Female authorship positions in health economic evaluations: a cross-sectional analysis.

OBJECTIVE: To investigate the gender of the authors who publish articles of health economic evaluations in medicine and healthcare journals. METHOD: We evaluated a random sample of economic evaluations indexed in MEDLINE during 2019. Gender of the first, last and corresponding author was determined by review of the author's first name. Data were summarized as frequency and percentage for categorical items and median and interquartile range (IQR) for continuous items. We also calculated the index of authors per paper. RESULTS: We included 200 studies with 1365 authors (median of 6 authors per paper; IQR: 4-9). Gender identification was possible for all authors in the study sample: 802 (59%) were men and 563 (41%) were women. The number of female first, last, and corresponding authors respectively were 78 (39%), 68 (34%), and 80 (40%) for health economic evaluations. DISCUSSION: Female scientists were underrepresented as co-authors and in prominent authorship positions in health economic evaluations. This study serves as a call to action for the scientific community to actively work towards equity and inclusion.

Transparency, openness, and reproducible research practices are frequently underused in health economic evaluations.

OBJECTIVES: To investigate the extent to which articles of economic evaluations of healthcare interventions indexed in MEDLINE incorporate research practices that promote transparency, openness, and reproducibility. STUDY DESIGN AND SETTING: We evaluated a random sample of health economic evaluations indexed in MEDLINE during 2019. We included articles written in English reporting an incremental cost-effectiveness ratio in terms of costs per life years gained, quality-adjusted life years, and/or disability-adjusted life years. Reproducible research practices, openness, and transparency in each article were extracted in duplicate. We explored whether reproducible research practices were associated with self-report use of a guideline. RESULTS: We included 200 studies published in 147 journals. Almost half were published as open access articles (n = 93; 47%). Most studies (n = 150; 75%) were model-based economic evaluations. In 109 (55%) studies, authors self-reported use a guideline (e.g., for study conduct or reporting). Few studies (n = 31; 16%) reported working from a protocol. In 112 (56%) studies, authors reported the data needed to recreate the incremental cost-effectiveness ratio for the base case analysis. This percentage was higher in studies using a guideline than studies not using a guideline (72/109 [66%] with guideline vs. 40/91 [44%] without guideline; risk ratio 1.50, 95% confidence interval 1.15-1.97). Only 10 (5%) studies mentioned access to raw data and analytic code for reanalyses. CONCLUSION: Transparency, openness, and reproducible research practices are frequently underused in health economic evaluations. This study provides baseline data to compare future progress in the field.

A cross-sectional analysis identified co-authorship networks and scientific collaboration on reporting guidelines for health research.

OBJECTIVES: To investigate scientific collaboration and citation metrics of reporting guidelines for health research. STUDY DESIGN AND SETTING: A cross-sectional analysis of published articles of reporting guidelines for health research. A search of the EQUATOR (Enhancing the QUAlity and Transparency Of health Research) Network Library (from inception to January 21, 2021) was supplemented by searching websites of guideline developers. For each article, metadata (e.g., authors, institutions, countries, citations) were extracted from the Web of Science and Scopus (up to October 25, 2021). Descriptive analyses were conducted. Network analyses of collaborations were presented. RESULTS: We included 662 articles published in 332 journals. The BMJ (n = 50 articles; 8%), Annals of Internal Medicine (n = 29; 4%), and Journal of Clinical Epidemiology (n = 24; 4%) published the largest number of articles. Four thousand seven hundred twenty two authors, 1,647 institutions, and 83 countries were involved. The global productivity was led by the United States (n = 456 articles), the United Kingdom (n = 414), and Canada (n = 306). We found eight clusters of authors (e.g., one major group with 337 members) and three clusters of institutions (e.g., one major group with 256 members). The most prolific authors were affiliated with the Ottawa Hospital Research Institute (Canada), the University of Ottawa (Canada), the University of Oxford (the United Kingdom), and Stanford University (the United States). CONCLUSION: Our analysis identified key actors producing reporting guidelines, most intense collaborations, and 'citation classics' in the field. These results could potentially be used to strengthen collaborations for developing and disseminating reporting guidelines for health research.

Revisiting systematic geographical variations in tonsils surgery in children in the Spanish National Health System: spatiotemporal ecological study on hospital administrative data.

OBJECTIVE: To provide new evidence on how tonsils surgery in children has geographically varied over time in the context of the Spanish National Health System. DESIGN: Observational ecological spatiotemporal study on geographical variations in medical practice, using linked administrative datasets, including virtually all surgeries performed from 2003 to 2015. SETTING: The Spanish National Health System, a quasi-federal structure with 17 autonomous communities (ACs), and 203 healthcare areas (HCAs). PARTICIPANTS: Patients aged 19 and younger residing in the HCAs and ACs. INTERVENTIONS: Tonsillectomy with adenoidectomy (T&A); and tonsillectomies alone (T). MAIN ENDPOINTS: (1) Evolution of T&A and T rates; (2) spatiotemporal variation in the risk of receiving T&A or T surgery at regional level (ACs) and HCAs; and (3) the fraction of the variation (FV) attributed to each of the components of variation-ACs, HCAs, year and interaction ACs year. RESULTS: T&A age-sex standardised rates increased over the period of analysis from 15.2 to 20.9 (5.7 points per 10 000 inhabitants). T alone remained relatively lower than T&A rates, evolving from 3.6 in 2003 to 3.9 in 2015 (0.3 points per 10 000 inhabitants). Most of the risk variation was captured at the HCAs level in both procedures (FV: 55.3% in T&A and 72.5% in T). The ACs level explained 27.6% of the FV in the risk in T&A versus 8% in T. The interaction ACs year was similar in both procedures (FV: 15.5% in T&A and 17.5% in T). The average trend hardly explained 1.46% and 1.83% of the variation, respectively. CONCLUSION: Our study showed wide persistent variations with a steady increase in rates and risk of T&A and a stagnation of T alone, where most of the variation risk was explained at HCA level.

Design and methodological characteristics of studies using observational routinely collected health data for investigating the link between cancer and neurodegenerative diseases: protocol for a meta-research study.

INTRODUCTION: Health services generate large amounts of routine health data (eg, administrative databases, disease registries and electronic health records), which have important secondary uses for research. Increases in the availability and the ability to access and analyse large amounts of data represent a major opportunity for conducting studies on the possible relationships between complex diseases. The objective of this study will be to evaluate the design, methods and reporting of studies conducted using observational routinely collected health data for investigating the link between cancer and neurodegenerative diseases. METHODS AND ANALYSIS: This is the protocol for a meta-research study. We registered the study protocol within the Open Science Framework: https://osf.io/h2qjg. We will evaluate observational studies (eg, cohort and case-control) conducted using routinely collected health data for investigating the associations between cancer and neurodegenerative diseases (such as Alzheimer's disease, amyotrophic lateral sclerosis/motor neuron disease, Huntington's disease, multiple sclerosis and Parkinson's disease). The following electronic databases will be searched (from their inception onwards): MEDLINE, Embase and Web of Science Core Collection. Screening and selection of articles will be conducted by at least two researchers. Potential discrepancies will be resolved via discussion. Design, methods and reporting characteristics in each article will be extracted using a standardised data extraction form. Information on general, methodological and transparency items will be reported. We will summarise our findings with tables and graphs (eg, bar charts, forest plots). ETHICS AND DISSEMINATION: Due to the nature of the proposed study, no ethical approval will be required. We plan to publish the full study in an open access peer-reviewed journal and disseminate the findings at scientific conferences and via social media. All data will be deposited in a cross-disciplinary public repository.

Mortality in Persons With Autism Spectrum Disorder or Attention-Deficit/Hyperactivity Disorder: A Systematic Review and Meta-analysis.

IMPORTANCE: Autism spectrum disorder (ASD) and attention-deficit/hyperactivity disorder (ADHD) are childhood-onset disorders that may persist into adulthood. Several studies have suggested that they may be associated with an increased risk of mortality; however, the results are inconsistent. OBJECTIVE: To assess the risk of mortality among persons with ASD or ADHD and their first-degree relatives. DATA SOURCES: A search of MEDLINE, Embase, Scopus, Web of Science, and PsycINFO (published from inception to April 1, 2021) was supplemented by searching reference lists of the retrieved articles. STUDY SELECTION: Cohort and case-control studies that reported mortality rate ratios (RRs) in persons with ASD or ADHD and/or their first-degree relatives compared with the general population or those without ASD/ADHD were included. DATA EXTRACTION AND SYNTHESIS: Screening, data extraction, and quality assessment were performed by at least 2 researchers independently. A random-effects model was used to meta-analyze individual studies and assessed heterogeneity (I2). MAIN OUTCOMES AND MEASURES: All-cause mortality in association with ASD or ADHD. Secondary outcome was cause-specific mortality. RESULTS: Twenty-seven studies were included, with a total of 642 260 individuals. All-cause mortality was found to be higher for persons with ASD (154 238 participants; 12 studies; RR, 2.37; 95% CI, 1.97-2.85; I2, 89%; moderate confidence) and persons with ADHD (396 488 participants; 8 studies; RR, 2.13; 95% CI, 1.13-4.02; I2, 98%; low confidence) than for the general population. Among persons with ASD, deaths from natural causes (4 studies; RR, 3.80; 95% CI, 2.06-7.01; I2, 96%; low confidence) and deaths from unnatural causes were increased (6 studies; RR, 2.50; 95% CI, 1.49-4.18; I2, 95%; low confidence). Among persons with ADHD, deaths from natural causes were not significantly increased (4 studies; RR, 1.62; 95% CI, 0.89-2.96; I2, 88%; low confidence), but deaths from unnatural causes were higher than expected (10 studies; RR, 2.81; 95% CI, 1.73-4.55; I2, 92%; low confidence). CONCLUSIONS AND RELEVANCE: This systematic review and meta-analysis found that ASD and ADHD are associated with a significantly increased risk of mortality. Understanding the mechanisms of these associations may lead to targeted strategies to prevent avoidable deaths in high-risk groups. The substantial heterogeneity between studies should be explored further.

Reproducible research practices, openness and transparency in health economic evaluations: study protocol for a cross-sectional comparative analysis.

INTRODUCTION: There has been a growing awareness of the need for rigorously and transparent reported health research, to ensure the reproducibility of studies by future researchers. Health economic evaluations, the comparative analysis of alternative interventions in terms of their costs and consequences, have been promoted as an important tool to inform decision-making. The objective of this study will be to investigate the extent to which articles of economic evaluations of healthcare interventions indexed in MEDLINE incorporate research practices that promote transparency, openness and reproducibility. METHODS AND ANALYSIS: This is the study protocol for a cross-sectional comparative analysis. We registered the study protocol within the Open Science Framework (osf.io/gzaxr). We will evaluate a random sample of 600 cost-effectiveness analysis publications, a specific form of health economic evaluations, indexed in MEDLINE during 2012 (n=200), 2019 (n=200) and 2022 (n=200). We will include published papers written in English reporting an incremental cost-effectiveness ratio in terms of costs per life years gained, quality-adjusted life years and/or disability-adjusted life years. Screening and selection of articles will be conducted by at least two researchers. Reproducible research practices, openness and transparency in each article will be extracted using a standardised data extraction form by multiple researchers, with a 33% random sample (n=200) extracted in duplicate. Information on general, methodological and reproducibility items will be reported, stratified by year, citation of the Consolidated Health Economic Evaluation Reporting Standards (CHEERS) statement and journal. Risk ratios with 95% CIs will be calculated to represent changes in reporting between 2012-2019 and 2019-2022. ETHICS AND DISSEMINATION: Due to the nature of the proposed study, no ethical approval will be required. All data will be deposited in a cross-disciplinary public repository. It is anticipated the study findings could be relevant to a variety of audiences. Study findings will be disseminated at scientific conferences and published in peer-reviewed journals.

Prevalence and comorbidity of autism spectrum disorder in Spain: study protocol for a systematic review and meta-analysis of observational studies.

BACKGROUND: Autism spectrum disorder (ASD) is a complex developmental disorder characterised by impaired social interaction and communication, and restrictive and repetitive behaviour. Previous systematic reviews have traditionally assessed the prevalence of ASD on global or regional context, with very few meta-analyses at the country level. The objective of this study will be to systematically evaluate published and unpublished observational studies that present prevalence and comorbidity of ASD among children, adolescent and adult population in Spain. METHODS/DESIGN: We designed and registered a study protocol for a systematic review and meta-analysis of descriptive epidemiology data. Observational studies (cohort, cross-sectional) reporting the prevalence of ASD and conducted in a wide range of people (e.g. general population, outpatient and/or school settings) will be included. The primary outcome will be the prevalence of ASD. Secondary outcomes will be the prevalence of any physical or mental comorbidity in association with ASD. No limitations will be imposed on publication status, study conduct period, and language of dissemination. Comprehensive literature searches will be conducted in seven electronic databases (from January 1980 onwards), including PubMed/MEDLINE, EMBASE, Scopus, Web of Science, PsycINFO, IME-Spanish Medical Index and IBECS-Spanish Bibliographic Index of Health Sciences. Grey literature will be identified through searching dissertation databases, Google Scholar and conference abstracts. Two team members will independently screen all citations, full-text articles, and abstract data. Potential conflicts will be resolved through discussion. The study methodological quality (or bias) will be appraised using an appropriate tool. If feasible, we will conduct random effects meta-analysis of observational data. Prevalence estimates will be stratified according to gender, age and geographical location. Additional analyses will be conducted to explore the potential sources of heterogeneity (e.g. methodological quality, sample size, diagnostic criteria). DISCUSSION: This systematic review and meta-analysis of observational data will identify, evaluate and integrate the epidemiological knowledge underlying the prevalence of ASD in Spain. The results of this study will be of interest to multiple audiences including patients, their families, caregivers, healthcare professional, scientists and policy makers. Results will be published in a peer-reviewed journal. Implications for future epidemiological research will be discussed. SYSTEMATIC REVIEW REGISTRATION: PROSPERO CRD42018090372.

Association of Anorexia Nervosa With Risk of Cancer: A Systematic Review and Meta-analysis.

Importance: Anorexia nervosa is recognized as an important cause of morbidity in young people. However, the risk of cancer in people with anorexia nervosa remains uncertain. Objective: To evaluate the association of anorexia nervosa with the risk of developing or dying of cancer. Data Sources: MEDLINE, Scopus, Embase, and Web of Science from database inception to January 9, 2019. Study Selection: Published observational studies in humans examining the risk of cancer in people with anorexia nervosa compared with the general population or those without anorexia nervosa. Studies needed to report incidence or mortality rate ratios (RRs). Data Extraction and Synthesis: Screening, data extraction, and methodological quality assessment were performed by at least 2 researchers independently. A random-effects model was used to synthesize individual studies. Heterogeneity (I2) was assessed and 95% prediction intervals (PIs) were calculated. Main Outcomes and Measures: All cancer incidence and cancer mortality associated with anorexia nervosa. Secondary outcomes were site-specific cancer incidence and mortality. Results: Seven cohort studies published in 10 articles (42 602 participants with anorexia nervosa) were included. Anorexia nervosa was not associated with risk of developing any cancer (4 studies in women; RR, 0.97; 95% CI, 0.89-1.06; P = .53; I2, 0%; 95% PI, 0.80-1.18; moderate confidence). Anorexia nervosa was associated with decreased breast cancer incidence (5 studies in women; RR, 0.60; 95% CI, 0.50-0.80; P < .001; I2, 0%; 95% PI, 0.44-0.83; high confidence). Conversely, anorexia nervosa was associated with increased risk of developing lung cancer (3 studies in women; RR, 1.50; 95% CI, 1.06-2.12; P = .001; I2, 0%; 95% PI, 0.19-16.46; low confidence) and esophageal cancer (2 studies in women; RR, 6.10; 95% CI, 2.30-16.18; P < .001; I2, 0%; low confidence). Conclusions and Relevance: Among people with anorexia nervosa, risk of developing cancer did not differ compared with the general population, but a significantly reduced risk of breast cancer was observed. Understanding the mechanisms underlying these associations could have important preventive potential.

Apoyo a las guías de publicación para estudios de evaluación económica por parte de las revistas biomédicas españolas / Endorsement of guidelines for reporting economic evaluation studies by Spanish biomedical journals

Objetivo: Examinar el apoyo a las guías de publicación para estudios de evaluación económica, como la declaración CHEERS (Consolidated Health Economic Evaluation Reporting Standards), por parte de las revistas biomédicas españolas. Método: Análisis transversal de las normas de autoría de las revistas biomédicas españolas incluidas en Journal Citation Reports 2017. Dos autores examinaron y extrajeron la siguiente información: mención de alguna guía de publicación, declaración CHEERS, recomendaciones del International Committee of Medical Journal Editors (ICMJE) e iniciativa Enhancing the QUAlity and Transparency Of health Research (EQUATOR). Resultados: De las 28 revistas incluidas, 23 (82,1%; intervalo de confianza del 95% [IC95%]: 63,1-93,9%) mencionaron alguna guía. Una única revista mencionó la declaración CHEERS para estudios de evaluación económica. Veinticuatro revistas (85,7%; IC95%: 67,3-96,0%) mencionaron las recomendaciones del ICMJE y 8 (28,6%; IC95%: 13,2-48,7%) la red EQUATOR. La declaración CONSORT (Consolidated Standards of Reporting Trials) para ensayos clínicos fue la guía más mencionada (n=21; 75,0%; IC95%: 55,1-89,3%). Discusión: La mayoría de las normas de autoría no incorporan información sobre cómo presentar evaluaciones económicas. Las revistas deberían apoyar el cumplimiento de las guías de publicación por parte de las personas autoras y revisoras

Objective: To examine the endorsement of reporting guidelines for economic evaluation studies, such as the CHEERS (Consolidated Health Economic Evaluation Reporting Standards) statement, by Spanish biomedical journals. Method: Cross-sectional analysis of the instructions to authors of Spanish biomedical journals included in the Journal Citation Reports 2017. Two authors examined and extracted the following information: mention of any reporting guideline, the CHEERS statement, the recommendations of the International Committee of Medical Journal Directors (ICMJE) and the Enhancing the QUAlity and Transparency Of health Research (EQUATOR) network. Results: Of the 28 journals included, 23 (82.1%; 95% confidence interval [95%CI]: 63.1-93.9%) mentioned at least one reporting guideline in the instructions to authors. Only one journal mentioned the CHEERS statement for health economic evaluations. Twenty-four journals (85.7%; 95%CI: 67.3-96.0%) mentioned the ICMJE recommendations and 8 (28.6%; 95%CI: 13.2-48.7%) mentioned the EQUATOR network. The CONSORT (Consolidated Standards of Reporting Trials) statement for clinical trials was the most- mentioned reporting guideline (n=21; 75.0%; 95%CI: 55.1-89.3%). Discussion: Most of the instructions to authors do not provide guidance on how to report economic evaluations. Journals should support compliance with reporting guidelines by authors and peer-reviewers