bluuebunny/crossref_metadata_2025_split huggingface dataset:
Parental bereavement – impact of death of neonates and children under 12 years on personhood of parents: a systematic scoping review
Authors: Vig Prachi Simran , Lim Jia Yin , Lee Randal Wei Liang , Huang Huixin , Tan Xiu Hui , Lim Wei Qiang , Lim Marie Bernadette Xin Yi , Lee Alexia Sze Inn , Chiam Min , Lim Crystal , Baral Vijayendra Ranjan , Krishna Lalit Kumar Radha 
Published: September 2021

AbstractBackgroundLosing a child tragically impacts the well-being and functioning of parents. With these effects extending beyond emotional, physical morbidity and compromising self-perceptions, appropriate, longitudinal, timely and personalised support is key to effective care of bereaved parents. However, in the absence of a comprehensive understanding of parental bereavement, effective support of bereaved parents remains suboptimal. To address this gap, we scrutinise prevailing data on the effects of a child’s death, aged 0–12 years, through the lens of the Ring Theory of Personhood (RToP).MethodsTo study prevailing accounts of bereaved parents following the death of a child, we adopt Krishna’s Systematic Evidence Based Approach (SEBA) to structure our Systematic Scoping Review (SSR in SEBA).ResultsThree thousand seventy-four abstracts were reviewed, 160 full text articles were evaluated, and 111 articles were included and analysed using thematic and content analysis. Four themes/categories were identified relating to the four rings of the RToP. Findings reveal that static concepts of protective and risk factors for grief are misplaced and that the support of healthcare professionals is key to assisting bereaved parents.ConclusionIn the absence of consistent support of bereaved parents, this study highlights the need for effective training of healthcare professionals, beginning with an appreciation that every aspect of an individual parent’s personhood is impacted by the loss of their child. Acknowledging grief as a complex, evolving and personalised process subjected to parental characteristics, settings, context and available support, this SSR in SEBA calls attention to effective nurturing of the relationship between parents and healthcare professionals, and suggests use of the RToP to assess and direct personalised, timely, specific support of parents in evolving conditions. We believe the findings of this review also call for further studies to support healthcare professionals as they journey with bereaved parents.
Achieving inclusive research priority-setting: what do people with lived experience and the public think is essential?
Authors: Pratt Bridget 
Published: September 2021

Abstract
Background
Engagement of people with lived experience and members of the public is an ethically and scientifically essential component of health research. Authentic engagement means they are involved as full partners in research projects. Yet engagement as partnership is uncommon in practice, especially during priority-setting for research projects. What is needed for agenda-setting to be shared by researchers and people with lived experience and/or members of the public (or organisations representing them)? At present, little ethical guidance exists on this matter, particularly that which has been informed by the perspectives of people with lived experience and members of the public. This article provides initial evidence about what they think are essential foundations and barriers to shared decision-making in health research priority-setting and health research more broadly.

Methods
An exploratory, qualitative study was conducted in 2019. 22 semi-structured interviews were performed with key informants from the UK and Australia.

Results
Three main types of foundations were thought to be essential to have in place before shared decision-making can occur in health research priority-setting: relational, environmental, and personal. Collectively, the three types of foundations addressed many (but not all) of the barriers to power sharing identified by interviewees.

Conclusions
Based on study findings, suggestions are made for what researchers, engagement practitioners, research institutions, and funders should do in their policy and practice to support meaningful engagement. Finally, key international research ethics guidelines on community engagement are considered in light of study findings.
Journey mapping as a novel approach to healthcare: a qualitative mixed methods study in palliative care
Authors: Ly Stephanie , Runacres Fiona , Poon Peter 
Published: September 2021

Abstract
Background
Journey mapping involves the creation of visual narrative timelines depicting the multidimensional relationship between a consumer and a service. The use of journey maps in medical research is a novel and innovative approach to understanding patient healthcare encounters.

Objectives
To determine possible applications of journey mapping in medical research and the clinical setting. Specialist palliative care services were selected as the model to evaluate this paradigm, as there are numerous evidence gaps and inconsistencies in the delivery of care that may be addressed using this tool.

Methods
A purposive convenience sample of specialist palliative care providers from the Supportive and Palliative Care unit of a major Australian tertiary health service were invited to evaluate journey maps illustrating the final year of life of inpatient palliative care patients. Sixteen maps were purposively selected from a sample of 104 consecutive patients. This study utilised a qualitative mixed-methods approach, incorporating a modified Delphi technique and thematic analysis in an online questionnaire.

Results
Our thematic and Delphi analyses were congruent, with consensus findings consistent with emerging themes. Journey maps provided a holistic patient-centred perspective of care that characterised healthcare interactions within a longitudinal trajectory. Through these journey maps, participants were able to identify barriers to effective palliative care and opportunities to improve care delivery by observing patterns of patient function and healthcare encounters over multiple settings.

Conclusions
This unique qualitative study noted many promising applications of the journey mapping suitable for extrapolation outside of the palliative care setting as a review and audit tool, or a mechanism for providing proactive patient-centred care. This is particularly significant as machine learning and big data is increasingly applied to healthcare.
Renal hemodynamic effects differ between antidiabetic combination strategies: randomized controlled clinical trial comparing empagliflozin/linagliptin with metformin/insulin glargine
Authors: Ott Christian , Jung Susanne , Korn Manuel , Kannenkeril Dennis , Bosch Agnes , Kolwelter Julie , Striepe Kristina , Bramlage Peter , Schiffer Mario , Schmieder Roland E. 
Published: September 2021

Abstract
Background
Type 2 diabetes causes cardio-renal complications and is treated with different combination therapies. The renal hemodynamics profile of such combination therapies has not been evaluated in detail.

Methods
Patients (N = 97) with type 2 diabetes were randomized to receive either empagliflozin and linagliptin (E+L group) or metformin and insulin glargine (M+I group) for 3 months. Renal hemodynamics were assessed with para-aminohippuric acid and inulin for renal plasma flow (RPF) and glomerular filtration rate (GFR). Intraglomerular hemodynamics were calculated according the Gomez´ model.

Results
Treatment with E+L reduced GFR (p = 0.003), but RPF remained unchanged (p = 0.536). In contrast, M+I not only reduced GFR (p = 0.001), but also resulted in a significant reduction of RPF (p < 0.001). Renal vascular resistance (RVR) decreased with E+L treatment (p = 0.001) but increased with M+I treatment (p = 0.001). The changes in RPF and RVR were different between the two groups (both padjust < 0.001). Analysis of intraglomerular hemodynamics revealed that E+L did not change resistance of afferent arteriole (RA) (p = 0.116), but diminished resistance of efferent arterioles (RE) (p = 0.001). In M+I group RA was increased (p = 0.006) and RE remained unchanged (p = 0.538). The effects on RA (padjust < 0.05) and on RE (padjust < 0.05) differed between the groups.

Conclusions
In patients with type 2 diabetes and preserved renal function treatment with M+I resulted in reduction of renal perfusion and increase in vascular resistance, in contrast to treatment with E+I that preserved renal perfusion and reduced vascular resistance. Moreover, different underlying effects on the resistance vessels have been estimated according to the Gomez model, with M+I increasing RA and E+L predominantly decreasing RE, which is in contrast to the proposed sodium-glucose cotransporter 2 inhibitor effects.
Trial registration: The study was registered at www.clinicaltrials.gov (NCT02752113) on April 26, 2016
Molecular detection of blaCTX-M gene to predict phenotypic cephalosporin resistance and clinical outcome of Escherichia coli bloodstream infections in Vietnam
Authors: Son Trinh Van , Manh Nguyen Dang , Trung Ngo Tat , Quyen Dao Thanh , Meyer Christian G. , Phuong Nguyen Thi Kim , Hoan Phan Quoc , Sang Vu Viet , Nurjadi Dennis , Velavan Thirumalaisamy P. , Bang Mai Hong , Song Le Huu 
Published: September 2021

Abstract
Background
Blood stream infections (BSI) caused by Extended Spectrum Beta-Lactamases (ESBLs) producing Enterobacteriaceae is a clinical challenge leading to high mortality, especially in developing countries. In this study, we sought to describe the epidemiology of ESBL-producing Escherichia coli strains isolated from Vietnamese individuals with BSI, to investigate the concordance of genotypic-phenotypic resistance, and clinical outcome of ESBL E. coli BSI.

Methods
A total of 459 hospitalized patients with BSI were screened between October 2014 and May 2016. 115 E. coli strains from 115 BSI patients were isolated and tested for antibiotic resistance using the VITEK®2 system. The ESBL phenotype was determined by double disk diffusion method following the guideline of Clinical and Laboratory Standards Institute. Screening for beta-lactamase (ESBL and carbapenemase) genes was performed using a multiplex-PCR assay.

Results
58% (67/115) of the E. coli strains were ESBL-producers and all were susceptible to both imipenem and meropenem. Resistance to third-generation cephalosporin was common, 70% (81/115) were cefotaxime-resistant and 45% (52/115) were ceftazidime-resistant. blaCTX-M was the most common ESBL gene detected (70%; 80/115) The sensitivity and specificity of blaCTX-M-detection to predict the ESBL phenotype was 87% (76–93% 95% CI) and 54% (39–48% 95% CI), respectively. 28%% (22/80) of blaCTX-M were classified as non-ESBL producers by phenotypic testing for ESBL production. The detection of blaCTX-M in ESBL-negative E. coli BSI was associated with fatal clinical outcome (27%; 6/22 versus 8%; 2/26, p = 0.07).

Conclusion
A high prevalence of ESBL-producing E. coli isolates harbouring blaCTX-M was observed in BSI patients in Vietnam. The genotypic detection of blaCTX-M may have added benefit in optimizing and guiding empirical antibiotic therapy of E. coli BSI to improve clinical outcome.
Multiplex lateral flow immunochromatographic assay is an effective method to detect carbapenemases without risk of OXA-48-like cross reactivity
Authors: Kon Hadas , Abramov Shirin , Frenk Sammy , Schwartz David , Shalom Ohad , Adler Amos , Carmeli Yehuda , Lellouche Jonathan 
Published: September 2021

Abstract
Background
It is essential to detect carriers of carbapenemase-producing Enterobacterales in order to implement infection control measures. The objectives of this study was to evaluate the NG-Test® CARBA 5 (CARBA 5) assay for detection of five carbapenemases and to assess the cross reactivity of other OXA-type carbapenemases with the OXA-48-like specific antibodies.

Methods
A total of 197 Enterobacterales isolates were tested. To evaluate the cross reactivity, 73 carbapenem-resistant A. baumannii, harboring OXA-type variants, were tested. Polymerase chain reaction (PCR) served as gold standard for carbapenemase identification.

Results
Excellent agreement was found between PCR and CARBA 5, for all but one isolate. The single false positive result (a blaSME positive S. marcescens isolate) was incorrectly positive for blaOXA-48 by CARBA 5. No cross reactivity was observed. The sensitivity and specificity were 100.0% and 98.0%, respectively.

Conclusions
The CARBA 5 assay is highly sensitive and specific and is recommended as a tool for the detection of the main carbapenemases of interest in clinical microbiology laboratories.
A self-amplified nanocatalytic system for achieving “1 + 1 + 1 > 3” chemodynamic therapy on triple negative breast cancer
Authors: Zhou Lulu , Chen Jinjin , Sun Yunhao , Chai Keke , Zhu Zhounan , Wang Chunhui , Chen Mengyao , Han Wenmei , Hu Xiaochun , Li Ruihao , Yao Tianming , Li Hui , Dong Chunyan , Shi Shuo 
Published: September 2021

Abstract
Background
Chemodynamic therapy (CDT), employing Fenton or Fenton-like catalysts to convert hydrogen peroxide (H2O2) into toxic hydroxyl radicals (·OH) to kill cancer cells, holds great promise in tumor therapy due to its high selectivity. However, the therapeutic effect is significantly limited by insufficient intracellular H2O2 level in tumor cells. Fortunately, β-Lapachone (Lapa) that can exert H2O2-supplementing functionality under the catalysis of nicotinamide adenine dinucleotide (phosphate) NAD(P)H: quinone oxidoreductase-1 (NQO1) enzyme offers a new idea to solve this problem. However, extensive DNA damage caused by high levels of reactive oxygen species can trigger the “hyperactivation” of poly(ADP-ribose) polymerase (PARP), which results in the severe interruption of H2O2 supply and further the reduced efficacy of CDT. Herein, we report a self-amplified nanocatalytic system (ZIF67/Ola/Lapa) to co-deliver the PARP inhibitor Olaparib (Ola) and NQO1-bioactivatable drug Lapa for sustainable H2O2 production and augmented CDT (“1 + 1 + 1 > 3”).

Results
The effective inhibition of PARP by Ola can synergize Lapa to enhance H2O2 formation due to the continuous NQO1 redox cycling. In turn, the high levels of H2O2 further react with Co2+ to produce the highly toxic ·OH by Fenton-like reaction, dramatically improving CDT. Both in vitro and in vivo studies demonstrate the excellent antitumor activity of ZIF67/Ola/Lapa in NQO1 overexpressed MDA-MB-231 tumor cells. Importantly, the nanocomposite presents minimal systemic toxicity in normal tissues due to the low NQO1 expression.

Conclusions
This design of nanocatalytic system offers a new paradigm for combing PARP inhibitor, NQO1-bioactivatable drug and Fenton-reagents to obtain sustained H2O2 generation for tumor-specific self-amplified CDT.

Graphic Abstract
Lactic acidosis and hyperlactatemia associated with lamivudine accumulation and sepsis in a kidney transplant recipient—a case report and review of the literature
Authors: Hollinger Alexa , Cueni Nadine , Marzolini Catia , Dickenmann Michael , Landmann Emmanuelle , Battegay Manuel , Martinez Aurélien Emmanuel , Siegemund Martin , Leuppi-Taegtmeyer Anne 
Published: September 2021

Abstract
Background
We report a case of sudden, lethal metabolic acidosis in a 70-year-old man on long-term nucleoside reverse transcriptase inhibitor (NRTI) -based antiretroviral therapy (ART) who had developed atypical necrotizing fasciitis 1 month after kidney transplantation.

Case presentation
The HIV infection of the patient was treated for the last four months with an integrase strand inhibitor (dolutegravir 50 mg/d) plus a NRTI backbone including lamivudine (150 mg/d) and abacavir (600 mg/d). In this renal transplant patient we hypothesize that the co-existence of sepsis, renal failure and an accumulation of lamivudine led to the development of fatal metabolic acidosis and hyperlactatemia. Although lamivudine is only rarely associated with hyperlactatemia, there is evidence that overdose may be a risk factor for developing it. In our patient the lamivudine concentration two days after stopping and during hemodiafiltration was more than 50 times higher than therapeutic target trough concentrations. Likely reasons for this were renal impairment and concurrent treatment with trimethoprim, known to inhibit the renal elimination of lamivudine.

Conclusions
NRTIs could trigger the development of hyperlactatemia in septic patients. The use of NRTI sparing regimens might be considered in the presence of this critical condition.
The de novo FAIRification process of a registry for vascular anomalies
Authors: Groenen Karlijn H. J. , Jacobsen Annika , Kersloot Martijn G. , dos Santos Vieira Bruna , van Enckevort Esther , Kaliyaperumal Rajaram , Arts Derk L. , ‘t Hoen Peter A. C. , Cornet Ronald , Roos Marco , Kool Leo Schultze 
Published: September 2021

Abstract
Background
Patient data registries that are FAIR—Findable, Accessible, Interoperable, and Reusable for humans and computers—facilitate research across multiple resources. This is particularly relevant to rare diseases, where data often are scarce and scattered. Specific research questions can be asked across FAIR rare disease registries and other FAIR resources without physically combining the data. Further, FAIR implies well-defined, transparent access conditions, which supports making sensitive data as open as possible and as closed as necessary.

Results
We successfully developed and implemented a process of making a rare disease registry for vascular anomalies FAIR from its conception—de novo. Here, we describe the five phases of this process in detail: (i) pre-FAIRification, (ii) facilitating FAIRification, (iii) data collection, (iv) generating FAIR data in real-time, and (v) using FAIR data. This includes the creation of an electronic case report form and a semantic data model of the elements to be collected (in this case: the “Set of Common Data Elements for Rare Disease Registration” released by the European Commission), and the technical implementation of automatic, real-time data FAIRification in an Electronic Data Capture system. Further, we describe how we contribute to the four facets of FAIR, and how our FAIRification process can be reused by other registries.

Conclusions
In conclusion, a detailed de novo FAIRification process of a registry for vascular anomalies is described. To a large extent, the process may be reused by other rare disease registries, and we envision this work to be a substantial contribution to an ecosystem of FAIR rare disease resources.
Satisfaction with in-patient child and adolescent psychiatric treatment: development and psychometric properties of the BEST questionnaires for adolescents and for parents
Authors: Keller Ferdinand , Naumann Alexander , Fegert Jörg M. 
Published: September 2021

Abstract
Background
Interest in the assessment of patient satisfaction with in-patient psychiatric treatment has steadily increased and several measurement tools are available for the quantification of patients’ experience. However, they are often uni-dimensional or focus mainly on therapeutic relationship and environment, and neglect other important issues such as information about treatment and participation. The BEST questionnaires were developed as comprehensive instruments that include items on all of the mentioned topics. The present study evaluates the psychometric properties of the BEST in a version for adolescents and for parents. Furthermore, the dimensionality of the satisfaction ratings is analyzed.

Method
Descriptive statistics were applied to data of 1582 adolescents (mean age = 15.0 years, SD = 1.65; 62.4% female) and 1998 parents/guardians assessed in seven in-patient units across Germany. The factorial structure of the BEST questionnaires was determined by exploratory and confirmatory factor analyses, including a bifactor model.

Results
The psychometric quality of the scales was strong. Correlations with another assessment instrument of patient satisfaction were good to high, indicating good convergent validity. Exploratory factor analyses revealed three factors in adolescents that were labelled as: Therapeutic relationship, environment, and general satisfaction and treatment success. For parents, the same three factors could be distinguished. Confirmatory bifactor models suggested that the vast majority of variance was accounted for by the general factor; the three specific factors provided some additional information. Agreement between the subscales of adolescents and their parents was only moderate. Parents were usually more satisfied.

Conclusions
The BEST questionnaires can be considered as reliable and valid instruments to not only assess the “classical” aspects of patient satisfaction, but to also assess newer fundamental topics such as children’s rights and treatment participation. For scientific usage, the total score seems superior because of the high explained variance by the general factor, but the subscale scores provide further information. The use of single items seems advantageous for quality management purposes.
Cytogenomic characterization of a de novo 4q34.1 deletion in a girl with mild dysmorphic features and a coagulation disorder
Authors: Meza-Espinoza Juan Pablo , Contreras-Gutiérrez José Alfredo , Arámbula-Meraz Eliakym , González-García Juan Ramón , Domínguez-Quezada Ma. Guadalupe , García-Magallanes Noemí , Madueña-Molina Jesús , Benítez-Pascual Julio , Partida-Pérez Miriam , Picos-Cárdenas Verónica Judith 
Published: September 2021

Abstract
Background
4q deletion syndrome is a rare chromosomal disorder that mostly arises de novo. The syndrome is characterized by craniofacial dysmorphism, digital abnormalities, skeletal alterations, heart malformations, developmental delay, growth retardation, Pierre Robin sequence, autistic spectrum and attention deficit-hyperactivity disorder, although not every patient shows the same features. Array comparative genomic hybridization (aCGH) use improves the detection of tiny chromosomal deletions and allows for a better understanding of genotype–phenotype correlations in affected patients. We report the case of a 6-year-old female patient showing mild dysmorphic features, mild mental disabilities and a coagulation disorder as a consequence of a de novo del(4)(q34.1) characterized by aCGH.

Case presentation
A 6-year-old female patient exhibited special craniofacial features, such as backward-rotated ears, upslanted palpebral fissures, broad nasal bridges, anteverted nares, broad nasal alae, smooth philtrums, smooth nasolabial folds, thin lips, horizontal labial commissures, and retrognathia. In the oral cavity, maxillary deformation, a high arched palate, agenesis of both mandibular canines and fusion of two mandibular incisors were observed. She also displayed bilateral implantation of the proximal thumbs, widely spaced nipples, dorsal kyphosis, hyperlordosis, and clitoral hypertrophy. In addition, the patient presented with coagulopathy, psychomotor delay, attention deficit-hyperactivity disorder, and mild mental disability. A chromosomal study showed the karyotype 46,XX,del(4)(q34.1), while an aCGH analysis revealed an 18.9 Mb deletion of a chromosome 4q subtelomeric region spanning 93 known genes.

Conclusion
The clinical manifestations of this patient were similar to those reported in other individuals with 4q deletion syndrome. Although most of the patients with a 4q34 terminal deletion share similarities, variations in phenotype are also common. In general, clinical effects of chromosomal deletion syndromes depend on the length of the deleted chromosomal segment and, consequently, on the number of lost genes; however, in all of these syndromes, there is no simple correlation between the phenotype and the chromosomal region involved, particularly in cases of 4q deletion.