain and compared with measurements obtained from 18 gender and age matched healthy comparison subjects. The mean (SD) age of the patients was 44 (11) years, and the mean (SD) duration of their complaints was 8 (7) years. Compared with the healthy subjects, the duration of dynamic activities was not significantly reduced (p=0.10) in the patient group. Mean (SD) intensity of everyday physical activity was lower (p=0.03) in the patients than in the healthy comparison subjects (0.021 [0.006] g versus 0.026 [0.004] g), and patients spent more time lying down (47.0 [10.2]% versus 34.3 [5.6] %; p=0.000) and less time sitting (29.2 [8.9]% versus 36.4 [9.3]%; p=0.03) than the healthy comparison subjects. In spite of significant differences between patients and healthy comparison subjects for some aspects of the activity pattern (which may reflect pain behaviour), the impact of chronic pain on everyday physical activity was relatively small.

Objective measurement of upper limb activity and mobility during everyday behavior using ambulatory accelerometry: the upper limb activity monitor.

Ambulatory accelerometry is a technique that allows objective measurement of aspects of everyday human behavior. The aim of our research has been to develop, validate, and apply this technique, which recently resulted in an upper limb activity monitor (ULAM). The ULAM consists of body-mounted acceleration sensors connected to a waist-worn data recorder and allows valid and objective assessment of activity of both upper limbs during performance of also automatically detected mobility-related activities: lying, sitting, standing, walking, cycling, and general movement. The ULAM can be used to determine (limitations of) upper limb activity and mobility in freely moving subjects with upper limb disorders. This article provides a detailed description of its characteristics, summarizes the results of a feasibility study and four application studies in subjects having upper limb complex regional pain syndrome, discusses the most important practical, technical, and methodological issues that were encountered, and describes current and future research projects related to measuring (limitations of) upper limb activity.

Upper limb activity over time in complex regional pain syndrome type 1 as objectively measured with an upper limb-activity monitor: an explorative multiple case study.

BACKGROUND: An upper limb-activity monitor (ULAM) has been developed to determine activity limitations in complex regional pain syndrome type 1 (CRPS1). The ULAM is based on 24h ambulatory monitoring of body segment accelerations and enables valid and objective quantification of mobility and upper limb activity in transversal studies. AIMS: To explore upper limb activity over time in acute upper limb CRPS1 as measured with the ULAM in a longitudinal study, and to compare this to time courses of other outcome measures for activity limitations and impairments. METHODS: Four subjects were measured four times during a treatment protocol. Several ULAM outcome measures related to upper limb usage and mobility, three questionnaires (RASQ, DASH, RAND36), and six impairment outcome indicators (VAS-momentary pain, VAS-pain resulting from effort, volume, temperature, active range of motion, strength) were used. RESULTS: Objectively measured upper limb activity frequently improved; improvements of >5% were found for 63% of the ULAM outcome measures at final assessment. The ULAM outcome measures had a time course more similar to the body-part and CRPS1 specific questionnaire RASQ than the other questionnaires. The time course of impaired temperature was most often in accordance with the ULAM, and both VAS scores showed least accordance. CONCLUSIONS: Clear changes in upper limb activity over time were frequently found as objectively measured with the ULAM, and relationships among the time courses of the ULAM and other outcome measures were largely explainable. The ULAM can validly assess upper limb activity over time in CRPS1, but between-measurement variability needs careful consideration.

Correlation between a novel upper limb activity monitor and four other instruments to determine functioning in upper limb complex regional pain syndrome type I.

OBJECTIVE: To determine the place of a novel Upper Limb Activity Monitor in the field of instruments measuring functioning and health in upper limb complex regional pain syndrome type I, by exploring the correlation between the Upper Limb Activity Monitor and 4 questionnaires. METHOD: Subjects (n = 30) were measured at home and correlations were calculated between the Upper Limb Activity Monitor and 4 questionnaires; Sickness Impact Profile, RAND-36 Health Survey, Disabilities of Arm Shoulder Hand Questionnaire and Radboud Skills Questionnaire. RESULTS: Of the inter-questionnaire correlations 83% were significant, whereas 46% of the correlations between the Upper Limb Activity Monitor and the questionnaires were significant. The number and strength of the correlations between the Upper Limb Activity Monitor and questionnaires was dependent on the degree to which similar aspects of functioning were measured. CONCLUSION: The Upper Limb Activity Monitor has some correlation with other instruments related to functioning and health, but generally it does not measure the same areas.

Impairments and activity limitations in subjects with chronic upper-limb complex regional pain syndrome type I.

OBJECTIVE: To determine the degree of impairments and activity limitations and their interrelationship in complex regional pain syndrome type I (CRPS type I). DESIGN: Cross-sectional study interrelating impairments and objectively measured activity limitations. SETTING: Ambulatory and home environment. PARTICIPANTS: Thirty nonacute upper-limb CRPS type I subjects. INTERVENTIONS: Not applicable. Main outcome measures Sensory, motor, and autonomic impairments, as well as activity-limitation outcome measures. The latter were objectively measured with a novel upper-limb activity monitor (based on ambulatory accelerometry). RESULTS: All subjects were impaired to some degree but with a large variability with respect to magnitude. Regarding activity limitations, the involved upper limb was clearly less active (lower intensity and percentage of activity) than the noninvolved side. Impaired active range of motion (adjusted R(2) range, 18%-39%) and grip strength (adjusted R(2) range, 12%-45%) were the most important factors explaining variance in activity limitations. CONCLUSIONS: All subjects were still impaired nearly 3 years after the causative event. The involved upper limb was also clearly less active than the noninvolved side, especially when the subjects were sitting and when the dominant side was involved. The more impairments a subject had, especially motor impairments, the more activity limitations were present.

Impact of upper limb complex regional pain syndrome type 1 on everyday life measured with a novel upper limb-activity monitor.

Complex regional pain syndrome type 1 (CRPS1) often leads to serious activity limitations in everyday life. To date, however, limitations in patients with CRPS1 of an upper limb have not been objectively measured.Therefore, the aim of this study was to determine the long-term impact of upper limb CRPS1 on general mobility and upper limb usage during everyday life, as measured with a novel upper limb-activity monitor (ULAM). In ten female chronic CRPS1 patients and ten healthy control subjects, 24-h activity patterns were measured with the ULAM. This ULAM consists of body-fixed acceleration sensors, connected to a recorder worn around the waist. The ULAM automatically detects upper limb activity during mobility-related activities. Several outcome measures related to general mobility and upper limb usage were compared between patients and controls. The results showed that CRPSI in the dominant upper limb had modest impact on general mobility; i.e. on the percentages spent in body positions and body motions and on mean intensity of body activity. For upper limb usage outcome measures during sitting, there was a marked difference between CRPS1 patients and controls. Especially patients with dominant side involvement clearly showed less activity of their involved limb during sitting, indicated by significant differences for the mean intensity (P=0.014), percentage (P=0.004), and proportion (P=0.032) of upper limb activity. It is concluded that these ten chronic CRPS1 patients still had limitations in upper limb usage during everyday life, 3.7 years (average) after the causative event.