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1.
Dev Med Child Neurol ; 65(4): 509-516, 2023 04.
Article in English | MEDLINE | ID: mdl-36155917

ABSTRACT

AIM: To explore whether subgroups of adults with cerebral palsy (CP) with different fatigue diurnal profiles can be discerned, and to explore whether sleep, physical activity, or health-related fitness are associated with these profiles. METHOD: Thirty-two adults (median age 29 years 8 months; range 20-54 years; 11 males, 21 females) with spastic CP (Gross Motor Function Classification System levels I-III) with physical activity-related fatigue complaints participated. Real-time fatigue and physical activity were assessed for 7 consecutive days by short message service text four times during the day and by wearing an accelerometer respectively. Sleep was assessed by the Pittsburgh Sleep Quality Index, and fitness by assessing body composition and aerobic capacity. Latent class growth modelling was used to classify subgroups according to their diurnal profiles of real-time fatigue. Univariable multinomial logistic regression analysis explored whether participant characteristics, sleep, physical activity, or health-related fitness were associated with diurnal profiles. RESULTS: Three distinct fatigue diurnal profiles were identified: stable low (n = 10), increasing (n = 14), and stable high (n = 8). Only aerobic capacity was associated with fatigue profiles (odds ratio 1.15, 95% confidence interval 1.00-1.34; p = 0.05). INTERPRETATION: Fatigue in adults with CP may be low or high stable or may increase during the day. These findings indicate the relevance of assessing fatigue variability. WHAT THIS PAPER ADDS: We found three patterns of daily fatigue in adults with cerebral palsy (CP). Only aerobic capacity was associated with fatigue profiles in adults with CP. Moment-to-moment variations in fatigue can help with personalized fatigue management.


Subject(s)
Cerebral Palsy , Physical Fitness , Male , Female , Humans , Adult , Exercise , Fatigue , Sleep
2.
Br J Sports Med ; 55(20): 1125-1134, 2021 Oct.
Article in English | MEDLINE | ID: mdl-34187784

ABSTRACT

OBJECTIVE: To provide a comprehensive, evidence-based overview of the risk factors, prevention, diagnosis, imaging, treatment and prognosis for Achilles tendinopathy. To make clinical recommendations for healthcare practitioners and patients. DESIGN: Comprehensive multidisciplinary guideline process funded by the Quality Foundation of the Dutch Federation of Medical Specialists. This process included a development, commentary and authorisation phase. Patients participated in every phase. DATA SOURCES: Multiple databases and existing guidelines were searched up to May 2019. Information from patients, healthcare providers and other stakeholders were obtained using a digital questionnaire, focus group interview and invitational conference. STUDY ELIGIBILITY CRITERIA: Studies on both insertional and/or midportion Achilles tendinopathy were eligible. Specific eligibility criteria were described per module. DATA EXTRACTION AND SYNTHESIS: To appraise the certainty of evidence, reviewers extracted data, assessed risk of bias and used the Grading of Recommendations Assessment, Development and Evaluation method, where applicable. Important considerations were: patient values and preferences, costs, acceptability of other stakeholders and feasibility of implementation. Recommendations were made based on the results of the evidence from the literature and the considerations. PRIMARY OUTCOME MEASURE: The primary and secondary outcome measures were defined per module and defined based on the input of patients obtained in collaboration with the Netherlands Patient Federation and healthcare providers from different professions. RESULTS: Six specific modules were completed: risk factors and primary prevention, diagnosis, imaging, treatment prognosis and secondary prevention for Achilles tendinopathy. SUMMARY/CONCLUSION: Our Dutch multidisciplinary guideline on Achilles tendinopathy provides six modules developed according to the standards of the Dutch Federation of Medical Specialists. Evidence-based recommendations for clinical practice are given for risk factors, prevention, diagnosis, imaging, treatment and prognosis. This guideline can assist healthcare providers and patients in clinical practice.


Subject(s)
Achilles Tendon , Practice Guidelines as Topic , Tendinopathy , Achilles Tendon/physiopathology , Humans , Netherlands , Tendinopathy/diagnosis , Tendinopathy/therapy
3.
Pediatr Phys Ther ; 32(3): 202-209, 2020 07.
Article in English | MEDLINE | ID: mdl-32604360

ABSTRACT

PURPOSE: To examine whether general fatigue and fatigue during or after walking are related to energy demands during walking and physical fitness in children and young adults with physical disabilities. METHODS: Sixty-eight individuals with physical disabilities participated. General fatigue (Checklist Individual Strength [CIS8R] questionnaire), walking-induced fatigue (OMNI [OMNIwalk] scale after walking for 6 min), gross and net energy costs (ECs) of walking, physical strain of walking, and aerobic and anaerobic fitness were measured. RESULTS: Regression analyses showed no relations with the CIS8R. For all participants, a higher net EC was weakly related to an increased OMNIwalk. For teenagers only, low anaerobic fitness and high physical strain of walking values were moderately related to high OMNIwalk scores. CONCLUSION: Low anaerobic fitness and high physical strain values partly explain fatigue after walking in teenagers with cerebral palsy, but not in younger children. General fatigue was not explained by low fitness levels or high energy demands of walking.


Subject(s)
Disabled Persons/psychology , Exercise/physiology , Fatigue/physiopathology , Physical Fitness/physiology , Walking/physiology , Adolescent , Adult , Child , Cross-Sectional Studies , Female , Humans , Male , Netherlands , Regression Analysis , Surveys and Questionnaires , Young Adult
4.
Arch Phys Med Rehabil ; 100(12): 2389-2398, 2019 12.
Article in English | MEDLINE | ID: mdl-31128113

ABSTRACT

OBJECTIVE: To identify existing outcome measurement instruments (OMIs) assessing risk factors for cardiometabolic disease in adolescents and adults with cerebral palsy (CP) reported on in the literature or used in the field. DATA SOURCES: The COnsensus-based Standards for the selection of health Measurement Instruments database of systematic reviews and 4 electronic databases (Embase, MEDLINE/Ovid, MEDLINE/Pubmed, PsychINFO) were searched up to June 19, 2017, that yielded 2594 articles. Experts in the field were consulted to identify any additional OMIs. STUDY SELECTION: Two reviewers independently applied inclusion criteria to select eligible studies using or evaluating measurement properties of OMIs assessing 1 of 8 outcomes: cardiorespiratory endurance, body size, body composition, physical behavior, sleep, nutrition, blood pressure, and blood lipids and glucose. Studies with an experimental or observational design including ≥10 adolescents or adults with CP were included. DATA EXTRACTION: One reviewer extracted data that were summarized for study and sample characteristics, outcomes, OMIs used, and if applicable data on measurement properties. Two reviewers rated the methodological quality and the quality of the OMIs. Feasibility for clinical practice and research was rated by experts in the field. DATA SYNTHESIS: Ninety OMIs were identified from 56 included articles and by the experts. Seventy OMIs pertained to cardiorespiratory endurance, body size, body composition, and physical behavior, whereas only 5 were identified for sleep and nutrition. Overall synthesis revealed that there is moderate to poor evidence for good quality of OMIs in this population. Based on feasibility for clinical practice, experts agreed on a single OMI per outcome (and 2 for cardiorespiratory endurance) to be included in a core set. CONCLUSION: Despite the range of available OMIs to assess risk factors for cardiometabolic disease in adolescents and adults with CP, evidence of good quality is often lacking. Nonetheless, a preliminary core set of 9 OMIs was systematically developed.


Subject(s)
Cardiovascular Diseases/epidemiology , Cerebral Palsy/epidemiology , Metabolic Diseases/epidemiology , Adolescent , Adult , Biomarkers , Blood Glucose , Blood Pressure , Body Weights and Measures , Cardiorespiratory Fitness , Diet , Exercise , Female , Humans , Life Style , Lipids/blood , Male , Middle Aged , Multimorbidity , Risk Factors , Sleep , Surveys and Questionnaires/standards , Young Adult
5.
Spinal Cord ; 56(10): 1008-1016, 2018 Oct.
Article in English | MEDLINE | ID: mdl-29955089

ABSTRACT

STUDY DESIGN: Cross-sectional exploratory study. OBJECTIVES: To determine oxygen uptake (VO2), energy expenditure (EE), and muscle activity (MA) during lying (rest), sitting, standing, and walking among ambulatory individuals with spinal cord Injury (SCI) and to compare VO2, EE, and MA between individuals with different levels of ambulation. SETTING: Rehabilitation institution with a spinal cord injury unit. METHODS: A total of 22 adults with motor-incomplete SCI, ten in a low-ambulation group (non-functional or household walker) and 12 in a high-ambulation group (community or normal walker). VO2 was measured using indirect calorimetry. EE was expressed in metabolic equivalent of task (MET). MA was measured using a wireless surface electromyography device. RESULTS: Mean VO2 was 3.19 ml/kg/min. During lying and sitting, EE was below 1.5 METs for all participants. During standing, three participants of the low-ambulation group and none in the high-ambulation group showed MET values of >1.5. In the walking condition, all participants showed MET values above 1.5. MA during stance was higher compared to the sitting condition and significantly higher in the low-ambulation group compared to the high-ambulation group. CONCLUSION: Lying, supported- and unsupported sitting, without moving, appear to be sedentary behaviors for ambulatory individuals with a motor-incomplete SCI (MET values of <1.5 and a lack of MA). Walking, but not standing, is a moderate physical activity (>1.5 METs), which can be used by all individuals with motor-incomplete SCI to interrupt sedentary behavior.


Subject(s)
Energy Metabolism , Motor Activity/physiology , Muscle, Skeletal/physiopathology , Posture/physiology , Spinal Cord Injuries/physiopathology , Cross-Sectional Studies , Electromyography , Female , Humans , Male , Middle Aged , Oxygen Consumption , Wireless Technology
6.
Arch Phys Med Rehabil ; 98(12): 2507-2513, 2017 12.
Article in English | MEDLINE | ID: mdl-28596080

ABSTRACT

OBJECTIVES: To describe (1) physical strain of walking, (2) the proportion of participants walking above the anaerobic threshold, and (3) 4 phenotypes of physical strain of walking on the basis of deviations in aerobic capacity and walking energy cost (EC) in children and adolescents with cerebral palsy (CP). DESIGN: Cohort study. SETTING: Academic medical center. PARTICIPANTS: A sample (N=57) of participants (n=37; mean age, 13.5±4.0y) with CP (Gross Motor Function Classification System [GMFCS] levels I [n=13], II [n=17], and III [n=7]) and typically developing (TD) participants (n=20; mean age, 11.8±3.5y). INTERVENTIONS: Not applicable. MAIN OUTCOME MEASURES: Oxygen consumption (Vo2walk), speed, and EC were determined during walking at a comfortable speed. Peak oxygen consumption (Vo2peak) and anaerobic threshold were measured during a maximal cycling exercise test. Aerobic capacity was reduced if lower than the 10th percentile, and EC was increased if higher than 3SD. Physical strain was defined as follows: (Vo2walk/Vo2peak)×100. RESULTS: Participants with CP had a higher physical strain (GMFCS level I, 55%±12% GMFCS level II, 62%±17%; GMFCS level III, 78%±14%) than did TD participants (40%±11%) (P<.001). Forty-three percent of participants with CP showed a Vo2walk at or above their anaerobic threshold as compared with 10% of TD participants (P=.007). Phenotypes showed that a reduced Vo2peak (n=9) or an increased EC (n=9) lead to an 18% to 20% higher physical strain, whereas a combination (n=12) leads to a 40% increase. CONCLUSIONS: Children and adolescents with CP walk at a high physical strain, approximating intense exercise and a considerable proportion walks close to or above their anaerobic threshold, probably explaining fatigue and reduced walking distance. Both an increased EC and a reduced Vo2peak contribute to high physical strain in children or adolescents with CP. The different causes of high physical strain in individuals with CP require different intervention strategies.


Subject(s)
Cerebral Palsy/physiopathology , Oxygen Consumption/physiology , Walking Speed/physiology , Adolescent , Anaerobic Threshold/physiology , Child , Exercise Test , Female , Humans , Male
7.
Dev Med Child Neurol ; 58(8): 798-808, 2016 08.
Article in English | MEDLINE | ID: mdl-26853808

ABSTRACT

Physical activity and its promotion, as well as the avoidance of sedentary behaviour, play important roles in health promotion and prevention of lifestyle-related diseases. Guidelines for young people and adults with typical development are available from the World Health Organisation and American College of Sports Medicine. However, detailed recommendations for physical activity and sedentary behaviour have not been established for children, adolescents, and adults with cerebral palsy (CP). This paper presents the first CP-specific physical activity and exercise recommendations. The recommendations are based on (1) a comprehensive review and analysis of the literature, (2) expert opinion, and (3) extensive clinical experience. The evidence supporting these recommendations is based on randomized controlled trials and observational studies involving children, adolescents, and adults with CP, and buttressed by the previous guidelines for the general population. These recommendations may be used to guide healthcare providers on exercise and daily physical activity prescription for individuals with CP.


Subject(s)
Cerebral Palsy/physiopathology , Cerebral Palsy/rehabilitation , Exercise Therapy , Exercise/physiology , Adolescent , Child , Female , Humans , Male
8.
Dev Med Child Neurol ; 57(7): 660-667, 2015 Jul.
Article in English | MEDLINE | ID: mdl-25582163

ABSTRACT

AIM: The aim of this study was to determine the longitudinal associations among fitness components and between fitness and mobility capacity in children with cerebral palsy (CP). METHOD: Forty-six children (26 males, 20 females; mean age 9y 7mo [SD 1y 8mo]) with a bilateral (n=24) or a unilateral spastic CP (n=22) participated in aerobic and anaerobic fitness measurements on a cycle ergometer and isometric muscle strength tests (Gross Motor Function Classification System [GMFCS] level I [n=26], level II [n=12], level III [n=8]). Mobility capacity was assessed with the gross motor function measure (GMFM) and a walking capacity test. Associations over longitudinal measurements (three or four measurements over 1y) were determined since longitudinal data allow a more accurate estimation. The associations were determined using a mixed model with fixed effects (mobility capacity as dependent variables and fitness components as independent variables) and a random intercept. RESULTS: In children with bilateral CP, changes in aerobic fitness were associated with changes in anaerobic fitness (p<0.001), and changes in aerobic fitness showed an association with changes in muscle strength (p<0.05). Anaerobic fitness was not associated with muscle strength. No associations between fitness components were found in unilateral CP. Anaerobic fitness and muscle strength were significant determinants for GMFM and walking capacity in bilateral but not in unilateral CP. INTERPRETATION: The longitudinal associations between aerobic and anaerobic fitness and mobility indicate that increasing either aerobic or anaerobic fitness is associated with improvements in mobility in children with bilateral CP. While increasing anaerobic fitness might be beneficial for mobility capacity in children with bilateral CP, this is less likely for children with unilateral CP.

9.
Clin Rehabil ; 28(10): 972-82, 2014 Oct.
Article in English | MEDLINE | ID: mdl-24047644

ABSTRACT

OBJECTIVE: To determine the effects of a six-month physical activity stimulation programme on social participation, self-perception and quality of life in children with cerebral palsy. DESIGN: Multicentre randomized controlled trial with concealed allocation, blinded assessments and intention-to-treat analysis. SETTING: Paediatric physiotherapy practices, special schools for children with a disability, and the child's own home. SUBJECTS: Forty-nine children with spastic cerebral palsy (28 male), aged 7-13 years, able to walk with and without walking aids. INTERVENTIONS: The intervention group followed a six-month physical activity stimulation programme involving counselling through motivational interviewing, home-based physiotherapy and four months of fitness training. The control group continued regular paediatric physiotherapy. MAIN MEASURES: Outcomes included social participation in domestic life, social participation in recreation and leisure (Life-Habits for Children questionnaire and Children's Assessment of Participation and Enjoyment questionnaire), self-perception (Harter's Self-Perception Profile for Children) and parent-reported quality of life (Cerebral Palsy Quality of Life Questionnaire). Assessments were performed at baseline, at six months (except quality of life) and at twelve months. RESULTS: Intervention resulted in a positive effect on social participation in domestic life at twelve months (mean between-group difference = 0.9, 95% confidence interval (CI) = 0.1 to 1.7 [1-10 scale], P = 0.03), but not at six months. No significant effects were found for social participation in recreation and leisure, self-perception at six months and twelve months or for quality of life at twelve months. CONCLUSIONS: The combination of counselling, home-based physiotherapy and fitness training was not effective in improving social participation in recreation and leisure, self-perception or quality of life, but did show a potential for improving social participation in domestic life over the longer term.


Subject(s)
Cerebral Palsy/rehabilitation , Directive Counseling/methods , Motor Activity , Physical Therapy Modalities , Quality of Life , Self Concept , Social Participation/psychology , Adolescent , Cerebral Palsy/psychology , Child , Female , Humans , Male , Netherlands , Statistics, Nonparametric
10.
Ned Tijdschr Geneeskd ; 1682024 04 17.
Article in Dutch | MEDLINE | ID: mdl-38630094

ABSTRACT

Shoulder dislocations remain the most frequent of joint dislocations, with anterior displacement of the humeral head being the direction of dislocation seen most often (97%). Recently, the Dutch clinical guideline on shoulder dislocations has been revised on the basis of predetermined bottlenecks in clinical practice. In this paper, the guideline is translated to clinical practice by means of two fictional cases, in which the novel recommendations are incorporated. The following topics were systematically assessed based on the best available scientific evidence: primary diagnostics, reduction techniques, painmedication/ sedation surrounding reduction and the need for physiotherapy, stabilization surgery and immobilization. Also, a best practice care pathway is advocated. Since scientific evidence is often inconclusive to provide undebatable therapeutic rules, the committee graded the available evidence and additionally used expert opinion to carefully draft recommendations. The paper concludes with an overview of all the recommendations stated in the updated multidisciplinary guideline.


Subject(s)
Anesthesia , Joint Dislocations , Shoulder Dislocation , Humans , Shoulder , Shoulder Dislocation/therapy , Ethnicity
11.
Arch Phys Med Rehabil ; 94(2): 287-301, 2013 Feb.
Article in English | MEDLINE | ID: mdl-23022091

ABSTRACT

OBJECTIVE: To systematically evaluate the level of evidence of the clinimetric properties of measures of aerobic and anaerobic capacity used for children with cerebral palsy (CP). DATA SOURCES: A systematic search of databases PubMed, Embase, SPORTDiscus, and PsycINFO through April 2011 was performed. STUDY SELECTION: Two independent raters identified and examined studies that reported laboratory- or field-based measures of maximal aerobic or anaerobic capacity in children with CP aged 5 to 14 years. DATA EXTRACTION: The COnsensus-based Standards for the selection of health status Measurement INstruments (COSMIN) checklist was used by 2 independent raters to evaluate the methodologic quality of the included clinimetric studies and to identify measures used in these studies. DATA SYNTHESIS: Twenty-four studies that used a maximal aerobic or anaerobic capacity measure were identified. Five studies reported clinimetric properties for 5 measures (2 aerobic and 3 anaerobic measures). Methodologic quality was excellent in 3 studies, showing good validity and reliability of field-based aerobic (Shuttle Run Test) and anaerobic (Muscle Power Sprint Test) measures. The studies on laboratory-based measures were rated fair, mainly because of inadequate statistics. The level of evidence was strong for good validity and reliability of the field-based tests. The level of evidence was unknown for validity and low to moderate for good reliability of laboratory-based tests. CONCLUSIONS: There is a paucity of research on the clinimetric properties of measurement instruments to assess aerobic and anaerobic capacity for children with CP. Further clinimetric studies of laboratory-based measures in children with CP at all Gross Motor Function Classification System (GMFCS) levels, and clinimetric studies of field-based measures in children who are classified as GMFCS levels III to V are required.


Subject(s)
Cerebral Palsy/physiopathology , Exercise Test , Physical Fitness/physiology , Child , Humans , Oxygen Consumption/physiology
12.
Dev Med Child Neurol ; 54(5): 436-42, 2012 May.
Article in English | MEDLINE | ID: mdl-22414202

ABSTRACT

AIM: To assess ambulatory activity of children with cerebral palsy (CP), aged 7 to 13 years, and identify associated characteristics. METHOD: Sixty-two children with spastic CP (39 males, 23 females; mean age 10y 1mo, SD 1y 8mo; age range 7-13y), classified as Gross Motor Function Classification System (GMFCS) levels I to III, participated. Ambulatory activity was measured during 1 week with a StepWatch activity monitor as steps per day, and time spent at medium and high step rates. Multiple linear regression analyses were performed following a backward selection procedure until only independent variables with p<0.05 remained in the model. Ambulatory activity outcome parameters served as dependent variables, and disease, personal, and environmental characteristics as independent variables. Ambulatory activity was corrected for body height. RESULTS: Children took more steps during school days (5169 steps, SD 1641) than during weekend days (4158 steps, SD 2048; p<0.001). Higher GMFCS level, bilateral CP, and higher age were associated with lower ambulatory activity on school days (R(2) ranged from 43-53%), whereas bilateral CP, higher age, and no sport club participation were associated with lower ambulatory activity in the weekend (R(2) ranged from 21-42%). Correcting for body height decreased the association with age. INTERPRETATION: Interventions should focus at increasing physical activity at the weekend for children with bilateral spastic CP.


Subject(s)
Cerebral Palsy/rehabilitation , Disability Evaluation , Walking , Adolescent , Body Height , Cerebral Palsy/diagnosis , Child , Cross-Sectional Studies , Education, Special , Female , Fitness Centers , Humans , Male , Motor Activity , Physical Therapy Modalities , Reference Values , Resistance Training , Social Environment
14.
J Phys Act Health ; 16(10): 894-901, 2019 10 01.
Article in English | MEDLINE | ID: mdl-31382243

ABSTRACT

BACKGROUND: It is questionable whether postures that are regarded as sedentary behavior in able-bodied persons evoke comparable physiological responses in adults with stroke or cerebral palsy (CP). This study aimed to compare metabolic demand and muscle activity in healthy controls, adults with stroke, and adults with CP during sedentary behavior and light physical activities. METHODS: Seventy-one adults (45.6 [18.9] y, range 18-86) participated in this study, of which there were 18 controls, 31 with stroke, and 22 with CP. The metabolic equivalent of task (MET) and level of muscle activation were assessed for different sedentary positions (sitting supported and unsupported) and light physical activities (standing and walking). RESULTS: During sitting supported and unsupported, people with mild to moderate stroke and CP show comparable MET and electromyographic values as controls. While sitting unsupported, people with severe stroke show higher METs and electromyographic values (P < .001), and people with severe CP only show higher METs compared with controls (P < .05) but all below 1.5 METs. Standing increased electromyographic values in people with severe stroke or CP (P < .001) and reached values above 1.5 METs. CONCLUSIONS: Physiologic responses during sedentary behavior are comparable for controls and adults with mild to moderate stroke and CP, whereas higher metabolic demands and muscle activity (stroke only) were observed in severely affected individuals.


Subject(s)
Cerebral Palsy/physiopathology , Energy Metabolism/physiology , Exercise/physiology , Motor Activity/physiology , Muscle, Skeletal/physiology , Posture/physiology , Sedentary Behavior , Stroke/physiopathology , Walking/physiology , Adolescent , Adult , Aged , Aged, 80 and over , Case-Control Studies , Cerebral Palsy/complications , Disabled Persons , Female , Humans , Male , Middle Aged , Sitting Position , Standing Position , Stroke/complications , Young Adult
15.
Trials ; 20(1): 176, 2019 Mar 19.
Article in English | MEDLINE | ID: mdl-30890152

ABSTRACT

BACKGROUND: Estimates of multimorbidity, defined as the presence of at least two chronic conditions, some of which attributable to modifiable behaviours, are high in adults with cerebral palsy (CP). An assessment protocol evaluating multimorbidity risk is needed in order to develop and evaluate effective interventions to optimize lifelong health in individuals with CP. The aim of this protocol paper is to describe the development of a core outcome set (COS) for assessing multimorbidity risk in adolescents and adults with CP, to be used in clinic and research. METHODS: The expert consortium will first define the target population and outcomes to be measured. Through a process of literature review and an international Delphi survey with expert clinicians and researchers, we will then determine which outcome measurement instruments (OMIs) can best measure those outcomes. The resulting OMIs will be used in a feasibility study with adolescents and adults with CP from an international clinical research network. Finally, a face-to-face stakeholder meeting with adolescents and adults with CP, their families/caregivers and researchers and clinicians who are experts in CP, will be organized to reach final agreement on the COS. DISCUSSION: This COS will guide clinicians and researchers in assessing multimorbidity risk in adolescents and adults with CP. The inclusion of experts and individuals with CP from international locations for establishing the COS lends strong support to its generalizability. Evidence of its feasibility and approval from all stakeholders will enable implementation in clinical practice, and guide future research using the COS in individuals with CP.


Subject(s)
Cerebral Palsy/therapy , Multimorbidity , Outcome Assessment, Health Care , Risk Assessment , Adolescent , Adult , Biomedical Research , Delphi Technique , Humans , Sample Size
16.
Front Pediatr ; 6: 75, 2018.
Article in English | MEDLINE | ID: mdl-29632853

ABSTRACT

OBJECTIVE: To investigate the effects of a school-based once-a-week sports program on physical fitness, physical activity, and cardiometabolic health in children and adolescents with a physical disability. METHODS: This controlled clinical trial included 71 children and adolescents from four schools for special education [mean age 13.7 (2.9) years, range 8-19, 55% boys]. Participants had various chronic health conditions including cerebral palsy (37%), other neuromuscular (44%), metabolic (8%), musculoskeletal (7%), and cardiovascular (4%) disorders. Before recruitment and based on the presence of school-based sports, schools were assigned as sport or control group. School-based sports were initiated and provided by motivated experienced physical educators. The sport group (n = 31) participated in a once-a-week school-based sports program for 6 months, which included team sports. The control group (n = 40) followed the regular curriculum. Anaerobic performance was assessed by the Muscle Power Sprint Test. Secondary outcome measures included aerobic performance, VO2 peak, strength, physical activity, blood pressure, arterial stiffness, body composition, and the metabolic profile. RESULTS: A significant improvement of 16% in favor of the sport group was found for anaerobic performance (p = 0.003). In addition, the sport group lost 2.8% more fat mass compared to the control group (p = 0.007). No changes were found for aerobic performance, VO2 peak, physical activity, blood pressure, arterial stiffness, and the metabolic profile. CONCLUSION: Anaerobic performance and fat mass improved following a school-based sports program. These effects are promising for long-term fitness and health promotion, because sports sessions at school eliminate certain barriers for sports participation and adding a once-a-week sports session showed already positive effects for 6 months. CLINICAL TRIAL REGISTRATION: This trial was registered with the Dutch Trial Registry (NTR4698).

17.
JIMD Rep ; 42: 99-103, 2018.
Article in English | MEDLINE | ID: mdl-29380259

ABSTRACT

BACKGROUND: Biallelic mutations in DNAJC12 were recently identified as a BH4-responsive cause of hyperphenylalaninemia (HPA). Outcome was only favorable when treatment was initiated early in life. We report on a 15-year-old boy with HPA due to a homozygous deletion in DNAJC12 in whom - despite his advanced age - treatment was initiated. CASE: A boy with developmental delay, an extrapyramidal movement disorder, and persistently elevated plasma phenylalanine levels was diagnosed with DNAJC12 deficiency at the age of 15 years. Diagnosis was made upon exome reanalysis revealing a homozygous 6.9 kb deletion in DNAJC12 which had not been detected by the standard exome analysis pipeline. Treatment with the BH4 analog sapropterin dihydrochloride (10 mg/kg/day) was initiated and evoked a 50% reduction of the plasma phenylalanine levels. More strikingly, a marked improvement in daily functioning and improved exercise tolerance was noted. Additionally, gait analysis before and after treatment initiation revealed a partial normalization of his movement disorder. CONCLUSION: Patients with hyperphenylalaninemia due to DNAJC12 deficiency may benefit from treatment with a BH4 analog - even when introduced at a later age.

18.
Gait Posture ; 54: 119-126, 2017 05.
Article in English | MEDLINE | ID: mdl-28288332

ABSTRACT

AIM: This cross-sectional study into children and young adults with cerebral palsy (CP) aimed to assess the association of gross energy cost (EC), net EC and net nondimensional (NN) EC during walking with age and body height, compared to typically developing (TD) peers. METHOD: Data was collected in 128 participants with CP (mean age 11y9mo; GMFCS I,n=48; II,n=56; III, n=24) and in 63 TD peers (mean age 12y5mo). Energy cost was assessed by measuring the oxygen consumption during over-ground walking at comfortable speed. Outcome measures derived from the assessment included the gross and net EC, and NN EC. Differences between the groups in the association between gross, net and NN EC with age and body height, were investigated with regression analyses and interaction effects (p<0.05). RESULTS: Interaction effects for age and body height by group were not significant, indicating similar associations for gross, net and NN EC with age or body height among groups. The models showed a significant decline for gross, net and NN EC with increasing age per year (respectively -0.201Jkg-1m-1; -0.073Jkg-1m-1; -0.007) and body height per cm (respectively -0.057Jkg-1m-1; -0.021Jkg-1m-1; -0.002). INTERPRETATION: Despite higher gross and net EC values for CP compared to TD participants , similar declines in EC outcomes can be expected with growth for participants aged 4-22 years with CP. All energy cost outcomes showed a decline with growth, indicating that correcting for this decline is required when evaluating changes in gross EC, and, to a lesser extent, in net and NN EC in response to treatment or from natural course over time.


Subject(s)
Body Height/physiology , Cerebral Palsy/physiopathology , Energy Metabolism/physiology , Walking/physiology , Adolescent , Adult , Age Factors , Child , Child, Preschool , Cross-Sectional Studies , Female , Humans , Male , Oxygen Consumption/physiology , Regression Analysis , Young Adult
19.
Phys Ther ; 95(7): 996-1005, 2015 Jul.
Article in English | MEDLINE | ID: mdl-25655878

ABSTRACT

BACKGROUND: A vicious circle of decreased physical fitness, early fatigue, and low physical activity levels (PAL) is thought to affect children with cerebral palsy (CP). However, the relationship of changes in physical fitness to changes in PAL and fatigue is unclear. OBJECTIVE: The objective of this study was to investigate the associations among changes in physical fitness, walking-related PAL, and fatigue in children with CP. DESIGN: This study was a secondary analysis of a randomized controlled trial with measurements at baseline, 6 months (after the intervention period), and 12 months. METHODS: Twenty-four children with bilateral spastic CP and 22 with unilateral spastic CP, aged 7 to 13 years, all walking, participated in this study. Physical fitness was measured by aerobic capacity, anaerobic threshold, anaerobic capacity, and isometric and functional muscle strength. Walking-related PAL was measured using an ankle-worn activity monitor for 1 week. Fatigue was determined with the Pediatric Quality of Life (PedsQL) Multidimensional Fatigue Scale. Longitudinal associations were analyzed by random coefficient regression analysis. RESULTS: In children with bilateral CP, all fitness parameters showed a positive, significant association with walking-related PAL, whereas no associations between physical fitness and walking-related PAL were seen in children with unilateral CP. No clinically relevant association between physical fitness and fatigue was found. LIMITATIONS: Although random coefficient regression analysis can be used to investigate longitudinal associations between parameters, a causal relationship cannot be determined. The actual direction of the association between physical fitness and walking-related PAL, therefore, remains inconclusive. CONCLUSIONS: Children with bilateral spastic CP might benefit from improved physical fitness to increase their PAL or vice versa, although this is not the case in children with unilateral CP. There appears to be no relationship between physical fitness and self-reported fatigue in children with CP. Interventions aimed at improving PAL may be differently targeted in children with either bilateral or unilateral CP.


Subject(s)
Cerebral Palsy/physiopathology , Fatigue/physiopathology , Physical Endurance/physiology , Physical Fitness/physiology , Walking/physiology , Adolescent , Cerebral Palsy/complications , Cerebral Palsy/rehabilitation , Child , Exercise Therapy , Fatigue/etiology , Female , Humans , Longitudinal Studies , Male , Muscle Strength/physiology , Quality of Life
20.
Res Dev Disabil ; 45-46: 316-28, 2015.
Article in English | MEDLINE | ID: mdl-26296079

ABSTRACT

OBJECTIVE: To analyze the clinimetric properties of maximal aerobic and anaerobic fitness measurement protocols in adults with cerebral palsy (CP). DATA SOURCES: A systematic search through March 2015 of databases PubMed, Embase, SPORTDiscus and PsycINFO was performed with medical subject heading terms for 'cerebral palsy' combined with search terms adults or adolescents and multiple text words for fitness and exercise tests that yielded 864 articles. STUDY SELECTION: Abstracts were screened by two reviewers to identify use of maximal fitness measurements in adolescents (14-18yrs) or adults (>18yrs) with CP of all abilities. Ninety-four articles were reviewed. No studies of adolescent (14-18yrs) qualified. Eight articles reported clinimetric properties for adults with CP who walk or propel a wheelchair independently. Five articles reported on aerobic capacity, one reported on anaerobic capacity and two reported on both. DATA EXTRACTION: Methodological quality of the studies was rated using portions of the COSMIN (COnsensus-based Standards for the selection of health status Measurement INstruments) checklist. Quality of the measurement protocols was evaluated based on statistical strength of the clinimetrics. Synthesis of the overall evidence was based on the Cochrane review group guidelines which combine methodological quality and statistical strength. DATA SYNTHESIS: Eight articles reported on 4 aerobic and 1 anaerobic protocols. Overall synthesis revealed that for ambulatory adults with CP there is (i) moderate evidence for good reliability and good construct validity of maximal aerobic and anaerobic cycle tests, (ii) moderate evidence for good criterion validity of sub-maximal aerobic cycle tests, and (iii) strong evidence for poor criterion validity of the six-minute walk test as a maximal aerobic test. And for adults who propel a wheelchair there is limited evidence of good reliability for maximal aerobic wheelchair ergometer tests. CONCLUSIONS: Limited quality research exists on the clinimetric properties of aerobic and anaerobic capacity measures for adults with CP who have independent mobility. Quality aerobic and anaerobic measures for adults with more severe mobility impairments are absent.


Subject(s)
Anaerobic Threshold/physiology , Cerebral Palsy/physiopathology , Exercise Test , Exercise Tolerance/physiology , Physical Fitness/physiology , Adolescent , Adult , Humans , Oxygen Consumption/physiology , Physical Endurance/physiology , Reproducibility of Results , Young Adult
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