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- Durno, C., et al.
(författare)
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Survival Benefit for Individuals With Constitutional Mismatch Repair Deficiency Undergoing Surveillance
- 2021
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Ingår i: Journal of Clinical Oncology. - : American Society of Clinical Oncology (ASCO). - 0732-183X .- 1527-7755. ; 39:25
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Tidskriftsartikel (refereegranskat)abstract
- PURPOSE Constitutional mismatch repair deficiency syndrome (CMMRD) is a lethal cancer predisposition syndrome characterized by early-onset synchronous and metachronous multiorgan tumors. We designed a surveillance protocol for early tumor detection in these individuals. PATIENTS AND METHODS Data were collected from patients with confirmed CMMRD who were registered in the International Replication Repair Deficiency Consortium. Tumor spectrum, efficacy of the surveillance protocol, and malignant transformation of low-grade lesions were examined for the entire cohort. Survival outcomes were analyzed for patients followed prospectively from the time of surveillance implementation. RESULTS A total of 193 malignant tumors in 110 patients were identified. Median age of first cancer diagnosis was 9.2 years (range: 1.7-39.5 years). For patients undergoing surveillance, all GI and other solid tumors, and 75% of brain cancers were detected asymptomatically. By contrast, only 16% of hematologic malignancies were detected asymptomatically (P < .001). Eighty-nine patients were followed prospectively and used for survival analysis. Five-year overall survival (OS) was 90% (95% CI, 78.6 to 100) and 50% (95% CI, 39.2 to 63.7) when cancer was detected asymptomatically and symptomatically, respectively (P = .001). Patient outcome measured by adherence to the surveillance protocol revealed 4-year OS of 79% (95% CI, 54.8 to 90.9) for patients undergoing full surveillance, 55% (95% CI, 28.5 to 74.5) for partial surveillance, and 15% (95% CI, 5.2 to 28.8) for those not under surveillance (P < .0001). Of the 64 low-grade tumors detected, the cumulative likelihood of transformation from low-to high-grade was 81% for GI cancers within 8 years and 100% for gliomas in 6 years. CONCLUSION Surveillance and early cancer detection are associated with improved OS for individuals with CMMRD.
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| 4. |
- Bornhorst, C., et al.
(författare)
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Early Life Factors and Inter-Country Heterogeneity in BMI Growth Trajectories of European Children: The IDEFICS Study
- 2016
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Ingår i: Plos One. - San Francisco : Public Library of Science (PLoS). - 1932-6203. ; 11:2
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Tidskriftsartikel (refereegranskat)abstract
- Background Starting from birth, this explorative study aimed to investigate between-country differences in body mass index (BMI) trajectories and whether early life factors explain these differences. The sample included 7,644 children from seven European countries (Belgium, Cyprus, Germany, Hungary, Italy, Spain, Sweden) participating in the multi-centre IDEFICS study. Information on early life factors and in total 53,409 repeated measurements of height and weight from 0 to <12 years of age were collected during the baseline (2007/2008) and follow-up examination (2009/2010) supplemented by records of routine child health visits. Country-specific BMI growth curves were estimated using fractional polynomial mixed effects models. Several covariates focussing on early life factors were added to the models to investigate their role in the between-countries differences. Large between-country differences were observed with Italian children showing significantly higher mean BMI values at all ages >= 3 years compared to the other countries. For instance, at age 11 years mean BMI values in Italian boys and girls were 22.3 [21.9; 22.8; 99% confidence interval] and 22.0 [21.5; 22.4], respectively, compared to a range of 18.4 [18.1; 18.8] to 20.3 [19.8; 20.7] in boys and 18.2 [17.8; 18.6] to 20.3 [19.8; 20.7] in girls in the other countries. After adjustment for early life factors, differences between country-specific BMI curves became smaller. Maternal BMI was the factor being most strongly associated with BMI growth (p<0.01 in all countries) with associations increasing during childhood. Gestational weight gain (GWG) was weakly associated with BMI at birth in all countries. In some countries, positive associations between BMI growth and children not being breastfed, mothers' smoking during pregnancy and low educational level of parents were found. Early life factors seem to explain only some of the inter-country variation in growth. Maternal BMI showed the strongest association with children's BMI growth.
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| 5. |
- Bornhorst, C., et al.
(författare)
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The effects of hypothetical behavioral interventions on the 13-year incidence of overweight/obesity in children and adolescents
- 2023
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Ingår i: International Journal of Behavioral Nutrition and Physical Activity. - 1479-5868. ; 20:1
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Tidskriftsartikel (refereegranskat)abstract
- Background In view of the high burden of childhood overweight/obesity (OW/OB), it is important to identify targets for interventions that may have the greatest effects on preventing OW/OB in early life. Using methods of causal inference, we studied the effects of sustained behavioral interventions on the long-term risk of developing OW/OB based on a large European cohort.Methods Our sample comprised 10 877 children aged 2 to < 10 years at baseline who participated in the well-phenotyped IDEFICS/I.Family cohort. Children were followed from 2007/08 to 2020/21. Applying the parametric g-formula, the 13-year risk of developing OW/OB was estimated under various sustained hypothetical interventions on physical activity, screen time, dietary intake and sleep duration. Interventions imposing adherence to recommendations (e.g. maximum 2 h/day screen time) as well as interventions 'shifting' the behavior by a specified amount (e.g. decreasing screen time by 30 min/day) were compared to 'no intervention' (i.e. maintaining the usual or so-called natural behavior). Separately, the effectiveness of these interventions in vulnerable groups was assessed.Results The 13-year risk of developing OW/OB was 30.7% under no intervention and 25.4% when multiple interventions were imposed jointly. Meeting screen time and moderate-to-vigorous physical activity (MVPA) recommendations were found to be most effective, reducing the incidence of OW/OB by -2.2 [-4.4;-0.7] and -2.1 [-3.7;-0.8] percentage points (risk difference [95% confidence interval]), respectively. Meeting sleep recommendations (-0.6 [-1.1;-0.3]) had a similar effect as increasing sleep duration by 30 min/day (-0.6 [-0.9;-0.3]). The most effective intervention in children of parents with low/medium educational level was being member in a sports club; for children of mothers with OW/OB, meeting screen time recommendations and membership in a sports club had the largest effects.Conclusions While the effects of single behavioral interventions sustained over 13 years were rather small, a joint intervention on multiple behaviors resulted in a relative reduction of the 13-year OW/OB risk by between 10 to 26%. Individually, meeting MVPA and screen time recommendations were most effective. Nevertheless, even under the joint intervention the absolute OW/OB risk remained at a high level of 25.4% suggesting that further strategies to better prevent OW/OB are required.
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| 6. |
- Do, S., et al.
(författare)
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Associations Between Psychosocial Well-Being, Stressful Life Events and Emotion-Driven Impulsiveness in European Adolescents
- 2022
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Ingår i: Journal of Youth and Adolescence. - : Springer Science and Business Media LLC. - 0047-2891 .- 1573-6601. ; 51, s. 1106-1117
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Tidskriftsartikel (refereegranskat)abstract
- Knowing the extent to which mental well-being and stressful life events during adolescence contribute to personality characteristics related to risk-taking behaviors, such as emotion-driven impulsiveness, is highly relevant for the development of health promotion measures. This study examined whether psychosocial well-being and different stressful life events are associated with emotion-driven impulsiveness. In total, 3,031 adolescents (52% girls; M-age = 13.6 years) were included from the I. Family Study, a cross-sectional examination on lifestyle-related behaviors conducted across eight European countries in 2013/14. Linear mixed-effects regression models showed that higher psychosocial well-being was associated with lower emotion-driven impulsiveness independent of socio-demographic, health-related, and parental variables. A higher number of stressful life events was associated with higher emotion-driven impulsiveness. Psychosocial well-being and stressful life events need to be further considered in the development and tailoring of health promotion strategies that aim to reduce emotion-driven impulsiveness.
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| 7. |
- Hunsberger, Monica, 1973, et al.
(författare)
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Dietary Carbohydrate and Nocturnal Sleep Duration in Relation to Children's BMI: Findings from the IDEFICS Study in Eight European Countries
- 2015
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Ingår i: Nutrients. - : MDPI AG. - 2072-6643. ; 7:12, s. 10223-10236
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Tidskriftsartikel (refereegranskat)abstract
- Previous research has found an association between being overweight and short sleep duration. We hypothesized that this association could be modified by a high carbohydrate (HC) diet and that the timing and type (starch or sugar) of intake may be an important factor in this context. Participants in the prospective, eight-country European study IDEFICS were recruited from September 2007 to June 2008, when they were aged two to nine years. Data on lifestyle, dietary intake and anthropometry were collected on two occasions. This study included 5944 children at baseline and 4301 at two-year follow-up. For each meal occasion (morning, midday, and evening), starch in grams and sugar in grams were divided by total energy intake (EI), and quartiles calculated. HC-starch and HC-sugar intake categories were defined as the highest quartile for each meal occasion. In a mutually adjusted linear regression model, short sleep duration as well as HC-starch in the morning were positively associated with body mass index (BMI) z-scores at baseline. HC-starch at midday was positively associated with body mass index (BMI) z-scores in children with short sleep duration, and negatively associated with BMI z-scores in those with normal sleep. After adjustment for baseline BMI z-scores, associations between total HC from starch or sugar and high BMI z-scores at two-year follow-up did not persist. Our observations offer a perspective on optimal timing for macronutrient consumption, which is known to be influenced by circadian rhythms. Reduced carbohydrate intake, especially during morning and midday meals, and following nocturnal sleep duration recommendations are two modifiable factors that may protect children from being overweight in the future.
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| 8. |
- Iguacel, I., et al.
(författare)
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Associations between social vulnerabilities and dietary patterns in European children: the Identification and prevention of Dietary- and lifestyle-induced health EFfects In Children and infantS (IDEFICS) study
- 2016
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Ingår i: British Journal of Nutrition. - : Cambridge University Press (CUP). - 0007-1145 .- 1475-2662. ; 116:7, s. 1288-1297
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Tidskriftsartikel (refereegranskat)abstract
- Socio-economic inequalities in childhood can determine dietary patterns, and therefore future health. This study aimed to explore associations between social vulnerabilities and dietary patterns assessed at two time points, and to investigate the association between accumulation of vulnerabilities and dietary patterns. A total of 9301 children aged 2-9 years participated at baseline and 2-year follow-up examinations of the Identification and prevention of Dietary-and lifestyle-induced health EFfects In Children and infantS study. In all, three dietary patterns were identified at baseline and follow-up by applying the K-means clustering algorithm based on a higher frequency of consumption of snacks and fast food (processed), sweet foods and drinks (sweet), and fruits and vegetables (healthy). Vulnerable groups were defined at baseline as follows: children whose parents lacked a social network, children from single-parent families, children of migrant origin and children with unemployed parents. Multinomial mixed models were used to assess the associations between social vulnerabilities and children's dietary patterns at baseline and follow-up. Children whose parents lacked a social network (OR 1.31; 99% CI 1.01, 1.70) and migrants (OR 1.45; 99% CI 1.15, 1.83) were more likely to be in the processed cluster at baseline and follow-up. Children whose parents were homemakers (OR 0.74; 99% CI 0.60, 0.92) were less likely to be in the processed cluster at baseline. A higher number of vulnerabilities was associated with a higher probability of children being in the processed cluster (OR 1.78; 99% CI 1.21, 2.62). Therefore, special attention should be paid to children of vulnerable groups as they present unhealthier dietary patterns.
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| 9. |
- Iguacel, I., et al.
(författare)
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Prospective associations between socioeconomically disadvantaged groups and metabolic syndrome risk in European children. Results from the IDEFICS study
- 2018
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Ingår i: International Journal of Cardiology. - : Elsevier BV. - 0167-5273 .- 1874-1754. ; 272, s. 333-340
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Tidskriftsartikel (refereegranskat)abstract
- Objective: Socioeconomic disadvantages during childhood are hypothesised to have negative implications for health. We aimed to investigate the association between socioeconomic disadvantages and children's total metabolic syndrome (MetS) score at baseline and follow-up and the extent to which socioeconomic disadvantages over time and the accumulation of these socioeconomic disadvantages can affect children's MetS risk. Methods: The two-year longitudinal IDEFICS study included 2401 European children (aged 2.0-9.9) with complete information of the 16,229 participating at baseline. Sociodemographic variables, psychosocial factors and lifestyle were proxy-reported via questionnaires. Socioeconomically disadvantaged groups included children from families with low income, low education, migrant origin, unemployed parents, parents who lacked a social network, and from non-traditional families. MetS risk score was calculated as the sum of z-scores of waist circumference, blood pressure, lipids and insulin resistance. Linear mixed-effects models were used to study the association between social disadvantages and MetS risk. Models were adjusted for sex, age, well-being and lifestyle (fruit and vegetables consumption, physical activity, screen time). Results: At both time points, children from low-income families (0.20 [0.03-0.37]); (beta estimate and 99% confidence interval), children from non-traditional families (0.14 [0.02-026]), children whose parents were unemployed (0.31 [0.05-0.57]) and children who accumulated >3 disadvantages (0.21 [0.04-0.37]) showed a higher MetS score compared to non-socioeconomically disadvantaged groups. Conclusion: Children from socioeconomically disadvantaged families are at high metabolic risk independently of diet, physical activity, sedentary behaviours and well-being. Interventions focusing on these socioeconomically disadvantaged groups should be developed to tackle health disparities. (C) 2018 Elsevier B.V. All rights reserved.
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| 10. |
- Nobre, Liana, et al.
(författare)
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Outcomes of BRAF V600E pediatric gliomas treated with targeted BRAF inhibition
- 2020
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Ingår i: JCO Precision Oncology. - 2473-4284. ; 3, s. 561-571
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Tidskriftsartikel (refereegranskat)abstract
- © 2020 by American Society of Clinical Oncology PURPOSE Children with pediatric gliomas harboring a BRAF V600E mutation have poor outcomes with current chemoradiotherapy strategies. Our aim was to study the role of targeted BRAF inhibition in these tumors. PATIENTS AND METHODS We collected clinical, imaging, molecular, and outcome information from patients with BRAF V600E–mutated glioma treated with BRAF inhibition across 29 centers from multiple countries. RESULTS Sixty-seven patients were treated with BRAF inhibition (pediatric low-grade gliomas [PLGGs], n = 56; pediatric high-grade gliomas [PHGGs], n = 11) for up to 5.6 years. Objective responses were observed in 80% of PLGGs, compared with 28% observed with conventional chemotherapy (P, .001). These responses were rapid (median, 4 months) and sustained in 86% of tumors up to 5 years while receiving therapy. After discontinuation of BRAF inhibition, 76.5% (13 of 17) of patients with PLGG experienced rapid progression (median, 2.3 months). However, upon rechallenge with BRAF inhibition, 90% achieved an objective response. Poor prognostic factors in conventional therapies, such as concomitant homozygous deletion of CDKN2A, were not associated with lack of response to BRAF inhibition. In contrast, only 36% of those with PHGG responded to BRAF inhibition, with all but one tumor progressing within 18 months. In PLGG, responses translated to 3-year progression-free survival of 49.6% (95% CI, 35.3% to 69.5%) versus 29.8% (95% CI, 20% to 44.4%) for BRAF inhibition versus chemotherapy, respectively (P = .02). CONCLUSION Use of BRAF inhibition results in robust and durable responses in BRAF V600E–mutated PLGG. Prospective studies are required to determine long-term survival and functional outcomes with BRAF inhibitor therapy in childhood gliomas.
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