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| 2. |
- Ravens-Sieberer, Ulrike, et al.
(författare)
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Reliability, construct and criterion validity of the KIDSCREEN-10 score : a short measure for children and adolescents' well-being and health-related quality of life
- 2010
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Ingår i: Quality of Life Research. - Berlin : Springer. - 0962-9343 .- 1573-2649. ; 19:10, s. 1487-1500
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Tidskriftsartikel (refereegranskat)abstract
- BackgroundTo assess the criterion and construct validity of the KIDSCREEN-10 well-being and health-related quality of life (HRQoL) score, a short version of the KIDSCREEN-52 and KIDSCREEN-27 instruments.MethodsThe child self-report and parent report versions of the KIDSCREEN-10 were tested in a sample of 22,830 European children and adolescents aged 8–18 and their parents (n = 16,237). Correlation with the KIDSCREEN-52 and associations with other generic HRQoL measures, physical and mental health, and socioeconomic status were examined. Score differences by age, gender, and country were investigated.ResultsCorrelations between the 10-item KIDSCREEN score and KIDSCREEN-52 scales ranged from r = 0.24 to 0.72 (r = 0.27–0.72) for the self-report version (proxy-report version). Coefficients below r = 0.5 were observed for the KIDSCREEN-52 dimensions Financial Resources and Being Bullied only. Cronbach alpha was 0.82 (0.78), test–retest reliability was ICC = 0.70 (0.67) for the self- (proxy-)report version. Correlations between other children self-completed HRQoL questionnaires and KIDSCREEN-10 ranged from r = 0.43 to r = 0.63 for the KIDSCREEN children self-report and r = 0.22–0.40 for the KIDSCREEN parent proxy report. Known group differences in HRQoL between physically/mentally healthy and ill children were observed in the KIDSCREEN-10 self and proxy scores. Associations with self-reported psychosomatic complaints were r = −0.52 (−0.36) for the KIDSCREEN-10 self-report (proxy-report). Statistically significant differences in KIDSCREEN-10 self and proxy scores were found by socioeconomic status, age, and gender.ConclusionsOur results indicate that the KIDSCREEN-10 provides a valid measure of a general HRQoL factor in children and adolescents, but the instrument does not represent well most of the single dimensions of the original KIDSCREEN-52. Test–retest reliability was slightly below a priori defined thresholds.
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- Bullinger, Monika, et al.
(författare)
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Assessing the quality of life of health-referred children and adolescents with short stature: development and psychometric testing of the QoLISSY instrument.
- 2013
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Ingår i: Health and quality of life outcomes. - : Springer Science and Business Media LLC. - 1477-7525. ; 11:1
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Tidskriftsartikel (refereegranskat)abstract
- BACKGROUND: When evaluating the outcomes of treatment in paediatric endocrinology, the health-related quality of life (HrQoL) of the child is to be taken into consideration. Since few self--reported HrQoL instruments exist for children with diagnosed short stature (dSS), the objective of this study was to develop and psychometrically test a targeted HrQoL instrument for use in multinational clinical research. METHODS: The target population were short stature (height < -2 SDS) children and adolescents (age 8--12 and 13--18 years) with a diagnosis of growth hormone deficiency (GHD) or idiopathic short stature (ISS), differing in growth hormone treatment status. Focus group discussions for concept and item generation, piloting of the questionnaire with cognitive debriefing, and instrument field testing with a retest were conducted simultaneously in five countries. After qualitative and preliminary quantitative analyses, psychometric testing of field test data in terms of reliability and validity including, confirmatory factor analyses (CFA) was performed. RESULTS: Following item generation from focus group discussions, 124 items were included in a pilot test with a cognitive debriefing exercise providing preliminary feedback on item and domain operating characteristics. A field test with 268 participants showed high internal consistency reliabilities (alpha 0.82 -- 0.95), good correlations with generic measures (up to r = .58), significant known group differences (e.g. in height: F = 32, df 244, p < 0.001) and an acceptable CFA model fit suggesting construct validity of the three-domain core structure with 22 items, supplemented by three mediator domains with 29 items. CONCLUSIONS: The QoLISSY questionnaire is a promising step forward in assessing the impact of dSS on HrQoL. It is based on items generated from the subjective experience of short stature children referred for endocrine investigation, is validated for use in five languages and it is easy to administer in clinical and research settings.
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- Kreimeier, Simone, et al.
(författare)
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EQ-5D-Y-5L : developing a revised EQ-5D-Y with increased response categories.
- 2019
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Ingår i: Quality of Life Research. - : Springer. - 0962-9343 .- 1573-2649. ; 28:7, s. 1951-1961
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Tidskriftsartikel (refereegranskat)abstract
- PURPOSE: EQ-5D-Y is a generic measure of health status for children and adolescents aged 8-15 years. Originally, it has three levels of severity in each dimension (3L). This study aimed to develop a descriptive system of EQ-5D-Y with an increased number of severity levels and to test comprehensibility and feasibility.METHODS: The study was conducted in Germany, Spain, Sweden and the UK. In Phase 1, a review of existing instruments and focus group interviews were carried out to create a pool of possible labels for a modified severity classification. Participants aged 8-15 rated the severity of the identified labels in individual sorting and response scaling interviews. In Phase 2, preliminary 4L and 5L versions were constructed for further testing in cognitive interviews with healthy participants aged 8-15 years and children receiving treatment for a health condition.RESULTS: In Phase 1, a total of 233 labels was generated, ranging from 37 (UK) to 79 labels (Germany). Out of these, 7 to 16 possible labels for each dimension in the different languages were rated in 255 sorting and response scaling interviews. Labels covered an appropriate range of severity on the health continuum in all countries. In Phase 2, the 5L version was generally preferred (by 68-88% of the participants per country) over the 4L version.CONCLUSIONS: This multinational study has provided a version of the EQ-5D-Y with 5 severity levels in each dimension. This extended version (EQ-5D-Y-5L) requires testing its psychometric properties and its performance compared to that of the original EQ-5D-Y-3L.
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| 5. |
- Nmezi, Bruce, et al.
(författare)
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An oligodendrocyte silencer element underlies the pathogenic impact of lamin B1 structural variants
- 2023
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Ingår i: bioRxiv : the preprint server for biology. - : Cold Spring Harbor Laboratory Press (CSHL).
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Tidskriftsartikel (refereegranskat)abstract
- The role of non-coding regulatory elements and how they might contribute to tissue type specificity of disease phenotypes is poorly understood. Autosomal Dominant Leukodystrophy (ADLD) is a fatal, adult-onset, neurological disorder that is characterized by extensive CNS demyelination. Most cases of ADLD are caused by tandem genomic duplications involving the lamin B1 gene ( LMNB1 ) while a small subset are caused by genomic deletions upstream of the gene. Utilizing data from recently identified families that carry LMNB1 gene duplications but do not exhibit demyelination, ADLD patient tissues, CRISPR modified cell lines and mouse models, we have identified a novel silencer element that is lost in ADLD patients and that specifically targets overexpression to oligodendrocytes. This element consists of CTCF binding sites that mediate three-dimensional chromatin looping involving the LMNB1 and the recruitment of the PRC2 repressor complex. Loss of the silencer element in ADLD identifies a previously unknown role for silencer elements in tissue specificity and disease causation.
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| 6. |
- Ravens-Sieberer, Ulrike, et al.
(författare)
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Feasibility, reliability, and validity of the EQ-5D-Y : results from a multinational study
- 2010
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Ingår i: Quality of Life Research. - : Springer. - 0962-9343 .- 1573-2649. ; 19:6, s. 887-897
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Tidskriftsartikel (refereegranskat)abstract
- PurposeTo examine the feasibility, reliability, and validity of the newly developed EQ-5D-Y.MethodsThe EQ-5D-Y was administered in population samples of children and adolescents in Germany, Italy, South Africa, Spain, and Sweden. Percentages of missing values and reported problems were calculated. Test–retest reliability was determined. Spearman’s rank correlation coefficients with other generic measures of HRQOL were calculated. Known groups’ validity was examined by comparing groups with a priori expected differences in HRQOL.ResultsBetween 91 and 100% of the respondents provided valid scorings. Sweden had the lowest proportion of reported problems (1–24.9% across EQ-5D-Y dimensions), with the highest proportions in South Africa (2.8–47.3%) and Italy (4.3–39.0%). Percentages of agreement in test–retest reliability ranged between 69.8 and 99.7% in the EQ-5D-Y dimensions; Kappa coefficients were up to 0.67. Correlation coefficients with other measures of self-rated health indicated convergent validity (up to r = −0.56). Differences between groups classified according to presence of chronic conditions, self-rated overall health and psychological problems provided preliminary evidence of known groups’ validity.ConclusionsResults provide preliminary evidence of the instrument’s feasibility, reliability and validity. Further study is required in clinical samples and for possible future applications in economic analyses.
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| 7. |
- Wille, Nora, et al.
(författare)
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Development of the EQ-5D-Y : a child-friendly version of the EQ-5D
- 2010
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Ingår i: Quality of Life Research. - : Springer. - 0962-9343 .- 1573-2649. ; 19:6, s. 875-886
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Tidskriftsartikel (refereegranskat)abstract
- PurposeTo develop a self-report version of the EQ-5D for younger respondents, named the EQ-5D-Y (Youth); to test its comprehensibility for children and adolescents and to compare results obtained using the standard adult EQ-5D and the EQ-5D-Y.MethodsAn international task force revised the content of EQ-5D and wording to ensure relevance and clarity for young respondents. Children’s and adolescents’ understanding of the EQ-5D-Y was tested in cognitive interviews after the instrument was translated into German, Italian, Spanish and Swedish. Differences between the EQ-5D and the EQ-5D-Y regarding frequencies of reported problems were investigated in Germany, Spain and South Africa.ResultsThe content of the EQ-5D dimensions proved to be appropriate for the measurement of HRQOL in young respondents. The wording of the questionnaire had to be adapted which led to small changes in the meaning of some items and answer options. The adapted EQ-5D-Y was satisfactorily understood by children and adolescents in different countries. It was better accepted and proved more feasible than the EQ-5D. The administration of the EQ-5D and of the EQ-5D-Y causes differences in frequencies of reported problems.ConclusionsThe newly developed EQ-5D-Y is a useful tool to measure HRQOL in young people in an age-appropriate manner.
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