bluuebunny/crossref_metadata_2025_split huggingface dataset:
Organisation of care for people receiving drug-resistant tuberculosis treatment in South Africa: a mixed methods study
Authors: Dickson Lindy , Le Roux Sacha Roxanne , Mitrani Leila , Hill Jeremy , Jassat Waasila , Cox Helen , Mlisana Koleka , Black John , Loveday Marian , Grant Alison , Kielmann Karina , Ndjeka Norbert , Moshabela Mosa , Nicol Mark 
Published: November 2023

ObjectivesTreatment for multidrug-resistant/rifampicin-resistant tuberculosis (MDR/RR-TB) is increasingly transitioning from hospital-centred to community-based care. A national policy for decentralised programmatic MDR/RR-TB care was adopted in South Africa in 2011. We explored variations in the implementation of care models in response to this change in policy, and the implications of these variations for people affected by MDR/RR-TB.DesignA mixed methods study was done of patient movements between healthcare facilities, reconstructed from laboratory records. Facility visits and staff interviews were used to determine reasons for movements.Participants and settingPeople identified with MDR/RR-TB from 13 high-burden districts within South Africa.Outcome measuresGeospatial movement patterns were used to identify organisational models. Reasons for patient movement and implications of different organisational models for people affected by MDR/RR-TB and the health system were determined.ResultsAmong 191 participants, six dominant geospatial movement patterns were identified, which varied in average hospital stay (0–281 days), average patient distance travelled (12–198 km) and number of health facilities involved in care (1–5 facilities). More centralised models were associated with longer delays to treatment initiation and lengthy hospitalisation. Decentralised models facilitated family-centred care and were associated with reduced time to treatment and hospitalisation duration. Responsiveness to the needs of people affected by MDR/RR-TB and health system constraints was achieved through implementation of flexible models, or the implementation of multiple models in a district.ConclusionsUnderstanding how models for organising care have evolved may assist policy implementers to tailor implementation to promote particular patterns of care organisation or encourage flexibility, based on patient needs and local health system resources. Our approach can contribute towards the development of a health systems typology for understanding how policy-driven models of service delivery are implemented in the context of variable resources.
Protocol for a cluster randomised trial of a goal-oriented care approach for multimorbidity patients supported by a digital platform
Authors: Gil Conde Margarida , Peyroteo Mariana , Maria Ana , Maia Mélanie Raimundo , Gregório João , Paulo Marilia Silva , Alves Marta , Papoila Ana Luísa , Lapão Luís Velez , Heleno Bruno 
Published: November 2023

IntroductionHealth information systems represent an opportunity to improve the care provided to people with multimorbidity. There is a pressing need to assess their impact on clinical outcomes to validate this intervention. Our study will determine whether using a digital platform (Multimorbidity Management Health Information System, METHIS) to manage multimorbidity improves health-related quality of life (HR-QoL).Methods and analysisA superiority, cluster randomised trial will be conducted at primary healthcare practices (1:1 allocation ratio). All public practices in the Lisbon and Tagus Valley (LVT) Region, Portugal, not involved in a previous pilot trial, will be eligible. At the participant level, eligible patients will be people with complex multimorbidity, aged 50 years or older, with access to an internet connection and a communication technology device. Participants who cannot sign/read/write and who do not have access to an email account will not be included in the study. The intervention combines a training programme and a customised information system (METHIS). Both are designed to help clinicians adopt a goal-oriented care model approach and to encourage patients and carers to play a more active role in autonomous healthcare. The primary outcome is HR-QoL, measured at 12 months with the physical component scale of the 12-item Short Form questionnaire (SF-12). Secondary outcomes will also be measured at 12 months and include mental health (mental component Scale SF-12, Hospital Anxiety and Depression Scale). We will also assess serious adverse events during the trial, including hospitalisation and emergency services. Finally, at 18 months, we will ask the general practitioners for any potentially missed diagnoses.Ethics and disseminationThe Research and Ethics Committee (LVT Region) approved the trial protocol. Clinicians and patients will sign an informed consent. A data management officer will handle all data, and the publication of several scientific papers and presentations at relevant conferences/workshops is envisaged.Trial registration numberNCT05593835.
From symptom onset to treatment initiation: protocol for a narrative study exploring the journey of older adults with tuberculosis in the English Midlands, UK
Authors: Kidy Farah , McCarthy Noel , Seers Kate 
Published: November 2023

IntroductionTime from symptom onset to treatment initiation in tuberculosis (TB) remains stubbornly prolonged despite reductions in disease incidence. Delays may contribute to increased morbidity, mortality, onward spread of disease and poor patient experiences. Most delays occur prior to hospital referral. The average primary care healthcare provider in England is unlikely to see TB on a regular basis. Little is known about primary care diagnostic and referral challenges.Adults aged 65 years or older are more likely to experience delays. However, little is known about their journey from symptom onset to treatment initiation.Methods and analysisWe will carry out a narrative study including adults aged 65 years or older, living in the English Midlands and receiving treatment for active TB. Twelve English and 12 Urdu or Punjabi speakers will be recruited from TB clinics and interviewed. Their primary care records will be accessed, and the primary care story and secondary care letters will be extracted. Each of the data sources will be analysed using dialogical narrative analysis. Data will be triangulated within participants and across the data set.Ethics and disseminationThis study received approval from the Health Research Authority and the Research Ethics Committee in April 2022. Risk management and equity considerations have been made a priority. Findings will be disseminated through publication in open access peer-reviewed journals, presentations to policy makers, primary healthcare and secondary healthcare professionals, and through public facing materials developed in conjunction with patients, members of the pubic, TB services and charities.
Tight perioperative blood pressure management to reduce complications: a randomised feasibility trial
Authors: Li Kai , Hu Zhouting , Li Wangyu , Shah Karan , Sessler Daniel 
Published: November 2023

ObjectiveEvaluate the feasibility of a trial of perioperative hypotension and serious complications.DesignA patient and assessor-blinded randomised feasibility trial.SettingWe included patients in a tertiary university hospital.ParticipantsWe enrolled 80 adults scheduled for major non-cardiac surgery.InterventionsIn patients randomised to tight blood pressure control, intraoperative mean arterial pressure (MAP) was targeted to ≥85 mm Hg maintained with norepinephrine infusion, and restarting chronic antihypertensive medications was delayed until the third postoperative day. In the reference group, intraoperative blood pressure was managed per routine and antihypertensive medications were restarted immediately after surgery.Primary and secondary outcome measuresOur first co-primary outcome was the fraction of time when intraoperative MAP was >85 mm Hg, intraoperative area (time integral) of MAP >85 mm Hg and MAP <65 mm Hg. The second co-primary outcome was time until antihypertensive medications were restarted after surgery. Secondary outcomes were time-weighted average intraoperative MAP, cumulative minimum MAP for 10 min, average postoperative systolic blood pressure (SBP) and mean of the lowest three postoperative SBPs.ResultsForty patients in each group were analysed. The median for intraoperative area of MAP >85 mm Hg was 1303 (772–2419) mm Hg*min in routine blood pressure (BP) cases and 2425 (1926–3545) mm Hg*min in tight BP control. The area for intraoperative MAP <65 mm Hg was 7 (0–40) mm Hg*min with routine BP management, and 0 (0–0) mm Hg*min with tight BP control. The fraction of time with MAP >85 mm Hg was 0.52 (0.25) and 0.87 (0.15). Antihypertensive medications were restarted 2 (1–3) days later in tight BP control cases. However, postoperative SBPs were similar.ConclusionsTight BP management markedly increased intraoperative MAP and reduced the amount of hypotension. In contrast, delaying chronic antihypertensive medications had little effect on postoperative SBP. The full trial appears feasible and remains necessary but should not include postoperative antihypertensive management.Trial registrationNCT04789733.
Critical quality appraisal and algorithm development of clinical practice guidelines for management of poststroke sensory loss: a systematic review
Authors: Li Xiao , Xing Xingmin , Yao Nannan 
Published: November 2023

ObjectivesThis study aimed to assess the methodological quality of clinical practice guidelines (CPGs) associated with the management of poststroke sensory loss and develop an algorithm for health professionals.MethodsWe conducted a systematic review for relevant CPGs published between 2017 and 2022 using the Preferred Reporting Items for Systematic Reviews and Meta-Analyses Checklist. Appraisal of Guidelines for Research and Evaluation II instrument was used to assess methodological quality. Recommendations for managing poststroke sensory loss from high and average-quality CPGs were summarised and developed into an algorithm.ResultsFirst, 1458 records were identified from the database searches and other sources. Finally, four CPGs were included: three were rated as high quality and one as average quality. Twenty-two recommendations were summarised from these CPGs and used to develop a draft algorithm. Then, we revised the draft algorithm developed by the authors based on expert feedback to form the final version.ConclusionsThe four CPGs included in this study had good quality. Based on these CPGs, we developed an algorithm to facilitate health professionals’ adherence to CPGs and contribute to evidence-based medicine. In the future, more high-quality CPGs are required to give further scientific and convincing evidence to manage poststroke sensory loss.
Hospital variations in failure to rescue after abdominal surgery: a nationwide, retrospective observational study
Authors: Augestad Knut Magne , Skyrud Katrine Damgaard , Lindahl Anne Karin , Helgeland Jon 
Published: November 2023

ObjectivesThis study aims to determine hospital variation and intensive care unit characteristics associated with failure to rescue after abdominal surgery in Norway.DesignA nationwide retrospective observational study.SettingAll 52 hospitals in Norway performing elective and acute abdominal surgery.ParticipantsAll 598 736 patients undergoing emergency and elective abdominal surgery from 2011 to 2021.Primary outcome measurePrimary outcome was failure to rescue within 30 days (FTR30), defined as in-hospital or out-of-hospital death within 30 days of a surgical patient who developed at least one complication within 30 days of the surgery (FTR30). Other outcome variables were surgical complications and hospital FTR30 variation. Statistical analysis was conducted separately for general surgery and abdominal surgery.ResultsThe 30-day postoperative complication rate was 30.7 (183 560 of 598 736 surgeries). Of general surgical complications (n=25 775), circulatory collapse (n=6127, 23%), cardiac arrhythmia (n=5646, 21%) and surgical infections (n=4334, 16 %) were most common and 1507 (5.8 %) patients were reoperated within 30 days. One thousand seven hundred and forty patients had FTR30 (6.7 %). The severity of complications was strongly associated with FTR30. In multivariate analysis of general surgery, adjusted for patient characteristics, only the year of surgery was associated with FTR30, with an estimated linear trend of −0.31 percentage units per year (95% CI (−0.48 to –0.15)). The driving distance from local hospitals to the nearest referral intensive care unit was not associated with FTR30. Over the last decade, FTR30 rates have varied significantly among similar hospitals.ConclusionsHospital factors cannot explain Norwegian hospitals’ significant FTR variance when adjusting for patient characteristics. The national FTR30 measure has dropped around 30% without a corresponding fall in surgical complications. No association was seen between rural hospital location and FTR30. Policy-makers must address microsystem issues causing high FTR30 in hospitals.
Treatment of schizotypal disorder: a protocol for a systematic review of the evidence and recommendations for clinical practice
Authors: Gundersen Kristina Ballestad , Rasmussen Andreas Rosén , Sandström Katharina Oravsky , Albert Nikolai , Polari Andrea , Ebdrup Bjørn H , Nelson Barnaby , Glenthøj Louise Birkedal 
Published: November 2023

IntroductionSchizotypal disorder is associated with a high level of disability at an individual level and high societal costs. However, clinical recommendations for the treatment of schizotypal disorder are scarce and based on limited evidence. This review aims to synthesise the current evidence on treatment for schizotypal disorder making recommendations for clinical practice.Methods and analysisThis systematic review protocol follows the Preferred Reporting Items for Systematic Reviews and Meta-Analyses guidelines. A systematic literature search will be performed in PsychArticles, Embase, Medline and Cochrane Central Register of Controlled Trials. Additionally, we will search for relevant articles manually. Inclusion criteria are published studies including individuals diagnosed with schizotypal personality disorder according to Diagnostic and Statistical Manual of Mental Disorders (DSM) criteria, or schizotypal disorder according to International Classification of Diseases (ICD) criteria. We will include interventional studies comprising any pharmacological and non-pharmacological treatment trials for patients with schizotypal disorder, and all relevant outcome measures will be reported. Risk of bias will be assessed by Cochrane risk-of-bias tools. Data will be synthesised using narrative or thematic analysis and, if suitable, through meta-analysis.Ethics and disseminationNo original data will be collected as part of this study and ethics approval is, therefore, not applicable. The results will be disseminated through peer-reviewed publication and presented at international scientific meetings. We will aim at submitting the final paper for publication within 4 months of completion of analyses. Furthermore, this systematic review will inform clinicians and researchers on the current state of evidence on treatment for schizotypal disorder. Findings may guide proposals for further research and potentially guide recommendations for clinical practice using the Grading of Recommendations Assessment, Development and Evaluation.PROSPERO registration numberCRD42022375001.
HeterozygousCOL17A1variants are a frequent cause of amelogenesis imperfecta
Authors: Hany Ummey , Watson Christopher M , Liu Lu , Smith Claire E L , Harfoush Asmaa , Poulter James A , Nikolopoulos Georgios , Balmer Richard , Brown Catriona J , Patel Anesha , Simmonds Jenny , Charlton Ruth , Acosta de Camargo María Gabriela , Rodd Helen D , Jafri Hussain , Antanaviciute Agne , Moffat Michelle , Al-Jawad Maisoon , Inglehearn Chris F , Mighell Alan J 
Published: November 2023

BackgroundCollagen XVII is most typically associated with human disease when biallelicCOL17A1variants (>230) cause junctional epidermolysis bullosa (JEB), a rare, genetically heterogeneous, mucocutaneous blistering disease with amelogenesis imperfecta (AI), a developmental enamel defect. Despite recognition that heterozygous carriers in JEB families can have AI, and that heterozygousCOL17A1variants also cause dominant corneal epithelial recurrent erosion dystrophy (ERED), the importance of heterozygousCOL17A1variants causing dominant non-syndromic AI is not widely recognised.MethodsProbands from an AI cohort were screened by single molecule molecular inversion probes or targeted hybridisation capture (both a custom panel and whole exome sequencing) forCOL17A1variants. Patient phenotypes were assessed by clinical examination and analyses of affected teeth.ResultsNineteen unrelated probands with isolated AI (no co-segregating features) had 17 heterozygous, potentially pathogenicCOL17A1variants, including missense, premature termination codons, frameshift and splice site variants in both the endo-domains and the ecto-domains of the protein. The AI phenotype was consistent with enamel of near normal thickness and variable focal hypoplasia with surface irregularities including pitting.ConclusionThese results indicate thatCOL17A1variants are a frequent cause of dominantly inherited non-syndromic AI. Comparison of variants implicated in AI and JEB identifies similarities in type and distribution, with five identified in both conditions, one of which may also cause ERED. Increased availability of genetic testing means that more individuals will receive reports of heterozygousCOL17A1variants. We propose that patients with isolated AI or ERED, due toCOL17A1variants, should be considered as potential carriers for JEB and counselled accordingly, reflecting the importance of multidisciplinary care.
Update of the UMD-VHL database: classification of 164 challenging variants based on genotype–phenotype correlation among 605 entries
Authors: Mougel Gregory , Mohamed Amira , Burnichon Nelly , Giraud Sophie , Pigny Pascal , Bressac-de Paillerets Brigitte , Mirebeau-Prunier Delphine , Buffet Alexandre , Savagner Frédérique , Romanet Pauline , Arlot Yannick , Gardie Betty , Gimenez-Roqueplo Anne-Paule , Beroud Christophe , Richard Stephane , Barlier Anne 
Published: November 2023

BackgroundThe von Hippel-Lindau (VHL) disease is a hereditary tumour syndrome caused by germline mutations inVHLtumour suppressor gene. The identification ofVHLvariants requires accurate classification which has an impact on patient management and genetic counselling.MethodsThe TENGEN (French oncogenetics network of neuroendocrine tumors) and PREDIR (French National Cancer Institute network for Inherited predispositions to kidney cancer) networks have collectedVHLgenetic variants and clinical characteristics of all VHL-suspected patients analysed from 2003 to 2021 by one of the nine laboratories performingVHLgenetic testing in France. Identified variants were registered in a locus-specific database, the Universal Mutation Database-VHL database (http://www.umd.be/VHL/).ResultsHere we report the expert classification of 164 variants, including all missense variants (n=124), all difficult interpretation variants (n=40) and their associated phenotypes. After initial American College of Medical Genetics classification, first-round classification was performed by the VHL expert group followed by a second round for discordant and ambiguous cases. Overall, the VHL experts modified the classification of 87 variants including 30 variants of uncertain significance that were as (likely)pathogenic variants for 19, and as likely benign for 11.ConclusionConsequently, this work has allowed the diagnosis and influenced the genetic counselling of 45 VHL-suspected families and can benefit to the worldwide VHL community, through this review.
Rethinking leadership approaches for community-wide opioid crisis intervention: harnessing positive inquiry to unearth front-line insight
Authors: Huber Thomas Patrick 
Published: November 2023

Background/AimThe opioid crisis presents a complex and widespread health challenge for communities, necessitating a deeper exploration beyond simple solutions. To address this crisis, it is crucial to identify and disseminate best practices. In this study, we focused on positive deviance narratives from Columbus, Ohio, a community deeply affected by the opioid crisis.MethodsEmploying an appreciative inquiry framework, we conducted purposive sampling to interview 47 frontline professionals and individuals grappling with opioid use disorder (OUD).ResultsOur research revealed several key insights, highlighting successful themes through positive narratives. First, perceiving OUD as a chronic disease emerged as a critical perspective, acknowledging the long-term nature of the condition and the need for sustained care. Second, re-humanising and de-stigmatising opioid users played a vital role in facilitating their recovery and reintegration into society. Embracing inclusive care delivery was another important aspect, ensuring that individuals from diverse backgrounds receive equitable access to effective treatment. Moreover, re-energising professionals to combat burnout proved essential, emphasising the importance of supporting and motivating healthcare providers in their efforts. Finally, fostering cross-institutional relationship building and collaboration emerged as a significant factor, as it encouraged a coordinated approach to addressing the crisis. Designing adaptive organisational structures also played a crucial role, enabling healthcare institutions to respond effectively to evolving challenges.ConclusionsUsing a positive deviance approach to a challenging public health crisis like OUD can help us discover new and innovative care management approaches for community wide interventions.
Survival analysis from the INCREASE study in PH-ILD: evaluating the impact of treatment crossover on overall mortality
Authors: Nathan Steven D , Johri Shilpa , Joly Joanna M , King Christopher S , Raina Amresh , McEvoy Colleen A , Lee Dasom , Shen Eric , Smith Peter , Deng Chunqin , Waxman Aaron B 
Published: November 2023

ObjectiveA post-hoc analysis of the INCREASE trial and its open-label extension (OLE) was performed to evaluate whether inhaled treprostinil has a long-term survival benefit in patients with pulmonary hypertension associated with interstitial lung disease (PH-ILD).MethodsTwo different models of survival were employed; the inverse probability of censoring weighting (IPCW) and the rank-preserving structural failure time (RPSFT) models both allow construction of a pseudo-placebo group, thereby allowing for long-term survival evaluation of patients with PH-ILD receiving inhaled treprostinil. Time-varying stabilised weights were calculated by fitting Cox proportional hazards models based on the baseline and time-varying prognostic factors to generate weighted Cox regression models with associated adjusted HRs.ResultsIn the INCREASE trial, there were 10 and 12 deaths in the inhaled treprostinil and placebo arms, respectively, during the 16-week randomised trial. During the OLE, all patients received inhaled treprostinil and there were 29 and 33 deaths in the prior inhaled treprostinil arm and prior placebo arm, respectively. With a conventional analysis, the HR for death was 0.71 (95% CI 0.46 to 1.10; p=0.1227). Both models demonstrated significant reductions in death associated with inhaled treprostinil treatment with HRs of 0.62 (95% CI 0.39 to 0.99; p=0.0483) and 0.26 (95% CI 0.07 to 0.98; p=0.0473) for the IPCW and RPSFT methods, respectively.ConclusionTwo independent modelling techniques that have been employed in the oncology literature both suggest a long-term survival benefit associated with inhaled treprostinil treatment in patients with PH-ILD.