bluuebunny/crossref_metadata_2025_split huggingface dataset:
An Adaptive Response Surface Methodology Based on Active Subspaces for Mixed Random and Interval Uncertainties
Authors: Hu Xingzhi , Duan Yanhui , Wang Ruili , Liang Xiao , Chen Jiangtao 
Published: October 2019

Abstract
The popular use of response surface methodology (RSM) accelerates the solutions of parameter identification and response analysis issues. However, accurate RSM models subject to aleatory and epistemic uncertainties are still challenging to construct, especially for multidimensional inputs, which is widely existed in real-world problems. In this study, an adaptive interval response surface methodology (AIRSM) based on extended active subspaces is proposed for mixed random and interval uncertainties. Based on the idea of subspace dimension reduction, extended active subspaces are given for mixed uncertainties, and interval active variable representation is derived for the construction of AIRSM. A weighted response surface strategy is introduced and tested for predicting the accurate boundary. Moreover, an interval dynamic correlation index is defined, and significance check and cross validation are reformulated in active subspaces to evaluate the AIRSM. The effectiveness of AIRSM is demonstrated on two test examples: three-dimensional nonlinear function and speed reducer design. They both possess a dominant one-dimensional active subspace with small estimation error, and the accuracy of AIRSM is verified by comparing with full-dimensional Monte Carlo simulates, thus providing a potential template for tackling high-dimensional problems involving mixed aleatory and interval uncertainties.
Factors associated with patient-reported likelihood of using online self-care interventions: a Scleroderma Patient-centered Intervention Network (SPIN) cohort study
Authors: Kwakkenbos Linda , Cumin Julie , Carrier Marie-Eve , Bartlett Susan J , Malcarne Vanessa L , Mouthon Luc , Nielson Warren R , Rannou François , Welling Joep , Thombs Brett D 
Published: October 2019

ObjectivesThe Scleroderma Patient-centered Intervention Network (SPIN) Cohort uses the cohort multiple randomised controlled trial design to embed trials of online self-care interventions for people living with systemic sclerosis (SSc; scleroderma). To offer interventions to patients interested in using them, participants complete signalling items that query about the likelihood that patients would agree to participate in nine different hypothetical online programmes addressing common SSc-related problems. It is not known what factors influence patient-reported interest in participating in a particular online intervention and if intervention-specific signalling questions provide unique information or replicate broader characteristics, such as overall willingness to participate or self-efficacy. This study assessed factors that explain responses to intervention-specific signalling items.DesignCross-sectional survey.SettingSPIN Cohort participants enrolled at 42 centres from Canada, the USA, the UK, France, Spain and Mexico who completed study questionnaires from March 2014 to January 2018 were included.MeasuresDemographic and disease characteristics, self-efficacy and symptoms related to each specific intervention were completed in addition to signalling items. General likelihood of using interventions was calculating by taking the mean score of the remaining signalling questions.Participants1060 participants with complete baseline data were included in the analyses.ResultsFor all individual signalling questions, controlling for other variables, the mean of the remaining signalling questions was the strongest predictor (standardised regression coefficient β from 0.61 (sleep) to 0.80 (self-management)). Smaller, but statistically significant, associations were found with the symptom associated with the respective signalling question and with general self-efficacy for 7 of 9 signalling questions.ConclusionsThe main factor associated with patients’ interest in participating in a disease-specific online self-care intervention is their general interest in participating in online interventions. Factors that may influence this general interest should be explored and taken into consideration when inviting patients to try online interventions.
Preventive strategies and factors associated with surgically treated necrotising enterocolitis in extremely preterm infants: an international unit survey linked with retrospective cohort data analysis
Authors: Adams Mark , Bassler Dirk , Darlow Brian A , Lui Kei , Reichman Brian , Hakansson Stellan , Norman Mikael , Lee Shoo K , Helenius Kjell K , Lehtonen Liisa , San Feliciano Laura , Vento Maximo , Moroni Marco , Beltempo Marc , Yang Junmin , Shah Prakesh S 
Published: October 2019

ObjectivesTo compare necrotising enterocolitis (NEC) prevention practices and NEC associated factors between units from eight countries of the International Network for Evaluation of Outcomes of Neonates, and to assess their association with surgical NEC rates.DesignProspective unit-level survey combined with retrospective cohort study.SettingNeonatal intensive care units in Australia/New Zealand, Canada, Finland, Israel, Spain, Sweden, Switzerland and Tuscany (Italy).PatientsExtremely preterm infants born between 240to 286weeks’ gestation, with birth weights<1500 g, and admitted between 2014–2015.ExposuresNEC prevention practices (probiotics, feeding, donor milk) using responses of an on-line pre-piloted questionnaire containing 10 questions and factors associated with NEC in literature (antenatal steroids, c-section, indomethacin treated patent ductus arteriosus and sepsis) using cohort data.Outcome measuresSurgical NEC rates and death following NEC using cohort data.ResultsThe survey response rate was 91% (153 units). Both probiotic provision and donor milk availability varied between 0%–100% among networks whereas feeding initiation and advancement rates were similar in most networks. The 9792 infants included in the cohort study to link survey results and cohort outcomes, revealed similar baseline characteristics but considerable differences in factors associated with NEC between networks. 397 (4.1%) neonates underwent NEC surgery, ranging from 2.4%–8.4% between networks. Standardised ratios for surgical NEC were lower for Australia/New Zealand, higher for Spain, and comparable for the remaining six networks.ConclusionsThe variation in implementation of NEC prevention practices and in factors associated with NEC in literature could not be associated with the variation in surgical NEC incidence. This corroborates the current lack of consensus surrounding the use of preventive strategies for NEC and emphasises the need for research.
High-flow oxygen for children’sairway surgery: randomised controlledtrial protocol (HAMSTER)
Authors: Humphreys Susan , von Ungern-Sternberg Britta Sylvia , Skowno Justin , Williams Tara , Taylor Julia , Taverner Fiona , Gibbons Kristen , Burgoyne Laura , Sommerfield David , Stephens Philip , Hallett Ben , Vijayasekaran Shyan , Slee Nicola , Burns Hannah , Sowa Marcin , Davidson Andrew , Schibler Andreas 
Published: October 2019

IntroductionHypoxaemia during anaesthesia for tubeless upper airway surgery in children with abnormal airways is common due to the complexity of balancing adequate depth of anaesthesia with maintenance of spontaneous breathing and providing an uninterrupted field of view of the upper airway for the surgeon. High-flow nasal oxygenation (HIGH-FLOW) can prolong safe apnoea time and be used in children with abnormal airways but to date has not been compared with the alternative technique of low-flow nasal oxygenation (LOW-FLOW). The aim is to investigate if use of HIGH-FLOW can reduce the number of hypoxaemic events requiring rescue oxygenation compared with LOW-FLOW.Methods and analysisHigh-flow oxygen for children’sairway surgery: randomised controlledtrial (HAMSTER) is a multicentre, unmasked, randomised controlled, parallel group, superiority trial comparing two oxygenation techniques during anaesthesia. Children (n=530) aged >37 weeks to 16 years presenting for elective tubeless upper airway surgery who fulfil inclusion but not exclusion criteria will be randomised prior to surgery to HIGH-FLOW or LOW-FLOW post induction of anaesthesia. Maintenance of anaesthesia with HIGH-FLOW requires Total IntraVenous Anaesthesia (TIVA) and with LOW-FLOW, either inhalational or TIVA at discretion of anaesthetist. The primary outcome is the incidence of hypoxaemic events requiring interruption of procedure for rescue oxygenation by positive pressure ventilation and the secondary outcome includes total hypoxaemia time, adverse cardiorespiratory events and unexpected paediatric intensive care admission admission. Hypoxaemia is defined as Sp02<90%. Analysis will be conducted on an intention-to-treat basis.Ethics and disseminationEthical approval has been obtained by Children’s Health Queensland Human Research Ethics Committee (HREC/18/QRCH/130). The trial commenced recruitment in 2018. The primary manuscript will be submitted for publication in a peer-reviewed journal.Trial registration numberThe HAMSTER is registered with the Australia and New Zealand Clinical TrialsRegistry: ACTRN12618000949280.
Palliative cancer care: costs in a Brazilian quaternary hospital
Authors: Rozman Luciana Martins , Campolina Alessandro Gonçalves , Lopez Rossana Mendoza , Chiba Toshio , De Soárez Patricia Coelho 
Published: October 2019

Palliative care (PC) improves the quality of life of patients with diseases such as cancer, and several studies have shown a reduction in costs among patients who use PC services when compared with those receiving standard oncological treatments. Most studies on PC costs are carried out in high-income countries. There is a lack of these types of studies in middle-income and low-income countries and of better evidence about this intervention.ObjectiveTo describe resource utilisation and costs among patients with cancer in a Brazilian quaternary hospital by cancer localisation and per month of treatment before death.MethodsThis study is a description of retrospective costs to estimate the costs of formal healthcare sector associated with PCs, from the perspective of a public quaternary cancer hospital. Unit costs were estimated using microcosting and macrocosting approaches.Setting/ParticipantsPatients older than 18 years old who died from 2010 to 2013 and who had at least two visits in PC and/or made use of hospice care.ResultsAmong the 2985 patients included in the study, the average cost per patient was US$12 335, ranging from US$8269 for patients with pancreatic cancer to US$19 395 for patients with brain cancer. The main costing item was hospital admission (47.6% of the total cost), followed by hospice care (29.5%) and medical and other supplies (11.1%).ConclusionsThe study clarified the direct medical costs and the profile and use of resources of patients with cancer who need PC, and can help in the planning and allocation of resources in cancer care.
Clinical significance of ‘double-hit’ and ‘double-expression’ lymphomas
Authors: Ma Zhiping , Niu Jing , Cao Yanzhen , Pang Xuelian , Cui Wenli , Zhang Wei , Li Xinxia 
Published: October 2019

Background‘Double-hit’ lymphoma (DHL) and ‘double-expression’ lymphoma (DEL) involving gene rearrangement and protein expression of MYC and BCL2/BCL6 have recently become the most commonly used terms to describe the poor prognostic types of diffuse large B-cell lymphoma (DLBCL). However, the clinical and pathological spectra of these rare entities have not been well defined. The aim of this study was to determine the frequency of DHL and DEL in DLBCL and their prognostic impacts in the era of cyclophosphamide, doxorubicin, vincristine and prednisone plus rituximab therapy.MethodsThe data and tissues from 98 patients diagnosed with DLBCL were used in this study. Formalin-fixed and paraffin-embedded tissues were constructed for immunohistochemistry (IHC) and interphase fluorescene in situ hybridisation (FISH) analysis for MYC, BCL6 and BCL2 rearrangements.ResultsThere were 14 cases (14.29%, 14/98) and 34 cases (34.70%, 34/98) of lymphomas with the DHL and DEL of MYC and BCL2/BCL6, respectively. DLBCL patients withMYC/BCL2rearrangements more frequently had bone marrow involvement (p=0.002), and their tumours were commonly of the germinal centre B cell (GCB) subtype. Patients with MYC+/BCL2 +coexpression were more often assessed using the International Prognostic Index (IPI), (Performance Status) PS score and bone marrow involvement. Patients with MYC+/BCL6 +coexpression were associated with their IPI values, Eastern Cooperative Oncology Group scores and occurrence of B symptoms. Their lymphomas were more often of the non-GCB subtype (p=0.010). Multivariate analysis showed that IPI, bone marrow involvement, rearrangements of BCL2, BCL6 and MYC, expression concurrent with rearrangements and coexpression were all significantly associated with overall survival and progression-free survival, with the exception of MYC+/BCL6 +coexpression.ConclusionsMYC/BCL2DHL,MYC/BCL6DHL and MYC/BCL2 DEL are an aggressive B cell lymphoma and patients have a poor prognosis. IPI and bone marrow involvement were independent prognostic factors for DHL and DEL. BCL2, BCL6 and MYC rearrangements translocation, expression concurrent rearrangements and coexpression were were independent prognostic factors for survival.Potential implicationsThe present study analysed the genomic alterations and protein expression levels of MYC, BCL2 and BCL6 using FISH and IHC in Chinese patients with DLBCL.We assessed the frequency, clinicopathological features and the prognostic impacts of DHL and DEL in a cohort of de novo DLBCL patients and evaluated the role of each genetic translocation separately and in combination in order to provide reliable conclusions and practical recommendations for diagnostic workups and prognostic predictions.
Integration of Large-Scale Genomic Data Sources With Evolutionary History Reveals Novel Genetic Loci for Congenital Heart Disease
Authors: Fotiou Elisavet , Williams Simon , Martin-Geary Alexandra , Robertson David L. , Tenin Gennadiy , Hentges Kathryn E. , Keavney Bernard 
Published: October 2019

Background:
Most cases of congenital heart disease (CHD) are sporadic and nonsyndromic, with poorly understood etiology. Rare genetic variants have been found to affect the risk of sporadic, nonsyndromic CHD, but individual studies to date are of only moderate sizes, and none to date has incorporated the ohnolog status of candidate genes in the analysis. Ohnologs are genes retained from ancestral whole-genome duplications during evolution; multiple lines of evidence suggest ohnologs are overrepresented among dosage-sensitive genes. We integrated large-scale data on rare variants with evolutionary information on ohnolog status to identify novel genetic loci predisposing to CHD.

Methods:
We compared copy number variants present in 4634 nonsyndromic CHD cases derived from publicly available data resources and the literature, and >27 000 healthy individuals. We analyzed deletions and duplications independently and identified copy number variant regions exclusive to cases. These data were integrated with whole-exome sequencing data from 829 sporadic, nonsyndromic patients with Tetralogy of Fallot. We placed our findings in an evolutionary context by comparing the proportion of vertebrate ohnologs in CHD cases and controls.

Results:

Novel genetic loci in CHD cases were significantly enriched for ohnologs compared with the genome (χ
2
test,
P
<0.0001, OR =1.253 [95% CI, 1.199–1.309]). We identified 54 novel candidate protein-coding genes supported by both: (1) copy number variant and whole-exome sequencing data; and (2) ohnolog status.

Conclusions:
We have identified new CHD candidate loci, and show for the first time that ohnologs are overrepresented among CHD genes. Incorporation of evolutionary metrics may be useful in refining candidate genes emerging from large-scale genetic evaluations of CHD.
Coelogynopora schockaerti n. sp. (Proseriata: Coelogynoporidae), a remnant of a platyhelminth coldwater fauna in the northwestern Mediterranean?
Authors: JOUK PHILIPPE E.H. , REVIS NATHALIE J.P. , ARTOIS TOM 
Published: October 2019

Coelogynopora schockaerti n. sp. (Proseriata: Coelogynoporidae) is described from the northwestern Gulf of Lion, Mediterranean. Only a few representatives of this genus are reported from Mediterranean marine sandy shores and only from the northwestern to central Mediterranean, but all of these species also occur outside the Mediterranean. So far, C. schockaerti n. sp. is the only species exclusively known from the Mediterranean. The species is solely encountered in the Gulf of Lion, a coldwater pocket in the Mediterranean and can, therefore, be considered as a remnant of an ancient coldwater platyhelminth fauna. C. schockaerti n. sp. is characterized by the shape and size of the spines of the male copulatory organ and the presence, orientation, shape and size of the two accessory spines. An identification key is given for the entire genus.
Effectiveness of Canal Occlusion for Intractable Posterior Canal Benign Paroxysmal Positional Vertigo: A Systematic Review
Authors: Maas Britta D. P. J. , van der Zaag‐Loonen Hester J. , van Benthem Peter Paul G. , Bruintjes Tjasse D. 
Published: October 2019

ObjectivesA last resort for therapy for intractable benign paroxysmal positional vertigo (BPPV) is mechanical occlusion of the posterior semicircular canal. The aim of this review was to assess the effect of posterior canal occlusion for intractable posterior canal BPPV on vertigo and to determine the risk of loss of auditory or vestibular function.Data SourcesA systematic literature search according to the PRISMA statement was performed on PubMed, the Cochrane Library, Embase, Web of Science, and CINAHL. The last search was conducted in June 2018.Review MethodsCohort studies with original data and case reports describing >5 cases were included if they analyzed the effect of posterior semicircular canal obliteration in adults with intractable posterior BPPV on vertigo. Two authors screened titles and abstracts for eligibility. The first author screened full texts and analyzed the data.ResultsEight retrospective studies met the eligibility criteria. The quality of all individual studies was rated fair. Canal occlusion was performed on 196 patients. All studies reported complete resolution of BPPV in all patients (100%). Among postoperatively tested patients, total loss of auditory function and vestibular function was reported in 2 of 190 (1%) and 9 of 68 (13%), respectively.ConclusionPosterior semicircular canal plugging resulted in 100% resolution of BPPV in patients with intractable BPPV in all studies. However, the strength of evidence was weak. Potential serious complications, such as deafness and loss of vestibular function, should be taken into account.
Unpacking Representation in State Immigration Policy: Latino Composition, White Racial Threat, and Legislator Partisanship
Authors: Avery James M. , Fine Jeffrey A. 
Published: October 2019

AbstractMost research studying minority representation concludes that minorities enjoy better representation when they constitute a larger share of a constituency, but only through the partisanship and race/ethnicity of the representative. Other research finds that minorities receive worse representation when they constitute a larger share of a constituency. We argue that minority composition will have an independent effect on representation, but that this effect will differ depending on the representative's partisanship. We apply our theory to Latino composition and state legislative voting on immigration policy and find that Latino composition has no effect on voting among Democratic legislators, who are less likely to vote in a restrictive direction on immigration than Republicans regardless of the Latino composition in their district. However, Republicans are more likely to vote to restrict immigration as Latinos comprise a larger share of their district. Our findings suggest that scholars should consider the moderating effect of legislator partisanship when examining minority composition and representation.
Changes in inflammatory bowel disease patients’ perspectives on biosimilars: A follow‐up survey
Authors: Peyrin-Biroulet Laurent , Lönnfors Sanna , Avedano Luisa , Danese Silvio 
Published: October 2019

Background and aimsThe aim of this survey was to find out whether the perspectives of patients with inflammatory bowel disease concerning biosimilars have changed since the publication of our last survey carried out in 2014–2015.MethodsAn online survey consisting of 19 questions was made available by the European Federation of Crohn's and Ulcerative Colitis Associations between July 2018 and December 2018. Only respondents who had heard of biosimilars were asked to respond to all of the questions.ResultsIn total, 1619 patients with inflammatory bowel disease responded the questionnaire. Most respondents were from Europe (79%), followed by Asia (8%), South America (7%) and Africa (5%). Some 44% of them had heard of biosimilars, and only these respondents continued to the biosimilar‐specific questions. Respondents worried significantly more about biosimilars being less effective than the originator (50% in current and 39% in previous survey, p = 0.0004). However, respondents were more likely to believe that biosimilars will have an impact on the management of inflammatory bowel disease (75% in current and 62% in previous survey).ConclusionsMany patients with inflammatory bowel disease remain unfamiliar with biosimilars. Although patients still worry about different aspects regarding biosimilars, they also tend to be more confident that biosimilars will have an impact on the management of their disease. More patient education is still needed to raise awareness about biosimilars.
Retrospective Analysis of Chinese Chronic Myeloid Leukemia Patients Who Have Stopped Tyrosine Kinase Inhibitors
Authors: Zhu Xiaojian , You Yong , Zhang Yicheng , Duan Minghui , Zhu Yu , Liu Bingcheng , Du Xin , Chen Suning 
Published: October 2019

Abstract
Background
Although life-long tyrosine kinase inhibitors (TKIs) therapy is still the consensus recommendation, TKIs discontinuation has yet been proven in part of the patients who sustain the long-term treatment-free remission (TFR) which has become a new endpoint and is an emerging goal in the management of chronic myeloid leukemia (CML) currently. In the last ten years, constant clinical trials have demonstrated that about 40%-50% patients with stable deep molecular response for a long period time stayed in TFR after stopping TKIs.
Methods
We retrospective analyzed 109 CML patients who have stopped TKIs in seven centers. They were included according to the unified standard. Rates of TFR were calculated by Kaplan-Meier analysis and stratified groups were compared with the log-rank test. COX univariate analysis was used to predict the potential factors on relapse after discontinuation.
Results
Clinical features of the 93 patients were included in this work. 39 of 93 patients lost major molecular remission (MMR) after a median follow-up of 15 months and the estimated TFR was 52.6%. 31 of 39 patients who met the definition of molecular relapse resumed TKIs treatment immediately and regained the response with an average time to MMR of 4.34 months (range, 1-12 months). No significant difference was found in median course of imatinib therapy between the TFR group and the relapse. Similarly, time to MMR, age, gender and Sokal risk also showed no difference between the two groups. History of withdrawal is the only statistically significant factor in this retrospective analysis.
Conclusion
In summary, this is the first report to prove that TKIs might be safely stopped in partial Chinese CML patients.

Disclosures
No relevant conflicts of interest to declare.