Continuous admission to primary school and mental health problems

Background: Younger children in a school class have higher rates of mental health problems if admission to primary school occurs once a year. This study examines whether this relative age effect also occurs if children are admitted to school continuously throughout the year. Methods: We assessed mental health problems based on parent-reports (using the Child Behavior Checklist, CBCL) and on professional assessments, among two Dutch national samples of in total 12,221 children aged 5-15 years (response rate: 86.9%). Results: At ages 5-6, we found a higher occurrence of mental health problems in relatively young children, both for mean CBCL scores (p = 0.017) and for problems assessed by child health professionals (p <0.0001). At ages 7-15, differences by relative age did not reach statistical significance. Conclusion: Continuous admission to primary school does not prevent mental health problems among young children, but may do so at older ages. Its potential for the prevention of mental problems deserves further study

Survival is influenced by approaches to local treatment of Ewing sarcoma within an international randomised controlled trial: analysis of EICESS-92

Background: Two national clinical trial groups, United Kingdom Children's Cancer and Leukaemia Group (CCLG) and the German Paediatric Oncology and Haematology Group (GPOH) together undertook a randomised trial, EICESS-92, which addressed chemotherapy options for Ewing's sarcoma. We sought the causes of unexpected survival differences between the study groups. Methods: 647 patients were randomised. Cox regression analyses were used to compare event-free survival (EFS) and overall survival (OS) between the two study groups. Results: 5-year EFS rates were 43% (95% CI 36-50%) and 57% (95% CI 52-62) in the CCLG and GPOH patients, respectively; corresponding 5-year OS rates were 52% (95% CI 45-59%) and 66% (95% CI 61-71). CCLG patients were less likely to have both surgery and radiotherapy (18 vs. 59%), and more likely to have a single local therapy modality compared to the GPOH patients (72 vs. 35%). Forty-five percent of GPOH patients had pre-operative radiotherapy compared to 3% of CCLG patients. In the CCLG group local recurrence (either with or without metastases) was the first event in 22% of patients compared with 7% in the GPOH group. After allowing for the effects of age, metastases, primary site, histology and local treatment modality, the risk of an EFS event was 44% greater in the CCLG cohort (95% CI 10-89%, p = 0.009), and the risk of dying was 30% greater, but not statistically significant (95% CI 3-74%, p = 0.08). Conclusions: Unexpected differences in EFS and OS occurred between two patient cohorts recruited within an international randomised trial. Failure to select or deliver appropriate local treatment modalities for Ewing's sarcoma may compromise chances of cure.Trial registration Supported by Deutsche Krebshilfe (Grants No. DKH M43/92/Jü2 and DKH 70-2551 Jü3), and European Union Biomedicine and Health Programme (Grants No. BMH1-CT92-1341 and BMH4-983956), and Cancer Research United Kingdom. Clinical trial information can be found for the following: NCT0000251

Preimplantation genetic testing for Neurofibromatosis type 1:more than 20 years of clinical experience

Neurofibromatosis type 1 (NF1) is an autosomal dominant disorder that affects the skin and the nervous system. The condition is completely penetrant with extreme clinical variability, resulting in unpredictable manifestations in affected offspring, complicating reproductive decision-making. One of the reproductive options to prevent the birth of affected offspring is preimplantation genetic testing (PGT). We performed a retrospective review of the medical files of all couples (n = 140) referred to the Dutch PGT expert center with the indication NF1 between January 1997 and January 2020. Of the couples considering PGT, 43 opted out and 15 were not eligible because of failure to identify the underlying genetic defect or unmet criteria for in vitro fertilization (IVF) treatment. The remaining 82 couples proceeded with PGT. Fertility assessment prior to IVF treatment showed a higher percentage of male infertility in males affected with NF1 compared to the partners of affected females. Cardiac evaluations in women with NF1 showed no contraindications for IVF treatment or pregnancy. For 67 couples, 143 PGT cycles were performed. Complications of IVF treatment were not more prevalent in affected females compared to partners of affected males. The transfer of 174 (out of 295) unaffected embryos led to 42 ongoing pregnancies with a pregnancy rate of 24.1% per embryo transfer. There are no documented cases of misdiagnosis following PGT in this cohort. With these results, we aim to provide an overview of PGT for NF1 with regard to success rate and safety, to optimize reproductive counseling and PGT treatment for NF1 patients.</p

The World Starts With Me: using intervention mapping for the systematic adaptation and transfer of school-based sexuality education from Uganda to Indonesia

Evidence-based health promotion programmes, including HIV/AIDS prevention and sexuality education programmes, are often transferred to other cultures, priority groups and implementation settings. Challenges in this process include the identification of retaining core elements that relate to the programme’s effectiveness while making changes that enhances acceptance in the new context and for the new priority group. This paper describes the use of a systematic approach to programme adaptation using a case study as an example. Intervention Mapping, a protocol for the development of evidence-based behaviour change interventions, was used to adapt the comprehensive school-based sexuality education programme ‘The World Starts With Me’. The programme was developed for a priority population in Uganda and adapted to a programme for Indonesian secondary school students. The approach helped to systematically address the complexity and challenges of programme adaptation and to find a balance between preservation of essential programme elements (i.e. logic models) that may be crucial to the programme’s effectiveness, including key objectives and theoretical behaviour change methods, and the adaptation of the programme to be acceptable to the new priority group and the programme implementers

Rapid whole exome sequencing in pregnancies to identify the underlying genetic cause in fetuses with congenital anomalies detected by ultrasound imaging

Objective: The purpose of this study was to explore the diagnostic yield and clinical utility of trio-based rapid whole exome sequencing (rWES) in pregnancies of fetuses with a wide range of congenital anomalies detected by ultrasound imaging. Methods: In this observational study, we analyzed the first 54 cases referred to our laboratory for prenatal rWES to support clinical decision making, after the sonographic detection of fetal congenital anomalies. The most common identified congenital anomalies were skeletal dysplasia (n = 20), multiple major fetal congenital anomalies (n = 17) and intracerebral structural anomalies (n = 7). Results: A conclusive diagnosis was identified in 18 of the 54 cases (33%). Pathogenic variants were detected most often in fetuses with skeletal dysplasia (n = 11) followed by fetuses with multiple major fetal congenital anomalies (n = 4) and intracerebral structural anomalies (n = 3). A survey, completed by the physicians for 37 of 54 cases, indicated that the rWES results impacted clinical decision making in 68% of cases. Conclusions: These results suggest that rWES improves prenatal diagnosis of fetuses with congenital anomalies, and has an important impact on prenatal and peripartum parental and clinical decision making

Implementation of a guideline for local health policy making by regional health services: exploring determinants of use by a web survey.

Previous evaluation showed insufficient use of a national guideline for integrated local health policy by Regional Health Services (RHS) in the Netherlands. The guideline focuses on five health topics and includes five checklists to support integrated municipal health policies. This study explores the determinants of guideline use by regional Dutch health professionals