bluuebunny/crossref_metadata_2025_split huggingface dataset:
Development of a factorial survey for use in an international study examining clinicians’ likelihood to support the decision to initiate invasive long-term ventilation for a child (the TechChild study)
Authors: Quirke Mary Brigid , Alexander Denise , Masterson Kate , Greene Jo , Walsh Cathal , Leroy Piet , Berry Jay , Polikoff Lee , Brenner Maria 
Published: July 2022

Abstract
Background
The decision to initiate invasive long-term ventilation for a child with complex medical needs can be extremely challenging. TechChild is a research programme that aims to explore the liminal space between initial consideration of such technology dependence and the final decision. This paper presents a best practice example of the development of a unique use of the factorial survey method to identify the main influencing factors in this critical juncture in a child’s care.

Methods
We developed a within-subjects design factorial survey. In phase 1 (design) we defined the survey goal (dependent variable, mode and sample). We defined and constructed the factors and factor levels (independent variables) using previous qualitative research and existing scientific literature. We further refined these factors based on expert feedback from expert clinicians and a statistician. In phase two (pretesting), we subjected the survey tool to several iterations (cognitive interviewing, face validity testing, statistical review, usability testing). In phase three (piloting) testing focused on feasibility testing with members of the target population (n = 18). Ethical approval was obtained from the then host institution’s Health Sciences Ethics Committee.

Results
Initial refinement of factors was guided by literature and interviews with clinicians and grouped into four broad categories: Clinical, Child and Family, Organisational, and Professional characteristics. Extensive iterative consultations with clinical and statistical experts, including analysis of cognitive interviews, identified best practice in terms of appropriate: inclusion and order of clinical content; cognitive load and number of factors; as well as language used to suit an international audience. The pilot study confirmed feasibility of the survey. The final survey comprised a 43-item online tool including two age-based sets of clinical vignettes, eight of which were randomly presented to each participant from a total vignette population of 480.

Conclusions
This paper clearly explains the processes involved in the development of a factorial survey for the online environment that is internationally appropriate, relevant, and useful to research an increasingly important subject in modern healthcare. This paper provides a framework for researchers to apply a factorial survey approach in wider health research, making this underutilised approach more accessible to a wider audience.
Methodological perspectives on the study of the health effects of unemployment – reviewing the mode of unemployment, the statistical analysis method and the role of confounding factors
Authors: Norström Fredrik , Hammarström Anne 
Published: July 2022

Abstract
Introduction
Studying the relationship between unemployment and health raises many methodological challenges. In the current study, the aim was to evaluate the sensitivity of estimates based on different ways of measuring unemployment and the choice of statistical model.

Methods
The Northern Swedish cohort was used, and two follow-up surveys thereof from 1995 and 2007, as well as register data about unemployment. Self-reported current unemployment, self-reported accumulated unemployment and register-based accumulated unemployment were used to measure unemployment and its effect on self-reported health was evaluated. Analyses were conducted with G-computation, logistic regression and three estimators for the inverse probability weighting propensity scores, and 11 potentially confounding variables were part of the analyses. Results were presented with absolute differences in the proportion with poor self-reported health between unemployed and employed individuals, except when logistic regression was used alone.

Results
Of the initial 1083 pupils in the cohort, our analyses vary between 488–693 individuals defined as employed and 61–214 individuals defined as unemployed. In the analyses, the deviation was large between the unemployment measures, with a difference of at least 2.5% in effect size when unemployed was compared with employed for the self-reported and register-based unemployment modes. The choice of statistical method only had a small influence on effect estimates and the deviation was in most cases lower than 1%. When models were compared based on the choice of potential confounders in the analytical model, the deviations were rarely above 0.6% when comparing models with 4 and 11 potential confounders. Our variable for health selection was the only one that strongly affected estimates when it was not part of the statistical model.

Conclusions
How unemployment is measured is highly important when the relationship between unemployment and health is estimated. However, misspecifications of the statistical model or choice of analytical method might not matter much for estimates except for the inclusion of a variable measuring health status before becoming unemployed. Our results can guide researchers when analysing similar research questions. Model diagnostics is commonly lacking in publications, but they remain very important for validation of analyses.
Relationship between patient experience and hospital readmission: system-level survey with deterministic data linkage method
Authors: Wong Eliza Lai-Yi , Poon Chin-Man , Cheung Annie Wai-Ling , Chen Frank Youhua , Yeoh Eng-Kiong 
Published: July 2022

Abstract
Background
Linkage of public healthcare data provides powerful resources for studying from a comprehensive view of quality of care than information for a single administrative database. It is believed that positive patient experiences reflect good quality of health care and may reduce patient readmission. This study aimed to determine the relationship between patient experience and hospital readmission at a system level by linking anonymous experience survey data with de-identified longitudinal hospital administrative admissions data.

Methods
Data were obtained by linking two datasets with anonymised individual-level records from seven largest-scale acute public hospitals over seven geographical clusters in Hong Kong. Selected records in the two datasets involving patient experience survey (PES) (2013 survey dataset) and healthcare utilization (admissions dataset) were used. Following data cleaning and standardization, a deterministic data linkage algorithm was used to identify pairs of records uniquely matched for a list of identifiers (10 selected variables) between two datasets. If patient’s record from the survey dataset matched with the hospitalization records in the admissions dataset, they were included in the subsequent analyses. Bivariate analyses and multivariable logistic regression models were performed to evaluate the associations between hospital readmission in the next calendar month and patient experience.

Results
The overall matching rate was 62.1% (1746/2811) for PES participants aged 45 or above from the survey dataset. The average score for overall inpatient experience was 8.10 (SD = 1.53). There was no significant difference between matched patients and unmatched patients in terms of their score for the perception of overall quality of care received during hospitalization (X2 = 6.931, p-value = 0.14) and score for overall inpatient experience (X2 = 7.853, p-value = 0.25). In the multivariable model, readmission through the outpatient department (planned admission) in the next calendar month was significantly associated with a higher score given to the overall quality of care received (adjusted OR = 1.54, 95%CI = 1.09–2.17), while such association was absent for readmission through Accident and Emergency department (adjusted OR = 0.75, 95%CI = 0.50–1.12).

Conclusions
This study demonstrated the feasibility of routine record linkage, with the limited intrusion of patients’ confidentiality, for evaluating health care quality. It also highlights the significant association between readmission through planned readmission and a higher score for overall quality of care received. A possible explanation might be the perceived better co-ordination between outpatient departments and inpatient service and the well-informed discharge plan given to this group of patients.
Safety and efficacy of irinotecan, oxaliplatin, and capecitabine (XELOXIRI) regimen with or without targeted drugs in patients with metastatic colorectal cancer: a retrospective cohort study
Authors: Liu Xiu , Ou Kai , Ma Xiaoting , Gao Lizhen , Wang Qi , Zhang Haizeng , Yang Lin 
Published: July 2022

Abstract
Background
Five-fluorouracil, folinic acid, oxaliplatin and irinotecan (FOLFOXIRI) regimen is used as the first-line treatment for metastatic colorectal cancer (mCRC). The use of capecitabine, an oral fluoropyrimidine pro-drug, is feasible and safe; hence, it provides an interesting alternative to 5-fluorouracil in the abovementioned regimen. This study aimed to evaluate the efficacy and safety of capecitabine, oxaliplatin, and irinotecan (XELOXIRI) regimen use with or without targeted drugs in Chinese patients with mCRC.

Methods
We conducted a retrospective, longitudinal cohort study of patients with mCRC who received XELOXIRI regimen with or without targeted drugs (bevacizumab or cetuximab) every 2 weeks between January 2017 and November 2019 at the National Cancer Center/Cancer Hospital, the Chinese Academy of Medical Sciences, and Peking Union Medical College. Treatment efficacy was assessed by investigators by evaluating the objective response rate (ORR) and disease control rate (DCR). Overall survival (OS) was assessed using Cox proportional hazards models. The adverse events were also analyzed.

Results
Sixty-one consecutive patients were examined and followed up for survival. As of November 8, 2021, the median follow-up time was 35.4 months. Disease progression and death occurred in 50 (82%) and 38 (62%) patients, respectively. The median treatment duration of XELOXIRI with or without bevacizumab or cetuximab was 10 cycles (range, 1–12 cycles). The median OS and PFS were 32.2 months (95%CI [24.8–39.6]) and 9.3 months (95% CI [8.1–10.5]), respectively. The ORR of 48 patients with measurable lesions was 70.8%, and the DCR was 89.6%. RAS/BRAF wild-type (HR 0.39; 95% CI [0.16–0.96], p = 0.04) and metastatic organs > 2 (HR 3.25; 95% CI [1.34–7.87], p = 0.009) were independent prognostic factors for OS. The incidence of any grade of adverse events (AEs) was 96.7% (59/61). Grade ≥ 3 AEs included neutropenia (19.7%), leukopenia (9.8%), diarrhea (3.3%), vomiting (3.3%), febrile neutropenia (1.6%), and thrombocytopenia (1.6%). No treatment-related death occurred.

Conclusion
The use of the XELOXIRI regimen with or without a targeted drug was effective, with a manageable toxicity profile in Chinese patients with mCRC.
Cancer risk in persons with new-onset anaemia: a population-based cohort study in Denmark
Authors: Boennelykke Astrid , Jensen Henry , Østgård Lene Sofie Granfeldt , Falborg Alina Zalounina , Hansen Anette Tarp , Christensen Kaj Sparle , Vedsted Peter 
Published: July 2022

Abstract
Background
The time interval from first symptom and sign until a cancer diagnosis significantly affects the prognosis. Therefore, recognising and acting on signs of cancer, such as anaemia, is essential. Evidence is sparse on the overall risk of cancer and the risk of specific cancer types in persons with new-onset anaemia detected in an unselected general practice population. We aimed to assess the risk of cancer in persons with new-onset anaemia detected in general practice, both overall and for selected cancer types.

Methods
This observational population-based cohort study used individually linked electronic data from laboratory information systems and nationwide healthcare registries in Denmark. We included persons aged 40–90 years without a prior history of cancer and with new-onset anaemia (no anaemia during the previous 15 months) detected in general practice in 2014–2018. We measured the incidence proportion and standardised incidence ratios of a new cancer diagnosis (all cancers except for non-melanoma skin cancers) during 12 months follow-up.

Results
A total of 48,925 persons (median [interquartile interval] age, 69 [55–78] years; 55.5% men) were included in the study. In total, 7.9% (95% confidence interval (CI): 7.6 to 8.2) of men and 5.2% (CI: 4.9 to 5.5) of women were diagnosed with cancer during 12 months. Across selected anaemia types, the highest cancer incidence proportion was seen in women with ‘anaemia of inflammation’ (15.3%, CI: 13.1 to 17.5) (ferritin > 100 ng/mL and increased C-reactive protein (CRP)) and in men with ‘combined inflammatory iron deficiency anaemia’ (19.3%, CI: 14.5 to 24.1) (ferritin < 100 ng/mL and increased CRP). For these two anaemia types, the cancer incidence across cancer types was 10- to 30-fold higher compared to the general population.

Conclusions
Persons with new-onset anaemia detected in general practice have a high cancer risk; and markedly high for ‘combined inflammatory iron deficiency anaemia’ and ‘anaemia of inflammation’. Anaemia is a sign of cancer that calls for increased awareness and action. There is a need for research on how to improve the initial pathway for new-onset anaemia in general practice.
Effectiveness of resilience training intervention on psychological capital of the underprivileged widowed women of Fasa City, Iran
Authors: Asadollahi Abdolrahim , Karimpoor Leila , Kaveh Mohammad Hossein , Ghahremani Leila 
Published: July 2022

Abstract
Introduction
Women heads of households (widows or divorcees) are vulnerable groups in society who face various psychological problems and have less resilience than other women. Therefore, the present study was conducted to determine the effectiveness of resilience educational intervention on the psychological capital of poor widows in Fasa city, south Iran.

Materials and Methods
In this quasi-experimental study, 120 widows covered by Imam Khomeini Relief Foundation were selected by a simple random sampling method based on a random Efron algorithm (Efron coin) and randomly assigned into two interventions and control groups (60 people each) in the second half of 2021. About 8 training sessions on resilience and psychological capital were held for the experimental group through training clips, audio transmissions, and podcasts. Data were collected using demographic characteristics forms, widowers' resilience assessment questionnaires, and Luten's psychological capital scale for testing and control before and two months after the intervention. Data were analyzed with Chi-square test, independent and paired t-test using SPSS V. 26 software.

Results
There were differences between the two groups at pretest in demographic variables and psychological capital and its subscales, and resilience and its subscales (P ≥ 0.05).. But two months after the educational intervention, a statistically significant increase was observed in the experimental group in these variables compared to the control group (p < 0.05). It indicates that there was an improvement in the outcomes from pretest to post-test within the intervention and no change in the outcomes over time within the control group.

Discussion and Conclusion
Using a resilience-based educational approach as a novelty of this research to promote psychological capital and resilience can increase resilience and psychological capital in widows. Then, considering the positive effect of this educational approach and the low cost of this intervention, it seems that the implementation of such interventions should be included in the plans related to widows.
A Canadian survey of medical students and undergraduate deans on the management of patients living with obesity
Authors: Katz Nathan J. , Lovrics Olivia , Zevin Boris 
Published: July 2022

Abstract
Background
With over 26% of Canadian adults living with obesity, undergraduate medical education (UGME) should prepare medical students to manage this chronic disease. It is currently unknown how the management of patients living with obesity is taught within UGME curricula in Canada. This study (1) examined the knowledge and self-reported competence of final-year medical students in managing patients living with obesity, and (2) explored how this topic is taught within UGME curricula in Canada.

Methods
We distributed two online surveys: one to final-year medical students, and another to UGME deans at 9 English-speaking medical schools in Canada. The medical student survey assessed students’ knowledge and self-reported competence in managing patients living with obesity. The dean’s survey assessed how management of patients living with obesity is taught within the UGME curriculum.

Results
One hundred thirty-three (6.9%) and 180 (9.3%) out of 1936 eligible students completed the knowledge and self-reported competence parts of the survey, respectively. Mean knowledge score was 10.5 (2.1) out of 18. Students had greatest knowledge about etiology of obesity and goals of treatment, and poorest knowledge about physiology and maintenance of weight loss. Mean self-reported competence score was 2.5 (0.86) out of 4. Students felt most competent assessing diet for unhealthy behaviors and calculating body mass index. Five (56%) out of 9 deans completed the survey. A mean of 14.6 (5.0) curricular hours were spent on teaching management of patients living with obesity. Nutrition and bariatric surgery were most frequently covered topics, with education delivered most often via large-group sessions and clinical activities.

Conclusions
Canadian medical students lack adequate knowledge and feel inadequately prepared to manage patients living with obesity. Changes to UGME curricula may help address this gap in education.
A novel splice site variant c.1183 + 1 G > C in DFNA5 causing autosomal dominant nonsyndromic hearing loss in a Chinese family
Authors: Li Qiong , Wang Shujuan , Liang Pengfei , Li Wei , Wang Jian , Fan Bei , Yang Yang , An Xiaogang , Chen Jun , Zha Dingjun 
Published: July 2022

Abstract
Background
The most frequent clinical presentation of autosomal dominant nonsyndromic hearing loss (ADNSHL) is bilateral, symmetrical, postlingual progressive sensorineural hearing loss, which begins with impairment at high frequencies and eventually progresses to hearing loss at all frequencies. Autosomal dominant deafness-5 (DFNA5) is a subtype of ADNSHL caused by heterozygous variants in the gasdermin E (GSDME, also known as DFNA5) gene.

Methods
Deafness gene NGS panel analysis were performed on the proband of a six-generation Chinese family with hearing loss. The co-segregation analysis between the hearing loss and the novel variant was analyzed by Sanger sequencing and pure-tone audiometry. The minigene splicing assay was performed to evaluate the potential effect of the variant on messenger RNA splicing in vitro.

Results
The family exhibited autosomal dominant, progressive, postlingual, nonsyndromic sensorineural hearing loss, which was similar to that of the previously reported DFNA5 families. A novel heterozygous splice site variant in GSDME gene intron 8 was identified, which co-segregated with the hearing loss phenotype of the family. The variant caused skipping of exon 8 in the mutant transcript, leading to the direct linking of exons 7 and 9.

Conclusions
We identified a novel GSDME splice site variant c.1183 + 1 G > C in an extended Chinese family, which led to the skipping of exon 8. The results extended the pathogenic variants spectrum of the GSDME gene, provided further support for the 'gain-of-function' mechanism of DFNA5, and afforded a molecular interpretation for these patients with ADNSHL.
Peri-event plasma PCSK9 and hsCRP after an acute myocardial infarction correlate with early deterioration of left ventricular ejection fraction: a cohort study
Authors: Silva-Bermúdez Lina S. , Vargas-Villanueva Andrea , Sánchez-Vallejo Carlos A. , Palacio Ana C. , Buitrago Andrés F. , Mendivil Carlos O. 
Published: July 2022

Abstract
Background
It is important to identify patients at increased risk of worsening of left ventricular ejection fraction (LVEF) after a myocardial infarction (MI). We aimed to identify the association of various potential biomarkers with LVEF impairment after an MI in South American patients.

Methods
We studied adult patients admitted to a University Hospital and diagnosed with an acute MI. Plasma concentrations of high-sensitivity C-reactive protein (hsCRP), proprotein convertase subtilisin/kexin type 9 (PCSK9), N-terminal prohormone of brain natriuretic peptide (NT-proBNP) and heart-type fatty-acid-binding protein (FABP3) were determined in samples drawn shortly after the event. Participants had a follow-up visit at least 45 days after the event. The primary endpoint was defined as any decline in LVEF at follow-up relative to baseline.

Results
The study included 106 patients (77.4% men, 22.6% women), mean age was 64.1, mean baseline LVEF was 56.6, 19% had a prior MI. We obtained a follow-up evaluation in 100 (94.4%) of participants, mean follow-up time was 163 days. There was a significant correlation between baseline PCSK9 and hsCRP (r = 0.39, p < 0.001). Baseline hsCRP concentrations were higher in patients who developed the endpoint than in those who did not (32.1 versus 21.2 mg/L, p = 0.066). After multivariate adjustment, baseline PCSK9, male sex and age were significantly associated with impairment in LVEF. The absolute change in LVEF was inversely correlated with baseline hsCRP (standardized coefficient = − 0.246, p = 0.004).

Conclusion
High plasma levels of PCSK9 and hsCRP were associated with early decreases in LVEF after an MI in Latin American patients.