bluuebunny/crossref_metadata_2025_split huggingface dataset:
Feasibility of asynchronous video-based telemedicine in the diagnosis and management of paediatric blepharoptosis
Authors: Souverein Erik A , Kim Jonathan W , Loudin Natasha N , Johnston Julia , Stewart Carly , Reid Mark W , Lee Thomas C , Nallasamy Sudha 
Published: January 2021

Introduction The purpose of this study was to assess the validity of using video glasses as part of an asynchronous telemedicine screening protocol for paediatric blepharoptosis. Methods A physician assistant wearing Pivothead SMART Series glasses recorded videos of paediatric patients referred for blepharoptosis in primary, down and upgaze while holding a ruler next to the eyes. An oculoplastic surgeon viewed the stored videos and recorded margin-reflex distance 1 and levator function. Using these measurements, the surgeon determined whether surgical intervention was recommended and, if so, which procedure was recommended. The surgeon recorded the same parameters for each patient based on an in-person examination performed later that day. Videos were reviewed eight months later and the same parameters were recorded. Results Twenty-nine children ( n = 58 eyes) were enrolled. Margin-reflex distance 1 and levator function measurements based on same-day video review agreed with in-person examination 94.8% (intraclass correlation coefficient = 0.82) and 98.3% (intraclass correlation coefficient = 0.96) of the time, respectively. Margin-reflex distance 1 and levator function measurements based on later video review agreed with in-person examination 93.1% (intraclass correlation coefficient = 0.85) and 94.8% (intraclass correlation coefficient = 0.93) of the time, respectively. Agreement in identifying surgical candidates was almost perfect (= = 0.93) for same-day video review and substantial (= = 0.73) for later video review. Sensitivity of identifying surgical patients was 100% for both same-day video review and later video review; though specificity was lower at 94.1% for same-day video review and 76.5% for later video review. Discussion Asynchronous telemedicine encounters employing video glasses are a useful screening modality for identifying surgical paediatric blepharoptosis patients.
Interrater and Intrarater Reliability of the Beighton Score: A Systematic Review
Authors: Bockhorn Lauren N. , Vera Angelina M. , Dong David , Delgado Domenica A. , Varner Kevin E. , Harris Joshua D. 
Published: January 2021

Background: The Beighton score is commonly used to assess the degree of hypermobility in patients with hypermobility spectrum disorder. Since proper diagnosis and treatment in this challenging patient population require valid, reliable, and responsive clinical assessments such as the Beighton score, studies must properly evaluate efficacy and effectiveness. Purpose: To succinctly present a systematic review to determine the inter- and intrarater reliability of the Beighton score and the methodological quality of all analyzed studies for use in clinical applications. Study Design: Systematic review; Level of evidence, 3. Methods: A systematic review of the MEDLINE, Embase, CINAHL, and SPORTDiscus databases was performed. Studies that measured inter- or intrarater reliability of the Beighton score in humans with and without hypermobility were included. Non-English, animal, cadaveric, level 5 evidence, and studies utilizing the Beighton score self-assessment version were excluded. Data were extracted to compare scoring methods, population characteristics, and measurements of inter- and intrarater reliability. Risk of bias was assessed with the COSMIN (Consensus-Based Standards for the Selection of Health Measurement Instruments) 2017 checklist. Results: Twenty-four studies were analyzed (1333 patients; mean ± SD age, 28.19 ± 17.34 years [range, 4-71 years]; 640 females, 594 males, 273 unknown sex). Of the 24 studies, 18 reported raters were health care professionals or health care professional students. For interrater reliability, 5 of 8 (62.5%) intraclass correlation coefficients and 12 of 19 (63.2%) kappa values were substantial to almost perfect. Intrarater reliability was reported as excellent in all studies utilizing intraclass correlation coefficients, and 3 of the 7 articles using kappa values reported almost perfect values. Utilizing the COSMIN criteria, we determined that 1 study met “very good” criteria, 7 met “adequate,” 15 met “doubtful,” and 1 met “inadequate” for overall risk of bias in the reliability domain. Conclusion: The Beighton score is a highly reliable clinical tool that shows substantial to excellent inter- and intrarater reliability when used by raters of variable backgrounds and experience levels. While individual components of risk of bias among studies demonstrated large discrepancy, most of the items were adequate to very good.
Surgical Treatment of Isolated Meniscal Tears in Competitive Male Wrestlers: Reoperations, Outcomes, and Return to Sport
Authors: Marigi Erick M. , Keyt Lucas K. , LaPrade Matthew D. , Camp Christopher L. , Levy Bruce A. , Dahm Diane L. , Stuart Michael J. , Krych Aaron J. 
Published: January 2021

Background: Wrestlers are highly active, young athletes prone to meniscal injuries that often require surgery. However, there is a lack of data evaluating the results of meniscal repair or partial meniscectomy in this cohort. Purpose: To describe the outcomes (subjective function, return to play, complications, reoperation rates, and progression of osteoarthritis) for treatment (meniscectomy or repair) of meniscal injuries in a cohort of competitive wrestlers. Study Design: Case series; Level of evidence, 4. Methods: All competitive wrestlers (high school, collegiate, or professional leagues) with a history of a meniscal injury and isolated meniscal surgery at a single institution between 2001 and 2017 were retrospectively identified. Failure was defined as a reinjury of the operative meniscus by clinical or advanced imaging examination, reoperation, or any additional surgical treatment of the meniscus after the index procedure. All patients were contacted for determination of reinjury rates, current sport status, and International Knee Documentation Committee and Tegner activity scores. Results: Of 85 male wrestlers with isolated meniscal tears, 34% underwent a meniscal repair, and 66% received a partial meniscectomy. Index surgery failed for 9.4% of the cohort. Among wrestlers treated with initial meniscal repair, 21% required a subsequent partial meniscectomy at a mean 2.2 years, and of those treated with partial meniscectomy, 3% underwent a second operation ( P < .001). All secondary operations were revision partial meniscectomies occurring at a mean 3.2 years (95% CI, 0.01-6.4 years) after the index procedure. At final follow-up, 89% of patients were able to return to sport, with 65% returning to wrestling competition. There was significant improvement in the Tegner score from a mean 6.5 (95% CI, 5.9-7.2) preoperatively to 8.3 (95% CI 8.0-8.6) postoperatively ( P < .001). Conclusion: The reoperation rate after meniscal surgery in wrestlers was quite low, but only 65% returned to competitive wrestling. Meniscal repair and partial meniscectomy improved patient-reported outcomes and activity levels at short-term follow-up. However, 21% of wrestlers treated with initial meniscal repair required a subsequent partial meniscectomy at a mean 2.2 years.
Occurrence of anatase in reworking altered ash beds (K-bentonites and tonsteins) and discrimination of source magmas: a case study of terrestrial Permian–Triassic boundary successions in China
Authors: Hong Hanlie , Jin Xiaoxue , Wan Miao , Ji Kaipeng , Liu Chen , Algeo Thomas J. , Fang Qian 
Published: January 2021

AbstractPotential secondary influences on titanium distribution should be evaluated when using ash beds as volcanic source indicators and for correlation purposes. In this study, well-correlated altered ash beds in Permian–Triassic boundary (PTB) successions of various facies in South China were investigated to better understand their use in source discrimination and stratigraphic correlation. The ash beds deposited in lacustrine and paludal facies contain significantly more Ti relative to deposits in marine facies. Neoformed anatase grains nanometres to micrometres in size are associated closely with clay minerals, whereas detrital anatase was observed in the remnants of altered ash beds of terrestrial facies. Extraction of the clay fraction of altered ash beds may exclude significantly detrital accessory minerals such as anatase and rutile added during sediment reworking, and the concentrations of immobile elements in the clay fraction may therefore be used to interpret more effectively their source igneous rocks.
Genetic ancestry and skeletal toxicities among childhood acute lymphoblastic leukemia patients in the DFCI 05-001 cohort
Authors: Yao Song , Zhu Qianqian , Cole Peter D. , Stevenson Kristen , Harris Marian H. , Schultz Emily , Kahn Justine M. , Ladas Elena J. , Athale Uma H. , Clavell Luis A. , Laverdiere Caroline , Leclerc Jean-Marie , Michon Bruno , Schorin Marshall A. , Welch Jennifer J. G. , Sallan Stephen E. , Silverman Lewis B. , Kelly Kara M. 
Published: January 2021

Abstract
Hispanic children have a higher incidence of acute lymphoblastic leukemia (ALL) and inferior treatment outcomes relative to non-Hispanic White children. We previously reported that Hispanic children with ALL had lower risk of fracture and osteonecrosis. To unravel the genetic root of such ethnic differences, we genotyped 449 patients from the DFCI 05-001 cohort and analyzed their ancestry. Patients with discordant clinical and genetic ancestral groups were reclassified, and those with unknown ancestry were reassigned on the basis of genetic estimates. Both clinical and genetic ancestries were analyzed in relation to risk of bone toxicities and survival outcomes. Consistent with clinically reported race/ethnicity, genetically defined Hispanic and Black patients had significantly lower risk of fracture (Hispanic: subdistribution hazard ratio [SHR], 0.42; 95% confidence interval [CI], 0.22-0.81; P = .01; Black: SHR, 0.28; 95% CI, 0.10-0.75; P = .01), and osteonecrosis (Hispanic: SHR, 0.12; 95% CI, 0.02-0.93; P = .04; Black: SHR, 0.24; 95% CI, 0.08-0.78; P = .02). The lower risk was driven by African but not Native American or Asian ancestry. In addition, patients with a higher percentage of Native American ancestry had significantly poorer overall survival and event-free survival. Our study revealed that the lower risk of bone toxicities among Black and Hispanic children treated for ALL was attributed, in part, to the percentage of African ancestry in their genetic admixture. The findings provide suggestive evidence for the protective effects of genetic factors associated with African decent against bone damage caused by ALL treatment and clues for future studies to identify underlying biological mechanisms.
IL-17A from innate and adaptive lymphocytes contributes to inflammation and damage in cystic fibrosis lung disease
Authors: Hagner Matthias , Albrecht Melanie , Guerra Matteo , Braubach Peter , Halle Olga , Zhou-Suckow Zhe , Butz Simone , Jonigk Danny , Hansen Gesine , Schultz Carsten , Dittrich Anna-Maria , Mall Marcus A. 
Published: January 2021

BackgroundElevated levels of interleukin (IL)-17A were detected in the airways of patients with cystic fibrosis (CF), but its cellular sources and role in the pathogenesis of CF lung disease remain poorly understood. The aim of this study was to determine the sources of IL-17A and its role in airway inflammation and lung damage in CF.MethodsWe performed flow cytometry to identify IL-17A-producing cells in lungs and peripheral blood from CF patients and β-epithelial Na+ channel transgenic (Scnn1b-Tg) mice with CF-like lung disease, and determined the effects of genetic deletion of Il17a and Rag1 on the pulmonary phenotype of Scnn1b-Tg mice.ResultsT-helper 17 cells, CD3+CD8+ T-cells, γδ T-cells, invariant natural killer T-cells and innate lymphoid cells contribute to IL-17A secretion in lung tissue, lymph nodes and peripheral blood of patients with CF. Scnn1b-Tg mice displayed increased pulmonary expression of Il17a and elevated IL-17A-producing innate and adaptive lymphocytes with a major contribution by γδ T-cells. Lack of IL-17A, but not the recombination activating protein RAG1, reduced neutrophilic airway inflammation in Scnn1b-Tg mice. Genetic deletion of Il17a or Rag1 had no effect on mucus obstruction, but reduced structural lung damage and revealed an IL-17A-dependent macrophage activation in Scnn1b-Tg mice.ConclusionsWe identify innate and adaptive sources of IL-17A in CF lung disease. Our data demonstrate that IL-17A contributes to airway neutrophilia, macrophage activation and structural lung damage in CF-like lung disease in mice. These results suggest IL-17A as a novel target for anti-inflammatory therapy of CF lung disease.
High eosinophil counts predict decline in FEV1: results from the CanCOLD study
Authors: Tan Wan C. , Bourbeau Jean , Nadeau Gilbert , Wang Wendy , Barnes Neil , Landis Sarah H. , Kirby Miranda , Hogg James C. , Sin Don D. 
Published: January 2021

IntroductionThe aim of this study was to examine the association between blood eosinophil levels and the decline in lung function in individuals aged >40 years from the general population.MethodsThe study evaluated the eosinophil counts from thawed blood in 1120 participants (mean age 65 years) from the prospective population-based Canadian Cohort of Obstructive Lung Disease (CanCOLD) study. Participants answered interviewer-administered respiratory questionnaires and performed pre-/post-bronchodilator spirometric tests at 18-month intervals; computed tomography (CT) imaging was performed at baseline. Statistical analyses to describe the relationship between eosinophil levels and decline in forced expiratory volume in 1 s (FEV1) were performed using random mixed-effects regression models with adjustments for demographics, smoking, baseline FEV1, ever-asthma and history of exacerbations in the previous 12 months. CT measurements were compared between eosinophil subgroups using ANOVA.ResultsParticipants who had a peripheral eosinophil count of ≥300 cells·µL−1(n=273) had a greater decline in FEV1compared with those with eosinophil counts of <150 cells·µL−1(n=430; p=0.003) (reference group) and 150–<300 cells·µL−1(n=417; p=0.003). The absolute change in FEV1was −32.99 mL·year−1for participants with eosinophil counts <150 cells·µL−1; −38.78 mL·year−1for those with 150–<300 cells·µL−1and −67.30 mL·year−1for participants with ≥300 cells·µL−1. In COPD, higher eosinophil count was associated with quantitative CT measurements reflecting both small and large airway abnormalities.ConclusionA blood eosinophil count of ≥300 cells·µL−1is an independent risk factor for accelerated lung function decline in older adults and is related to undetected structural airway abnormalities.
Markers of disease activity in COPD: an 8-year mortality study in the ECLIPSE cohort
Authors: Celli Bartolome , Locantore Nicholas , Yates Julie C. , Bakke Per , Calverley Peter M.A. , Crim Courtney , Coxson Harvey O. , Lomas David A. , MacNee William , Miller Bruce E. , Mullerova Hana , Rennard Stephen I. , Silverman Edwin K. , Wouters Emiel , Tal-Singer Ruth , Agusti Alvar , Vestbo Jørgen 
Published: January 2021

RationaleThere are no validated measures of disease activity in COPD. Since “active” disease is expected to have worse outcomes (e.g.mortality), we explored potential markers of disease activity in patients enrolled in the ECLIPSE cohort in relation to 8-year all-cause mortality.MethodsWe investigated 1) how changes in relevant clinical variables over time (1 or 3 years) relate to 8-year mortality; 2) whether these variables inter-relate; and 3) if any clinical, imaging and/or biological marker measured cross-sectionally at baseline relates to any activity component.ResultsResults showed that 1) after 1 year, hospitalisation for COPD, exacerbation frequency, worsening of body mass index, airflow obstruction, dyspnoea and exercise (BODE) index or health status (St George's Respiratory Questionnaire (SGRQ)) and persistence of systemic inflammation were significantly associated with 8-year mortality; 2) at 3 years, the same markers, plus forced expiratory volume in 1 s (FEV1) decline and to a lesser degree computed tomography (CT) emphysema, showed association, thus qualifying as markers of disease activity; 3) changes in FEV1, inflammatory cytokines and CT emphysema were not inter-related, while the multidimensional indices (BODE and SGRQ) showed modest correlations; and 4) changes in these markers could not be predicted by any baseline cross-sectional measure.ConclusionsIn COPD, 1- and 3-year changes in exacerbation frequency, systemic inflammation, BODE and SGRQ scores and FEV1decline are independent markers of disease activity associated with 8-year all-cause mortality. These disease activity markers are generally independent and not predictable from baseline measurements.
TLR2-mediated innate immune priming boosts lung anti-viral immunity
Authors: Girkin Jason , Loo Su-Ling , Esneau Camille , Maltby Steven , Mercuri Francesca , Chua Brendon , Reid Andrew T. , Veerati Punnam Chander , Grainge Chris L. , Wark Peter A.B. , Knight Darryl , Jackson David , Demaison Christophe , Bartlett Nathan W. 
Published: January 2021

BackgroundWe assessed whether Toll-like receptor (TLR)2 activation boosts the innate immune response to rhinovirus infection, as a treatment strategy for virus-induced respiratory diseases.MethodsWe employed treatment with a novel TLR2 agonist (INNA-X) prior to rhinovirus infection in mice, and INNA-X treatment in differentiated human bronchial epithelial cells derived from asthmatic-donors. We assessed viral load, immune cell recruitment, cytokines, type I and III interferon (IFN) production, as well as the lung tissue and epithelial cell immune transcriptome.ResultsWe show,in vivo, that a single INNA-X treatment induced innate immune priming characterised by low-level IFN-λ, Fas ligand, chemokine expression and airway lymphocyte recruitment. Treatment 7 days before infection significantly reduced lung viral load, increased IFN-β/λ expression and inhibited neutrophilic inflammation. Corticosteroid treatment enhanced the anti-inflammatory effects of INNA-X. Treatment 1 day before infection increased expression of 190 lung tissue immune genes. This tissue gene expression signature was absent with INNA-X treatment 7 days before infection, suggesting an alternate mechanism, potentiallyviaestablishment of immune cell-mediated mucosal innate immunity.In vitro, INNA-X treatment induced a priming response defined by upregulated IFN-λ, chemokine and anti-microbial gene expression that preceded an accelerated response to infection enriched for nuclear factor (NF)-κB-regulated genes and reduced viral loads, even in epithelial cells derived from asthmatic donors with intrinsic delayed anti-viral immune response.ConclusionAirway epithelial cell TLR2 activation induces prolonged innate immune priming, defined by early NF-κB activation, IFN-λ expression and lymphocyte recruitment. This response enhanced anti-viral innate immunity and reduced virus-induced airway inflammation.
Machine learning can predict disease manifestations and outcomes in lymphangioleiomyomatosis
Authors: Chernbumroong Saisakul , Johnson Janice , Gupta Nishant , Miller Suzanne , McCormack Francis X. , Garibaldi Jonathan M. , Johnson Simon R. 
Published: January 2021

BackgroundLymphangioleiomyomatosis (LAM) is a rare multisystem disease with variable clinical manifestations and differing rates of progression that make management decisions and giving prognostic advice difficult. We used machine learning to identify clusters of associated features which could be used to stratify patients and predict outcomes in individuals.Patients and methodsUsing unsupervised machine learning we generated patient clusters using data from 173 women with LAM from the UK and 186 replication subjects from the US National Heart, Lung, and Blood Institute (NHLBI) LAM registry. Prospective outcomes were associated with cluster results.ResultsTwo- and three-cluster models were developed. A three-cluster model separated a large group of subjects presenting with dyspnoea or pneumothorax from a second cluster with a high prevalence of angiomyolipoma symptoms (p=0.0001) and tuberous sclerosis complex (TSC) (p=0.041). Patients in the third cluster were older, never presented with dyspnoea or pneumothorax (p=0.0001) and had better lung function. Similar clusters were reproduced in the NHLBI cohort. Assigning patients to clusters predicted prospective outcomes: in a two-cluster model the future risk of pneumothorax was 3.3 (95% CI 1.7–5.6)-fold greater in cluster 1 than cluster 2 (p=0.0002). Using the three-cluster model, the need for intervention for angiomyolipoma was lower in clusters 2 and 3 than cluster 1 (p<0.00001). In the NHLBI cohort, the incidence of death or lung transplant was much lower in clusters 2 and 3 (p=0.0045).ConclusionsMachine learning has identified clinically relevant clusters associated with complications and outcome. Assigning individuals to clusters could improve decision making and prognostic information for patients.
Long-term exposure to low-level air pollution and incidence of asthma: the ELAPSE project
Authors: Liu Shuo , Jørgensen Jeanette Therming , Ljungman Petter , Pershagen Göran , Bellander Tom , Leander Karin , Magnusson Patrik K.E. , Rizzuto Debora , Hvidtfeldt Ulla A. , Raaschou-Nielsen Ole , Wolf Kathrin , Hoffmann Barbara , Brunekreef Bert , Strak Maciej , Chen Jie , Mehta Amar , Atkinson Richard W. , Bauwelinck Mariska , Varraso Raphaëlle , Boutron-Ruault Marie-Christine , Brandt Jørgen , Cesaroni Giulia , Forastiere Francesco , Fecht Daniela , Gulliver John , Hertel Ole , de Hoogh Kees , Janssen Nicole A.H. , Katsouyanni Klea , Ketzel Matthias , Klompmaker Jochem O. , Nagel Gabriele , Oftedal Bente , Peters Annette , Tjønneland Anne , Rodopoulou Sophia P. , Samoli Evangelia , Kristoffersen Doris Tove , Sigsgaard Torben , Stafoggia Massimo , Vienneau Danielle , Weinmayr Gudrun , Hoek Gerard , Andersen Zorana Jovanovic 
Published: January 2021

BackgroundLong-term exposure to ambient air pollution has been linked to childhood-onset asthma, although evidence is still insufficient. Within the multicentre project Effects of Low-Level Air Pollution: A Study in Europe (ELAPSE), we examined the associations of long-term exposures to particulate matter with a diameter <2.5 µm (PM2.5), nitrogen dioxide (NO2) and black carbon (BC) with asthma incidence in adults.MethodsWe pooled data from three cohorts in Denmark and Sweden with information on asthma hospital diagnoses. The average concentrations of air pollutants in 2010 were modelled by hybrid land-use regression models at participants’ baseline residential addresses. Associations of air pollution exposures with asthma incidence were explored with Cox proportional hazard models, adjusting for potential confounders.ResultsOf 98 326 participants, 1965 developed asthma during a mean follow-up of 16.6 years. We observed associations in fully adjusted models with hazard ratios of 1.22 (95% CI 1.04–1.43) per 5 μg·m−3 for PM2.5, 1.17 (95% CI 1.10–1.25) per 10 µg·m−3 for NO2 and 1.15 (95% CI 1.08–1.23) per 0.5×10−5 m−1 for BC. Hazard ratios were larger in cohort subsets with exposure levels below the European Union and US limit values and possibly World Health Organization guidelines for PM2.5 and NO2. NO2 and BC estimates remained unchanged in two-pollutant models with PM2.5, whereas PM2.5 estimates were attenuated to unity. The concentration–response curves showed no evidence of a threshold.ConclusionsLong-term exposure to air pollution, especially from fossil fuel combustion sources such as motorised traffic, was associated with adult-onset asthma, even at levels below the current limit values.