Machine learning to optimize literature screening in medical guideline development.

OBJECTIVES: In a time of exponential growth of new evidence supporting clinical decision-making, combined with a labor-intensive process of selecting this evidence, methods are needed to speed up current processes to keep medical guidelines up-to-date. This study evaluated the performance and feasibility of active learning to support the selection of relevant publications within medical guideline development and to study the role of noisy labels. DESIGN: We used a mixed-methods design. Two independent clinicians' manual process of literature selection was evaluated for 14 searches. This was followed by a series of simulations investigating the performance of random reading versus using screening prioritization based on active learning. We identified hard-to-find papers and checked the labels in a reflective dialogue. MAIN OUTCOME MEASURES: Inter-rater reliability was assessed using Cohen's Kappa (ĸ). To evaluate the performance of active learning, we used the Work Saved over Sampling at 95% recall (WSS@95) and percentage Relevant Records Found at reading only 10% of the total number of records (RRF@10). We used the average time to discovery (ATD) to detect records with potentially noisy labels. Finally, the accuracy of labeling was discussed in a reflective dialogue with guideline developers. RESULTS: Mean ĸ for manual title-abstract selection by clinicians was 0.50 and varied between - 0.01 and 0.87 based on 5.021 abstracts. WSS@95 ranged from 50.15% (SD = 17.7) based on selection by clinicians to 69.24% (SD = 11.5) based on the selection by research methodologist up to 75.76% (SD = 12.2) based on the final full-text inclusion. A similar pattern was seen for RRF@10, ranging from 48.31% (SD = 23.3) to 62.8% (SD = 21.20) and 65.58% (SD = 23.25). The performance of active learning deteriorates with higher noise. Compared with the final full-text selection, the selection made by clinicians or research methodologists deteriorated WSS@95 by 25.61% and 6.25%, respectively. CONCLUSION: While active machine learning tools can accelerate the process of literature screening within guideline development, they can only work as well as the input given by human raters. Noisy labels make noisy machine learning.

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.

Study protocol of a national multicentre prospective evaluation study assessing the validity and impact of the Dutch Paediatric Early Warning Score (PEWS) in the Netherlands.

INTRODUCTION: Early recognition of clinical deterioration and timely intervention are important to improve morbidity and mortality in paediatric care. The Paediatric Early Warning Score (PEWS) is a scoring system aiming to identify hospitalised children at risk for deterioration. Currently, there is a large heterogeneity of PEWS systems in the Netherlands, with a considerable number remaining unvalidated or self-designed. Therefore, a consensus-based Dutch PEWS has been developed in a national study using the Core Outcome Measures in Effectiveness Trials initiative. The Dutch PEWS is a uniform system that integrates a core set of vital parameters together with pre-existing risk factors and uses risk stratification to proactively follow-up on patients at risk (so-called 'watcher patients'). This study aims to validate the Dutch PEWS and to determine its impact on improving patient safety in various hospital settings. METHODS AND ANALYSIS: This national study will be a large multicentre evaluation study, in which the Dutch PEWS will be implemented and evaluated in 12 hospitals in the Netherlands. In this study, a mixed methods methodology will be used and evaluated on predefined outcome measures. To examine the validity of the Dutch PEWS, statistical analyses will be undertaken on quantitative data retrieved from electronic health records. Surveys among physicians and nurses; semistructured interviews with healthcare providers and parents; and daily evaluation forms are being conducted to determine the impact of the Dutch PEWS. The study is being conducted from December 2020 to June 2024.

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.

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.

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.

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.

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.

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.

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.

Respiratory muscle fatigability in patients with spinal muscular atrophy.

BACKGROUND: Respiratory failure is a major cause of morbidity and mortality in patients with Spinal Muscular Atrophy (SMA). Lack of endurance, or "fatigability," is an important symptom of SMA. In addition to respiratory muscle weakness, respiratory function in SMA may be affected by Respiratory Muscle Fatigability (RMF). AIM: The purpose of this study was to explore RMF in patients with SMA. METHODS: We assessed a Respiratory Endurance Test (RET) in 19 children (median age [years]: 11) and 36 adults (median age [years]: 34) with SMA types 2 and 3. Participants were instructed to breath against an inspiratory threshold load at either 20%, 35%, 45%, 55%, or 70% of their individual maximal inspiratory mouth pressure (PImax). RMF was defined as the inability to complete 60 consecutive breaths. Respiratory fatigability response was determined by change in maximal inspiratory mouth pressure (ΔPImax) and perceived fatigue (∆perceived fatigue). RESULTS: The probability of RMF during the RET increased by 59%-69% over 60 breaths with every 10% increase in inspiratory threshold load (%PImax). Fatigability response was characterized by a large variability in ΔPImax (-21% to +16%) and a small increase in perceived fatigue (p = 0.041, range 0 to +3). CONCLUSION AND KEY FINDINGS: Patients with SMA demonstrate a dose-dependent increase in RMF without severe increase in exercise-induced muscle weakness or perceived fatigue. Inspiratory muscle loading in patients with SMA seems feasible and its potential to stabilize or improve respiratory function in patients with SMA needs to be determined in further research.

[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.

Development and internal validation of prediction models for future hospital care utilization by patients with multimorbidity using electronic health record data.

OBJECTIVE: To develop and internally validate prediction models for future hospital care utilization in patients with multiple chronic conditions. DESIGN: Retrospective cohort study. SETTING: A teaching hospital in the Netherlands (542 beds). PARTICIPANTS: All adult patients (n = 18.180) who received care at the outpatient clinic in 2017 for two chronic diagnoses or more (including oncological diagnoses) and who returned for hospital care or outpatient clinical care in 2018. Development and validation using a stratified random split-sample (n = 12.120 for development, n = 6.060 for internal validation). OUTCOMES: ≥2 emergency department visits in 2018, ≥1 hospitalization in 2018 and ≥12 outpatient visits in 2018. STATISTICAL ANALYSIS: Multivariable logistic regression with forward selection. RESULTS: Evaluation of the models' performance showed c-statistics of 0.70 (95% CI 0.69-0.72) for the hospitalization model, 0.72 (95% CI 0.70-0.74) for the ED visits model and 0.76 (95% 0.74-0.77) for the outpatient visits model. With regard to calibration, there was agreement between lower predicted and observed probability for all models, but the models overestimated the probability for patients with higher predicted probabilities. CONCLUSIONS: These models showed promising results for further development of prediction models for future healthcare utilization using data from local electronic health records. This could be the first step in developing automated alert systems in electronic health records for identifying patients with multimorbidity with higher risk for high healthcare utilization, who might benefit from a more integrated care approach.

The effects of wheelchair mobility skills and exercise training on physical activity, fitness, skills and confidence in youth using a manual wheelchair.

PURPOSE: To evaluate the effects of a combination of wheelchair mobility skills (WMS) training and exercise training on physical activity (PA), WMS, confidence in wheelchair mobility, and physical fitness. METHODS: Youth using a manual wheelchair (n = 60) participated in this practice-based intervention, with a waiting list period (16 weeks), exercise training (8 weeks), WMS training (8 weeks), and follow-up (16 weeks). Repeated measures included: PA (Activ8), WMS (Utrecht Pediatric Wheelchair Mobility Skills Test), confidence in wheelchair mobility (Wheelchair Mobility Confidence Scale), and physical fitness (cardiorespiratory fitness, (an)aerobic performance) and were analysed per outcome parameter using a multilevel model analyses. Differences between the waiting list and training period were determined with an unpaired sample t-test. RESULTS: Multilevel model analysis showed significant positive effects for PA (p = 0.01), WMS (p < 0.001), confidence in wheelchair mobility (p < 0.001), aerobic (p < 0.001), and anaerobic performance (p < 0.001). Unpaired sample t-tests underscored these effects for PA (p < 0.01) and WMS (p < 0.001). There were no effects on cardiorespiratory fitness. The order of training (exercise before WMS) had a significant effect on confidence in wheelchair mobility. CONCLUSIONS: A combination of exercise and WMS training appears to have significant positive long-term effects on PA, WMS, confidence in wheelchair mobility, and (an)aerobic performance in youth using a manual wheelchair.Implications for rehabilitationExercise training and wheelchair mobility skills (WMS) training can lead to a sustained improvement in physical activity (PA) in youth using a manual wheelchair.These combined trainings can also lead to a sustained increase in WMS, confidence in wheelchair mobility, and (an)aerobic performance.More attention is needed in clinical practice and in research towards improving PA in youth using a manual wheelchair.

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.

Motor unit reserve capacity in spinal muscular atrophy during fatiguing endurance performance.

OBJECTIVE: To investigate the availability of any motor unit reserve capacity during fatiguing endurance testing in patients with spinal muscular atrophy (SMA). METHODS: We recorded surface electromyography (sEMG) of various muscles of upper- and lower extremities of 70 patients with SMA types 2-4 and 19 healthy controls performing endurance shuttle tests (ESTs) of arm and legs. We quantitatively evaluated the development of fatigability and motor unit recruitment using time courses of median frequencies and amplitudes of sEMG signals. Linear mixed effect statistical models were used to evaluate group differences in median frequency and normalized amplitude at onset and its time course. RESULTS: Normalized sEMG amplitudes at onset of upper body ESTs were significantly higher in patients compared to controls, yet submaximal when related to maximal voluntary contractions, and showed an inverse correlation to SMA phenotype. sEMG median frequencies decreased and amplitudes increased in various muscles during execution of ESTs in patients and controls. CONCLUSIONS: Decreasing median frequencies and increasing amplitudes reveal motor unit reserve capacity in individual SMA patients during ESTs at submaximal performance intensities. SIGNIFICANCE: Preserving, if not expanding motor unit reserve capacity may present a potential therapeutic target in clinical care to reduce fatigability in individual patients with SMA.

Correlates of Fatigability in Patients With Spinal Muscular Atrophy.

OBJECTIVE: To determine the associations between fatigability and muscle strength, motor function, neuromuscular junction (NMJ) function, and perceived fatigue in spinal muscular atrophy (SMA), we assessed 61 patients with SMA. METHODS: Fatigability was defined as the inability to continue a 20-minute submaximal repetitive task of either walking or proximal or distal arm function and expressed as drop-out on the Endurance Shuttle Test Combined Score (ESTCS). We assessed muscle strength with the Medical Research Council (MRC) sum score, motor function with the Hammersmith Functional Motor Scale Expanded (HFMSE) and Motor Function Measure (MFM), NMJ function with repetitive nerve stimulation of the accessory and ulnar nerve, and perceived fatigue with the PROMIS Fatigue Short Form questionnaire in 61 children and adults with SMA types 2-4. We applied Cox regression analysis to explore the associations between fatigability and these factors. RESULTS: The hazard of drop-out on the ESTCS decreased 0.8%, 2%, and 1.3% for each point increase in the MRC sum score, the HFMSE score, and the MFM percentual score, respectively. However, we observed prominent fatigability with preserved muscle function and vice versa in 13%-16% of patients. We did not find an association between NMJ dysfunction of the accessory (p = 0.37) and ulnar nerve (p = 0.063) and fatigability, which could be due to a large number of missing values. Perceived fatigue in SMA was comparable to reference values and was not associated with fatigability (p = 0.52). CONCLUSION: Fatigability in SMA is associated with, yet not equivalent to, muscle strength and motor function.

Examining Causes and Prevention Strategies of Adverse Events in Deceased Hospital Patients: A Retrospective Patient Record Review Study in the Netherlands.

OBJECTIVE: To improve patient safety and possibly prevent mortality from adverse events (AEs) in hospitals, it is important to gain insight in their underlying causes. We aimed to examine root causes and potential prevention strategies of AEs in deceased hospital patients. METHODS: Data on 571 AEs were used from two retrospective patient record review studies of patients who died during hospitalization in the Netherlands. Trained reviewers assessed contributing factors and potential prevention strategies. The results were analyzed together with data on preventability of the AE and the relationship of the AE with the death of the patient. RESULTS: In 47% of the AEs, patient-related causes were identified, in 35% human causes, in 9% organizational causes, and in 3% technical causes. Preventable AEs were caused by technical, organizational, and human causes (78%, 74%, and 74%, respectively) more often than by patient-related causes (33%). In addition, technical factors caused AEs leading to preventable death (78%) relatively often. Recommended strategies to prevent AEs were quality assurance/peer review, evaluation of safety behavior, improving procedures, and improving information and communication structures. CONCLUSIONS: Human failures played an important role in the causation of AEs in Dutch hospitals, because they occurred frequently and they were frequently the cause of preventable AEs. To a lesser extent, latent organizational and technical factors were identified. Patient-related factors were often identified, but the preventability of the AEs with these causes was low. For future research into causes of AEs, we recommend combining record review with interviewing.

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.