7 research outputs found

    Multicenter research priorities in pediatric CMR: results of a collaborative wiki survey

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    Multicenter studies in pediatric cardiovascular magnetic resonance (CMR) improve statistical power and generalizability. However, a structured process for identifying important research topics has not been developed. We aimed to (1) develop a list of high priority knowledge gaps, and (2) pilot the use of a wiki survey to collect a large group of responses. Knowledge gaps were defined as areas that have been either unexplored or under-explored in the research literature. High priority goals were: (1) feasible and answerable from a multicenter research study, and (2) had potential for high impact on the field of pediatric CMR. Seed ideas were contributed by a working group and imported into a pairwise wiki survey format which allows for new ideas to be uploaded and voted upon (https://allourideas.org). Knowledge gaps were classified into 2 categories: ‘Clinical CMR Practice’ (16 ideas) and ‘Disease Specific Research’ (22 ideas). Over a 2-month period, 3,658 votes were cast by 96 users, and 2 new ideas were introduced. The 3 highest scoring sub-topics were myocardial disorders (9 ideas), translating new technology & techniques into clinical practice (7 ideas), and normal reference values (5 ideas). The highest priority gaps reflected strengths of CMR (e.g., myocardial tissue characterization; implementation of technologic advances into clinical practice), and deficiencies in pediatrics (e.g., data on normal reference values). The wiki survey format was effective and easy to implement, and could be used for future surveys

    Multicenter research priorities in pediatric CMR: results of a collaborative wiki survey

    Get PDF
    Multicenter studies in pediatric cardiovascular magnetic resonance (CMR) improve statistical power and generalizability. However, a structured process for identifying important research topics has not been developed. We aimed to (1) develop a list of high priority knowledge gaps, and (2) pilot the use of a wiki survey to collect a large group of responses. Knowledge gaps were defined as areas that have been either unexplored or under-explored in the research literature. High priority goals were: (1) feasible and answerable from a multicenter research study, and (2) had potential for high impact on the field of pediatric CMR. Seed ideas were contributed by a working group and imported into a pairwise wiki survey format which allows for new ideas to be uploaded and voted upon ( https://allourideas.org ). Knowledge gaps were classified into 2 categories: 'Clinical CMR Practice' (16 ideas) and 'Disease Specific Research' (22 ideas). Over a 2-month period, 3,658 votes were cast by 96 users, and 2 new ideas were introduced. The 3 highest scoring sub-topics were myocardial disorders (9 ideas), translating new technology & techniques into clinical practice (7 ideas), and normal reference values (5 ideas). The highest priority gaps reflected strengths of CMR (e.g., myocardial tissue characterization; implementation of technologic advances into clinical practice), and deficiencies in pediatrics (e.g., data on normal reference values). The wiki survey format was effective and easy to implement, and could be used for future surveys

    Multicenter research priorities in pediatric CMR: results of a collaborative wiki survey

    No full text
    Abstract Multicenter studies in pediatric cardiovascular magnetic resonance (CMR) improve statistical power and generalizability. However, a structured process for identifying important research topics has not been developed. We aimed to (1) develop a list of high priority knowledge gaps, and (2) pilot the use of a wiki survey to collect a large group of responses. Knowledge gaps were defined as areas that have been either unexplored or under-explored in the research literature. High priority goals were: (1) feasible and answerable from a multicenter research study, and (2) had potential for high impact on the field of pediatric CMR. Seed ideas were contributed by a working group and imported into a pairwise wiki survey format which allows for new ideas to be uploaded and voted upon ( https://allourideas.org ). Knowledge gaps were classified into 2 categories: ‘Clinical CMR Practice’ (16 ideas) and ‘Disease Specific Research’ (22 ideas). Over a 2-month period, 3,658 votes were cast by 96 users, and 2 new ideas were introduced. The 3 highest scoring sub-topics were myocardial disorders (9 ideas), translating new technology & techniques into clinical practice (7 ideas), and normal reference values (5 ideas). The highest priority gaps reflected strengths of CMR (e.g., myocardial tissue characterization; implementation of technologic advances into clinical practice), and deficiencies in pediatrics (e.g., data on normal reference values). The wiki survey format was effective and easy to implement, and could be used for future surveys

    Tool use without a tool:Kinematic characteristics of pantomiming as compared to actual use and the effect of brain damage

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    Movement goals and task mechanics differ substantially between actual tool use and corresponding pantomimes. In addition, apraxia seems to be more severe during pantomime than during actual tool use. Comparisons of these two modes of action execution using quantitative methods of movement analyses are rare. In the present study, repetitive scooping movements with a ladle from a bowl into a plate were recorded and movement kinematics was analyzed. Brain-damaged patients using their ipsilesional hand and healthy control subjects were tested in three conditions: pantomime, demonstration with the tool only, and actual use in the normal context. Analysis of the hand trajectories during the transport component revealed clear differences between the tasks, such as slower actual use and moderate deficits in patients with left brain damage (LBD). LBD patients were particularly impaired in the scooping component: LBD patients with apraxia exhibited reduced hand rotation at the bowl and the plate. The deficit was most obvious during pantomime but actual use was also affected, and reduced hand rotation was consistent across conditions as indicated by strong pair-wise correlations between task conditions. In healthy control subjects, correlations between movement parameters were most evident between the pantomime and demonstration conditions but weak in correlation pairs involving actual use. From these findings and published neuroimaging evidence, we conclude that for a specific tool-use action, common motor schemas are activated but are adjusted and modified according to the actual task constraints and demands. An apraxic LBD individual can show a deficit across all three action conditions, but the severity can differ substantially between conditions

    Diversité des prises en charge hospitalières de l'anorexie mentale en psychiatrie en France

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    International audienceBackground: Hospitalization is rare in anorexia nervosa (AN) and local application of indications is heterogeneous. However, no study has evaluated the effect of these different treatment modalities on the mean length of stay. Our objective was to describe the context and modalities of a wide range of hospital care programs offered to patients with anorexia nervosa in eleven specialized French psychiatric centers for patients from childhood to adulthood. This work was carried out within the framework of the EVHAN (Evaluation of Hospitalization for AN, Eudract number: 2007-A01110-53, registered in Clinical trials) research program. The EVHAN program comprises five main lines: weight objectives at discharge, the practice or not of a separation period, the use of clear nutritional dietary objectives (cognitive/behavioral), the intensity of family involvement in treatment, and the existence or absence of a stabilization phase before ending inpatient treatment. These main lines will make it possible to study the impact of treatment modalities on the future of patients in the short and medium term (at discharge and at 1-year follow-up). Methods: The eleven centers are located in France (Bordeaux, Nantes, Paris and Ile-de-France, Rouen and Saint-Étienne). Various staff members (psychiatrists, somatic doctors, nurses, dieticians, psychologists) from each center were interviewed using a semi-structured questionnaire. Data on operating modalities and context of care were collected and analyzed. Results: Four of the eleven centers were exclusively child/adolescent centers, and seven of 11 were adolescent (from 11, 13 or 16 years) and young adult centers. All centers offered a graduated approach from outpatient to full hospitalization. The majority had a number of beds allocated for patients with eating disorders. The criteria for hospitalization were homogeneous with respect to somatic and/or psychic severity prefiguring the consensus criteria defined by the French Health Authority (HAS) in 2010. Child/adolescent units used the weight curve to set weight objectives at discharge (between the 10th and 50th percentiles). Most adult units used weight objectives at discharge corresponding to a body mass index between 17 and 20 kg/m2. Nine centers used a written or oral care contract. One unit did not separate the patient from her/his usual environment, the others had a practice gradient of partial separation and total separation times. These were either short, lasting a maximum of 3 weeks, or long, lasting more than 3 weeks. Conversely, patients were not isolated within the unit, and benefited from a rich social life, depending on her/his physical condition. The patient's family was in contact with the team and fully supported. The longest periods of separation involved adolescents and adults. Nutritional support varied from a group approach (meals in the dining room, standardized meals of the care center) to very individualized approaches within a specific framework. All the units reported meeting with families at least once during the hospitalization; with the patient's parents for child/adolescent patients and/or unmarried patients and with the husband/wife for married patients. The majority of the centers requested a phase of weight stabilization, whatever the age before hospital discharge. Conclusion: There is international and national consensus regarding the indications for hospitalization, and the main lines of multidisciplinary care to be developed within this framework. However, local application of these indications was heterogeneous resulting in diverse modalities of hospital care for anorexia nervosa in France. Specialized teams have developed management strategies arising from their “team culture”. The complexity of the anorexic pathology, due to the psyche-soma intrication and the diversity of age groups, highlights the complexity of care available. The impact of this diversity of hospital care on patient outcomes will be studied as a result of this work

    Admixture Mapping in Lupus Identifies Multiple Functional Variants within IFIH1 Associated with Apoptosis, Inflammation, and Autoantibody Production

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    Systemic lupus erythematosus (SLE) is an inflammatory autoimmune disease with a strong genetic component. African-Americans (AA) are at increased risk of SLE, but the genetic basis of this risk is largely unknown. To identify causal variants in SLE loci in AA, we performed admixture mapping followed by fine mapping in AA and European-Americans (EA). Through genome-wide admixture mapping in AA, we identified a strong SLE susceptibility locus at 2q22-24 (LOD=6.28), and the admixture signal is associated with the European ancestry (ancestry risk ratio ~1.5). Large-scale genotypic analysis on 19,726 individuals of African and European ancestry revealed three independently associated variants in the IFIH1 gene: an intronic variant, rs13023380 [P(meta) = 5.20×10(-14); odds ratio, 95% confidence interval = 0.82 (0.78-0.87)], and two missense variants, rs1990760 (Ala946Thr) [P(meta) = 3.08×10(-7); 0.88 (0.84-0.93)] and rs10930046 (Arg460His) [P(dom) = 1.16×10(-8); 0.70 (0.62-0.79)]. Both missense variants produced dramatic phenotypic changes in apoptosis and inflammation-related gene expression. We experimentally validated function of the intronic SNP by DNA electrophoresis, protein identification, and in vitro protein binding assays. DNA carrying the intronic risk allele rs13023380 showed reduced binding efficiency to a cellular protein complex including nucleolin and lupus autoantigen Ku70/80, and showed reduced transcriptional activity in vivo. Thus, in SLE patients, genetic susceptibility could create a biochemical imbalance that dysregulates nucleolin, Ku70/80, or other nucleic acid regulatory proteins. This could promote antibody hypermutation and auto-antibody generation, further destabilizing the cellular network. Together with molecular modeling, our results establish a distinct role for IFIH1 in apoptosis, inflammation, and autoantibody production, and explain the molecular basis of these three risk alleles for SLE pathogenesis

    Admixture Mapping in Lupus Identifies Multiple Functional Variants within IFIH1 Associated with Apoptosis, Inflammation, and Autoantibody Production

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