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Unlabelled: A 5-year-old girl presented with multiple tumours of the central nervous system. As on the first MRI scan bilateral vestibular schwannomas were not detected due to their small size, she initially did not meet the criteria for neurofibromatosis type 2 (NF2), although her clinical symptoms were highly suggestive for the diagnosis. Using molecular studies, a mutation in the NF2 gene was found confirming the clinical suspicion at an early age and indicating the value of molecular analysis. Follow-up MRI 3 years later demonstrated bilateral vestibular schwannomas more clearly, since they had increased in size.Conclusion: In children, magnetic resonance imaging can be inconclusive for the diagnosis of neurofibromatosis type 2, since very small vestibular schwannomas may be missed. In these cases molecular studies may provide additional evidence for the diagnosis. We propose guidelines for a screening protocol for children at risk for having neurofibromatosis type 2. [ABSTRACT FROM AUTHOR]

Unlabelled: In this double-blind randomised placebo-controlled trial it was investigated during a two-year follow-up whether oral bisphosphonates (Olpadronate 10 mg/m2/day) influence quality of life in children with osteogenesis imperfecta (OI). Thirty-four children with OI (classified according to Sillence criteria), aged 3 to 18 years of age, with a restricted level of ambulation were included. Randomisation was performed using a list of computer generated random numbers to allocate patients to receive Olpadronate or placebo. Quality of life was measured using self-perception profile for children (SPPC) and health-utility index (HUI). Differences between baseline measurements and measurements at two years follow-up were analysed within the Olpadronate and placebo group using a student's t-test. Differences in HUI and SPPC regression coefficients were analysed by random-effects repeated measures analysis (SAS, Proc Mixed, version 8.2), adjusted for age, gender and type of OI. Within the Olpadronate group there was a significant decrease in pain utility; however, difference in six months' regression coefficients between the placebo and Olpadronate group were not significant. Within the placebo group there was a significant increase in scholastic competence and behavioural conduct. The item behavioural conduct showed a steeper annual regression coefficient favourable for the placebo group. In the other SPPC items none of the annual regression coefficients showed a significant difference between the Olpadronate and the placebo group.Conclusion: We found only slight differences in quality of life in favour of the bisphosphonate group. A small but not significant decrease in pain was detected in the bisphosphonate group. [ABSTRACT FROM AUTHOR]

Objective: Epicardial adipose tissue thickness (EATT) is suggested to play a role in the development of cardiovascular disease. In adolescents it is correlated with BMI z-score, cardiovascular risk factors, and pro- and anti-inflammatory markers. EATT of overweight/obese children was compared with EATT of normal weight peers (cross-sectional design). We investigated the association between EATT, cardiovascular risk factors and pro- and anti-inflammatory markers and the effect of a one year, multidisciplinary, treatment program on EATT in overweight/obese children (longitudinal design). Methods: EATT was measured by echocardiography (25 obese, 8 overweight and 15 normal weight children; median age 5.1 years). In the overweight/obese children blood pressure, lipid profile, glucose, insulin, high sensitive CRP, and adiponectin concentrations were measured. In overweight/obese children participating in a multidisciplinary treatment program, measurements were repeated after 4 and 12 months. Results: EATT was significantly higher in the overweight (median 1.38mm) and obese (median 1.57mm) children compared to normal weight children (median 0.87mm). Among obese children EATT was significantly inversely associated with adiponectin (r = −0.485). Conclusions: EATT is increased in overweight/obese children and is inversely associated with adiponectin. Echocardiographic measurement of EATT is easy and might serve as a simple tool for cardio-metabolic risk stratification. [ABSTRACT FROM AUTHOR]

Introduction: Childhood obesity has serious health risks including the development of metabolic syndrome, cardiovascular disease and mortality later in life. The critical growth period from 3 to 7 years provides a window of opportunity for interventions. The goal of this study is to evaluate a one year, multidisciplinary, low-intensity treatment program for young obese children, complemented with web-based modules, called "AanTafel!", on body composition, cardiometabolic risk profile, quality of life (HRQoL), eating behavior and physical activity. Methods: In the pre-post-test design all measures were taken at baseline, 4 months, at the end of treatment and 3 years after baseline. Results: Thirteen boys and 27 girls with median BMI z-score of, respectively, 4.2 and 3.3 aged 3 to 8 started "AanTafel!". Eighty percent (n = 32) completed treatment. BMI z-score decreased with 0.45 (end of treatment) and sustained after 3 years. At the start, 16.7% of the children had all four components of metabolic syndrome which decreased to 0%. HDL cholesterol significantly increased. Concentrations of the markers IL18, e-selectin, and sICAM significantly decreased indicating a reduction of inflammation. Conclusion: "AanTafel!" is effective in improving health of obese young children. The reduction of overweight is clinically relevant and sustained after 3 years. [ABSTRACT FROM AUTHOR]

Background: What is the appropriate time to start complementary feeding for preterm infants? The answer to this question is yet under debate. The timing of initiating complementary feeding may be associated with overweight in term infants. This systematic review aimed to study the effect of the timing of initiating complementary feeding on overweight in preterm infants. Predefined search items included preterm infants, complementary feeding, overweight, and their synonyms. Summary: The search identified 15,749 articles, of which 5 articles were included. Three studies presented data of randomized controlled trials and 2 studies were cohort studies. Two randomized controlled trials found no significant difference in body mass index (BMI) Z-score between the intervention groups at 12 months of age. One randomized controlled trial presented a significant greater mean rate of growth in length per week until 12 months in the preterm weaning strategy-group compared with the current best practices. One observational study concluded that each month the infants received complementary food later, the Z-score for length and weight was reduced by 0.1. Key Messages: No clear conclusion could be drawn from the included studies. This review illustrates the need for further research to access the effect of the timing of initiating complementary feeding on overweight in preterm infants. [ABSTRACT FROM AUTHOR]

Background: Childhood obesity prevalence has increased worldwide and is an important risk factor for type 2 diabetes (T2D) and cardiovascular disease (CVD). The production of inflammatory adipokines by obese adipose tissue contributes to the development of T2D and CVD. While levels of circulating adipokines such as adiponectin and leptin have been established in obese children and adults, the expression of adiponectin and leptin receptors on circulating immune cells can modulate adipokine signalling, but has not been studied so far. Here, we aim to establish the expression of adiponectin and leptin receptors on circulating immune cells in obese children pre and post-lifestyle intervention compared to normal weight control children. Methods: 13 obese children before and after a 1-year lifestyle intervention were compared with an age and sex-matched normal weight control group of 15 children. Next to routine clinical and biochemical parameters, circulating adipokines were measured, and flow cytometric analysis of adiponectin receptor 1 and 2 (AdipoR1, AdipoR2) and leptin receptor expression on peripheral blood mononuclear cell subsets was performed. Results: Obese children exhibited typical clinical and biochemical characteristics compared to controls, including a higher BMI-SD, blood pressure and circulating leptin levels, combined with a lower insulin sensitivity index (QUICKI). The 1-year lifestyle intervention resulted in stabilization of their BMI-SD. Overall, circulating leukocyte subsets showed distinct adipokine receptor expression profiles. While monocytes expressed high levels of all adipokine receptors, NK and iNKT cells predominantly expressed AdipoR2, and B-lymphocytes and CD4+ and CD8+ T-lymphocyte subsets expressed AdipoR2 as well as leptin receptor. Strikingly though, leukocyte subset numbers and adipokine receptor expression profiles were largely similar in obese children and controls. Obese children showed higher naïve B-cell numbers, and pre-intervention also higher numbers of immature transition B-cells and intermediate CD14++CD16+ monocytes combined with lower total monocyte numbers, compared to controls. Furthermore, adiponectin receptor 1 expression on nonclassical CD14+CD16++ monocytes was consistently upregulated in obese children pre-intervention, compared to controls. However, none of the differences in leukocyte subset numbers and adipokine receptor expression profiles between obese children and controls remained significant after multiple testing correction. Conclusions: First, the distinct adipokine receptor profiles of circulating leukocyte subsets may partly explain the differential impact of adipokines on leukocyte subsets. Second, the similarities in adipokine receptor expression profiles between obese children and normal weight controls suggest that adipokine signaling in childhood obesity is primarily modulated by circulating adipokine levels, instead of adipokine receptor expression. [ABSTRACT FROM AUTHOR]

Background: In term infants, there is evidence that early complementary feeding is a risk factor for childhood obesity. Therefore, timely introduction of complementary feeding during infancy is necessary. The World Health Organization (WHO) and European Society for Paediatric Gastroenterology Hepatology and Nutrition (ESPGHAN) both developed recommendations for the start of complementary feeding for term-born infants. However, these guidelines cannot be directly translated to preterm infants. Recent literature looking at the introduction of complementary feeding in preterm infants gives contrasting information. Given these contrasting reports on the introduction of solid foods in premature born infants, a systematic review is needed. The primary objective of this study is to analyze the effect of the time starting complementary feeding on overweight (including obesity) in preterm infants. Methods: An electronic systematic literature search with pre-defined terms will be conducted in Cochrane, PubMed, EMBASE, Web of Science, Scopus, and CINAHL. There will be no restriction for time period. Primarily, data from randomized controlled trials (RCTs) will be included in this systematic review. Search terms will include preterm infants, complementary feeding, overweight, and their synonyms. Article selection, including risk of bias assessment, will be performed by three reviewers independently. Body mass index standard deviation score (BMI-SDS or BMI-Z-score) will be used to compare studies. The consistency of results across the studies will influence the decision whether or not to combine results in a meta-analysis. Studies that cannot be included in the meta-analysis will be described in a narrative analysis. Discussion: This systematic review will give an overview of the existing knowledge on the timing of complementary feeding in preterm infants and the effect on overweight. It will form a basis for future guidelines for complementary feeding for preterm infants. Systematic review registration: PROSPERO CRD42015014215 [ABSTRACT FROM AUTHOR]