Shared decision-making with older adults with cancer: Adaptation of a model through literature review and expert opinion.

OBJECTIVE: To provide a literature overview of characteristics of Shared Decision Making (SDM) with specific importance to the older adult population with cancer and to tailor an existing model of SDM in patients with cancer to the needs of older adults. METHODS: A systematic search of several databases was conducted. Eligible studies described factors influencing SDM concerning cancer treatment with adults aged 65 years or above, with any type of cancer. We included qualitative or mixed-methods studies. Themes were identified and discussed in an expert panel, including a patient-representative, until consensus was reached on an adjusted model. RESULTS: Overall 29 studies were included and nine themes were identified from the literature. The themes related to the importance of goal setting, need for tailored information provision, the role of significant others, uncertainty of evidence, the importance of time during and outside of consultations, the possible ill-informed preconceptions that health care professionals (HCPs) might have about older adults and the specific competencies they need to engage in the SDM process with older adults. No new themes emerged from discussion with expert panel. This study presents a visual model of SDM with older patients with cancer based on the identified themes. CONCLUSIONS: Our model shows key elements that are specific to SDM with older adults. Further research needs to focus on how to educate HCPs on the competencies needed to engage in SDM with older patients, and how to implement the model into everyday practice.

Development of the national Dutch PEWS: the challenge against heterogeneity and implementation difficulties of PEWS in the Netherlands.

BACKGROUND: For the early recognition of deteriorating patients several Pediatric Early Warning Score (PEWS) systems have been developed with the assumption that early detection can prevent further deterioration. Although PEWS are widely being used in hospitals in the Netherlands, there is no national consensus on which score to use and how to embed the score into a PEWS system. This resulted in a substantial heterogeneity of PEWS systems, of which many are unvalidated or self-designed. The primary objective of this study was to develop a pragmatic consensus-based PEWS system that can be utilized in all Dutch hospitals (University Medical Centers, teaching hospitals, and general hospitals). METHODS: This study is an iterative mixed-methods study. The methods from the Core Outcome Measures in Effectiveness Trials (COMET) initiative were used and consisted of two Delphi rounds, two inventories set out to all Dutch hospitals and a focus group session with parents. The study was guided by five expert meetings with different stakeholders and a final consensus meeting that resulted in a core PEWS set. RESULTS: The first Delphi round was completed by 292 healthcare professionals, consisting of pediatric nurses and physicians. In the second Delphi round 217 healthcare professionals participated. Eventually, the core PEWS set was been developed comprising of the parameters work of breathing, respiratory rate, oxygen therapy, heart rate and capillary refill time, and AVPU (Alert, Verbal, Pain, and Unresponsive). In addition, risk stratification was added to the core set with standardized risk factors consisting of [1] worried signs from healthcare professionals and parents and [2] high-risk treatment, with the option to add applicable local defined risk factors. Lastly, the three categories of risk stratification were defined (standard, medium, and high risk) in combination with standardized actions of the professionals for each category. CONCLUSION: This study demonstrates a way to end a country's struggle with PEWS heterogeneity by co-designing a national Dutch PEWS system. Currently, the power of the system is being investigated in a large multi-center study in the Netherlands.

Clusters of medical specialties around patients with multimorbidity - employing fuzzy c-means clustering to explore multidisciplinary collaboration.

BACKGROUND: Hospital care organization, structured around medical specialties and focused on the separate treatment of individual organ systems, is challenged by the increasing prevalence of multimorbidity. To support the hospitals' realization of multidisciplinary care, we hypothesized that using machine learning on clinical data helps to identify groups of medical specialties who are simultaneously involved in hospital care for patients with multimorbidity. METHODS: We conducted a cross-sectional study of patients in a Dutch general hospital and used a fuzzy c-means clustering algorithm for the analysis. We explored the patients' membership degrees in each cluster to identify subgroups of medical specialties that provide care to the same patients with multimorbidity. We used retrospectively collected electronic health record data from 2017. We extracted data from 22,133 patients aged ≥18 years who had received outpatient clinical care for two or more chronic and/ or oncological diagnoses. RESULTS: We found six clusters of medical specialties and identified 22 subgroups. The clusters were labeled based on the specialties that most characterized them: 1. dermatology/ plastic surgery, 2. six specialties (gynecology/ rheumatology/ orthopedic surgery/ urology/ gastroenterology/ otorhinolaryngology), 3. pulmonology, 4. internal medicine/ cardiology/ geriatrics, 5. neurology/ physiatry (rehabilitation)/ anesthesiology, and 6. internal medicine. Most patients had a full or dominant membership to one of these clusters of medical specialties (11 subgroups), whereas fewer patients had a membership to two clusters. The prevalence of specific diagnosis groups, patient characteristics, and healthcare utilization differed between subgroups. CONCLUSION: Our study shows that clusters and subgroups of medical specialties simultaneously involved in hospital care for patients with multimorbidity can be identified with fuzzy c-means cluster analysis using clinical data. Clusters and subgroups differed regarding the involved medical specialties, diagnoses, patient characteristics, and healthcare utilization. With this strategy, hospitals and medical specialists can further analyze which subgroups are target populations that might benefit from improved multidisciplinary collaboration.

Sports Participation, Physical Activity, and Health-Related Fitness in Youth With Chronic Diseases or Physical Disabilities: The Health in Adapted Youth Sports Study.

ABSTRACT: Lankhorst, K, Takken, T, Zwinkels, M, van Gaalen, L, Velde, St, Backx, F, Verschuren, O, Wittink, H, and de Groot, J. Sports participation, physical activity, and health-related fitness in youth with chronic diseases or physical disabilities: the health in adapted youth sports study. J Strength Cond Res 35(8): 2327-2337, 2021-Youth with chronic diseases or physical disabilities (CDPD) often show reduced fitness and physical activity (PA) levels and participate less in organized sports compared with healthy peers. The purpose of this study was to examine the associations between participation in sports and health-related fitness and PA in youth with CDPD. A total of 163 subjects (mean age 14 years; range 8-19 years) with CDPD were included in this cross-sectional study, with 81 participating in organized sports and 82 not. Subjects were recruited between October 2014 and November 2016. Aerobic and anaerobic fitness, agility, and muscle strength were assessed in the laboratory, whereas PA was monitored in daily life using accelerometry during 1 week. Linear regression analyses were used to assess the associations of sports participation (independent variable) with health-related fitness and PA (dependent variables). Results show that youth with CDPD participating in organized sports 2 times a week performed better on all outcome measures. They reached a higher peak oxygen uptake (difference of 4.9 ml O2·kg-1·min-1, P = 0.001) compared with their peers not participating in sports. Also, anaerobic fitness, agility, muscle strength, and PA were all positively associated with sports participation. Moreover, the association between sports participation and aerobic fitness was mediated by PA for 31% (P = 0.045). In conclusion, participation in sports is associated with both higher levels of PA and health-related fitness in youth with CDPD. Promotion and stimulation of participation in sports seems a good way to promote health-related fitness as well as a healthy active lifestyle in youth with CDPD.

The Preliminary Criterion Validity of the Activ8 Activity Monitor to Measure Physical Activity in Youth Using a Wheelchair.

PURPOSE: The aim of this study was to assess the criterion validity of the Activ8 to detect "active wheelchair use" and to distinguish 6 types of wheelchair activities using video recordings as a gold standard. METHODS: Ten participants who use a manual wheelchair were participants in this study. Data of 2 participants were used for adapting the algorithm and data of 7 participants were used for assessing the criterion validity. RESULTS: Criterion validity for detecting "active wheelchair use" had a relative time difference of 7.4%, agreement of 96%, sensitivity of 98.3%, and positive predictive value of 90%. Results for distinguishing 6 types of wheelchair activities had an agreement of 73%, sensitivity of 67.1%, and positive predictive value of 65.5%. CONCLUSIONS: The Activ8 is able to detect "active wheelchair use" in youth using a manual wheelchair. Further development of the algorithm is necessary to distinguish between different types of wheelchair activities.

Instruments Measuring Physical Activity in Individuals Who Use a Wheelchair: A Systematic Review of Measurement Properties.

OBJECTIVE: To systematically review the evidence evaluating validity or reliability of self-reported and device-based instruments, to measure physical activity (PA) in individuals who use a wheelchair, and to make recommendations for the selection of PA outcomes tools. DATA SOURCES: PubMed, Embase, and CINAHL were systematically searched. STUDY SELECTION: Studies reporting measurement properties of instruments to assess PA in individuals who use a wheelchair. DATA EXTRACTION: The Consensus-Based Standards for the Selection of Health Status Measurement Instruments checklist was used to assess the methodological quality of the included studies. The measurement properties of instruments assessing PA were examined. DATA SYNTHESIS: The search yielded 5341 records, 61 were considered relevant, 21 articles were included. A best evidence synthesis was performed on 9 studies including 4 self-reported instruments and 13 studies including 8 device-based instruments. One study evaluated both self-reported and device-based instruments. The overall methodological quality of all studies ranged from poor to excellent. Variable levels of evidence were found for both the validity and reliability for self-reported instruments and for criterion validity for device-based instruments. CONCLUSIONS: The Physical Activity Scale for Individuals with Disabilities (PASIPD) and The Physical Activity Recall Assessment for People with Spinal Cord Injury (PARA-SCI) seem the most promising self-reported instruments for measuring the intensity of PA. Device-based instruments that can be used for measuring both the intensity and type of PA are the GENEActiv, Actigraph GT3X+, Actiheart, or the Physical Activity Monitor System (PAMS), showing moderate evidence for a positive rating of criterion validity. For measuring the type of PA, the PAMS and VitaMove are suitable, showing both good evidence for a positive rating of criterion validity.

Sleep quantity and its relation with physical activity in children with cerebral palsy; insights using actigraphy.

AIM: To objectively assess the sleep quantity, and explore the relationships between sleep quantity and quality, and physical activity and sedentary behaviour in children and adolescents with cerebral palsy (CP). METHODS: An observational cross-sectional study was conducted. In total, 36 children with spastic CP (mean age 15y 4mo, SD 2y 6mo; classified as Gross Motor Function Classification System levels I (25), II (9), III (1) and IV (1)) were included. Active time, sedentary time and sleep quantity were measured using an activity monitor for 7 consecutive days. RESULTS: Total sleep duration of children with CP ranged between 7.2 and 11.2 h. No significant correlations were found between active time and sleep quantity for total week, weekdays, and weekend days. Moderate negative correlations were found between sedentary time and sleep quantity during total week (r = -0.456, P = 0.005), weekdays (r = -0.453, P = 0.006) and weekend days (r = -0.48, P = 0.003). CONCLUSIONS: Our findings suggest that children with CP are getting the recommended sleep duration, and that sedentary behaviour is correlated with sleep quantity in children with CP and may be more applicable to children with better motor functions. Future studies using more elaborate, objective sleep quantity and quality measures are recommended.

Assessment of fatigability in patients with spinal muscular atrophy: development and content validity of a set of endurance tests.

BACKGROUND: Fatigability has emerged as an important dimension of physical impairment in patients with Spinal Muscular Atrophy (SMA). At present reliable and valid outcome measures for both mildly and severely affected patients are lacking. Therefore the primary aim of this study is the development of clinical outcome measures for fatigability in patients with SMA across the range of severity. METHODS: We developed a set of endurance tests using five methodological steps as recommended by the 'COnsensus-based Standards for the selection of health Measurement INstruments (COSMIN). In this iterative process, data from multiple sources were triangulated including a scoping review of scientific literature, input from a scientific and clinical multidisciplinary expert panel and three pilot studies including healthy persons (N = 9), paediatric patients with chronic disorders (N = 10) and patients with SMA (N = 15). RESULTS: Fatigability in SMA was operationalised as the decline in physical performance. The following test criteria were established; one method of testing for patients with SMA type 2-4, a set of outcome measures that mimic daily life activities, a submaximal test protocol of repetitive activities over a longer period; external regulation of pace. The scoping review did not generate suitable outcome measures. We therefore adapted the Endurance Shuttle Walk Test for ambulatory patients and developed the Endurance Shuttle Box and Block Test and the - Nine Hole Peg Test for fatigability testing of proximal and distal arm function. Content validity was established through input from experts and patients. Pilot testing showed that the set of endurance tests are comprehensible, feasible and meet all predefined test criteria. CONCLUSIONS: The development of this comprehensive set of endurance tests is a pivotal step to address fatigability in patients with SMA.

Physical activity and sedentary behaviour in children with spina bifida.

AIM: To describe active and sedentary time in children with spina bifida and to compare their physical activity on weekdays versus weekends. METHOD: In this exploratory cross-sectional study, data from 13 Canadian and 22 Dutch children with spina bifida (14 females, 21 males; mean age 10y 11mo, standard deviation [SD] 3y 6mo, range 5y 6mo-18y; Hoffer classification distribution: community [n=28], household [n=3], non-functional [n=3], and non-ambulator [n=1]) were analysed. Objective measures of physical activity and sedentary behaviour were obtained by using ActiGraph or Actiheart activity monitors. Data for the participants wearing the ActiGraph were compared with age- and sex-matched controls that were developing typically using independent-samples t-tests. Activity data collected on weekdays was compared to those on weekends. RESULTS: ActiGraph data demonstrated children with spina bifida spent more time sedentary (mean [SD] 49.5min/h [5.78]) and less time in moderate to vigorous physical activity (mean [SD] 2.33min/h [1.61]) compared with the typically developing group (mean [SD] 41.0min/h [5.76] and 5.46min/h [2.13], p=0.001 and p<0.001 respectively). For both ActiGraph- and Actiheart-derived data, physical activity and sedentary time were not significantly different between weekdays and weekends. INTERPRETATION: Children with spina bifida have reduced levels of physical activity and increased sedentary behaviour, with no statistical differences seen between weekdays and weekends. Several methodological issues related to activity monitoring warrant consideration when choosing the appropriate method to quantify physical activity and sedentary behaviour. WHAT THIS PAPER ADDS: Reduced levels of physical activity and sedentary time were quantified in children with spina bifida. Objective quantification of physical behaviour in ambulatory and non-ambulatory school-aged children with spina bifida is possible.

ACTIVIDAD FÍSICA Y COMPORTAMIENTO SEDENTARIO EN NIÑOS CON ESPINA BÍFIDA: OBJETIVO: Describir el tiempo activo y sedentario en niños con espina bífida y comparar su actividad física entre semana y fines de semana. MÉTODO: En este estudio exploratorio de corte transversal, datos de 13 niños canadienses y 22 holandeses con espina bífida (14 mujeres, 21 varones; edad media 10 años 11 meses, desviación estándar [DE] 3 años 6 meses, rango 5 años 6 meses - 18 años; se analizaron en base a la distribución basada en la clasificación de Hoffer: comunidad [n = 28], hogar [n = 3], no funcional [n = 3] y no ambulatorios [n = 1]). Se obtuvieron medidas objetivas de actividad física y comportamiento sedentario mediante el uso de monitores de actividad ActiGraph o Actiheart. Los datos de los participantes que usaban el ActiGraph se compararon con los niños con desarrollo típicos como controles, apareados por edad y sexo usando pruebas t de muestras independientes. Los datos de actividad recopilados durante la semana se compararon con los de los fines de semana. RESULTADOS: Los datos de ActiGraph demostraron que los niños con espina bífida pasaron más tiempo sedentarios (media [DE] 49,5min / h [5,78]) y menos tiempo en actividades físicas moderadas a intensas (media [DE] 2,33min / h [1,61]) en comparación con el grupo control (media [DE] 41,0min / h [5,76] y 5,46min / h [2,13], p = 0,001 y p <0,001 respectivamente). Tanto para los datos derivados de ActiGraph como de Actiheart, la actividad física y el tiempo sedentario no fueron significativamente diferentes entre semana y fines de semana. INTERPRETACIÓN: Los niños con espina bífida tienen niveles reducidos de actividad física y un mayor comportamiento sedentario, sin diferencias estadísticas observadas entre semana y fines de semana. Al elegir un método apropiado para cuantificar la actividad física y el comportamiento sedentario se deben considerar varios problemas metodológicos relacionados con el monitoreo de la actividad en esta población.

ATIVIDADE FÍSICA E COMPORTAMENTO SEDENTÁRIO EM CRIANÇAS COM ESPINHA BÍFIDA: OBJETIVO: Descrever o tempo ativo e sedentário de crianças com espinha bífida e comparar sua atividade física durante a semana versus fins de semana. MÉTODO: Neste estudo exploratório transversal, dados de 13 crianças canadenses e 22 crianças holandesas com espinha bífida (14 do sexo feminino, 21 do sexo masculino; médida de idade 10a 11m, desvio padrão [DP] 3a 6m, variação 5a 6m-18a; Distribuição da classificação de Hoffer: comunidade [n=28], domiciliar [n=3], não funcional [n=3], e não ambulatório [n=1]) foram analisados. Medidas objetivas de atividade física e comportamento sedentário foram obtidas por meio de monitores de atividade ActiGraph ou Actiheart. Dados para os participantes que usaram ActiGraph foram comparados com controles pareados por idade e sexo que tinha desenvolvimento típico usando testes t para amostras independentes. Dados de atividade coletados durante a semana foram comparados com os dados de finais de semana. RESULTADOS: Os dados do ActiGraph demonstraram que crianças com espinha bífida passaram mais tempo sedentário (média [DP] 49,5min/h [5,78]) e menos tempo em atividade física vigorosa (média [DP] 2,33min/h [1,61]) comparados com o grupo com desenvolvimento típico (média [DP] 41,0min/h [5,76] e 5,46min/h [2,13], p=0,001 and p<0,001 respectivamente). Para os dados derivados do ActiGraph- e Actiheart, a atividade física e o tempo sedentário não foram significativamente diferentes comparando os dias da semana com os fins de semana. INTERPRETAÇÃO: Crianças com espinha bífida têm níveis reduzidos de atividade física e comportamento sedentário aumentado, sem diferenças estatísticas quando comparados os dias da semana. Várias questões metodológicas relacionadas ao monitoramento de atividades indicam necessidade de refletir quanto à escolha do método apropriado para quantificar a ativiade física e comportamento sedentário.

Physical exercise training for type 3 spinal muscular atrophy.

BACKGROUND: Physical exercise training might improve muscle and cardiorespiratory function in spinal muscular atrophy (SMA). Optimization of aerobic capacity or other resources in residual muscle tissue through exercise may counteract the muscle deterioration that occurs secondary to motor neuron loss and inactivity in SMA. There is currently no evidence synthesis available on physical exercise training in people with SMA type 3. OBJECTIVES: To assess the effects of physical exercise training on functional performance in people with SMA type 3, and to identify any adverse effects. SEARCH METHODS: On 8 May 2018, we searched the Cochrane Neuromuscular Specialised Register, Cochrane Central Register of Controlled Trials, MEDLINE, Embase, CINAHL, AMED, and LILACS. On 25 April 2018 we searched NHSEED, DARE, and ClinicalTrials.gov and WHO ICTRP for ongoing trials. SELECTION CRITERIA: We included randomized controlled trials (RCTs) or quasi-RCTs lasting at least 12 weeks that compared physical exercise training (strength training, aerobic exercise training, or both) to placebo, standard or usual care, or another type of non-physical intervention for SMA type 3. Participants were adults and children from the age of five years with a diagnosis of SMA type 3 (Kugelberg-Welander syndrome), confirmed by genetic analysis. DATA COLLECTION AND ANALYSIS: We used standard Cochrane methodological procedures. MAIN RESULTS: We included one RCT that studied the effects of a six-month, home-based, combined muscle strength and recumbent cycle ergometry training program versus usual care in 14 ambulatory people with SMA. The age range of the participants was between 10 years and 48 years. The study was evaluator-blinded, but personnel and participants could not be blinded to the intervention, which placed the results at a high risk of bias. Participants performed strength training as prescribed, but 50% of the participants did not achieve the intended aerobic exercise training regimen. The trial used change in walking distance on the six-minute walk test as a measure of function; a minimal detectable change is 24.0 m. The change from baseline to six months' follow-up in the training group (9.4 m) was not detectably different from the change in the usual care group (-0.14 m) (mean difference (MD) 9.54 m, 95% confidence interval (CI) -83.04 to 102.12; N = 12). Cardiopulmonary exercise capacity, assessed by the change from baseline to six months' follow-up in peak oxygen uptake (VO2max) was similar in the training group (-0.12 mL/kg/min) and the usual care group (-1.34 mL/kg/min) (MD 1.22 mL/kg/min, 95% CI -2.16 to 4.6; N = 12). A clinically meaningful increase in VO2max is 3.5 mL/kg/min.The trial assessed function on the Hammersmith Functional Motor Scale - Expanded (HFMSE), which has a range of possible scores from 0 to 66, with an increase of 3 or more points indicating clinically meaningful improvement. The HFMSE score in the training group increased by 2 points from baseline to six months' follow-up, with no change in the usual care group (MD 2.00, 95% CI -2.06 to 6.06; N = 12). The training group showed a slight improvement in muscle strength, expressed as the manual muscle testing (MMT) total score, which ranges from 28 (weakest) to 280 (strongest). The change from baseline in MMT total score was 6.8 in the training group compared to -5.14 in the usual care group (MD 11.94, 95% CI -3.44 to 27.32; N = 12).The trial stated that training had no statistically significant effects on fatigue and quality of life. The certainty of evidence for all outcomes was very low because of study limitations and imprecision. The study did not assess the effects of physical exercise training on physical activity levels. No study-related serious adverse events or adverse events leading to withdrawal occurred, but we cannot draw wider conclusions from this very low-certainty evidence. AUTHORS' CONCLUSIONS: It is uncertain whether combined strength and aerobic exercise training is beneficial or harmful in people with SMA type 3, as the quality of evidence is very low. We need well-designed and adequately powered studies using protocols that meet international standards for the development of training interventions, in order to improve our understanding of the exercise response in people with SMA type 3 and eventually develop exercise guidelines for this condition.

Physical activity in wheelchair-using youth with spina bifida: an observational study.

BACKGROUND: Even though typically developing youth are already at risk for physical inactivity, youth with spina bifida may be even at higher risk as a consequence of their reduced mobility. No objective data is available for youth with spina bifida who use a manual wheelchair, so the seriousness of the problem is unknown. The purpose of this observational study was to quantify physical activity in wheelchair-using youth with spina bifida and evaluate the intensity of activities. METHODS: Fifty-three children and adolescents (5-19 years) with spina bifida who use a manual wheelchair for daily life, long distances or sports were included. To assess time spent in several types of activities VitaMove data of 34 participants were used and were presented as time spent sedentary and time spent physically active. This was compared to reference data of typically developing youth. To assess time spent in several intensities Actiheart data of 36 participants were used. The intensities were categorized according to the American College of Sports Medicine, ranging from very light intensity to near to maximal intensity. Data of 25 participants were used to combine type of activity and intensity. RESULTS: Children and adolescents with spina bifida who use a manual wheelchair were more sedentary (94.3% versus 78.0% per 24 h, p < 0.000) and less physically active (5.0% versus 12.2% per 24 h, p < 0.000) compared to typically developing peers. Physical activity during weekend days was worse compared to school days; 19% met the Guidelines of Physical Activity during school days and 8% during weekend days. The intensities per activity varied extensively between participants. CONCLUSIONS: Children and adolescents with spina bifida who use a manual wheelchair are less physically active and more sedentary than typically developing youth. The physical activity levels on school days seem to be more favorable than the physical activity levels on a weekend day. The low levels of physical activity need our attention in pediatric rehabilitation practice. The different intensities during activities indicate the importance of individually tailored assessments and interventions.

Effects of High-Intensity Interval Training on Fitness and Health in Youth With Physical Disabilities.

PURPOSE: To investigate the effects of high-intensity interval training (HIT) on physical fitness and cardiometabolic health in youth with physical disabilities. METHODS: For this quasi-experimental study 70 participants were recruited from schools for special education and divided into runners and users of wheelchairs. HIT was performed for 8 weeks, twice a week, containing 30 seconds all-out exercises. RESULTS: Exercise adherence was 84.5%. Following HIT, there were improvements in anaerobic performance, agility, aerobic performance, and systolic and diastolic blood pressure. There were no changes in peak oxygen uptake ((Equation is included in full-text article.)O2peak), arterial stiffness, body composition, lipid profile, and fasting glucose. CONCLUSIONS: Both anaerobic and aerobic performance improved after HIT, with no changes in (Equation is included in full-text article.)O2peak. There were no effects on cardiometabolic health, except for a decrease in blood pressure.

Evidence for increasing physical activity in children with physical disabilities: a systematic review.

AIM: To summarize the best evidence of interventions for increasing physical activity in children with physical disabilities. METHOD: A systematic review was conducted using an electronic search executed in Academic Search Elite, Academic Search Premier, CINAHL, Embase, MEDLINE, PEDro, PsychINFO, and SPORTDiscus up to February 2016. The selection of articles was performed independently by two researchers according to predetermined eligibility criteria. Data extraction, methodological quality, and levels of evidence were independently assessed by two researchers using a data-collection form from the Cochrane Collaboration and according to the guidelines of the American Academy for Cerebral Palsy and Developmental Medicine. RESULTS: Seven studies were included. Five randomized controlled trials ranged from strong level I to weak level II studies, and two pre-post design studies were classified as level IV. There is level I evidence for no effect of physical training on objectively measured physical activity, conflicting level II evidence for interventions with a behavioural component on the increase of objectively measured physical activity directly after the intervention, and level II evidence for no effect during follow-up. Results are limited to children with cerebral palsy as no other diagnoses were included. INTERPRETATION: Increasing physical activity in children with physical disabilities is very complex and demands further development and research.

Validity and Reliability of Skill-Related Fitness Tests for Wheelchair-Using Youth With Spina Bifida.

OBJECTIVES: To determine content validity of the Muscle Power Sprint Test (MPST), and construct validity and reliability of the MPST, 10×5 Meter Sprint Test (10×5MST), slalom test, and One Stroke Push Test (1SPT) in wheelchair-using youth with spina bifida (SB). DESIGN: Clinimetric study. SETTING: Rehabilitation centers, SB outpatient services, and private practices. PARTICIPANTS: A convenience sample of children and adolescents (N=53; 32 boys, 21 girls; age range, 5-19y) with SB who use a manual wheelchair. Participants were recruited through rehabilitation centers, SB outpatient services, pediatric physical therapists, and the BOSK (Association of Physically Disabled Persons and their Parents). INTERVENTIONS: Not applicable. MAIN OUTCOME MEASURES: Construct validity of the MPST was determined by comparing results with the arm-cranking Wingate Anaerobic Test (WAnT) using paired t tests and Pearson correlation coefficients, while content validity was assessed using time-based criteria for anaerobic testing. Construct validity of the 10×5MST, slalom test, and 1SPT was analyzed by hypothesis testing using Pearson correlation coefficients and multiple regression. For reliability, intraclass correlation coefficients (ICCs) and smallest detectable changes (SDCs) were calculated. RESULTS: For the MPST, the mean ± SD exercise time of 4 sprints was 28.1±6.6 seconds. Correlations between the MPST and arm-cranking WAnT were high (r>.72, P<.01). Excellent correlations were found between the 10×5MST and slalom test (r=.93, P<.01), while correlations between the 10×5MST or slalom test and MPST and 1SPT were moderate (r=-.56 to -.70; r=.56, P<.01). The variation of the 1SPT was explained for 38% by wheelchair mass (ß=-.489) and total upper muscle strength (ß=.420). All ICCs were excellent (ICCs>.95), but the SDCs varied widely. CONCLUSIONS: The MPST is a valid and reliable test in wheelchair-using youth with SB for measuring anaerobic performance. The 10×5MST and slalom test are valid and reliable for measuring agility. For the 1SPT, both validity and reliability are questionable.

Development of a wheelchair mobility skills test for children and adolescents: combining evidence with clinical expertise.

BACKGROUND: Wheelchair mobility skills (WMS) training is regarded by children using a manual wheelchair and their parents as an important factor to improve participation and daily physical activity. Currently, there is no outcome measure available for the evaluation of WMS in children. Several wheelchair mobility outcome measures have been developed for adults, but none of these have been validated in children. Therefore the objective of this study is to develop a WMS outcome measure for children using the current knowledge from literature in combination with the clinical expertise of health care professionals, children and their parents. METHODS: Mixed methods approach. Phase 1: Item identification of WMS items through a systematic review using the 'COnsensus-based Standards for the selection of health Measurement Instruments' (COSMIN) recommendations. Phase 2: Item selection and validation of relevant WMS items for children, using a focus group and interviews with children using a manual wheelchair, their parents and health care professionals. Phase 3: Feasibility of the newly developed Utrecht Pediatric Wheelchair Mobility Skills Test (UP-WMST) through pilot testing. RESULTS: Phase 1: Data analysis and synthesis of nine WMS related outcome measures showed there is no widely used outcome measure with levels of evidence across all measurement properties. However, four outcome measures showed some levels of evidence on reliability and validity for adults. Twenty-two WMS items with the best clinimetric properties were selected for further analysis in phase 2. Phase 2: Fifteen items were deemed as relevant for children, one item needed adaptation and six items were considered not relevant for assessing WMS in children. Phase 3: Two health care professionals administered the UP-WMST in eight children. The instructions of the UP-WMST were clear, but the scoring method of the height difference items needed adaptation. The outdoor items for rolling over soft surface and the side slope item were excluded in the final version of the UP-WMST due to logistic reasons. CONCLUSIONS: The newly developed 15 item UP-WMST is a validated outcome measure which is easy to administer in children using a manual wheelchair. More research regarding reliability, construct validity and responsiveness is warranted before the UP-WMST can be used in practice.

Personal and environmental factors to consider when aiming to improve participation in physical activity in children with Spina Bifida: a qualitative study.

BACKGROUND: Youth with spina bifida (SB) are less fit and active than other groups with childhood disability. While recent studies have shown benefits of exercise training, the increased fitness levels do not sustain or lead to increased levels of physical activity (PA) in these children. Therefore, it seems important to explore which factors are associated with participation in PA (or lack of) in youth with SB. The objective of this study is to describe both personal and environmental factors that are important for participation in physical activity as experienced by these children and their parents, in order to better develop intervention strategies to improve participation in PA in youth with SB. METHODS: Eleven semi-structured interviews with parents of children with SB aged 4-7 years, nine focus groups with youth with SB (n = 33, age 8-18 years) and eight focus groups with their parents (n = 31) were conducted, recorded and transcribed verbatim. Two independent researchers analyzed the data. Central themes for physical activity were constructed, using the model for Physical Activity for Persons with a Disability (PAD model) as a background scheme. RESULTS: Data showed that youth with SB encountered both personal and environmental factors associated with participation in PA on all levels of the PAD model. Bowel and bladder care, competence in skills, sufficient fitness, medical events and self-efficacy were important personal factors. Environmental factors that were associated with physical activity included the contact with and support from other people, the use of assistive devices for mobility and care, adequate information regarding possibilities for adapted sports and accessibility of playgrounds and sports facilities. CONCLUSIONS: Our findings suggest that a variety of both personal and environmental factors were either positively or negatively associated with participation in PA. An individual approach, assessing possibilities rather than overcoming barriers within and surrounding the child may be a good starting point when setting up intervention programs to improve participation in PA. Therefore, assessment of both personal and environmental factors associated with physical activity should be standard care within multidisciplinary intervention programs aimed to encourage healthy active lifestyles in youth with SB.

Factors associated with physical activity in children and adolescents with a physical disability: a systematic review.

AIM: The aim of this review was to summarize the important factors associated with participation in physical activity in children and adolescents with physical disabilities. METHOD: A systematic mixed-studies review was conducted using the databases Academic Search Elite, CINAHL, The Cochrane Library, EMBASE, PEDro, PsycINFO, PubMed, and SPORTDiscus, searching for studies conducted from January 2000 to May 2013. The studies were identified by two independent researchers following predetermined inclusion and exclusion criteria. The methodological quality was determined using the McMaster University critical review forms for qualitative or quantitative research and was numerically rated according to the criteria developed by Imms. RESULTS: The initial electronic search yielded 10 161 articles, of which six were qualitative and 12 were quantitative studies. These studies showed that a diverse range of positive and negative factors were associated with participation in physical activity, such as self-efficacy, physical fitness, increasing age, and the availability of equipment and local facilities. INTERPRETATION: Future intervention studies could use these results, within the context of an individual child and his or her environment, as the basis for increasing physical activity levels, starting in early childhood and continuing throughout adolescence and into adulthood. An increased awareness of and focus on providing appropriate equipment and adapted sports in the child's own environment by policy makers might increase physical activity levels.

Validation of an activity monitor for children who are partly or completely wheelchair-dependent.

BACKGROUND: Children who are wheelchair-dependent are at risk for developing unfavorable physical behavior; therefore, assessment, monitoring and efforts to improve physical behavior should start early in life. VitaMove is an accelerometer-based activity monitor and can be used to detect and distinguish different categories of physical behavior, including activities performed in a wheelchair and activities using the legs. The purpose of this study was to assess the validity of the VitaMove activity monitor to quantify physical behavior in children who are partly or completely wheelchair-dependent. METHODS: Twelve children with spina bifida (SB) or cerebral palsy (CP) (mean age, 14 ± 4 years) performed a series of wheelchair activities (wheelchair protocol) and, if possible, activities using their legs (n = 5, leg protocol). Activities were performed at their own home or school. In children who were completely wheelchair-dependent, VitaMove monitoring consisted of one accelerometer-based recorder attached to the sternum and one to each wrist. For children who were partly ambulatory, an additional recorder was attached to each thigh. Using video-recordings as a reference, primary the total duration of active behavior, including wheeled activity and leg activity, and secondary agreement, sensitivity and specificity scores were determined. RESULTS: Detection of active behaviour with the VitaMove activity monitor showed absolute percentage errors of 6% for the wheelchair protocol and 10% for the leg protocol. For the wheelchair protocol, the mean agreement was 84%, sensitivity was 80% and specificity was 85%. For the leg protocol, the mean agreement was 83%, sensitivity was 78% and specificity was 90%. Validity scores were lower in severely affected children with CP. CONCLUSIONS: The VitaMove activity monitor is a valid device to quantify physical behavior in children who are partly or completely wheelchair-dependent, except for severely affected children and for bicycling.

Cardiopulmonary Exercise Testing in Children and Adolescents With Dystrophinopathies: A Pilot Study.

PURPOSE: To determine exercise response during cardiopulmonary exercise testing in children and adolescents with dystrophinopathies. METHODS: Exercise response on the cardiopulmonary exercise test (CPET) was compared with a standard care test protocol. RESULTS: Nine boys (aged 10.8 ± 4.7 years) with Becker muscular dystrophy (n = 6) and Duchenne muscular dystrophy (n = 3) were included. The feasibility of the CPET was similar to a standard care test protocol, and no serious adverse events occurred. In 67% of the subjects with normal or only mildly impaired functional capacity, the CPET could be used to detect moderate to severe cardiopulmonary exercise limitations. CONCLUSIONS: The CPET seems to be a promising outcome measure for cardiopulmonary exercise limitations in youth with mild functional limitations. Further research with larger samples is warranted to confirm current findings and investigate the additional value of the CPET to longitudinal follow-up of cardiomyopathy and the development of safe exercise programs for youth with dystrophinopathies.