Objectives Tonsillectomy and adenoidectomy in children are controversial subjects with large regional variation in surgical rates, partly explained by cultural differences and lack of high-quality... Show moreObjectives Tonsillectomy and adenoidectomy in children are controversial subjects with large regional variation in surgical rates, partly explained by cultural differences and lack of high-quality evidence on indications for surgery. A quality of care cycle was executed on this topic in the Netherlands. The objective of this study was to estimate changes in healthcare utilisation for paediatric tonsil surgery in the Netherlands.Methods Population-based data on tonsillectomies and adenoidectomies in children up to age 10 were retrieved retrospectively from Dutch administrative databases between 2005 and 2018. A change point analysis was performed to detect the most pivotal change point in surgical rates. We performed univariate analyses to compare surgical patients' characteristics before and after the pivotalpoint . Impact on healthcare budget and societal costs were estimated using current prices and data from cost-effectiveness analyses.Results The annual number of adenotonsillectomies reduced by 10 952 procedures (-39%; from 129 per 10 000 children to 87 per 10 000 children) between 2005 and 2018, and the number of adenoidectomies by 14 757 procedures (-49%; from 138 per 10 000 children to 78 per 10 000 children). The most pivotal change point was observed around 2012, accompanied by small changes in patient selection for surgery before and after 2012. An estimated euro5.3 million per year was saved on the healthcare budget and euro10.4 million per year on societal costs.Conclusion The quality of care cycle resulted in fewer operations, with a concomitant reduction of costs. We suggest that part of these savings be invested in new research to maintain the quality of care cycle. Show less
Purpose Knowledge of post-hematopoietic cell transplantation (HCT) non-hematological autoimmune disease (AD) is far from satisfactory. Method This multicenter retrospective study focuses on... Show morePurpose Knowledge of post-hematopoietic cell transplantation (HCT) non-hematological autoimmune disease (AD) is far from satisfactory. Method This multicenter retrospective study focuses on incidence, risk factors, and outcomes of post-HCT AD in 596 children with primary immunodeficiency (PID) who were transplanted from 2009 to 2018. Results The indications of HCT were severe combined immunodeficiency (SCID, n = 158, 27%) and non-SCID PID (n = 438, 73%). The median age at HCT was 2.3 years (range, 0.04 to 18.3 years). The 5-year overall survival for the entire cohort was 79% (95% cumulative incidence (CIN), 74-83%). The median follow-up of surviving patients was 4.3 years (0.08 to 14.7 years). The CIN of post-HCT AD was 3% (2-5%) at 1 year post-HCT, 7% (5-11%) at 5 years post-HCT, and 11% (7-17%) at 8 years post-HCT. The median onset of post-HCT AD was 2.2 years (0.12 to 9.6 years). Autoimmune thyroid disorder (n = 19, 62%) was the most common post-HCT AD, followed by neuromuscular disorders (n = 7, 22%) and rheumatological manifestations (n = 5, 16%). All patients but one required treatment for post-HCT AD. After multivariate analysis, age at transplant (p = 0.01) and T cell-depleted graft (p < 0.001) were significant predictors of post-HCT AD. None of the T cell-depleted graft recipients developed post-HCT AD. Patients with a lower CD3+ count at 6 months post-HCT had a significant higher incidence of post-HCT AD compared to disease controls. Graft-versus-host disease, viral infection, and donor chimerism had no association with post-HCT AD. Conclusion Post-HCT AD occurred in 11% at 8 years post-HCT and its occurrence was associated with older age at HCT and unmanipulated graft. Show less
Dommelen, P. van; Zoonen, R. van; Vlasblom, E.; Wit, J.M.; Beltman, M.; Expert Comm 2020
Aim To develop a guideline for preventive child healthcare professionals in order to improve early detection of pathological disorders associated with short stature (or growth faltering) or tall... Show moreAim To develop a guideline for preventive child healthcare professionals in order to improve early detection of pathological disorders associated with short stature (or growth faltering) or tall stature (or accelerated growth).Methods We updated the previous Dutch guideline for short stature in children aged 0-9 years and extended it to adolescents (10-17 years), and added a guideline for tall stature, based on literature and input from an expert committee. Specificities were calculated in a cohort of healthy Dutch children aged 0-9 years (n = 970). We investigated the impact of a late onset of puberty on height standard deviation score based on the Dutch growth charts.Results Growth parameters of the guideline include height, the distance between height and target height and change of height over time. Other parameters include diagnostic clues from medical history and physical examination, for example behavioural problems, precocious or delayed puberty, body disproportion and dysmorphic features.Conclusion Preventive child healthcare professionals now have an updated guideline for referring short or tall children to specialist care. Further research is needed on the diagnostic yield after referral and specificity at field level. Show less
Ruszczynski, M.; Ambrozej, D.; Adamiec, A.; Ryczaj, K.; Elenius, V.; Cavkaytar, O.; ... ; Feleszko, W. 2020
Background Asthma-like symptoms in preschool children, such as wheezing and dyspnea, are common time- and resource-consuming diagnostic and management challenges. Quality of wheezing and asthma... Show moreBackground Asthma-like symptoms in preschool children, such as wheezing and dyspnea, are common time- and resource-consuming diagnostic and management challenges. Quality of wheezing and asthma recommendations varies. The purpose of this study, carried out by the European Academy of Allergy and Clinical Immunology (EAACI) Task Force for Preschool Wheeze, was to systematically review and assess the quality of guidelines for diagnosis and treatment of preschool wheezing and/or asthma. Methods The Cochrane Library, MEDLINE, and EMBASE were searched until June 2018. The methodological rigor, quality, and transparency of relevant guidelines were assessed with the use of the Appraisal of Guidelines for Research and Evaluation (AGREE II) tool. Results We identified 26 guidelines. The quality scores for each domain varied. Of all domains, clarity and presentation had the highest mean score, whereas applicability and stakeholder involvement had the lowest. The scores (median) for individual domains were as follows: score and purpose 86%; stakeholder involvement 49%; rigor of development 54%; clarity of presentation 85%; applicability 51%; and editorial independence 63%. Conclusion Although several guidelines on asthma management in children are available, however, their quality varies. Additionally, there is a considerable gap in reliable recommendations on the management and treatment of non-asthmatic preschool wheeze. Show less
Diet and body mass index (BMI) have been shown to affect the gut microbiota of children, but studies are largely performed in developed countries. Here, we conducted a cross-sectional investigation... Show moreDiet and body mass index (BMI) have been shown to affect the gut microbiota of children, but studies are largely performed in developed countries. Here, we conducted a cross-sectional investigation on the differences in the bacterial gut microbiota between normal-weight and overweight urban Filipino children, and determined the relationship between their energy, macronutrient and dietary fiber intakes, and their gut microbiota composition and diversity. Forty-three children (normal-weight,n= 32; overweight,n= 11) participated in the study. Energy and fiber intakes were collected using a semi-quantitative Food Frequency Questionnaire (FFQ). The gut microbiota was profiled using 16S rRNA gene amplicon sequencing of the V3-V4 region. The diet of the children was a mixture of traditional and Western patterns. There were no significant differences in energy, macronutrients and energy-adjusted fiber intakes between the normal-weight and overweight groups, but there were significantly more children meeting the recommended fiber intake in the overweight group. Alpha and beta bacterial diversities did not significantly differ between weight groups. Relative abundance ofBifidobacterium,TuricibacterandClostridiaceae 1were higher in the normal-weight than overweight children, andLachnospirawas higher in overweight children. Show less
Benitez-Paez, A.; Olivares, M.; Szajewska, H.; Piescik-Lech, M.; Polanco, I.; Castillejo, G.; ... ; Sanz, Y. 2020
Celiac disease (CeD) is an immune-mediated disorder triggered by exposure to dietary gluten proteins in genetically predisposed individuals. In addition to the host genome, the microbiome has... Show moreCeliac disease (CeD) is an immune-mediated disorder triggered by exposure to dietary gluten proteins in genetically predisposed individuals. In addition to the host genome, the microbiome has recently been linked to CeD risk and pathogenesis. To progress in our understanding of the role of breast milk microbiota profiles in CeD, we have analyzed samples from a sub-set of mothers (n= 49) included in the PreventCD project, whose children did or did not develop CeD. The results of the microbiota data analysis indicated that neither the BMI, HLA-DQ genotype, the CeD condition nor the gluten-free diet of the mothers could explain the human milk microbiota profiles. Nevertheless, we found that origin country, the offspring's birth date and, consequently, the milk sampling date influenced the abundance and prevalence of microbes in human milk, undergoing a transition from an anaerobic to a more aerobic microbiota, including potential pathogenic species. Furthermore, certain microbial species were more abundant in milk samples from mothers whose children went on to develop CeD compared to those that remained healthy. These included increases in facultative methylotrophs such asMethylobacterium komagataeandMethylocapsa palsarumas well as in species such asBacteroides vulgatus, that consumes fucosylated-oligosaccharides present in human milk, and other breast-abscess associated species. Theoretically, these microbiota components could be vertically transmitted from mothers-to-infants during breastfeeding, thereby influencing CeD risk. Show less
Seizure detection devices can improve epilepsy care, but wearables are not always tolerated. We previously demonstrated good performance of a real-time video-based algorithm for detection of... Show moreSeizure detection devices can improve epilepsy care, but wearables are not always tolerated. We previously demonstrated good performance of a real-time video-based algorithm for detection of nocturnal convulsive seizures in adults with learning disabilities. The algorithm calculates the relative frequency content based on the group velocity reconstruction from video-sequence optical flow. We aim to validate the video algorithm on nocturnal motor seizures in a pediatric population. We retrospectively analyzed the algorithm performance on a database including 1661 full recorded nights of 22 children (age = 3-17 years) with refractory epilepsy at home or in a residential care setting. The algorithm detected 118 of 125 convulsions (median sensitivity per participant = 100%, overall sensitivity = 94%, 95% confidence interval = 61%-100%) and identified all 135 hyperkinetic seizures. Most children had no false alarms; 81 false alarms occurred in six children (median false alarm rate [FAR] per participant per night = 0 [range = 0-0.47], overall FAR = 0.05 per night). Most false alarms (62%) were behavior-related (eg, awake and playing in bed). Our noncontact detection algorithm reliably detects nocturnal epileptic events with only a limited number of false alarms and is suitable for real-time use. Show less
Background: Mitotic rate is a strong predictor of outcome in adult patients with primary cutaneous melanoma, but for children and adolescent patients this is unknown.Objective: We sought to assess... Show moreBackground: Mitotic rate is a strong predictor of outcome in adult patients with primary cutaneous melanoma, but for children and adolescent patients this is unknown.Objective: We sought to assess the prognostic value of primary tumor mitotic rate in children and adolescents with primary melanoma.Methods: This was a cohort study of 156 patients who were <20 years of age and who had clinically localized cutaneous melanoma. Patients <12 years of age were classified as children and those 12 to 19 years of age as adolescents. Clinicopathologic and outcome data were collected. Recurrence-free and melanoma-specific survival were calculated. Univariable and multivariable analyses were performed using Cox proportional hazard models.Results: Thirteen of 156 patients (8%) were children. The mitotic rate was >= 1/mm(2) in 104 patients (67%) and correlated with increasing Breslow thickness. A positive sentinel node was found in 23 of 61 patients (38%) in whom a sentinel lymph node biopsy specimen was obtained. The median follow-up was 61 months. Five-year melanoma-specific and recurrence-free survival rates were 91% and 84%, respectively. Mitotic rate was a stronger predictor of outcome than tumor thickness and was the only factor independently associated with recurrence-free survival.Limitations: This research was conducted at a single institution and the sample size was small.Conclusion: Mitotic rate is an independent predictor of recurrence-free survival in children and adolescents with clinically localized melanoma. Show less
Interethnic prejudice in children has been studied mostly in the United States, but less often in Europe, where the public discourse is increasingly negative about ethnic minorities, especially the... Show moreInterethnic prejudice in children has been studied mostly in the United States, but less often in Europe, where the public discourse is increasingly negative about ethnic minorities, especially the Muslim minority. This study examined in-group favoritism (White preference) and out-group rejection of children of Middle Eastern descent (representing the Muslim minority) among White children in the Netherlands. Social preference for and rejection of children of Middle Eastern descent are compared with preference for and rejection of Black children. Social preference and rejection were measured using a task in which participants were presented with pictures of children with different ethnic appearances, and asked to select who they wanted to (not) play with, (not) sit next to, and invite for their birthday party. In addition, maternal implicit prejudice against people of Middle Eastern descent and explicit attitudes toward their children’s interethnic contact were assessed. The study included 140 children aged 6 to 8 years (M = 7.26, SD = 0.77) and their mothers. The results reveal both in-group favoritism and out-group rejection. The Middle Eastern out-group was preferred less than the Black out-group. Reporting absolutely no reservations about children’s interethnic contact by mothers was associated with less rejection of children of Middle Eastern descent. Findings reveal that young children already show interethnic prejudice and that particularly people of Middle Eastern descent are devalued. The results show that maternal acceptance of child interethnic contact seems to play a role, and provide starting points for further investigation of the relation between parental and child interethnic attitudes. Show less
Outcome of 333 children with acute myeloid leukaemia relapsing after a first allogeneic haematopoietic stem cell transplantation was analyzed. Four-year probability of overall survival (4y-pOS) was... Show moreOutcome of 333 children with acute myeloid leukaemia relapsing after a first allogeneic haematopoietic stem cell transplantation was analyzed. Four-year probability of overall survival (4y-pOS) was 14%. 4y-pOS for 122 children receiving a second haematopoietic stem cell transplantation was 31% and 3% for those that did not (P = <0 center dot 0001). Achievement of a subsequent remission impacted survival (P = <0 center dot 0001). For patients receiving a second transplant survival with or without achieving a subsequent remission was comparable. Graft source (bone marrow vs. peripheral blood stem cells, P = 0 center dot 046) and donor choice (matched family vs. matched unrelated donor, P = 0 center dot 029) positively impacted survival after relapse. Disease recurrence and non-relapse mortality at four years reached 45% and 22%. Show less
Heart rate variability (HRV) is a non-invasive indicator of autonomic nervous system function. HRV recordings show artefacts due to technical and/or biological issues. The Kubios software is one of... Show moreHeart rate variability (HRV) is a non-invasive indicator of autonomic nervous system function. HRV recordings show artefacts due to technical and/or biological issues. The Kubios software is one of the most used software to process HRV recordings, offering different levels of threshold-based artefact correction (i.e., Kubios filters). The aim of the study was to analyze the impact of different Kubios filters on the quantification of HRV derived parameters from short-term recordings in three independent human cohorts. A total of 312 participants were included: 107 children with overweight/obesity (10.0 +/- 1.1 years, 58% men), 132 young adults (22.2 +/- 2.2 years, 33% men) and 73 middle-aged adults (53.6 +/- 5.2 years, 48% men). HRV was assessed using a heart rate monitor during 10-15 min, and the Kubios software was used for HRV data processing using all the Kubios filters available (i.e., 6). Repeated-measures analysis of variance indicated significant differences in HRV derived parameters in the time-domain (all p < 0.001) across the Kubios filters in all cohorts, moreover similar results were observed in the frequency-domain. When comparing two extreme Kubios filters, these statistical differences could be clinically relevant, e.g. more than 10 ms in the standard deviation of all normal R-R intervals (SDNN). In conclusion, the results of the present study suggest that the application of different Kubios filters had a significant impact on HRV derived parameters obtained from short-term recordings in both time and frequency-domains. Show less