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.

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.

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.

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.

A Grounded Theory of Interdisciplinary Communication and Collaboration in the Outpatient Setting of the Hospital for Patients with Multiple Long-Term Conditions.

Interdisciplinary communication and collaboration are crucial in the care of people with multiple long-term conditions (MLTCs) yet are often experienced as insufficient. Through the lens of complexity science, this study aims to explain how healthcare professionals (HCPs) adapt to emerging situations in the care of patients with MLTC by examining interdisciplinary communication and collaboration in the outpatient hospital setting. We used the constant comparative method to analyze transcribed data from seven focus groups with twenty-one HCPs to generate a constructivist grounded theory of 'interdisciplinary communication and collaboration in the outpatient setting of the hospital for patients with multiple long-term conditions'. Our theory elucidates the various pathways of communication and collaboration. Why, when, and how team members choose to collaborate influences if and to what degree tailored care is achieved. There is great variability and unpredictability to this process due to internalized rules, such as beliefs on the appropriateness to deviate from guidelines, and the presence of an interprofessional identity. We identified organizational structures that influence the dynamics of the care team such as the availability of time and financial compensation for collaboration. As we strive for tailored care for patients with MLTC, our theory provides promising avenues for future endeavors.

A hospital care coordination team intervention for patients with multimorbidity: A practice-based, participatory pilot study.

OBJECTIVES: This study aims to develop and pilot a hospital care coordination team intervention for patients with multimorbidity and identify key uncertainties. METHODS: Practice-based, participatory pilot study with mixed methods in a middle-large teaching hospital. We included adult patients who had visited seven or more outpatient specialist clinics in 2018. The intervention consisted of an intake, a comprehensive review by a dedicated care coordination team, a consultation to discuss results and two follow-up appointments. We collected both quantitative and qualitative data. RESULTS: Out of 131 invited patients, 28 participants received the intake and comprehensive review. The intervention resulted in mixed outputs and short-term outcomes. Among the 28 participants, 21 received recommendations for at least two out of three categories (medication, involved medical specialists, other). Patients' experienced effects ranged from no to very large effects. Key uncertainties were how to identify patients with a need for care coordination and the minimum of required data that can be collected during regular clinical care with feasible effort. DISCUSSION: Recruitment and selection for hospital care coordination should be refined to include patients with multimorbidity who might benefit most. Outcomes of research and clinical care should align and first focus on evaluating the results of care coordination before evaluating health-related outcomes.

A Delphi consensus checklist helped assess the need to develop rapid guideline recommendations.

BACKGROUND AND OBJECTIVES: We aimed to develop a checklist to aid guideline developers in determining which scientific or societal cause ("triggers") are relevant when considering to initiate a rapid recommendation procedure. METHODS: We conducted a two-round modified Delphi procedure with a panel of Dutch guideline experts, clinicians, and patient representatives. A previously conducted systematic literature review and semistructured interviews with four science journalists were used to generate a list of potential items. This item list was submitted to the panel for discussion, reduction and refinement into a checklist. RESULTS: Thirteen experts took part. Two questionnaires were completed in which participants scored an initial list of 64 items based on relevance. During two online meetings, the scores were discussed, irrelevant items were removed, and relevant items were reformulated into seven questions. The final "quickscan assessment of the need for a rapid recommendation" covers user perspective, scientific evidence, clinical relevance, clinical practice variation, applicability, quality of care and public health outcomes, and ethical/legal considerations. CONCLUSION: The quickscan aids guideline developers in systematically assessing whether a trigger expresses a valid need for developing a rapid recommendation. Future research could focus on the applicability and validity of the checklist within guideline development programs.

Supporting older patients in making healthcare decisions: The effectiveness of decision aids; A systematic review and meta-analysis.

OBJECTIVE: To systematically review randomized controlled trials and clinical controlled trials evaluating the effectiveness of Decision Aids (DAs) compared to usual care or alternative interventions for older patients facing treatment, screening, or care decisions. METHODS: A systematic search of several databases was conducted. Eligible studies included patients ≥ 65 years or reported a mean of ≥ 70 years. Primary outcomes were attributes of the choice made and decision making process, user experience and ways in which DAs were tailored to older patients. Meta-analysis was conducted, if possible, or outcomes were synthesized descriptively. RESULTS: Overall, 15 studies were included. Using DAs were effective in increasing knowledge (SMD 0.90; 95% CI [0.48, 1.32]), decreasing decisional conflict (SMD -0.15; 95% CI [-0.29, -0.01]), improving patient-provider communication (RR 1.67; 95% CI [1.21, 2.29]), and preparing patients to make an individualized decision (MD 35.7%; 95% CI [26.8, 44.6]). Nine studies provided details on how the DA was tailored to older patients. CONCLUSION: This review shows a number of favourable results for the effectiveness of DAs in decision making with older patients. PRACTICE IMPLICATIONS: Current DAs can be used to support shared decision making with older patients when faced with treatment, screening or care decisions.

Randomized double-blind placebo-controlled crossover trial with pyridostigmine in spinal muscular atrophy types 2-4.

Hereditary proximal spinal muscular atrophy causes weakness and increased fatigability of repetitive motor functions. The neuromuscular junction is anatomically and functionally abnormal in patients with spinal muscular atrophy. Pharmacological improvement of neuromuscular transmission may therefore represent a promising additional treatment strategy. We conducted a Phase II, monocentre, placebo-controlled, double-blind, cross-over trial with the acetylcholinesterase inhibitor pyridostigmine in treatment-naïve patients with spinal muscular atrophy types 2-4. We investigated the safety and efficacy of pyridostigmine on fatigability and motor function. Each participant received pyridostigmine and a placebo for 8 weeks, in random order. Primary outcomes were the repeated nine-hole peg test for fatigability and motor function measure. Secondary outcomes were patient-reported effects, endurance shuttle test combined scores and adverse events. We included 35 patients. For the repeated nine-hole peg test, the mean difference was 0.17 s/trial (95% confidence interval: -1.17-1.49; P = 0.8), favouring placebo, and for the motor function measure, 0.74% (95% confidence interval: 0.00-1.49; P = 0.05), favouring pyridostigmine. Around 74% of patients reported medium-to-large beneficial effects of pyridostigmine on fatigability, compared with 29.7% in the placebo arm. This was paralleled by a reduced dropout risk of 70% on the endurance shuttle test combined scores (hazard ratio: 0.30; 95% confidence interval: 0.15-0.58) under pyridostigmine. Adverse events, mostly mild and self-limiting, occurred more frequently under pyridostigmine. No serious adverse events related to the study medication were observed. Patients with spinal muscular atrophy tolerated pyridostigmine well. There were no significant differences in primary outcomes, but the self-reported reduction of fatigability and improved endurance shuttle test combined score performance suggest that pyridostigmine may be useful as an additional therapy to survival motor neuron-augmenting drugs. Trial registration number: EudraCT: 2011-004369-34, NCT02941328.

[Guidelines for customised integrated care in multimorbidity]. / Richtlijnen voor passende zorg bij multimorbiditeit.

In patients with multimorbidity, healthcare providers follow various disease-specific guidelines. Besides the fact that simultaneous treatment of several chronic diseases can be intensive for the patient, there is also the risk of contradictory advice or interactions when all recommendations are applied simultaneously. There are a number of developments to make guidelines more applicable to the growing target group of multimorbid patients. The 'Methodology for senior-proof guidelines' describes how to pay attention to patients with multimorbidity in all phases of guideline development. In addition, integrated guideline use for multimorbidity is being developed through a new modular structure with the use of interconnections. The future doctor will have to acquire knowledge and skills in translating treatment goals of patients with multimorbidity into an integral and coordinated tailor-made plan in cooperation with other professionals. A guideline for the treatment of multimorbidity can provide support in working across domains without directly applicable evidence.

[Coordinating and tailoring hospital care for patients with multimorbidity: who will take the lead?] / Regie bij multimorbiditeit in het ziekenhuis.

OBJECTIVE: To gain insight in medical specialists' and nurse practitioners' opinions on multimorbidity and coordination and tailoring of hospital care. DESIGN: Exploratory mixed-method design. METHOD: From August 2018 until January 2019, 35 Dutch medical associations were asked to forward a digital survey with open- and close-ended questions to their members. We used qualitative and quantitative methods to analyze the data. The main themes were identified with inductive, thematic analysis. RESULTS: There were 554 respondents from 22 associations, 43% of the medical specialist respondents were internist (n=221). The qualitative analysis of the answers regarding what is required in hospital care for patients with multimorbidity resulted in eight themes at the patient's, professional's and hospital organization's level. To the open question about who should take the lead, respondents most often answered the geriatrician or internist, followed by the general practitioner, 'the care professional who is treating the main problem', a nurse practitioner/physician assistant and the 'attending physician of the primary team'. All geriatricians and almost all internists felt they possessed the competencies to take the lead in hospital care for patients with multimorbidity. CONCLUSION: Medical specialists' and nurse practitioners' diverse ideas about who should take the lead in hospital care for patients with multimorbidity were a noteworthy finding. It is important to start local conversations about how to divide roles and responsibilities regarding the coordination and tailoring of hospital care for patients with multimorbidity.