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Xin Fan,1 Rashid Menhas,2 Rizwan Ahmed Laar1 1College of Physical Education, Hubei Normal University, Huangshi, 435002, People’s Republic of China; 2Research Center of Sports Social Sciences, College of Physical Education and Sports, Soochow University, Suzhou, Jiangsu Province, People’s Republic of ChinaCorrespondence: Rashid Menhas, Research Center of Sports Social Sciences, College of Physical Education and Sports, Soochow University, Suzhou, Jiangsu Province, People’s Republic of China, Email menhas.r@yahoo.com Rizwan Ahmed Laar, College of Physical Education, Hubei Normal University, Huangshi, 435002, People’s Republic of China, Email rizwan_laar@yahoo.comBackground: Pandemic prevention measures increased physical inactivity due to travel restrictions that led to negative physical fitness, health behavior, psychological health, and general well-being. The mediating role of coping behavior must be identified before developing interventional strategies for this pandemic.Objective: The study investigates the mediating role of coping behavior to mitigate the Coronavirus disease impact on physical fitness, health behavior, psychological health, and general well-being.Methods: A web-based survey was used under convenience sampling to collect the primary data. Smart-PLS 3.0 was used to analyze the collected data.Results: All 14 direct correlations (H1-H14) were correct, and coping behavior’s mediating impact was shown to be statistically significant (H9a- H14d).Conclusion: Our study findings indicate that mediating role of coping behavior in mitigating the pandemic impacts was statistically significant. It is concluded that coping behavior is a healthy adaptation to protecting the barrier against COVID-19 adverse effects on health.Keywords: COVID-19, lockdown, impact, strategies

Ping Fang,1 Shusheng Shi,1 Rashid Menhas,2 Rizwan Ahmed Laar,3 Muhammad Muddasar Saeed4 1School of Sports Science and Physical Education, Nanjing Normal University, Nanjing, Jiangsu Province, People’s Republic of China; 2Research Center of Sports Social Sciences, School of Physical Education and Sports, Soochow University, Suzhou, Jiangsu Province, People’s Republic of China; 3College of Physical Education, Hubei Normal University, Huangshi, Hubei Province, People’s Republic of China; 4International Education College Chinese and Western Medicine Clinic, Dalian Medical University, Dalian, Liaoning, People’s Republic of ChinaCorrespondence: Rashid Menhas, Research Center of Sports Social Sciences, School of Physical Education and Sports, Soochow University, Suzhou, Jiangsu Province, People’s Republic of China, Email menhas.r@yahoo.com Rizwan Ahmed Laar, College of Physical Education, Hubei Normal University, Huangshi, Hubei Province, People’s Republic of China, Email rizwan_laar@yahoo.comBackground: E-health apps play a vital role in the era of the COVID-19 pandemic. More people across the globe have started to use fitness and health apps to mitigate the COVID-19 negative impacts on the health-related quality of life. E-health treatments are becoming more common these days.Aim: The present research aim was to explore the mediating role of digital platforms for physical activity and fitness with demographic characteristics among Chinese people during the pandemic.Methods: The primary data was collected through an online survey across China under the snowball sampling technique. A total of 5351 respondents took part in the online questionnaire survey. After quantification and quality check of the data, a total of 5000 respondents’ responses were added in the final analysis. The collected data were analyzed by using SPSS-25 and SmartPLS 3.0 software.Results: The structural equation model analysis H1 (β = 0.227, t = 18.725, p = < 0.000), H2 (β = 0.225, t = 17.892, p = < 0.000), H3 (β = 0.229, t = 17.845, p = < 0.000), H4 (β = 0.54, t = 60.928, p= < 0.000), H5 (β = 0.553, t = 57.291, p = < 0.000), H6 (β = 0.559, t = 56.956, p = < 0.000), H7 (β = 0.358, t = 24.779, p = < 0.000), H8 (β = 0.259, t = 19.617, p = < 0.000) and H9 (β = 0.288, t = 19.92, p = < 0.000) results confirm the proposed hypothesis of the study.Conclusion: E-health apps are playing a vital role in maintaining an active lifestyle through physical activity. Fitness and health apps can promote physical activity and improve health-related quality of life. Results of the current study concluded that E-health apps are promising to promote physical activity and fitness during the COVID-19 preventive measures.Keywords: COVID-19, digital platforms, demographic characteristics, physical activity, Chinese people

ObjectiveInborn errors of immunity (IEI) are a heterogeneous group of disorders, affecting different components of the immune system. Over 450 IEI related genes have been identified, with new genes continually being recognized. This makes the early application of next-generation sequencing (NGS) as a diagnostic method in the evaluation of IEI a promising development. We aimed to provide an overview of the diagnostic yield and time to diagnosis in a cohort of patients suspected of IEI and evaluated by an NGS based IEI panel early in the diagnostic trajectory in a multicenter setting in the Netherlands.Study DesignWe performed a prospective observational cohort study. We collected data of 165 patients with a clinical suspicion of IEI without prior NGS based panel evaluation that were referred for early NGS using a uniform IEI gene panel. The diagnostic yield was assessed in terms of definitive genetic diagnoses, inconclusive diagnoses and patients without abnormalities in the IEI gene panel. We also assessed time to diagnosis and clinical implications.ResultsFor children, the median time from first consultation to diagnosis was 119 days versus 124 days for adult patients (U=2323; p=0.644). The median turn-around time (TAT) of genetic testing was 56 days in pediatric patients and 60 days in adult patients (U=1892; p=0.191). A definitive molecular diagnosis was made in 25/65 (24.6%) of pediatric patients and 9/100 (9%) of adults. Most diagnosed disorders were identified in the categories of immune dysregulation (n=10/25; 40%), antibody deficiencies (n=5/25; 20%), and phagocyte diseases (n=5/25; 20%). Inconclusive outcomes were found in 76/165 (46.1%) patients. Within the patient group with a genetic diagnosis, a change in disease management occurred in 76% of patients.ConclusionIn this cohort, the highest yields of NGS based evaluation for IEI early in the diagnostic trajectory were found in pediatric patients, and in the disease categories immune dysregulation and phagocyte diseases. In cases where a definitive diagnosis was made, this led to important disease management implications in a large majority of patients. More research is needed to establish a uniform diagnostic pathway for cases with inconclusive diagnoses, including variants of unknown significance.

Having a clear and stable sense of how one performs in a field is a key contributor to goal pursuit. Performance feedback is often considered a crucial resource for developing this clear and stable self-knowledge but may be less optimally integrated when feedback is considered inaccurate or dishonest. The current paper investigates how such feedback perceptions may limit the development of people's ability self-concept, and how workplace contexts can restrict communication. A 2-week daily diary study among 197 junior researchers working in STEM-fields (N = 1,353 data points) showed that those in more competitive (vs. more collaborative) work environments overall perceived feedback as more inaccurate and dishonest (but not as more positively inflated). These results did not differ for men and women, showing that both face negative consequences of working in a more competitive context in terms of their ability to get high-quality feedback. On the daily level, results showed that days on which people received more inaccurate and dishonest (but not positively inflated) feedback were also days on which they reported higher imposter feelings, and lower ability self-esteem, self-concept clarity, and self-concept stability. In turn, days on which people felt more like an imposter and reported lower ability self-esteem, self-concept clarity, and self-concept stability, were also days on which motivation was lower. Ability self-esteem and self-concept clarity (but not imposter feelings and self-concept stability) were also related to lower daily risk-taking tendencies. Together, these results show that an important contextual factor--the perceived competitiveness of one's work environment--influences feedback inaccuracy and dishonesty, with consequences for the ability to develop a clear, stable and certain ability self-concept.

A repeat expansion mutation in the C9orf72 gene is the leading known genetic cause of FTD and ALS. The C9orf72-ALS/FTD field has been plagued by a lack of reliable tools to monitor this genomic locus and its RNA and protein products. We have validated assays that quantify C9orf72 pathobiology at the DNA, RNA and protein levels using knock-out human iPSC lines as controls. Here we show that single-molecule sequencing can accurately measure the repeat expansion and faithfully report on changes to the C9orf72 locus in what has been a traditionally hard to sequence genomic region. This is of particular value to sizing and phasing the repeat expansion and determining changes to the gene locus after gene editing. We developed ddPCR assays to quantify two major C9orf72 transcript variants, which we validated by selective excision of their distinct transcriptional start sites. Using validated knock-out human iPSC lines, we validated 4 commercially available antibodies (of 9 tested) that were specific for C9orf72 protein quantification by Western blot, but none were specific for immunocytochemistry. We tested 15 combinations of antibodies against dipeptide repeat proteins (DPRs) across 66 concentrations using MSD immunoassay, and found two (against poly-GA and poly-GP) that yielded a 1.5-fold or greater signal increase in patient iPSC-motor neurons compared to knock-out control, and validated them in human postmortem and transgenic mouse brain tissue. Our validated DNA, RNA and protein assays are applicable to discovery research as well as clinical trials.

This paper explores the concept of e-resilience in educational systems, particularly in Public Affairs Education, in response to the COVID-19 pandemic. We investigate the drivers and challenges faced by actors from an educational system perspective, which is conceptualized by adjusting Bronfenbrenners' ecological model using dimensions of "Technology, Human," and "Intersections" and levels "Macro, Meso" and "Micro," during this global shock. Utilizing a multiple case study approach across Argentina, Nigeria, South Africa, the Netherlands, and the United States, we examine e-resilience through 36 group interviews involving 41 students, staff, administration, and program directors. Our findings reveal that challenges within the technological system impact overall system performance, while human-level challenges disrupt learning and pose greater obstacles for the system. Despite these challenges, each context exhibits various drivers, providing insights for building more robust educational systems. By better understanding the dynamics of e-resilience within each educational system context, we can enhance educational systems in the face of unprecedented shocks.

This research explores the effect of implementing educational open microcredentials on student motivation, engagement, and completion in open access online courses. Our case study reviews the impact on the Community of Learning for African PhD Fellows, a capacity-building project supporting PhD fellows in Sub-Saharan Africa. It builds on an analysis of data from learning analytics, surveys, and semi-structured interviews. Our case study findings indicate that course completion was low, in course offering rounds with and without online certification. Main hurdles to completion are lack of time and lack of direct career benefits or academic value attached to the course completion. We found that, while open access online courses are appreciated by PhD fellows, the implementation of open microcredentials did not provide an incentive towards completion of online courses for this population. Hard and soft copy certificates at this point are more appreciated.

This study examined the impact of two questionnaire characteristics, scale position and questionnaire length, on the prevalence of random responders in the TIMSS 2015 eighth-grade student questionnaire. While there was no support for an absolute effect of questionnaire length, we did find a positive effect for scale position, with an increase of 5% in random responding over the course of the questionnaire (in both the shorter and the longer version). However, scale character turned out to be an unexpected but more important determinant. Scales about students' confidence in mathematics or science showed an increase of 9% in random responding, which is double the impact of scale position. Potential mechanisms underlying the confidence case and general implications of the results for questionnaire design are discussed.

Questionnaires in educational research assessing students' attitudes and beliefs are low-stakes for the students. As a consequence, students might not always consistently respond to a questionnaire scale but instead provide more random response patterns with no clear link to items' contents. We study inter-individual differences in students' intra-individual random responding profile across 19 questionnaire scales in the TIMSS 2015 eighth-grade student questionnaire in seven countries. A mixture IRT approach was used to assess students' random responder status on a questionnaire scale. A follow-up latent class analysis across the questionnaire revealed four random responding profiles that generalized across countries: A majority of consistent nonrandom responders, intermittent moderate random responders, frequent random responders, and students that were exclusively triggered to respond randomly on the confidence scales in the questionnaire. We discuss the implications of our findings in light of general data-quality concerns and the potential ineffectiveness of early-warning monitoring systems in computer-based surveys.

To the editor: Human RAD50 deficiency is a very rare genomic instability syndrome associated with microcephaly and stunted growth (1). Here, we describe RAD50 deficiency in two siblings harboring a [...]

In his theory of criticism, D. N. Walton presupposes that an opponent either critically questions an argument, without supplementing this questioning with any reasoning of her own, or that she puts forward a critical question and supplements it with a counterargument, that is, with reasoning in defense of an opposite position of her own. In this paper, I show that there is a kind of in-between critical option for the opponent that needs to be taken into account in any classification of types of criticism, and that should not be overlooked in a system of dialogue norms, nor in a procedure for developing a strategically expedient critique. In this third option, an opponent questions and overtly doubts a statement of the proponent and supplements her doubts with a counterconsideration that explains and motivates her position of critical doubt, yet without supporting any opposite thesis, thereby assisting, as it were, the proponent in his attempt to develop a responsive argumentation, tailor-made to convince this particular opponent. First, I elaborate on the notion of an explanatory counterconsideration. Second, I discuss Walton’s distinction between premises that can be challenged by mere questioning (“ordinary premises” and “assumptions”) and premises that must be challenged by incurring the obligation to offer counter-argumentation (somewhat confusingly labeled “exceptions”). I contend that the latter type of premises, that I would label “normality premises,” can be attacked without incurring a genuine burden of proof. Instead, it can be attacked by means of incurring a burden of criticism (Van Laar and Krabbe, 2013) that amounts to the obligation to offer an explanatory counterconsideration, rather than a convincing ex concessis argument. Of course, providing the opponent with the right to discharge her burden of criticism with explanatory counterconsiderations brings a clear strategic ad- vantage to her. It is much less demanding to motivate one’s doubts regarding proposition P, than to convince the proponent of not-P. If we want to encourage opponents to act critically, and proponents to develop responsive arguments, the importance of the notions of an explanatory counterconsideration and of a motivated doubt should be emphasized in the theory of criticism.