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2.
  • Ades, A. E., et al. (författare)
  • Zika virus infection in pregnancy : a protocol for the joint analysis of the prospective cohort studies of the ZIKAlliance, ZikaPLAN and ZIKAction consortia
  • 2020
  • Ingår i: BMJ Open. - : BMJ PUBLISHING GROUP. - 2044-6055. ; 10:12
  • Tidskriftsartikel (refereegranskat)abstract
    • Introduction: Zika virus (ZIKV) infection in pregnancy has been associated with microcephaly and severe neurological damage to the fetus. Our aim is to document the risks of adverse pregnancy and birth outcomes and the prevalence of laboratory markers of congenital infection in deliveries to women experiencing ZIKV infection during pregnancy, using data from European Commission-funded prospective cohort studies in 20 centres in 11 countries across Latin America and the Caribbean.Methods and analysis: We will carry out a centre-by-centre analysis of the risks of adverse pregnancy and birth outcomes, comparing women with confirmed and suspected ZIKV infection in pregnancy to those with no evidence of infection in pregnancy. We will document the proportion of deliveries in which laboratory markers of congenital infection were present. Finally, we will investigate the associations of trimester of maternal infection in pregnancy, presence or absence of maternal symptoms of acute ZIKV infection and previous flavivirus infections with adverse outcomes and with markers of congenital infection. Centre-specific estimates will be pooled using a two-stage approach.Ethics and dissemination: Ethical approval was obtained at each centre. Findings will be presented at international conferences and published in peer-reviewed open access journals and discussed with local public health officials and representatives of the national Ministries of Health, Pan American Health Organization and WHO involved with ZIKV prevention and control activities.
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3.
  • Elsik, Christine G., et al. (författare)
  • The Genome Sequence of Taurine Cattle : A Window to Ruminant Biology and Evolution
  • 2009
  • Ingår i: Science. - : American Association for the Advancement of Science (AAAS). - 0036-8075 .- 1095-9203. ; 324:5926, s. 522-528
  • Tidskriftsartikel (refereegranskat)abstract
    • To understand the biology and evolution of ruminants, the cattle genome was sequenced to about sevenfold coverage. The cattle genome contains a minimum of 22,000 genes, with a core set of 14,345 orthologs shared among seven mammalian species of which 1217 are absent or undetected in noneutherian (marsupial or monotreme) genomes. Cattle-specific evolutionary breakpoint regions in chromosomes have a higher density of segmental duplications, enrichment of repetitive elements, and species-specific variations in genes associated with lactation and immune responsiveness. Genes involved in metabolism are generally highly conserved, although five metabolic genes are deleted or extensively diverged from their human orthologs. The cattle genome sequence thus provides a resource for understanding mammalian evolution and accelerating livestock genetic improvement for milk and meat production.
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4.
  • Langefeld, Carl D., et al. (författare)
  • Transancestral mapping and genetic load in systemic lupus erythematosus
  • 2017
  • Ingår i: Nature Communications. - : NATURE PUBLISHING GROUP. - 2041-1723. ; 8
  • Tidskriftsartikel (refereegranskat)abstract
    • Systemic lupus erythematosus (SLE) is an autoimmune disease with marked gender and ethnic disparities. We report a large transancestral association study of SLE using Immunochip genotype data from 27,574 individuals of European (EA), African (AA) and Hispanic Amerindian (HA) ancestry. We identify 58 distinct non-HLA regions in EA, 9 in AA and 16 in HA (similar to 50% of these regions have multiple independent associations); these include 24 novel SLE regions (P < 5 x 10(-8)), refined association signals in established regions, extended associations to additional ancestries, and a disentangled complex HLA multigenic effect. The risk allele count (genetic load) exhibits an accelerating pattern of SLE risk, leading us to posit a cumulative hit hypothesis for autoimmune disease. Comparing results across the three ancestries identifies both ancestry-dependent and ancestry-independent contributions to SLE risk. Our results are consistent with the unique and complex histories of the populations sampled, and collectively help clarify the genetic architecture and ethnic disparities in SLE.
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5.
  • Bright, Chloe J, et al. (författare)
  • Risk of Soft-Tissue Sarcoma Among 69 460 Five-Year Survivors of Childhood Cancer in Europe
  • 2018
  • Ingår i: Journal of the National Cancer Institute. - : Oxford University Press (OUP). - 1460-2105 .- 0027-8874. ; 110:6
  • Tidskriftsartikel (refereegranskat)abstract
    • Background: Childhood cancer survivors are at risk of subsequent primary soft-tissue sarcomas (STS), but the risks of specific STS histological subtypes are unknown. We quantified the risk of STS histological subtypes after specific types of childhood cancer.Methods: We pooled data from 13 European cohorts, yielding a cohort of 69 460 five-year survivors of childhood cancer. Standardized incidence ratios (SIRs) and absolute excess risks (AERs) were calculated.Results: Overall, 301 STS developed compared with 19 expected (SIR = 15.7, 95% confidence interval [CI] = 14.0 to 17.6). The highest standardized incidence ratios were for malignant peripheral nerve sheath tumors (MPNST; SIR = 40.6, 95% CI = 29.6 to 54.3), leiomyosarcomas (SIR = 29.9, 95% CI = 23.7 to 37.2), and fibromatous neoplasms (SIR = 12.3, 95% CI = 9.3 to 16.0). SIRs for MPNST were highest following central nervous system tumors (SIR = 80.5, 95% CI = 48.4 to 125.7), Hodgkin lymphoma (SIR = 81.3, 95% CI = 35.1 to 160.1), and Wilms tumor (SIR = 76.0, 95% CI = 27.9 to 165.4). Standardized incidence ratios for leiomyosarcoma were highest following retinoblastoma (SIR = 342.9, 95% CI = 245.0 to 466.9) and Wilms tumor (SIR = 74.2, 95% CI = 37.1 to 132.8). AERs for all STS subtypes were generally low at all years from diagnosis (AER < 1 per 10 000 person-years), except for leiomyosarcoma following retinoblastoma, for which the AER reached 52.7 (95% CI = 20.0 to 85.5) per 10 000 person-years among patients who had survived at least 45 years from diagnosis of retinoblastoma.Conclusions: For the first time, we provide risk estimates of specific STS subtypes following childhood cancers and give evidence that risks of MPNSTs, leiomyosarcomas, and fibromatous neoplasms are particularly increased. While the multiplicative excess risks relative to the general population are substantial, the absolute excess risk of developing any STS subtype is low, except for leiomyosarcoma after retinoblastoma. These results are likely to be informative for both survivors and health care providers.
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6.
  • Byrne, Julianne, et al. (författare)
  • The PanCareSurFup consortium : research and guidelines to improve lives for survivors of childhood cancer
  • 2018
  • Ingår i: European Journal of Cancer. - : ELSEVIER SCI LTD. - 0959-8049 .- 1879-0852. ; 103:Nov, s. 238-248
  • Tidskriftsartikel (refereegranskat)abstract
    • Background: Second malignant neoplasms and cardiotoxicity are among the most serious and frequent adverse health outcomes experienced by childhood and adolescent cancer survivors (CCSs) and contribute significantly to their increased risk of premature mortality. Owing to differences in health-care systems, language and culture across the continent, Europe has had limited success in establishing multi-country collaborations needed to assemble the numbers of survivors required to clarify the health issues arising after successful cancer treatment. PanCareSurFup (PCSF) is the first pan-European project to evaluate some of the serious long-term health risks faced by survivors. This article sets out the overall rationale, methods and preliminary results of PCSF. Methods: The PCSF consortium pooled data from 13 cancer registries and hospitals in 12 European countries to evaluate subsequent primary malignancies, cardiac disease and late mortality in survivors diagnosed between ages 0 and 20 years. In addition, PCSF integrated radiation dosimetry to sites of second malignancies and to the heart, developed evidence-based guidelines for long-term care and for transition services, and disseminated results to survivors and the public. Results: We identified 115,596 individuals diagnosed with cancer, of whom 83,333 were 5-year survivors and diagnosed from 1940 to 2011. This single data set forms the basis for cohort analyses of subsequent malignancies, cardiac disease and late mortality and case-control studies of subsequent malignancies and cardiac disease in 5-year survivors. Conclusions: PCSF delivered specific estimates of risk and comprehensive guidelines to help survivors and care-givers. The expected benefit is to provide every European CCS with improved access to care and better long-term health.
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7.
  • Celse, Tristan, et al. (författare)
  • OTX2 duplications : a recurrent cause of oculo-auriculo-vertebral spectrum
  • 2023
  • Ingår i: Journal of Medical Genetics. - : BMJ Publishing Group Ltd. - 0022-2593 .- 1468-6244. ; 60:6, s. 620-626
  • Tidskriftsartikel (refereegranskat)abstract
    • Background Oculo-auriculo-vertebral spectrum (OAVS) is the second most common cause of head and neck malformations in children after orofacial clefts. OAVS is clinically heterogeneous and characterised by a broad range of clinical features including ear anomalies with or without hearing loss, hemifacial microsomia, orofacial clefts, ocular defects and vertebral abnormalities. Various genetic causes were associated with OAVS and copy number variations represent a recurrent cause of OAVS, but the responsible gene often remains elusive.Methods We described an international cohort of 17 patients, including 10 probands and 7 affected relatives, presenting with OAVS and carrying a 14q22.3 microduplication detected using chromosomal microarray analysis. For each patient, clinical data were collected using a detailed questionnaire addressed to the referring clinicians. We subsequently studied the effects of OTX2 overexpression in a zebrafish model.Results We defined a 272 kb minimal common region that only overlaps with the OTX2 gene. Head and face defects with a predominance of ear malformations were present in 100% of patients. The variability in expressivity was significant, ranging from simple chondromas to severe microtia, even between intrafamilial cases. Heterologous overexpression of OTX2 in zebrafish embryos showed significant effects on early development with alterations in craniofacial development.Conclusions Our results indicate that proper OTX2 dosage seems to be critical for the normal development of the first and second branchial arches. Overall, we demonstrated that OTX2 genomic duplications are a recurrent cause of OAVS marked by auricular malformations of variable severity.
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8.
  • Fidler, Miranda M., et al. (författare)
  • Risk of Subsequent Bone Cancers Among 69 460 Five-Year Survivors of Childhood and Adolescent Cancer in Europe
  • 2018
  • Ingår i: Journal of the National Cancer Institute. - : Oxford University Press (OUP). - 1460-2105 .- 0027-8874. ; 110:2
  • Tidskriftsartikel (refereegranskat)abstract
    • Introduction: We investigate the risks of subsequent primary bone cancers after childhood and adolescent cancer in 12 European countries. For the first time, we satisfactorily address the risks beyond 40 years from diagnosis and beyond 40 years of age among all survivors.Methods: This largest-ever assembled cohort comprises 69 460 five-year survivors of cancer diagnosed before age 20 years. Standardized incidence ratios, absolute excess risks, and multivariable-adjusted relative risks and relative excess risks were calculated. All statistical tests were two-sided.Results: Overall, survivors were 21.65 times (95% confidence interval = 18.97 to 24.60 times) more likely to be diagnosed with a subsequent primary bone cancer than expected from the general population. The greatest excess numbers of bone cancers were observed after retinoblastoma, bone sarcoma, and soft tissue sarcoma. The excess number of bone cancers declined linearly with both years since diagnosis and attained age (all P < .05). Beyond 40 years from diagnosis and age 40 years, there were at most 0.45 excess bone cancers among all survivors per 10 000 person-years at risk; beyond 30 years from diagnosis and age 30 years, there were at most 5.02 excess bone cancers after each of retinoblastoma, bone sarcoma, and soft tissue sarcoma, per 10 000 person-years at risk.Conclusions: For all survivors combined and the cancer groups with the greatest excess number of bone cancers, the excess numbers observed declined with both age and years from diagnosis. These results provide novel, reliable, and unbiased information about risks and risk factors among long-term survivors of childhood and adolescent cancer.
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9.
  • Kottyan, Leah C., et al. (författare)
  • The IRF5-TNPO3 association with systemic lupus erythematosus has two components that other autoimmune disorders variably share.
  • 2015
  • Ingår i: Human Molecular Genetics. - : Oxford University Press (OUP). - 0964-6906 .- 1460-2083. ; 24:2, s. 582-596
  • Tidskriftsartikel (refereegranskat)abstract
    • Exploiting genotyping, DNA sequencing, imputation and trans-ancestral mapping, we used Bayesian and frequentist approaches to model the IRF5-TNPO3 locus association, now implicated in two immunotherapies and seven autoimmune diseases. Specifically, in systemic lupus erythematosus (SLE), we resolved separate associations in the IRF5 promoter (all ancestries) and with an extended European haplotype. We captured 3230 IRF5-TNPO3 high-quality, common variants across 5 ethnicities in 8395 SLE cases and 7367 controls. The genetic effect from the IRF5 promoter can be explained by any one of four variants in 5.7 kb (P-valuemeta = 6 × 10(-49); OR = 1.38-1.97). The second genetic effect spanned an 85.5-kb, 24-variant haplotype that included the genes IRF5 and TNPO3 (P-valuesEU = 10(-27)-10(-32), OR = 1.7-1.81). Many variants at the IRF5 locus with previously assigned biological function are not members of either final credible set of potential causal variants identified herein. In addition to the known biologically functional variants, we demonstrated that the risk allele of rs4728142, a variant in the promoter among the lowest frequentist probability and highest Bayesian posterior probability, was correlated with IRF5 expression and differentially binds the transcription factor ZBTB3. Our analytical strategy provides a novel framework for future studies aimed at dissecting etiological genetic effects. Finally, both SLE elements of the statistical model appear to operate in Sjögrens syndrome and systemic sclerosis whereas only the IRF5-TNPO3 gene-spanning haplotype is associated with primary biliary cirrhosis, demonstrating the nuance of similarity and difference in autoimmune disease risk mechanisms at IRF5-TNPO3.
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10.
  • Lessard, Christopher J., et al. (författare)
  • Identification of IRF8, TMEM39A, and IKZF3-ZPBP2 as Susceptibility Loci for Systemic Lupus Erythematosus in a Large-Scale Multiracial Replication Study
  • 2012
  • Ingår i: American Journal of Human Genetics. - : Elsevier BV. - 0002-9297 .- 1537-6605. ; 90:4, s. 648-660
  • Tidskriftsartikel (refereegranskat)abstract
    • Systemic lupus erythematosus (SLE) is a chronic heterogeneous autoimmune disorder characterized by the loss of tolerance to self-antigens and dysregulated interferon responses. The etiology of SLE is complex, involving both heritable and environmental factors. Candidate-gene studies and genome-wide association (GWA) scans have been successful in identifying new loci that contribute to disease susceptibility; however, much of the heritable risk has yet to be identified. In this study, we sought to replicate 1,580 variants showing suggestive association with SLE in a previously published GWA scan of European Americans; we tested a multiethnic population consisting of 7,998 SLE cases and 7,492 controls of European, African American, Asian, Hispanic, Gullah, and Amerindian ancestry to find association with the disease. Several genes relevant to immunological pathways showed association with SLE. Three loci exceeded the genome-wide significance threshold: interferon regulatory factor 8 (IRF8; rs11644034; p(meta-Euro) = 2.08 x 10(-10)), transmembrane protein 39A (TMEM39A; rs1132200; p(meta-all) 8.62 x 10(-9)), and 17q21 (rs1453560; p(meta-all) = 3.48 x 10(-10)) between IKAROS family of zinc finger 3 (AIOLOS; IKZF3) and zona pellucida binding protein 2 (ZPBP2). Fine mapping, resequencing, imputation, and haplotype analysis of IRF8 indicated that three independent effects tagged by rs8046526, rs450443, and rs4843869, respectively, were required for risk in individuals of European ancestry. Eleven additional replicated effects (5 x 10(-8) < p(meta-Euro) < 9.99 x 10(-5)) were observed with CFHR1, CADM2, LOC730109/IL12A, LPP, LOC63920, SLU7, ADAMTSL1, C10orf64, OR8D4 FAM19A2, and STXBP6. The results of this study increase the number of confirmed SLE risk loci and identify others warranting further investigation.
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11.
  • Lotze, Heike K., et al. (författare)
  • Global ensemble projections reveal trophic amplification of ocean biomass declines with climate change
  • 2019
  • Ingår i: Proceedings of the National Academy of Sciences of the United States of America. - : Proceedings of the National Academy of Sciences. - 0027-8424 .- 1091-6490. ; 116:26, s. 12907-12912
  • Tidskriftsartikel (refereegranskat)abstract
    • While the physical dimensions of climate change are now routinely assessed through multimodel intercomparisons, projected impacts on the global ocean ecosystem generally rely on individual models with a specific set of assumptions. To address these single-model limitations, we present standardized ensemble projections from six global marine ecosystem models forced with two Earth system models and four emission scenarios with and without fishing. We derive average biomass trends and associated uncertainties across the marine food web. Without fishing, mean global animal biomass decreased by 5% (+/- 4% SD) under low emissions and 17% (+/- 11% SD) under high emissions by 2100, with an average 5% decline for every 1 degrees C of warming. Projected biomass declines were primarily driven by increasing temperature and decreasing primary production, and were more pronounced at higher trophic levels, a process known as trophic amplification. Fishing did not substantially alter the effects of climate change. Considerable regional variation featured strong biomass increases at high latitudes and decreases at middle to low latitudes, with good model agreement on the direction of change but variable magnitude. Uncertainties due to variations in marine ecosystem and Earth system models were similar. Ensemble projections performed well compared with empirical data, emphasizing the benefits of multimodel inference to project future outcomes. Our results indicate that global ocean animal biomass consistently declines with climate change, and that these impacts are amplified at higher trophic levels. Next steps for model development include dynamic scenarios of fishing, cumulative human impacts, and the effects of management measures on future ocean biomass trends.
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12.
  • Matias, Marisa, et al. (författare)
  • Profiles of Parental Burnout Around the Globe : Similarities and Differences Across 36 Countries
  • 2023
  • Ingår i: Cross-cultural research. - 1069-3971 .- 1552-3578. ; 57:5, s. 499-538
  • Tidskriftsartikel (refereegranskat)abstract
    • Parental burnout (PB) is a pervasive phenomenon. Parenting is embedded in cultural values, and previous research has shown the role of individualism in PB. In this paper, we reanalyze previously collected data to identify profiles based on the four dimensions of PB, and explore whether these profiles vary across countries’ levels of collectivistic-individualistic (COL-IND) values. Our sample comprised 16,885 individuals from 36 countries (73% women; 27% men), and we used a latent profile approach to uncover PB profiles. The findings showed five profiles: Fulfilled, Not in PB, Low risk of PB, High risk of PB and Burned out. The profiles pointed to climbing levels of PB in the total sample and in each of the three country groups (High COL/Low IND, Medium COL-IND, Low COL/High IND). Exploratory analyses revealed that distinct dimensions of PB had the most prominent roles in the climbing pattern, depending on the countries’ levels of COL/IND. In particular, we found contrast to be a hallmark dimension and an indicator of severe burnout for individualistic countries. Contrary to our predictions, emotional distance and saturation did not allow a clear differentiation across collectivistic countries. Our findings support several research avenues regarding PB measurement and intervention.
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13.
  • Miranda, Elizabeth, et al. (författare)
  • Recording Patient Data in Burn Unit Logbooks in Rwanda : Who and What Are We Missing?
  • 2021
  • Ingår i: Journal of Burn Care & Research. - : Oxford University Press. - 1559-047X .- 1559-0488. ; 42:3, s. 526-532
  • Tidskriftsartikel (refereegranskat)abstract
    • Systematic data collection in high-income countries has demonstrated a decreasing burn morbidity and mortality, whereas lack of data from low- and middle-income countries hinders a global overview of burn epidemiology. In low- and middle-income countries, dedicated burn registries are few. Instead, burn data are often recorded in logbooks or as one variable in trauma registries, where incomplete or inconsistently recorded information is a known challenge. The University Teaching Hospital of Kigali hosts the only dedicated burn unit in Rwanda and has collected data on patients admitted for acute burn care in logbooks since 2005. This study aimed to assess the data registered between January 2005 and December 2019, to evaluate the extent of missing data, and to identify possible factors associated with "missingness." All data were analyzed using descriptive statistics, Fishers exact test, and Wilcoxon Rank Sum test. In this study, 1093 acute burn patients were included and 64.2% of them had incomplete data. Data completeness improved significantly over time. The most commonly missing variables were whether the patient was referred from another facility and information regarding whether any surgical intervention was performed. Missing data on burn mechanism, burn degree, and surgical treatment were associated with in-hospital mortality. In conclusion, missing data is frequent for acute burn patients in Rwanda, although improvements have been seen over time. As Rwanda and other low- and middle-income countries strive to improve burn care, ensuring data completeness will be essential for the ability to accurately assess the quality of care, and hence improve it.
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14.
  • Mukagaju, Francoise, et al. (författare)
  • What is Known About Burns in East Africa? A Scoping Review
  • 2021
  • Ingår i: Journal of Surgical Research. - : ACADEMIC PRESS INC ELSEVIER SCIENCE. - 0022-4804 .- 1095-8673. ; 266, s. 113-124
  • Forskningsöversikt (refereegranskat)abstract
    • Background: Bums are a global public health concern, with the majority of the disease burden affecting low-and middle-income countries. Yet, as suggested by previous publications, there is a widespread belief that literature about burns in low-and middle-income countries is lacking. Therefore, we aimed to assess with a scoping review, the extent of the literature output on bums in East Africa, and to investigate patient demographics, injury characteristics, treatment and outcomes, as reported from the existing publications. Methods: Studies discussing bums in East Africa were identified by searching PubMed / Medline (National Library of Medicine), EMBASE (Elsevier), Global Health Database (EBSCO), and Global Index Medicus on December 12, 2019. Controlled vocabulary terms (i.e., MeSH, EMTREE, Global Health thesaurus terms) were included when available and appropriate. No year restrictions were applied. Results: A total of 1,044 records were retrieved from the database searches, from which 40 articles from 6 countries published between 1993 and 2019 were included in the final review. No studies were found from five East African countries with the lowest GDP. Most papers focused on pediatric trauma patients or tertiary hospital settings. The total number of burn patients recorded was 44,369, of which the mean proportion of males was 56%. The most common cause of injury was scalds (61%), followed by open flame (17%). Mortality rate ranged from 0-67%. The mean length of stay in hospital was between 9-60 d. Conclusions: Burn data is limited in the East African region, with socio-economically weak countries being particularly underrepresented. This scoping review has identified the largest set of literature on burns in East Africa to date, indicating the importance of reviewing data at a regional or local level, as "global" studies tend to be dominated by high-income country data. Data collection in specific registries is needed to better characterize the exact burden of burn injuries in East Africa. (c) 2021 Elsevier Inc. All rights reserved.
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15.
  • Nezerwa, Yves, et al. (författare)
  • Referral of Burn Patients in the Absence of Guidelines: A Rwandan Study
  • 2022
  • Ingår i: Journal of Surgical Research. - : ACADEMIC PRESS INC ELSEVIER SCIENCE. - 0022-4804 .- 1095-8673. ; 278, s. 216-222
  • Tidskriftsartikel (refereegranskat)abstract
    • Introduction: The management of severe burns and pediatric burns requires an organized system of care delivery, which includes referral guidelines. In Rwanda, the burn unit at the University Teaching Hospital of Kigali (CHUK) is the only dedicated burn unit in the country and admits patients of all ages referred from the other provinces. However, since there are no official referral guidelines, it is unknown whether patients with burns are appropriately referred. This study aims to analyze referral patterns among burn patients admitted at the CHUK burn unit and their adherence to the referral criteria listed by the American Burn Association (ABA), comparing patients transferred to the burn unit from facilities within Kigali and those referred from facilities outside Kigali. Methods: This retrospective study included all patients with acute burns admitted to the CHUK burn unit with data available on the province of origin from 2005 to 2019. Patients with burns younger than 16 y were defined as pediatric burns, as per the CHUK routines. Characteristics of all burns referred from a facility within Kigali (Pat-K) and facilities outside Kigali (Pat-O) were compared using Fishers exact test for categorical variables or Wilcoxon rank-sum test for continuous variables. The adherence to ABA referral criteria was assessed for variables with available data, which were total burnt surface area %, burn thickness, cause of burn, and age. Results: The study population consisted of 1093 patients, of which 1064 had data regarding if they were referred from other facilities to CHUK. Overall, the median age was 3 y (2-16 y), with Pat-O being older than Pat-K (P < 0.001). Scalds were the dominant cause of injury in both groups; flame was more common among Pat-O than among Pat-K (in 25.5% versus 10.6%, P < 0.01). Burns of larger size and depth were more common among Pat-O, as compared to the Pat-K group [median total burnt surface area % = 19% versus 15.5%, respectively ( P < 0.001); presence of full-thickness burns = 55.6% versus 29.7 %, respectively (P < 0.001)]. Hospitalization was longer and in-hospital mortality higher in Pat-O than in Pat-K [LOS = 42 d (interquartile range 11-164) versus 28 d (interquartile range 9-132), P < 0.05; in-hospital mortality = 18.9% versus 10.1%, P < 0.001]. Among Pat-O, 85% had characteristics that mirrored one or more of the analyzed ABA criteria. Conclusions: Although there are no clear guidelines, referral patterns indicate that patients are being appropriately referred for specialized burn care in Rwanda. Compared to patients from Kigali, rural patients had larger and more severe injuries, needed more surgical interventions, and had worse outcomes. Most of referred patients matched criteria listed among international referral guidelines. However, the statement of national referral criteria is essential to improve timely access to adequate care. (C) 2022 Elsevier Inc. All rights reserved.
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16.
  • Niyigena, Anne, et al. (författare)
  • The true costs of cesarean delivery for patients in rural Rwanda : Accounting for post-discharge expenses in estimated health expenditures
  • 2022
  • Ingår i: International Journal for Equity in Health. - : Springer Science and Business Media LLC. - 1475-9276. ; 21:1
  • Tidskriftsartikel (refereegranskat)abstract
    • Introduction: While it is recognized that there are costs associated with postoperative patient follow-up, risk assessments of catastrophic health expenditures (CHEs) due to surgery in sub-Saharan Africa rarely include expenses after discharge. We describe patient-level costs for cesarean section (c-section) and follow-up care up to postoperative day (POD) 30 and evaluate the contribution of follow-up to CHEs in rural Rwanda. Methods: We interviewed women who delivered via c-section at Kirehe District Hospital between September 2019 and February 2020. Expenditure details were captured on an adapted surgical indicator financial survey tool and extracted from the hospital billing system. CHE was defined as health expenditure of ≥ 10% of annual household expenditure. We report the cost of c-section up to 30 days after discharge, the rate of CHE among c-section patients stratified by in-hospital costs and post-discharge follow-up costs, and the main contributors to c-section follow-up costs. We performed a multivariate logistic regression using a backward stepwise process to determine independent predictors of CHE at POD30 at α ≤ 0.05. Results: Of the 479 participants in this study, 90% were classified as impoverished before surgery and an additional 6.4% were impoverished by the c-section. The median out-of-pocket costs up to POD30 was US$122.16 (IQR: $102.94, $148.11); 63% of these expenditures were attributed to post-discharge expenses or lost opportunity costs (US$77.50; IQR: $67.70, $95.60). To afford c-section care, 64.4% borrowed money and 18.4% sold possessions. The CHE rate was 27% when only considering direct and indirect costs up to the time of discharge and 77% when including the reported expenses up to POD30. Transportation and lost household wages were the largest contributors to post-discharge costs. Further, CHE at POD30 was independently predicted by membership in community-based health insurance (aOR = 3.40, 95% CI: 1.21,9.60), being a farmer (aOR = 2.25, 95% CI:1.00,3.03), primary school education (aOR = 2.35, 95% CI:1.91,4.66), and small household sizes had 0.22 lower odds of experiencing CHE compared to large households (aOR = 0.78, 95% CI:0.66,0.91). Conclusion: Costs associated with surgical follow-up are often neglected in financial risk calculations but contribute significantly to the risk of CHE in rural Rwanda. Insurance coverage for direct medical costs is insufficient to protect against CHE. Innovative follow-up solutions to reduce costs of patient transport and compensate for household lost wages need to be considered.
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17.
  • Nkurunziza, Theoneste, et al. (författare)
  • mHealth-community health worker telemedicine intervention for surgical site infection diagnosis : a prospective study among women delivering via caesarean section in rural Rwanda
  • 2022
  • Ingår i: BMJ Global Health. - : BMJ Publishing Group. - 2059-7908. ; 7:7
  • Tidskriftsartikel (refereegranskat)abstract
    • Background Surgical site infections (SSIs) cause a significant global public health burden in low and middle-income countries. Most SSIs develop after patient discharge and may go undetected. We assessed the feasibility and diagnostic accuracy of an mHealth-community health worker (CHW) home-based telemedicine intervention to diagnose SSIs in women who delivered via caesarean section in rural Rwanda. Methods This prospective cohort study included women who underwent a caesarean section at Kirehe District Hospital between September 2019 and March 2020. At postoperative day 10 (+/- 3 days), a trained CHW visited the woman at home, provided wound care and transmitted a photo of the wound to a remote general practitioner (GP) via WhatsApp. The GP reviewed the photo and made an SSI diagnosis. The next day, the woman returned to the hospital for physical examination by an independent GP, whose SSI diagnosis was considered the gold standard for our analysis. We describe the intervention process indicators and report the sensitivity and specificity of the telemedicine-based diagnosis. Results Of 787 women included in the study, 91.4% (n=719) were located at their home by the CHW and all of them (n=719, 100%) accepted the intervention. The full intervention was completed, including receipt of GP telemedicine diagnosis within 1 hour, for 79.0% (n=623). The GPs diagnosed 30 SSIs (4.2%) through telemedicine and 38 SSIs (5.4%) through physical examination. The telemedicine sensitivity was 36.8% and specificity was 97.6%. The negative predictive value was 96.4%. Conclusions Implementation of an mHealth-CHW home-based intervention in rural Rwanda and similar settings is feasible. Patients acceptance of the intervention was key to its success. The telemedicine-based SSI diagnosis had a high negative predictive value but a low sensitivity. Further studies must explore strategies to improve accuracy, such as accompanying wound images with clinical data or developing algorithms using machine learning.
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19.
  • Reulen, Raoul C, et al. (författare)
  • Risk Factors for Primary Bone Cancer After Childhood Cancer : A PanCare Childhood and Adolescent Cancer Survivor Care and Follow-Up Studies Nested Case-Control Study
  • 2023
  • Ingår i: Journal of clinical oncology : official journal of the American Society of Clinical Oncology. - 0732-183X .- 1527-7755. ; 41:21, s. 3735-3746
  • Tidskriftsartikel (refereegranskat)abstract
    • PURPOSE: Radiation to the bone and exposure to alkylating agents increases the risk of bone cancer among survivors of childhood cancer, but there is uncertainty regarding the risks of bone tissue radiation doses below 10 Gy and the dose-response relationship for specific types of chemotherapy.METHODS: Twelve European countries contributed 228 cases and 228 matched controls to a nested case-control study within a cohort of 69,460 5-year survivors of childhood cancer. Odds ratios (ORs) of developing bone cancer for different levels of cumulative radiation exposure and cumulative doses of specific types of chemotherapy were calculated. Excess ORs were calculated to investigate the shape and extent of any dose-response relationship.RESULTS: The OR associated with bone tissue exposed to 1-4 Gy was 4.8-fold (95% CI, 1.2 to 19.6) and to 5-9 Gy was 9.6-fold (95% CI, 2.4 to 37.4) compared with unexposed bone tissue. The OR increased linearly with increasing dose of radiation ( P trend < .001) up to 78-fold (95% CI, 9.2 to 669.9) for doses of ≥40 Gy. For cumulative alkylating agent doses of 10,000-19,999 and ≥20,000 mg/m 2, the radiation-adjusted ORs were 7.1 (95% CI, 2.2 to 22.8) and 8.3 (95% CI, 2.8 to 24.4), respectively, with independent contributions from each of procarbazine, ifosfamide, and cyclophosphamide. Other cytotoxics were not associated with bone cancer. CONCLUSION: To our knowledge, we demonstrate-for the first time-that the risk of bone cancer is increased 5- to 10-fold after exposure of bone tissue to cumulative radiation doses of 1-9 Gy. Alkylating agents exceeding 10,000 mg/m 2 increase the risk 7- to 8-fold, particularly following procarbazine, ifosfamide, and cyclophosphamide. These substantially elevated risks should be used to develop/update clinical follow-up guidelines and survivorship care plans.
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20.
  • Roskam, Isabelle, et al. (författare)
  • Parental Burnout Around the Globe : a 42-Country Study
  • 2021
  • Ingår i: Affective science. - : Springer Science and Business Media LLC. - 2662-205X .- 2662-2041. ; 2, s. 58-79
  • Tidskriftsartikel (refereegranskat)abstract
    • High levels of stress in the parenting domain can lead to parental burnout, a condition that has severe consequences for both parents and children. It is not yet clear, however, whether parental burnout varies by culture, and if so, why it might do so. In this study, we examined the prevalence of parental burnout in 42 countries (17,409 parents; 71% mothers; Mage = 39.20) and showed that the prevalence of parental burnout varies dramatically across countries. Analyses of cultural values revealed that individualistic cultures, in particular, displayed a noticeably higher prevalence and mean level of parental burnout. Indeed, individualism plays a larger role in parental burnout than either economic inequalities across countries, or any other individual and family characteristic examined so far, including the number and age of children and the number of hours spent with them. These results suggest that cultural values in Western countries may put parents under heightened levels of stress.Supplementary Information: The online version contains supplementary material available at 10.1007/s42761-020-00028-4.
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21.
  • Shephard, Kerry, et al. (författare)
  • Empowering Students in Higher-Education to Teach and Learn
  • 2017
  • Ingår i: New Zealand journal of educational studies. - : Springer Science and Business Media LLC. - 0028-8276 .- 2199-4714. ; 52:1, s. 41-55
  • Tidskriftsartikel (refereegranskat)abstract
    • We explored opportunities, advantages and barriers to enabling students to establish student-led learning events at a New Zealand university. We used an action-research approach to explore if students felt empowered to use the infrastructure of this university to realise something that they themselves set out to achieve. We discovered that, in achieving a series of open discussions about sustainability, students adopted a democratic, distributed form of decision-making, not unlike a typical academic model, with leaders taking temporary roles that included passing on responsibility to those who followed. Students were proud of the events they created and identified the discussion format as something different from their experience as undergraduate students in our institution. This article, co-authored by staff and students, considers whether higher education processes that do empower students do so adequately and the extent to which students are prepared by higher education to take on powerful roles after they graduate.
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22.
  • Shyaka, Ian, et al. (författare)
  • Estimating mortality risk in burn patients admitted at Rwanda's largest referral hospital
  • 2024
  • Ingår i: International Journal of Burns and Trauma. - : E-CENTURY PUBLISHING CORP. - 2160-2026. ; 14:1, s. 25-31
  • Tidskriftsartikel (refereegranskat)abstract
    • Background: Burns is a disease of poverty, disproportionately affecting populations in low- and middle-income countries, where most of the injuries and the deaths caused by burns occurs. In Sub-Saharan Africa, it is estimated that one fifth of burn victims die from their injuries. Mortality prediction indexes are used to estimate outcomes after provided burn care, which has been used in burn services of high-income countries over the last 60 years. It remains to be seen whether these are reliable in low-income settings. This study aimed to analyze inhospital mortality and to apply mortality estimation indexes in burn patients admitted to the only specialized burn unit in Rwanda. Methods: This retrospective study included all patients with burns admitted at the burn unit (BU) of the University Teaching Hospital in Kigali (CHUK) between 2005 and 2019. Patient data were collected from the BU logbook. Descriptive statistics were calculated with frequency (%) and median (interquartile range, IQR). Association between burns characteristics and in-hospital mortality was calculated with Fisher's exact test, and Wilcoxon rank, as appropriate. Mortality estimation analysis, including Baux score, Lethal Area 50 (LA50), and point of futility, was calculated in those patients with complete data on age and TBSA. LA50 and point-of-futility were calculated using logistic regression. Results: Among the 1093 burn patients admitted at the CHUK burn unit during the study period, 49% (n=532) had complete data on age and TBSA. Their median age, TBSA, and Baux score were 3.4 years (IQR 1.9-17.1), 15% (IQR 11-25), and 24 (IQR 16-38), respectively. Overall, reported in-hospital mortality was 13% (n=121/931), LA50 for Baux score was 89.9 (95% CI 76.2-103.7), and the point-of-futility was at a Baux score of 104. Conclusion: Mortality estimation indexes based on age and TBSA are feasible to use in low-income settings. However, implementation of systematic data collection would contribute to a more accurate calculation of the mortality risk.
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23.
  • Tittensor, Derek P., et al. (författare)
  • A protocol for the intercomparison of marine fishery and ecosystem models : Fish-MIP v1.0
  • 2018
  • Ingår i: Geoscientific Model Development. - : Copernicus GmbH. - 1991-959X .- 1991-9603. ; 11:4, s. 1421-1442
  • Tidskriftsartikel (refereegranskat)abstract
    • Model intercomparison studies in the climate and Earth sciences communities have been crucial to building credibility and coherence for future projections. They have quantified variability among models, spurred model development, contrasted within- and among-model uncertainty, assessed model fits to historical data, and provided ensemble projections of future change under specified scenarios. Given the speed and magnitude of anthropogenic change in the marine environment and the consequent effects on food security, biodiversity, marine industries, and society, the time is ripe for similar comparisons among models of fisheries and marine ecosystems. Here, we describe the Fisheries and Marine Ecosystem Model Intercomparison Project protocol version 1.0 (Fish-MIP v1.0), part of the Inter-Sectoral Impact Model Intercomparison Project (ISIMIP), which is a cross-sectoral network of climate impact modellers. Given the complexity of the marine ecosystem, this class of models has substantial heterogeneity of purpose, scope, theoretical underpinning, processes considered, parameterizations, resolution (grain size), and spatial extent. This heterogeneity reflects the lack of a unified understanding of the marine ecosystem and implies that the assemblage of all models is more likely to include a greater number of relevant processes than any single model. The current Fish-MIP protocol is designed to allow these heterogeneous models to be forced with common Earth System Model (ESM) Coupled Model Intercomparison Project Phase 5 (CMIP5) outputs under prescribed scenarios for historic (from the 1950s) and future (to 2100) time periods; it will be adapted to CMIP phase 6 (CMIP6) in future iterations. It also describes a standardized set of outputs for each participating Fish-MIP model to produce. This enables the broad characterization of differences between and uncertainties within models and projections when assessing climate and fisheries impacts on marine ecosystems and the services they provide. The systematic generation, collation, and comparison of results from Fish-MIP will inform an understanding of the range of plausible changes in marine ecosystems and improve our capacity to define and convey the strengths and weaknesses of model-based advice on future states of marine ecosystems and fisheries. Ultimately, Fish-MIP represents a step towards bringing together the marine ecosystem modelling community to produce consistent ensemble medium- and long-term projections of marine ecosystems.
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24.
  • Wilder-Smith, Annelies, et al. (författare)
  • The legacy of ZikaPLAN: a transnational research consortium addressing Zika
  • 2021
  • Ingår i: Global Health Action. - : Taylor & Francis. - 1654-9716 .- 1654-9880. ; 14
  • Forskningsöversikt (refereegranskat)abstract
    • Global health research partnerships with institutions from high-income countries and low- and middle-income countries are one of the European Commission's flagship programmes. Here, we report on the ZikaPLAN research consortium funded by the European Commission with the primary goal of addressing the urgent knowledge gaps related to the Zika epidemic and the secondary goal of building up research capacity and establishing a Latin American-European research network for emerging vector-borne diseases. Five years of collaborative research effort have led to a better understanding of the full clinical spectrum of congenital Zika syndrome in children and the neurological complications of Zika virus infections in adults and helped explore the origins and trajectory of Zika virus transmission. Individual-level data from ZikaPLAN`s cohort studies were shared for joint analyses as part of the Zika Brazilian Cohorts Consortium, the European Commission-funded Zika Cohorts Vertical Transmission Study Group, and the World Health Organization-led Zika Virus Individual Participant Data Consortium. Furthermore, the legacy of ZikaPLAN includes new tools for birth defect surveillance and a Latin American birth defect surveillance network, an enhanced Guillain-Barre Syndrome research collaboration, a de-centralized evaluation platform for diagnostic assays, a global vector control hub, and the REDe network with freely available training resources to enhance global research capacity in vector-borne diseases.
  •  
25.
  • Wilder-Smith, Annelies, et al. (författare)
  • ZikaPLAN : addressing the knowledge gaps and working towards a research preparedness network in the Americas
  • 2019
  • Ingår i: Global Health Action. - : Taylor & Francis. - 1654-9716 .- 1654-9880. ; 12:1
  • Tidskriftsartikel (refereegranskat)abstract
    • Zika Preparedness Latin American Network (ZikaPLAN) is a research consortium funded by the European Commission to address the research gaps in combating Zika and to establish a sustainable network with research capacity building in the Americas. Here we present a report on ZikaPLAN`s mid-term achievements since its initiation in October 2016 to June 2019, illustrating the research objectives of the 15 work packages ranging from virology, diagnostics, entomology and vector control, modelling to clinical cohort studies in pregnant women and neonates, as well as studies on the neurological complications of Zika infections in adolescents and adults. For example, the Neuroviruses Emerging in the Americas Study (NEAS) has set up more than 10 clinical sites in Colombia. Through the Butantan Phase 3 dengue vaccine trial, we have access to samples of 17,000 subjects in 14 different geographic locations in Brazil. To address the lack of access to clinical samples for diagnostic evaluation, ZikaPLAN set up a network of quality sites with access to well-characterized clinical specimens and capacity for independent evaluations. The International Committee for Congenital Anomaly Surveillance Tools was formed with global representation from regional networks conducting birth defects surveillance. We have collated a comprehensive inventory of resources and tools for birth defects surveillance, and developed an App for low resource regions facilitating the coding and description of all major externally visible congenital anomalies including congenital Zika syndrome. Research Capacity Network (REDe) is a shared and open resource centre where researchers and health workers can access tools, resources and support, enabling better and more research in the region. Addressing the gap in research capacity in LMICs is pivotal in ensuring broad-based systems to be prepared for the next outbreak. Our shared and open research space through REDe will be used to maximize the transfer of research into practice by summarizing the research output and by hosting the tools, resources, guidance and recommendations generated by these studies. Leveraging on the research from this consortium, we are working towards a research preparedness network.
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