Bimanual Capacity of Children With Cerebral Palsy: Intra- and Interrater Reliability of a Revised Edition of the Bimanual Fine Motor Function Classification.

AIMS: To develop a revised edition of the Bimanual Fine Motor Function (BFMF 2), as a classification of fine motor capacity in children with cerebral palsy (CP), and establish intra- and interrater reliability of this edition. METHODS: The content of the original BFMF was discussed by an expert panel, resulting in a revised edition comprising the original description of the classification levels, but in addition including figures with specific explanatory text. Four professionals classified fine motor function of 79 children (3-17 years; 45 boys) who represented all subtypes of CP and Manual Ability Classification levels (I-V). Intra- and inter-rater reliability was assessed using overall intra-class correlation coefficient (ICC), and Cohen's quadratic weighted kappa. RESULTS: The overall ICC was 0.86. Cohen's weighted kappa indicated high intra-rater (кw: >0.90) and inter-rater (кw: >0.85) reliability. CONCLUSIONS: The revised BFMF 2 had high intra- and interrater reliability. The classification levels could be determined from short video recordings (<5 minutes), using the figures and precise descriptions of the fine motor function levels included in the BFMF 2. Thus, the BFMF 2 may be a feasible and useful classification of fine motor capacity both in research and in clinical practice.

Bimanual Fine Motor Function (BFMF) Classification in Children with Cerebral Palsy: Aspects of Construct and Content Validity.

UNLABELLED: The Bimanual Fine Motor Function (BFMF) is currently the principal classification of hand function recorded by the Surveillance of Cerebral Palsy in Europe (SCPE) register. The BFMF is used in a number of epidemiological studies, but has not yet been validated. AIMS: To examine aspects of construct and content validity of the BFMF. METHODS AND RESULTS: Construct validity of the BFMF was assessed by comparison with the Manual Ability Classification System (MACS) using register-based data from 539 children born 1999-2003 (304 boys; 4-12 years). The high correlation with the MACS (Spearman's rho = 0.89, CI: 0.86-0.91, p<.001) supports construct validity of the BFMF. The content of the BFMF was appraised through literature review, and by using the ICF-CY as a framework to compare the BFMF and MACS. The items hold, grasp and manipulate were found to be relevant to describe increasingly advanced fine motor abilities in children with CP, but the description of the BFMF does not state whether it is a classification of fine motor capacity or performance. CONCLUSION: Our results suggest that the BFMF may provide complementary information to the MACS regarding fine motor function and actual use of the hands, particularly if used as a classification of fine motor capacity.

Stability of motor function and associated impairments between childhood and adolescence in young people with cerebral palsy in Europe.

AIM: The aim of the study was to investigate whether impairments associated with cerebral palsy were stable between childhood and adolescence. METHOD: The Study of Participation of Children with Cerebral Palsy Living in Europe (SPARCLE) longitudinal study was conducted in nine European regions. In total, 818 children aged 8 to 12 years were randomly selected from population-based registers; 594 (73%) were followed up at the age of 13 to 17 years (344 males, 250 females; median age 10 y 4 mo) Research associates visited them in their homes and recorded their motor function and additional impairments. Stability of impairment was assessed using the weighted kappa coefficient. RESULTS: The proportion of participants whose level of impairment remained unchanged varied from 63% for fine motor function to 98% for hearing. For gross motor function, communication, and cognitive level, the kappa and the lower bound of its 95% confidence interval (CI) were above 0.75, indicating stability between childhood and adolescence; for fine motor function and feeding, the kappa was above 0.75 but the lower bound of the 95% CI was below 0.75, indicating probable stability; for seizures and vision, the kappa was below 0.75, although the upper bound of the 95% CI was above 0.75, indicating possible change; for hearing the kappa and its entire CI were below 0.75, indicating change. Overall, 81% of participants had no seizures in childhood, of whom 93% were seizure-free in adolescence. INTERPRETATION: Motor function and additional impairments were generally stable between childhood and adolescence.

Association between participation in life situations of children with cerebral palsy and their physical, social, and attitudinal environment: a cross-sectional multicenter European study.

OBJECTIVE: To evaluate how participation of children with cerebral palsy (CP) varied with their environment. DESIGN: Home visits to children. Administration of Assessment of Life Habits and European Child Environment Questionnaires. Structural equation modeling of putative associations between specific domains of participation and environment, while allowing for severity of child's impairments and pain. SETTING: European regions with population-based registries of children with CP. PARTICIPANTS: Children (n=1174) aged 8 to 12 years were randomly selected from 8 population-based registries of children with CP in 6 European countries. Of these, 743 (63%) agreed to participate; 1 further region recruited 75 children from multiple sources. Thus, there were 818 children in the study. INTERVENTIONS: Not applicable. MAIN OUTCOME MEASURE: Participation in life situations. RESULTS: For the hypothesized associations, the models confirmed that higher participation was associated with better availability of environmental items. Higher participation in daily activities-mealtimes, health hygiene, personal care, and home life-was significantly associated with a better physical environment at home (P<.01). Mobility was associated with transport and physical environment in the community. Participation in social roles (responsibilities, relationships, recreation) was associated with attitudes of classmates and social support at home. School participation was associated with attitudes of teachers and therapists. Environment explained between 14% and 52% of the variation in participation. CONCLUSIONS: The findings confirmed the social model of disability. The physical, social, and attitudinal environment of disabled children influences their participation in everyday activities and social roles.

Pain, balance, activity, and participation in children with hypermobility syndrome.

PURPOSE: To describe hypermobility, balance, pain, activity, and participation in children with hypermobility and compare these characteristics with those of a control group. METHOD: Twenty children aged 8 to 16 years with hypermobility syndrome (HMS) or Ehlers-Danlos syndrome and a control group of 24 children of the same age participated in the study. Hypermobility was assessed according to the Del Mar scale, balance was assessed with the Bruininks-Oseretsky test of motor proficiency, and participation in daily life activities was assessed with the frequency of participation questionnaire. Pain and physical activity were assessed in a diary. RESULTS: In comparison with the control group, the children with hypermobility had significantly more hypermobile joints and more pain and scored lower in the balance test, and their activity was affected on a daily basis. CONCLUSION: Pain appears to affect activity and participation in children with HMS. Balance is decreased in children with HMS compared with healthy controls.

Bone health in children and adolescents with juvenile idiopathic arthritis and the influence of short-term physical exercise.

PURPOSE: To study bone mineral density (BMD) in 54 children and adolescents with juvenile idiopathic arthritis before and after a short-term exercise program. METHODS: Fifty-four children, 41 girls and 13 boys, median age 13.9 years, participated in a 12-week exercise program, with 33 children in an exercise group. The program consisted of one hundred 2-footed jumps with a rope and standardized muscle strength exercise. Both BMD and bone mineral content were assessed with dual-energy x-ray absorptiometry (DXA) and DXA Laser Calscan for the heel at the start and after 3 and 6 months. RESULTS: The study group had BMD measurements within the reference range compared with normative data with Z score at start. Bone mineral density values in total body, but not Z score, increased significantly (P = .012) in the exercise group. CONCLUSIONS: The study group had BMD measurements within the reference range. Twelve weeks of exercise increases BMD in children with juvenile idiopathic arthritis.

Are infants with torticollis at risk of a delay in early motor milestones compared with a control group of healthy infants?

Recently it has been claimed that infants with congenital muscular torticollis (CMT) are at risk of a delay in early motor milestones. The aim of the present study was to investigate whether infants with CMT are indeed at risk in comparison with a control group of healthy infants. A second aim was to investigate whether the time spent in a prone position and plagiocephaly had any influence on motor development. Eighty-two infants with CMT (35 females and 47 males) were compared with 40 healthy infants (18 females and 22 males). Motor development was assessed with the Alberta Infant Motor scale (AIMS). Multiple regression showed that infants in the CMT group had a significantly lower AIMS score than the control group at 2 months (p=0.03) and 6 months of age (p=0.05). Infants who spent at least three occasions daily in a prone position when awake had significantly higher AIMS scores than infants who spent less time prone at 2 months (p=0.001), 6 months (p<0.001), and 10 months of age (p<0.001). The CMT group achieved early motor milestones significantly later than the control group until the age of 10 months, but the risk of delay seems to be more strongly associated with little or no time prone when awake than with CMT.

Reliability and validity of the Child Health QuestionnairePF-50 for European children with cerebral palsy.

OBJECTIVE: To evaluate the psychometric performance of the Child Health Questionnaire (CHQ) in children with cerebral palsy (CP). METHOD: 818 parents of children with CP, aged 8-12 from nine regions of Europe completed the CHQ (parent form 50 items). Functional abilities were classified using the five-level Gross Motor Function Classification Scheme (Levels I-III as ambulant; Level IV-V as nonambulant CP). RESULTS: Ceiling effects were observed for a number of subscales and summary scores across all Gross Motor Function Classification System levels, whilst floor effects occurred only in the physical functioning scale (Level V CP). Reliability was satisfactory overall. Confirmatory factor analysis (CFA) revealed a seven-factor structure for the total sample of children with CP but with different factor structures for ambulant and nonambulant children. CONCLUSION: The CHQ has limited applicability in children with CP, although with judicious use of certain domains for ambulant and nonambulant children can provide useful and comparable data about child health status for descriptive purposes.

Self-reported quality of life of 8-12-year-old children with cerebral palsy: a cross-sectional European study.

BACKGROUND: Little is known about the quality of life (QoL) of disabled children. We describe self-reported QoL of children with cerebral palsy, factors that influence it, and how it compares with QoL of the general population. METHODS: 1174 children aged 8-12 years were randomly selected from eight population-based registers of children with cerebral palsy in six European countries and 743 (63%) agreed to participate; one further region recruited 75 children from multiple sources. Researchers visited these 818 children. 318 (39%) with severe intellectual impairment could not self-report; 500 (61%) reported their QoL using KIDSCREEN, an instrument with scores in ten domains, each with SD=10. Multivariable regression was used to relate QoL to impairments, pain, and sociodemographic characteristics. Comparisons were made with QoL data from the general population. FINDINGS: Impairments were not significantly associated with six KIDSCREEN domains. Comparison of least and most able groups showed that severely limited self-mobility was significantly associated with reduced mean score for physical wellbeing (7.6, 95% CI 2.7-12.4); intellectual impairment with reduced mean for moods and emotions (3.7, 1.5-5.9) and autonomy (3.3, 0.9-5.7); and speech difficulties with reduced mean for relationships with parents (4.5, 1.9-7.1). Pain was common and associated with lower QoL on all domains. Impairments and pain explained up to 3% and 7%, respectively, of variation in QoL. Children with cerebral palsy had similar QoL to children in the general population in all domains except schooling, in which evidence was equivocal, and physical wellbeing, in which comparison was not possible. INTERPRETATION: Parents can be reassured that most children aged 8-12 years with cerebral palsy will have similar QoL to other children. This finding should guide social and educational policy to ensure that disabled children participate fully in society. Because of its association with QoL, children's pain should be carefully assessed.

Psychological problems in children with cerebral palsy: a cross-sectional European study.

OBJECTIVES: To describe psychological symptoms in 8-12-year-old children with cerebral palsy; to investigate predictors of these symptoms and their impact on the child and family. DESIGN: A cross-sectional multi-centre survey. PARTICIPANTS: Eight hundred and eighteen children with cerebral palsy, aged 8-12 years, identified from population-based registers of cerebral palsy in eight European regions and from multiple sources in one further region. MAIN OUTCOME MEASURES: The Strengths and Difficulties Questionnaire (SDQ)(P4-16) and the Total Difficulties Score (TDS) dichotomised into normal/borderline (TDS < or = 16) versus abnormal (TDS > 16). STATISTICAL ANALYSIS: Multilevel, multivariable logistic regression to relate the presence of psychological symptoms to child and family characteristics. RESULTS: About a quarter of the children had TDS > 16 indicating significant psychological symptoms, most commonly in the domain Peer Problems. Better gross motor function, poorer intellect, more pain, having a disabled or ill sibling and living in a town were independently associated with TDS > 16. The risk of TDS > 16 was odds ratio (OR) = .2 (95% CI: .1 to .3) comparing children with the most and least severe functional limitations; OR = 3.2 (95%CI: 2.1 to 4.8) comparing children with IQ < 70 and others; OR = 2.7 (95% CI: 1.5 to 4.6) comparing children in severe pain and others; OR = 2.7 (95% CI:1.6 to 4.6) comparing children with another disabled sibling or OR = 1.8 (95%CI: 1.2 to 2.8) no siblings and others; OR = 1.8 (95% CI: 1.1 to 2.8) comparing children resident in a town and others. Among parents who reported their child to have psychological problems, 95% said they had lasted over a year, 37% said they distressed their child and 42% said they burdened the family at least 'quite a lot'. CONCLUSIONS: A significant proportion of children with cerebral palsy have psychological symptoms or social impairment sufficiently severe to warrant referral to specialist services. Care must be taken in the assessment and management of children with cerebral palsy to ensure psychological problems are not overlooked and potentially preventable risk factors like pain are treated effectively. The validity of the SDQ for children with severe disability warrants further assessment.

Behaviour in children with cerebral palsy with and without epilepsy.

The aim of the study was to describe behavioural problems in children with cerebral palsy (CP) with and without epilepsy. The children were sampled from the Western Sweden CP register and were part of a European Union project. The Strength and Difficulties Questionnaire and questions on epilepsy were answered by one parent of each child. Medical records were reviewed. Parents of 83 children (44 males, 39 females) age range participated: 30 at Gross Motor Function Classification System levels I and II, and 53 at levels III to V; 60 had spastic age range 8 to 12 years (bilateral 42, unilateral 18) and 23 dyskinetic CP; 34 children had active epilepsy. The proportion of children with normal behaviour on the total difficulties score (TDS) of the Strength and Difficulties Questionnaire was significantly lower than normative data (57% vs 80%, p<0.001). Parents of 21 children (25%) considered their child's behaviour to be abnormal. Children with CP and epilepsy had a significantly higher median TDS (p=0.03) than seizure-free children. In children with aided or no walking ability, the TDS was significantly higher in those with epilepsy (p=0.04). Parents of 32 children (39%) considered their children's behaviour to have an impact on themselves and others. We conclude that behavioural problems are common in children with CP, and even more when epilepsy is present. Parents identify these problems, and professionals need to address them.

Muscle strength training to improve gait function in children with cerebral palsy.

The aim of the study was to investigate the influence of muscle strength training on gait outcomes in children with cerebral palsy. Sixteen children (two females, 14 males, Gross Motor Function Classification System levels I-II, mean age 12y 6mo, range 9y 4mo-15y 4mo) underwent muscle strength measurement using a handheld device, Gross Motor Function Measure (GMFM) assessment, three-dimensional gait analysis, joint range of motion assessment, and grading of spasticity before and after 8 weeks of training. All participants had a diagnosis of spastic diplegia and could walk without aids. Training consisted of exercises for lower extremity muscles with free weights, rubber bands, and body weight for resistance, three times a week. Values for muscle strength below normal were identified in all children; this was most pronounced at the ankle, followed by the hip muscles. After training, muscle strength and GMFM scores increased, velocity was unchanged, stride length increased, and cadence was reduced. There was an increase in hip extensor moment and power generated at push off. Eight weeks of muscle strength training can increase muscle strength and improve gait function.

Walking ability is related to muscle strength in children with cerebral palsy.

The purpose of this study was to assess the relation between muscle strength and walking ability in children with bilateral spastic cerebral palsy at GMFCS levels I-III. 55 children (mean age 10.7, range 5-15) were tested for muscle strength in eight lower limb muscle groups with a handheld myometer. They were also tested with the Gross Motor Function Measure domains for standing and walking, running and jumping. Muscle strength in the legs was below normative predicted value in most of the children, with muscle weakness most pronounced around the ankle, followed by the hip muscles. There was a significant difference in muscle strength between GMFCS levels. There was also a moderate to high correlation between muscle strength and the GMFM, indicating that muscle weakness affects walking ability. Independently walking children had more than 50% of predicted muscle strength values.

Reference values for range of motion and muscle function of the neck in infants.

PURPOSE: To determine reference values for cervical range of motion (ROM) in rotation and lateral flexion and for muscle function in the lateral neck flexors in a sample of infants who were healthy. METHOD: ROM was measured, and muscle function was estimated in 38 infants at the ages of 2, 4, 6, and 10 months. RESULTS: For rotation the mean ROM was 110 degrees and for lateral flexion it was 70 degrees. Infants of 2 months of age had a median muscle function score of 1 (interquartile range, 1-2). Muscle function increased to score 3 to 4 by 10 months. CONCLUSION: Infants below 1 year of age have good ROM in rotation (> or = 100 degrees ) and lateral flexion (> or = 65 degrees ) of the neck. These reference values for passive ROM and muscle function of the neck may have clinical utility in assessing and documenting the initial evaluation and progress of infants with congenital muscular torticollis.

Bilateral spastic cerebral palsy--prevalence through four decades, motor function and growth.

UNLABELLED: The aim was to depict changes in the prevalence and severity of bilateral spastic cerebral palsy (CP) over a 40-year period. Another objective was to characterise the group born in 1991-1998 with respect to gross motor function, spasticity and growth. Data were obtained from the CP register of western Sweden and rehabilitation records. RESULTS: After a rise to 1.27 per 1000 live births in 1983-1986, the prevalence decreased significantly, in children born both preterm and at term, to 0.69 in 1995-1998. After 1975, more children were born preterm than at term. There was a significant decrease in severe bilateral spastic CP during the same period, mainly in children born at term. In all, 46% of the children born at term and 33% of those born preterm had a severe motor impairment, i.e. no walking ability. In the 167 children born in 1991-1998, the gross motor function classification system (GMFCS) level was I in 14%, II in 34%, III in 10%, IV in 25% and V in 17%. The GMFCS level correlated with the gross motor function measure (GMFM) and the Ashworth spasticity scores, as well as with the deviation in postnatal weight and height. We conclude that the prevalence of bilateral spastic CP has decreased since the mid-1980s, parallel to a reduction in the severity of the motor impairment. Children born preterm have predominated since the mid-1970s. The severity of the motor impairment correlated with the degree of spasticity, GMFM and growth. The percentage of children who were underweight was substantial.

Assessment of data quality in a multi-centre cross-sectional study of participation and quality of life of children with cerebral palsy.

BACKGROUND: SPARCLE is a cross-sectional survey in nine European regions, examining the relationship of the environment of children with cerebral palsy to their participation and quality of life. The objective of this report is to assess data quality, in particular heterogeneity between regions, family and item non-response and potential for bias. METHODS: 1,174 children aged 8-12 years were selected from eight population-based registers of children with cerebral palsy; one further centre recruited 75 children from multiple sources. Families were visited by trained researchers who administered psychometric questionnaires. Logistic regression was used to assess factors related to family non-response and self-completion of questionnaires by children. RESULTS: 431/1,174 (37%) families identified from registers did not respond: 146 (12%) were not traced; of the 1,028 traced families, 250 (24%) declined to participate and 35 (3%) were not approached. Families whose disabled children could walk unaided were more likely to decline to participate. 818 children entered the study of which 500 (61%) self-reported their quality of life; children with low IQ, seizures or inability to walk were less likely to self-report. There was substantial heterogeneity between regions in response rates and socio-demographic characteristics of families but not in age or gender of children. Item non-response was 2% for children and ranged from 0.4% to 5% for questionnaires completed by parents. CONCLUSION: While the proportion of untraced families was higher than in similar surveys, the refusal rate was comparable. To reduce bias, all analyses should allow for region, walking ability, age and socio-demographic characteristics. The 75 children in the region without a population based register are unlikely to introduce bias.

Children who had congenital torticollis as infants are not at higher risk for a delay in motor development at preschool age.

OBJECTIVE: To investigate whether congenital muscular torticollis (CMT) or the time in a prone position as an infant had any influence on motor development at preschool age. DESIGN: A case-control study. PARTICIPANTS: Eighty-one children who had participated in a previous study that investigated motor development in infants with CMT and a control group of infants without CMT. METHOD: A follow-up at the age of 3.5-5 years; the Movement Assessment Battery for Children was used with the earlier CMT group and the control group to assess their motor development. An independent physiotherapist, who was blinded of the children's previous group belonging, assessed the children. MAIN OUTCOME MEASUREMENTS: Percentile scores of motor development. RESULTS: Multiple regression showed no impact on earlier group belonging or the amount of time spent in a prone position as an infant. The left-handed children had a significantly (P < .01) lower percentile in the Movement Assessment Battery for Children. CONCLUSION: Neither CMT nor spending limited periods of time as an infant in the prone position when awake have any significant long-term effects on motor development. Children who had CMT as infants were not at higher risk for a delay in motor development at preschool age.

A pilot study on changes in passive range of motion in the cervical spine, for children aged 0-5 years.

BACKGROUND: The aim of this study was to investigate the values for passive range of motion (PROM) in rotation and lateral flexion of the neck, for children aged 3.5-5 years, and to compare the results with the measurements carried out on the same group of children as infants. METHOD: Twenty-three children (10 females) were assessed in PROM in rotation and lateral flexion of the neck. Both measurements were performed with the children in a lying position using a large protractor. The dependent t-test was used to compare PROM with the PROM from an earlier study when the children were infants. RESULT: PROM of the neck was found to be a mean 100° in rotation and 70° in lateral flexion at the age of 3.5-5 years. There was a significant decrease in rotation when comparing the measurements with values from infancy. CONCLUSIONS: These data suggest that there is a decrease in PROM in rotation during the first 5 years of life, and that PROM in lateral flexion remains unchanged.

Muscle strength, physical fitness and well-being in children and adolescents with juvenile idiopathic arthritis and the effect of an exercise programme: a randomized controlled trial.

BACKGROUND: Decreased muscle strength, fitness and well-being are common in children and adolescents with juvenile idiopathic arthritis (JIA) compared to healthy peers. Biological drugs have improved health in children with JIA, but despite this pain is still a major symptom and bone health is reported as decreased in the group. The improvement made by the biological drugs makes it possible to more demanding exercises. To jump is an exercise that can improve bone heath, fitness and muscle strength. The aim of the study was to see if an exercise programme with jumps had an effect on muscle strength, physical fitness and well-being and how it was tolerated. METHODS: Muscle strength and well-being were studied before and after a 12-week exercise programme in 54 children and adolescents with JIA, 9-21 years old. The participants were randomized into an exercise and a control group. Muscle strength, fitness and well-being were documented before and after the training period and at follow-up after 6 months. Physical activity in leisure time was documented in diaries. The fitness/exercise programme was performed at home three times a week and included rope skipping and muscle strength training exercises.Assessment included measurement of muscle strength with a handheld device, and with Grip-it, step-test for fitness with documentation of heart rate and pain perception and two questionnaires (CHAQ, CHQ) on well-being. RESULTS: There were no differences between exercise and control group regarding muscle strength, grip strength, fitness or well-being at base line. Muscle weakness was present in hip extensors, hip abductors and handgrip. For the exercise group muscle strength in hip and knee extensors increased after the 12-week exercise programme and was maintained in knee extensors at follow-up. There was no change in fitness tested with the individually adapted step-test. The CHQ questionnaire showed that pain was common in the exercise group and in the control group. There were only small changes in the CHAQ and CHQ after the training period. The fitness/exercise programme was well tolerated and pain did not increase during the study. CONCLUSIONS: A weight bearing exercise programme, with muscle strength training with free weights and rope skipping was well tolerated without negative consequences on pain. It also improved muscle strength in the legs and can be recommended for children and adolescents with JIA.