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1.	Sundh, Karin Jerhamre, et al. (författare) Cancer in children and young adults born after assisted reproductive technology: a Nordic cohort study from the Committee of Nordic ART and Safety (CoNARTaS). 2014 Ingår i: Human reproduction (Oxford, England). - : Oxford University Press (OUP). - 1460-2350 .- 0268-1161. ; 29:9, s. 2050-7 Tidskriftsartikel (refereegranskat)abstract Do children and young adults born after assisted reproductive technology (ART) have an increased risk of cancer?
2.	Adel Fahmideh, Maral, et al. (författare) A Weighted Genetic Risk Score of Adult Glioma Susceptibility Loci Associated with Pediatric Brain Tumor Risk. 2019 Ingår i: Scientific reports. - : Springer Science and Business Media LLC. - 2045-2322. ; 9:1 Tidskriftsartikel (refereegranskat)abstract Genetic risk score (GRS) is used to demonstrate the genetic variants contributing to the polygenic architecture of complex diseases. By using a GRS, we have investigated the additive impact of the known adult glioma susceptibility loci on the pediatric brain tumor (PBT) risk and assessed the proportion of PBT heritability attributable to these susceptibility loci. A GRS was generated for PBTs based on the alleles and associated effect sizes derived from a previously published genome-wide association study on adult glioma. The GRS was calculated in CEFALO, a population-based case-control study of brain tumors in children and adolescents including saliva DNA of 245 cases and 489 controls. The unconditional logistic regression model was used to investigate the association between standardized GRS and risk of PBTs. To measure the variance explained by the effect of GRS, Nagelkerke pseudo-R2 was calculated. The GRS for adult brain tumors was associated with an increased risk of PBTs (OR 1.25 [95% CI 1.06-1.49], p=0.009) and 0.3% of the variance in PBTs could be explained by the effect of GRS on the liability scale. This study provides evidence that heritable risks of PBTs are in-part attributable to some common genetic variants associated with adult glioma.
3.	Adel Fahmideh, Maral, et al. (författare) Parental age and risk of genetic syndromes predisposing to nervous system tumors: nested case-control study. 2018 Ingår i: Clinical epidemiology. - 1179-1349. ; 10, s. 729-738 Tidskriftsartikel (refereegranskat)abstract Phacomatoses are genetic syndromes that are associated with increased risk of developing nervous system tumors. Phacomatoses are usually inherited, but many develop de novo, with unknown etiology. In this population-based study, we investigated the effect of parental age on the risk of phacomatoses in offspring.The study was a population-based nested case-control study. All individuals born and residing in Sweden between January 1960 and December 2010 were eligible for inclusion. Using the Patient Register, 4625 phacomatosis cases were identified and further classified as familial or nonfamilial. Ten matched controls per case were randomly selected from the eligible population. Data were analyzed using conditional logistic regression. Analyses were conducted for neurofibromatosis alone (n=2089) and other phacomatoses combined (n=2536).Compared with offspring of fathers aged 25-29 years, increased risk estimates of nonfamilial neurofibromatosis were found for offspring of fathers aged 35-39 years (odds ratio [OR]=1.43 [95% CI 1.16-1.74]) and ≥40 years (OR =1.74 [95% CI 1.38-2.19]). For other nonfamilial phacomatoses, the risk estimate for offspring of fathers aged ≥40 years was OR =1.23 (95% CI 1.01-1.50). Paternal age was not associated with familial phacomatoses, and no consistent association was observed with maternal age.The findings show a consistent increase in risk of de novo occurrence of phacomatoses predisposing to nervous system tumors in offspring with increasing paternal age, most pronounced for neurofibromatosis, while maternal age did not seem to influence the risk. These findings suggest an increasing rate of new mutations in the NF1 and NF2 genes in spermatozoa of older fathers.
4.	Anclair, Malin, et al. (författare) Parental fears following their child's brain tumor diagnosis and treatment. 2009 Ingår i: Journal of pediatric oncology nursing : official journal of the Association of Pediatric Oncology Nurses. - : SAGE Publications. - 1043-4542 .- 1532-8457. ; 26:2, s. 68-74 Tidskriftsartikel (refereegranskat)abstract The objective of this study is to portray the illness-related threats experienced by parents of children after the diagnosis of central nervous system (CNS) tumor. Parents were asked to rate the extent to which they experienced a set of specific fears related to their child's brain tumor and its treatment. Outcomes for parents of CNS tumor patients (n = 82) were compared with those of reference parents of patients treated for acute lymphoblastic leukemia (n = 208). The fears about an illness recurrence and the late effects of treatment were most prominent among parents of CNS tumor patients. For 7 out of 11 kinds of fear, parents of CNS tumor patients expressed a stronger fear than the reference group. More than a quarter of the parents of children treated for CNS tumors feared a complete decline of the child. Parents of CNS tumor patients experience relatively heightened cancer related fears in several domains. The fear of devastating consequences felt by one fourth of parents signals the need of individualized psychological support and information at diagnosis and follow-up to facilitate parental coping with the posttreatment situation.
5.	Andersen, T V, et al. (författare) Patterns of exposure to infectious diseases and social contacts in early life and risk of brain tumours in children and adolescents: an International Case-Control Study (CEFALO). 2013 Ingår i: British journal of cancer. - : Springer Science and Business Media LLC. - 1532-1827 .- 0007-0920. ; 108, s. 2346-2353 Tidskriftsartikel (refereegranskat)abstract Background:Infectious diseases and social contacts in early life have been proposed to modulate brain tumour risk during late childhood and adolescence.Methods:CEFALO is an interview-based case-control study in Denmark, Norway, Sweden and Switzerland, including children and adolescents aged 7-19 years with primary intracranial brain tumours diagnosed between 2004 and 2008 and matched population controls.Results:The study included 352 cases (participation rate: 83%) and 646 controls (71%). There was no association with various measures of social contacts: daycare attendance, number of childhours at daycare, attending baby groups, birth order or living with other children.Cases of glioma and embryonal tumours had more frequent sick days with infections in the first 6 years of life compared with controls. In 7-19 year olds with 4+ monthly sick day, the respective odds ratios were 2.93 (95% confidence interval: 1.57-5.50) and 4.21 (95% confidence interval: 1.24-14.30).Interpretation:There was little support for the hypothesis that social contacts influence childhood and adolescent brain tumour risk. The association between reported sick days due to infections and risk of glioma and embryonal tumour may reflect involvement of immune functions, recall bias or inverse causality and deserve further attention.British Journal of Cancer advance online publication 7 May 2013; doi:10.1038/bjc.2013.201 www.bjcancer.com.
6.	Aydin, Denis, et al. (författare) Mobile phone use and brain tumors in children and adolescents: a multicenter case-control study. 2011 Ingår i: Journal of the National Cancer Institute. - : Oxford University Press (OUP). - 1460-2105 .- 0027-8874. ; 103:16, s. 1264-76 Tidskriftsartikel (refereegranskat)abstract It has been hypothesized that children and adolescents might be more vulnerable to possible health effects from mobile phone exposure than adults. We investigated whether mobile phone use is associated with brain tumor risk among children and adolescents.
7.	Blomstrand, Malin, et al. (författare) Estimated clinical benefit of protecting neurogenesis in the developing brain during radiation therapy for pediatric medulloblastoma. 2012 Ingår i: Neuro-Oncology. - : Oxford University Press (OUP). - 1523-5866 .- 1522-8517. ; 14:7, s. 882-889 Tidskriftsartikel (refereegranskat)abstract We sought to assess the feasibility and estimate the benefit of sparing the neurogenic niches when irradiating the brain of pediatric patients with medulloblastoma (MB) based on clinical outcome data. Pediatric MB survivors experience a high risk of neurocognitive adverse effects, often attributed to the whole-brain irradiation that is part of standard management. Neurogenesis is very sensitive to radiation, and limiting the radiation dose to the hippocampus and the subventricular zone (SVZ) may preserve neurocognitive function. Radiotherapy plans were created using 4 techniques: standard opposing fields, intensity-modulated radiotherapy (IMRT), intensity-modulated arc therapy (IMAT), and intensity-modulated proton therapy (IMPT). Mean dose to the hippocampus and SVZ (mean for both sites) could be limited to 88.3% (range, 83.6%-91.0%), 77.1% (range, 71.5%-81.3%), and 42.3% (range, 26.6%-51.2%) with IMAT, IMRT, and IMPT, respectively, while maintaining at least 95% of the prescribed dose in 95% of the whole-brain target volume. Estimated risks for developing memory impairment after a prescribed dose of 23.4 Gy were 47% (95% confidence interval [CI], 21%-69%), 44% (95% CI, 21%-65%), 41% (95% CI, 22%-60%), and 33% (95% CI, 23%-44%) with opposing fields, IMAT, IMRT, and IMPT, respectively. Neurogenic niche sparing during cranial irradiation of pediatric patients with MB is feasible and is estimated to lower the risks of long-term neurocognitive sequelae. Greatest sparing is achieved with intensity-modulated proton therapy, thus making this an attractive option to be tested in a prospective clinical trial.
8.	Boman, Krister K, et al. (författare) Disability, body image and sports/physical activity in adult survivors of childhood CNS tumors: population-based outcomes from a cohort study. 2013 Ingår i: Journal of neuro-oncology. - : Springer Science and Business Media LLC. - 1573-7373 .- 0167-594X. ; 112:1, s. 99-106 Tidskriftsartikel (refereegranskat)abstract Childhood CNS tumor survivors risk health and functional impairments that threaten normal psychological development and self-perception. This study investigated the extent to which health and functional ability predict adult survivors' body image (BI) and self-confidence regarding sports and physical activity. The study cohort covered 708 eligible ≥18year old CNS tumor survivors, and data from 528 (75%) were analyzed. Disability was estimated using the Health Utilities Index™ Mark2/3, a multidimensional self-report instrument. Physical self-confidence in terms of BI and sports/physical activity-related self-confidence (SPAS) were assessed using the BI and the Sports/Athletics modules of a standardized self-report assessment scale. In adjusted regression models, global health and functional status (GHFS) predicted BI (B=0.94, 95% CI 0.69-1.19) and SPAS (B=0.79, 95% CI 0.55-1.04). Emotion and pain, and to a lesser degree cognition, speech and vision disability, were associated with poorer BI and SPAS. Gender, sub-diagnosis, and time since diagnosis influenced the relationship between health status and physical self-confidence outcomes. Females had poorer GHFS, BI and SPAS than males. Decreased health and functional ability following childhood CNS cancer intrudes on physical self-confidence, with females being at heightened risk for both disability and negative self-confidence. Identified disability and gender-related risk calls for a follow-up plan that integrates treatment of psychological sequelae in lifetime monitoring of childhood CNS tumor survivors to restore and protect self-image and self-confidence, essential mental health correlates. An expanded plan should recognize the need for such services, optimizing life-long quality of survival for CNS tumor survivors.
9.	Boman, Krister K., et al. (författare) Generalizing approaches to surveillance for complex social outcomes in broad‐range patient populations— : The cost in terms of lost information and subgroup utility 2022 Ingår i: Cancer. - : John Wiley & Sons. - 0008-543X .- 1097-0142. ; 128:13, s. 2400-2404 Tidskriftsartikel (refereegranskat)abstract Complex social outcomes, including those related to education and employment, depend on compound combinations of background factors that are significantly different for childhood and adult cancers. Medical and psychosocial prerequisites related to the age at cancer diagnosis and treatment require surveillance for complex social outcomes that meets the particularized needs of corresponding patient subgroups.
10.	Boman, K K, et al. (författare) Health and persistent functional late effects in adult survivors of childhood CNS tumours: a population-based cohort study. 2009 Ingår i: European journal of cancer (Oxford, England : 1990). - : Elsevier BV. - 1879-0852 .- 0959-8049. ; 45:14, s. 2552-61 Tidskriftsartikel (refereegranskat)abstract Survivors of central nervous system (CNS) tumours are particularly vulnerable to tumour- and treatment-related disability. We present the incidence of specific and overall functional and health-related late effects in a national adult survivor cohort. Diagnostic subgroups at particular risk for persistent sequels are identified. Data collection targeted 708 eligible >18 years old survivors, 708 parent proxies and 1000 general population controls. Functional disability including sensory and cognitive impairment, emotional status and pain was assessed using the Health Utilities Index Mark 2/3 (HUI2/3). Survivors and controls, and diagnostic subgroups were contrasted to identify the general and relative risk for late effects by sub-diagnosis. Survivors had persistent late effects in sensation, mobility, self-care and cognition. Deficits in these domains indicated clinically important disability in overall health, although indices of emotion and pain were unaffected compared to controls. Late effects tended to aggravate with time, and female survivors had poorer health. Oligodendroglioma, mixed/unspecified glioma, intracranial germ cell tumour and medulloblastoma survivors had poorest overall health. Least late effects were found for other specified/unspecified CNS tumours (including meningeoma and nerve sheath tumours), and for astrocytoma. An impact on educational, vocational and family-related outcomes, and higher utilisation of social insurance or government subsidies validated health-related sequelae in adulthood. Comparisons with controls confirm persistent disability in multiple functional domains in adult CNS tumour survivors. The heightened proportion of survivors presenting severe disability is a factor that specifically differentiates survivors from controls, although diagnostic subgroups differ significantly regarding the amount and severity of late effects.
11.	Brodin, N Patrik, et al. (författare) Hippocampal sparing radiotherapy for pediatric medulloblastoma: impact of treatment margins and treatment technique. 2014 Ingår i: Neuro-oncology. - : Oxford University Press (OUP). - 1523-5866 .- 1522-8517. ; 16:4, s. 594-602 Tidskriftsartikel (refereegranskat)abstract BackgroundWe investigated how varying the treatment margin and applying hippocampal sparing and proton therapy impact the risk of neurocognitive impairment in pediatric medulloblastoma patients compared with current standard 3D conformal radiotherapy.MethodsWe included 17 pediatric medulloblastoma patients to represent the variability in tumor location relative to the hippocampal region. Treatment plans were generated using 3D conformal radiotherapy, hippocampal sparing intensity-modulated radiotherapy, and spot-scanned proton therapy, using 3 different treatment margins for the conformal tumor boost. Neurocognitive impairment risk was estimated based on dose-response models from pediatric CNS malignancy survivors and compared among different margins and treatment techniques.ResultsMean hippocampal dose and corresponding risk of cognitive impairment were decreased with decreasing treatment margins (P < .05). The largest risk reduction, however, was seen when applying hippocampal sparing proton therapy-the estimated risk of impaired task efficiency (95% confidence interval) was 92% (66%-98%), 81% (51%-95%), and 50% (30%-70%) for 3D conformal radiotherapy, intensity-modulated radiotherapy, and proton therapy, respectively, for the smallest boost margin and 98% (78%-100%), 90% (60%-98%), and 70% (39%-90%) if boosting the whole posterior fossa. Also, the distance between the closest point of the planning target volume and the center of the hippocampus can be used to predict mean hippocampal dose for a given treatment technique.ConclusionsWe estimate a considerable clinical benefit of hippocampal sparing radiotherapy. In choosing treatment margins, the tradeoff between margin size and risk of neurocognitive impairment quantified here should be considered.
12.	Brodin, N Patrik, et al. (författare) Radiobiological risk estimates of adverse events and secondary cancer for proton and photon radiation therapy of pediatric medulloblastoma. 2011 Ingår i: Acta oncologica (Stockholm, Sweden). - 1651-226X. ; 50:6, s. 806-16 Tidskriftsartikel (refereegranskat)abstract Abstract Introduction. The aim of this model study was to estimate and compare the risk of radiation-induced adverse late effects in pediatric patients with medulloblastoma (MB) treated with either three-dimensional conformal radiotherapy (3D CRT), inversely-optimized arc therapy (RapidArc(®) (RA)) or spot-scanned intensity-modulated proton therapy (IMPT). The aim was also to find dose-volume toxicity parameters relevant to children undergoing RT to be used in the inverse planning of RA and IMPT, and to use in the risk estimations. Material and methods. Treatment plans were created for all three techniques on 10 pediatric patients that have been treated with craniospinal irradiation (CSI) at our institution in 2007-2009. Plans were generated for two prescription CSI doses, 23.4 Gy and 36 Gy. Risk estimates were based on childhood cancer survivor data when available and secondary cancer (SC) risks were estimated as a function of age at exposure and attained age according to the organ-equivalent dose (OED) concept. Results. Estimates of SC risk was higher for the RA plans and differentiable from the estimates for 3D CRT at attained ages above 40 years. The risk of developing heart failure, hearing loss, hypothyroidism and xerostomia was highest for the 3D CRT plans. The risks of all adverse effects were estimated as lowest for the IMPT plans, even when including secondary neutron (SN) irradiation with high values of the neutron radiation weighting factors (WR(neutron)). Conclusions. When comparing RA and 3D CRT treatment for pediatric MB it is a matter of comparing higher SC risk against higher risks of non-cancer adverse events. Considering time until onset of the different complications is necessary to fully assess patient benefit in such a comparison. The IMPT plans, including SN dose contribution, compared favorably to the photon techniques in terms of all radiobiological risk estimates.
13.	Brolin, Inger, et al. (författare) [Comparison between hysterosalpingographic findings and lesions observed by laparoscopy and laparotomy (author's transl)]. : Vergleich zwischen Hysterosalpingographie, Laparoskopie und Laparotomie. 1980 Ingår i: RöFo : Fortschritte auf dem Gebiete der Röntgenstrahlen und der Nuklearmedizin. - : Georg Thieme Verlag KG. - 1438-9029 .- 1438-9010. ; 133:5, s. 510-3 Tidskriftsartikel (refereegranskat)abstract Laparoscopy and/or laparotomy had been performed in 91 patients also examined by hysterosalpingography (HSG) during two years. The comparison of the radiological and the operative findings shows, that total occlusion and hydrosalpinx are radiological signs of adhesions in the pelvis. A depot of contrast medium remaining around the fimbriated end of the oviduct is also a sign of adhesions. More than half of the patients with adhesions surrounding the oviducts and/or endometriosis had however normal hysterosalpingograms. Thus, the radiological examination, hysterosalpingography, has a low sensitivity rate concerning pelvic abnormalities.
14.	Câmara-Costa, Hugo, et al. (författare) Neuropsychological Outcome of Children Treated for Standard Risk Medulloblastoma in the PNET4 European Randomized Controlled Trial of Hyperfractionated Versus Standard Radiation Therapy and Maintenance Chemotherapy. 2015 Ingår i: International journal of radiation oncology, biology, physics. - : Elsevier BV. - 1879-355X .- 0360-3016. ; 92:5, s. 978-85 Tidskriftsartikel (refereegranskat)abstract In the European HIT-SIOP PNET4 randomized controlled trial, children with standard risk medulloblastoma were allocated to hyperfractionated radiation therapy (HFRT arm, including a partially focused boost) or standard radiation therapy (STRT arm), followed, in both arms, by maintenance chemotherapy. Event-free survival was similar in both arms. Previous work showed that the HFRT arm was associated with worse growth and better questionnaire-based executive function, especially in children <8years of age at diagnosis. Therefore, the aim of this study was to compare performance-based cognitive outcomes between treatment arms.
15.	Camara-Costa, H., et al. (författare) Quality of survival and cognitive performance in children treated for medulloblastoma in the PNET 4 randomized controlled trial 2017 Ingår i: Neuro-Oncology Practice. - : Oxford University Press (OUP). - 2054-2577 .- 2054-2585. ; 4:3, s. 161-170 Tidskriftsartikel (refereegranskat)abstract Background. The relationship between direct assessments of cognitive performance and questionnaires assessing quality of survival (QoS) is reported to be weak-to-nonexistent. Conversely, the associations between questionnaires evaluating distinct domains of QoS tend to be strong. This pattern remains understudied. Methods. In the HIT-SIOP PNET4 randomized controlled trial, cognitive assessments, including Full Scale, Verbal and Performance IQ, Working Memory, and Processing Speed, were undertaken in 137 survivors of standard-risk medulloblastoma from 4 European countries. QoS questionnaires, including self-reports and/or parent reports of the Behavior Rating Inventory of Executive Function (BRIEF), the Health Utilities Index, the Strengths and Difficulties Questionnaire, and the Pediatric Quality of Life Inventory, were completed for 151 survivors. Correlations among direct cognitive assessments, QoS questionnaires, and clinical data were examined in participants with both assessments available (n = 86). Results. Correlations between direct measures of cognitive performance and QoS questionnaires were weak, except for moderate correlations between the BRIEF Metacognition Index (parent report) and working memory (r = .32) and between health status (self-report) and cognitive outcomes (r = .35-.44). Correlations among QoS questionnaires were moderate to strong both for parent and self-report (r = .39-.76). Principal Component Analysis demonstrated that questionnaires and cognitive assessments loaded on 2 separate factors. Conclusions. We hypothesize that the strong correlations among QoS questionnaires is partially attributable to the positive/negative polarity of all questions on the questionnaires, coupled with the relative absence of diseasespecific questions. These factors may be influenced by respondents' personality and emotional characteristics, unlike direct assessments of cognitive functioning, and should be taken into account in clinical trials.
16.	Christensen, J. S., et al. (författare) Brain tumors in children and adolescents and exposure to animals and farm life: a multicenter case-control study (CEFALO) 2012 Ingår i: Cancer Causes & Control. - : Springer Science and Business Media LLC. - 0957-5243 .- 1573-7225. ; 23:9, s. 1463-1473 Tidskriftsartikel (refereegranskat)abstract The etiology of brain tumors in children and adolescents is largely unknown, and very few environmental risk factors have been identified. The aim of this study was to examine the relationship between pre- or postnatal animal contacts or farm exposures and the risk of childhood brain tumors (CBTs), since infectious agents may pose a risk factor and a proposed mechanism is transferral of infectious agents from animals to humans. The case-control study conducted in Denmark, Norway, Sweden, and Switzerland included brain tumor cases diagnosed from 2004 to 2008 aged 7-19 years at diagnosis. Three hundred and fifty-two cases (83 % participation rate) were matched to 646 population-based controls (71 % participation rate). Conditional logistic regression was used to estimate odds ratios. Maternal farm residence during pregnancy was inversely related to all CBTs combined (adjusted odds ratio (aOR) = 0.40, 95 % confidence interval (CI) = 0.19-0.88), as was the child's farm residence but not statistically significantly so (aOR = 0.57, 95 % CI = 0.28-1.17). Exposure to animals was in general not related to CBT risk except postnatal contact with birds showing reduced aORs of all CBTs (0.67, 95 % CI = 0.46-0.97) and primitive neuroectodermal tumor (0.28, 95 % CI = 0.10-0.83). Sensitivity analyses focusing on early exposure of the child did not change the associations observed for the entire exposure period with the exception of exposure to goats and sheep which was associated with reduced risks of both all CBTs (aOR = 0.48, 95 % CI = 0.24-0.97) and astrocytomas (aOR = 0.29, 95 % CI = 0.10-0.87). Altogether, our data indicate an inverse association between the mother during pregnancy or the child living on a farm and CBT risk, which contrasts with the existing literature and merits further attention. With respect to exposure to animals, we did not observe any systematic pattern. This suggests that a potential protective effect of farm residence is mediated by some other factor than animal contact.
17.	Clifford, Steven C, et al. (författare) Biomarker-driven stratification of disease-risk in non-metastatic medulloblastoma: Results from the multi-center HIT-SIOP-PNET4 clinical trial. 2015 Ingår i: Oncotarget. - : Impact Journals, LLC. - 1949-2553. ; 6:36, s. 38827-39 Tidskriftsartikel (refereegranskat)abstract To improve stratification of risk-adapted treatment for non-metastatic (M0), standard-risk medulloblastoma patients by prospective evaluation of biomarkers of reported biological or prognostic significance, alongside clinico-pathological variables, within the multi-center HIT-SIOP-PNET4 trial.
18.	Danielsson, Anna, 1973, et al. (författare) MethPed: a DNA methylation classifier tool for the identification of pediatric brain tumor subtypes 2015 Ingår i: Clinical Epigenetics. - : Springer Science and Business Media LLC. - 1868-7083 .- 1868-7075. ; 7 Tidskriftsartikel (refereegranskat)abstract Background: Classification of pediatric tumors into biologically defined subtypes is challenging, and multifaceted approaches are needed. For this aim, we developed a diagnostic classifier based on DNA methylation profiles. Results: Methylation data generated by the Illumina Infinium HumanMethylation 450 BeadChip arrays were downloaded from the Gene Expression Omnibus (n = 472). Using the data, we built MethPed, which is a multiclass random forest algorithm, based on DNA methylation profiles from nine subgroups of pediatric brain tumors. DNA from 18 regional samples was used to validate MethPed. MethPed was additionally applied to a set of 28 publically available tumors with the heterogeneous diagnosis PNET. MethPed could successfully separate individual histology tumor types at a very high accuracy (kappa = 0.98). Analysis of a regional cohort demonstrated the clinical benefit of MethPed, as confirmation of diagnosis of tumors with clear histology but also identified possible differential diagnoses in tumors with complicated and mixed type morphology. Conclusions: We demonstrate the utility of methylation profiling of pediatric brain tumors and offer MethPed as an easy-to-use toolbox that allows researchers and clinical diagnosticians to test single samples as well as large cohorts for subclass prediction of pediatric brain tumors. This will immediately aid clinical practice and importantly increase our molecular knowledge of these tumors for further therapeutic development.
19.	Dobsicek Trefna, Hana, 1979, et al. (författare) Antenna Applicator for Microwave Hyperthermia Treatment of Pediatric Brain Cancer 2014 Ingår i: 8th European Conference on Antennas and Propagation, EuCAP 2014, The Hague, The Netherlands 6-11 April 2014. - 9788890701849 Konferensbidrag (övrigt vetenskapligt/konstnärligt)abstract A novel antenna applicator for microwave hyperthermia allowing treatment of deep brain tumors is proposed. The applicator consists of up to 16 antennas placed around the head in a helmet-like set-up and operates at a frequency range of 430-1000~MHz. The self-grounded bow-tie antennas are housed in a molded plastic enclosure with the shape of a truncated cone. The inner space of the enclosure is filled with distilled water. The antennas are attached to a perimetric water bolus with a thickness of 2 cm and aligned with the head shape. The focusing ability of the applicator was investigated on a homogeneous SAM model and on a model of a 13-year old patient containing a spherical tumor of 2 cm radius. Two different tumor positions were investigated: the right frontal lobe and the central brain. The obtained SAR distributions are favorable, although a relatively high level of energy is also absorbed on the surface of the body. This heating is however not expected to cause problems as it can be cooled by blood perfusion and water bolus. Our results show that focused microwave heating in the brain is feasible and warrants further verification on phantoms.
20.	Dobsicek Trefna, Hana, 1979, et al. (författare) Development of focused microwave hyperthermia of pediatric brain cancer 2015 Ingår i: RADIOTHERAPY AND ONCOLOGY. - 0167-8140. ; 115, s. S685-S686, s. 708-709 Konferensbidrag (övrigt vetenskapligt/konstnärligt)
21.	Fahmideh, M. A., et al. (författare) CCDC26, CDKN2BAS, RTEL1 and TERT Polymorphisms in pediatric brain tumor susceptibility 2015 Ingår i: Carcinogenesis. - : Oxford University Press (OUP). - 0143-3334 .- 1460-2180. ; 51, s. S163-S163 Tidskriftsartikel (refereegranskat)abstract The role of genetic polymorphisms in pediatric brain tumor (PBT) etiology is poorly understood. We hypothesized that single nucleotide polymorphisms (SNPs) identified in genome-wide association studies (GWAS) on adult glioma would also be associated with PBT risk. The study is based on the Cefalo study, a population-based multicenter case-control study. Saliva DNA from 245 cases and 489 controls, aged 7-19 years at diagnosis/reference date, was extracted and genotyped for 29 SNPs reported by GWAS to be significantly associated with risk of adult glioma. Data were analyzed using unconditional logistic regression. Stratified analyses were performed for two histological subtypes: astrocytoma alone and the other tumor types combined. The results indicated that four SNPs, CDKN2BAS rs4977756 (p = 0.036), rs1412829 (p = 0.037), rs2157719 (p = 0.018) and rs1063192 (p = 0.021), were associated with an increased susceptibility to PBTs, whereas the TERT rs2736100 was associated with a decreased risk (p = 0.018). Moreover, the stratified analyses showed a decreased risk of astrocytoma associated with RTEL1 rs6089953, rs6010620 and rs2297440 (p(trend) = 0.022, p(trend) = 0.042, p(trend) = 0.029, respectively) as well as an increased risk of this subtype associated with RTEL1 rs4809324 (p(trend) = 0.033). In addition, SNPs rs10464870 and rs891835 in CCDC26 were associated with an increased risk of non-astrocytoma tumor subtypes (p(trend) = 0.009, p(trend) = 0.007, respectively). Our findings indicate that SNPs in CDKN2BAS, TERT, RTEL1 and CCDC26 may be associated with the risk of PBTs. Therefore, we suggest that pediatric and adult brain tumors might share common genetic risk factors and similar etiological pathways.
22.	Fukuda, A., et al. (författare) Age-dependent sensitivity of the developing brain to irradiation is correlated with the number and vulnerability of progenitor cells 2005 Ingår i: J Neurochem. ; 92:3, s. 569-84 Tidskriftsartikel (refereegranskat)abstract In a newly established model of unilateral, irradiation (IR)-induced injury we compared the outcome after IR to the immature and juvenile brain, using rats at postnatal days 9 or 23, respectively. We demonstrate that (i) the immature brains contained more progenitors in the subventricular zone (SVZ) and subgranular zone (SGZ) compared with the juvenile brains; (ii) cellular injury, as judged by activation of caspase 3 and p53, as well as nitrotyrosine formation, was more pronounced in the SVZ and SGZ in the immature brains 6 h after IR; (iii) the number of progenitor and immature cells in the SVZ and SGZ decreased 6 h and 7 days post-IR, corresponding to acute and subacute effects in humans, respectively, these effects were more pronounced in immature brains; (iv) myelination was impaired after IR at both ages, and much more pronounced after IR to immature brains; (v) the IR-induced changes remained significant for at least 10 weeks, corresponding to late effects in humans, and were most pronounced after IR to immature brains. It appears that IR induces both an acute loss of progenitors through apoptosis and a perturbed microenvironment incompatible with normal proliferation and differentiation, and that this is more pronounced in the immature brain.
23.	Fukuda, Aya, et al. (författare) Progenitor cell injury after irradiation to the developing brain can be modulated by mild hypothermia or hyperthermia 2005 Ingår i: J Neurochem. ; 94:6, s. 1604-19 Tidskriftsartikel (refereegranskat)abstract Ionizing radiation induced acute cell death in the dentate gyrus subgranular zone (SGZ) and the subventricular zone (SVZ). Hypomyelination was also observed. The effects of mild hypothermia and hyperthermia for 4 h after irradiation (IR) were studied in postnatal day 9 rats. One hemisphere was irradiated with a single dose of 8 Gy and animals were randomized to normothermia (rectal temperature 36 degrees C for 4 h), hypothermia (32 degrees C for 4 h) or hyperthermia (39 degrees C for 4 h). Cellular injury, e.g. chromatin condensation and nitrotyrosine formation, appeared to proceed faster when the body temperature was higher. Caspase-3 activation was more pronounced in the hyperthermia group and nuclear translocation of p53 was less pronounced in the hypothermia group 6 h after IR. In the SVZ the loss of nestin-positive progenitors was more pronounced (48%) and the size was smaller (45%) in the hyperthermia group 7 days post-IR. Myelination was not different after hypo- or hyperthermia. This is the first report to demonstrate that hypothermia may be beneficial and that hyperthermia may aggravate the adverse side-effects after radiation therapy to the developing brain.
24.	Fukuda, Hirotsugu, et al. (författare) Irradiation-induced progenitor cell death in the developing brain is resistant to erythropoietin treatment and caspase inhibition 2004 Ingår i: Cell Death Differ. - Univ Gothenburg, Dept Physiol, Perinatal Ctr, SE-40530 Gothenburg, Sweden. Osaka Univ, Sch Med, Dept Obstet & Gynecol, Suita, Osaka 5650871, Japan. Zhengzhou Univ, Affiliated Hosp 3, Dept Pediat, Zhengzhou 450052, Peoples R China. Uppsala Univ, Dept Neurosci, SE-75123 Uppsala, Sweden. Sahlgrens Univ Hosp, Dept Radiat Phys, SE-41345 Gothenburg, Sweden. H Lundbeck & Co AS, Mol Dis Biol, DK-2500 Copenhagen, Denmark. Sahlgrens Univ Hosp, Dept Oncol, SE-41345 Gothenburg, Sweden. Queen Silvia Childrens Hosp, Dept Pediat, SE-41685 Gothenburg, Sweden. : NATURE PUBLISHING GROUP. - 1350-9047 .- 1476-5403. ; 11:11, s. 1166-78 Tidskriftsartikel (refereegranskat)abstract One hemisphere of postnatal day 8 (P8) rats or P10 mice was irradiated with a single dose of 4-12 Gy, and animals were killed from 2 h to 8 weeks after irradiation (IR). In the subventricular zone (SVZ) and the granular cell layer (GCL) of the dentate gyrus, harboring neural and other progenitor cells, nitrosylation and p53 peaked 2-12 h after IR, followed by markers for active caspase-3, apoptosis-inducing factor and TUNEL (6-24 h). Ki67-positive (proliferating) cells had disappeared by 12 h and partly reappeared by 7 days post-IR. The SVZ and GCL areas decreased approximately 50% 7 days after IR. The development of white matter was hampered, resulting in 50-70% less myelin basic protein staining. Pretreatment with erythropoietin did not confer protection against IR. Caspase inhibition by overexpression of XIAP prevented caspase-9 and caspase-3 activation but not cell death, presumably because of increased caspase-independent cell death.
25.	Goschzik, T., et al. (författare) Prognostic effect of whole chromosomal aberration signatures in standard-risk, non-WNT/non-SHH medulloblastoma: a retrospective, molecular analysis of the HIT-SIOP PNET 4 trial 2018 Ingår i: Lancet Oncology. - : Elsevier BV. - 1470-2045. ; 19:12, s. 1602-1616 Tidskriftsartikel (refereegranskat)abstract Background Most children with medulloblastoma fall within the standard-risk clinical disease group defined by absence of high-risk features (metastatic disease, large-cell/anaplastic histology, and MYC amplification), which includes 50-60% of patients and has a 5-year event-free survival of 75-85%. Within standard-risk medulloblastoma, patients in the WNT subgroup are established as having a favourable prognosis; however, outcome prediction for the remaining majority of patients is imprecise. We sought to identify novel prognostic biomarkers to enable improved risk-adapted therapies. Methods The HIT-SIOP PNET 4 trial recruited 338 patients aged 4-21 years with medulloblastoma between Jan 1, 2001, and Dec 31, 2006, in 120 treatment institutions in seven European countries to investigate hyperfractionated radiotherapy versus standard radiotherapy. In this retrospective analysis, we assessed the remaining tumour samples from patients in the HIT-SIOP PNET 4 trial (n=136). We assessed the clinical behaviour of the molecularly defined WNT and SHH subgroups, and identified novel independent prognostic markers and models for standard-risk patients with non-WNT/non-SHH disease. Because of the scarcity and low quality of available genomic material, we used a mass spectrometry-minimal methylation classifier assay (MS-MIMIC) to assess methylation subgroup and a molecular inversion probe array to detect genome-wide copy number aberrations. Prognostic biomarkers and models identified were validated in an independent, demographically matched cohort (n=70) of medulloblastoma patients with non-WNT/non-SHH standard-risk disease treated with conventional therapies (maximal surgical resection followed by adjuvant craniospinal irradiation [all patients] and chemotherapy [65 of 70 patients], at UK Children's Cancer and Leukaemia Group and European Society for Paediatric Oncology (SIOPE) associated treatment centres between 1990 and 2014. These samples were analysed by Illumina 450k DNA methylation microarray. HIT-SIOP PNET 4 is registered with ClinicalTrials. gov, number NCT01351870. Findings We analysed methylation subgroup, genome-wide copy number aberrations, and mutational features in 136 assessable tumour samples from the HIT-SIOP PNET 4 cohort, representing 40% of the 338 patients in the trial cohort. This cohort of 136 samples consisted of 28 (21%) classified as WNT, 17 (13%) as SHH, and 91 (67%) as non-WNT/non-SHH (we considered Group3 and Group4 medulloblastoma together in our analysis because of their similar molecular and clinical features). Favourable outcomes for WNT tumours were confirmed in patients younger than 16 years, and all relapse events in SHH (four [24%] of 17) occurred in patients with TP53 mutation (TP53(mut)) or chromosome 17p loss. A novel whole chromosomal aberration signature associated with increased ploidy and multiple non-random whole chromosomal aberrations was identified in 38 (42%) of the 91 samples from patients with non-WNT/non-SHH medulloblastoma in the HIT-SIOP PNET 4 cohort. Biomarkers associated with this whole chromosomal aberration signature (at least two of chromosome 7 gain, chromosome 8 loss, and chromosome 11 loss) predicted favourable prognosis. Patients with non-WNT/non-SHH medulloblastoma could be reclassified by these markers as having favourable-risk or high-risk disease. In patients in the HIT-SIOP PNET4 cohort with non-WNT/non-SHH medulloblastoma, with a median follow-up of 6.7 years (IQR 5.8-8.2), 5-year event-free survival was 100% in the favourable-risk group and 68% (95% CI 57.5-82.7; p=0.00014) in the high-risk group. In the validation cohort, with a median follow-up of 5.6 years (IQR 3.1-8.1), 5-year event-free survival was 94.7% (95% CI 85.2-100) in the favourable-risk group and 58.6% (95% CI 45.1-76.1) in the high-risk group (hazard ratio 9.41, 95% CI 1.25-70.57; p=0.029). Our comprehensive molecular investigation identified subgroup-specific risk models which allowed 69 (51%) of 134 accessible patients from the standard-risk medulloblastoma HIT-SIOP PNET 4 cohort to be assigned to a favourable-risk group. Interpretation We define a whole chromosomal signature that allows the assignment of non-WNT/non-SHH medulloblastoma patients normally classified as standard-risk into favourable-risk and high-risk categories. In addition to patients younger than 16 years with WNT tumours, patients with non-WNT/non-SHH tumours with our defined whole chromosomal aberration signature and patients with SHH-TP53wild-type tumours should be considered for therapy de-escalation in future biomarker-driven, risk-adapted clinical trials. The remaining subgroups of patients with high-risk medulloblastoma might benefit from more intensive therapies. Copyright (c) 2018 The Author(s). Published by Elsevier Ltd.
26.	Gudrunardottir, Thora, et al. (författare) Treatment developments and the unfolding of the quality of life discussion in childhood medulloblastoma: a review. 2014 Ingår i: Child's nervous system : ChNS : official journal of the International Society for Pediatric Neurosurgery. - : Springer Science and Business Media LLC. - 1433-0350. ; 30:6, s. 979-990 Forskningsöversikt (refereegranskat)abstract To describe how the quality of life (QOL) discussion in childhood medulloblastoma (MB) relates to treatment developments, survival and sequelae from 1920 to 2014. Articles containing "childhood medulloblastoma" and "quality of life" were identified in PubMed. Those containing phrases pertaining to psychological, emotional, behavioral or social adjustment in the title, abstract or keywords were selected. Inclusion of relevant older publications was assured by cross-checking references. 1920-1930s: suction, electro-surgery, kilovolt (KV) irradiation. Survival = months. Focus on operative mortality, symptoms and survival. 1940s: radiotherapy improved. 1950s: chemotherapy and intubation. Survival = years. Opinions oscillated between optimism/awareness of physical sequelae of radiotherapy. 1960s: magnified vision, ventriculo-peritoneal (VP) shunts, megavolt (MV) irradiation. Long-term survival shifted the attention towards neurological problems, disability and carcinogenesis of radiotherapy. 1970s: CT, microscope, bipolar coagulation, shunt filters, neuroanesthesia, chemotherapy trials and staging studies. Operative mortality decreased and many patients (re)entered school; emphasis on neuropsychological sequelae, IQ and academic performance. 1980s: magnetic resonance imaging (MRI), Cavitron ultrasonic aspiration (CUSA), laser surgery, hyper-fractionated radiotherapy (HFRT). Cerebellar mutism, psychological and social issues. 1990s: pediatric neurosurgery, proton beams, stem cell rescue. Reflections on QOL as such. 21st century: molecular genetics. Premature aging, patterns of decline, risk- and resilience factors. QOL is a critical outcome measure. Focus depends on survival and sequelae, determined after years of follow-up. Detailed measurements are limited by time, money and human resources, and self-reporting questionnaires represent a crude measure limited by subjectivity. Therapeutic improvements raise the question of QOL versus cure. QOL is a potential primary research endpoint; multicenter international studies are needed, as are web-based tools that work across cultures.
27.	Hoven, E., et al. (författare) Information needs of survivors and families after childhood CNS tumor treatment: a population-based study 2018 Ingår i: Acta Oncologica. - : Informa UK Limited. - 0284-186X .- 1651-226X. ; 57:5, s. 649-657 Tidskriftsartikel (refereegranskat)abstract Background: This study examines information needs and satisfaction with provided information among childhood central nervous system (CNS) tumor survivors and their parents.Material and methods: In a population-based sample of 697 adult survivors in Sweden, 518 survivors and 551 parents provided data. Information needs and satisfaction with information were studied using a multi-dimensional standardized questionnaire addressing information-related issues.Results: Overall, 52% of the survivors and 48% of the parents reported no, or only minor, satisfaction with the extent of provided information, and 51% of the survivors expressed a need for more information than provided. The information received was found useful (to some extent/very much) by 53%, while 47% did not find it useful, or to a minor degree only. Obtaining written material was associated with greater satisfaction and usefulness of information. Dissatisfaction with information was associated with longer time since diagnosis, poorer current health status and female sex. The survivors experienced unmet information needs vis-a-vis late effects, illness education, rehabilitation and psychological services. Overall, parents were more dissatisfied than the survivors.Conclusion: These findings have implications for improvements in information delivery. Information in childhood CNS tumor care and follow-up should specifically address issues where insufficiency was identified, and recognize persistent and with time changing needs at the successive stages of long-term survivorship.
28.	Hovén, Emma, 1983-, et al. (författare) Persistent impact of illness on families of adult survivors of childhood central nervous system tumors: a population-based cohort study. 2013 Ingår i: Psycho-oncology. - : Wiley. - 1099-1611 .- 1057-9249. ; 22:1, s. 160-167 Tidskriftsartikel (refereegranskat)abstract OBJECTIVE: This study aims to determine the long-term impact on families of adult survivors of childhood central nervous system tumors. Illness-related family consequences were studied in relation to modifying determinants. METHODS: In a population-based cohort of parents of 697 survivors 18years and older, 551 parents provided data. The impact of cancer on the families was evaluated in four domains using the Impact on Family Scale (economic situation, personal burden, social life, sibling impact). The results were analyzed in relation to survivors' health assessed using the Health Utilities Index™, parent satisfaction with information about illness and treatment, and perceived health-care needs of their child. RESULTS: Despite an established mild-to-moderate impact on the group level, outcomes provided evidence of substantial cancer-related family consequences even once the child had reached adulthood. About one fifth of parents reported psychological and financial difficulties exceeding the cutoff limit for a significant impact still ≥5years after diagnosis. A stronger total family impact was associated with poorer health of survivors (F[3,302]=56.65, p < 0.001), and unmet informational - (F[3,231]=14.06, p < 0.001) and health-care needs (t(218) =5.31, p < 0.001). The impact was unrelated to survivors' age at follow-up and time since diagnosis. CONCLUSIONS: Adverse cancer-related consequences affect a considerable portion of families of childhood survivors of central nervous system tumor, even after reaching adulthood. The impact is aggravated by lasting sequelae and perceived shortcomings of long-term follow-up, factors that partly are avoidable. Improved clinical follow-up should particularly address illness information and long-term health-care needs to reduce the impact on families of survivors suffering from chronic health conditions. Copyright © 2011 John Wiley & Sons, Ltd.
29.	Hovén, Emma, 1983-, et al. (författare) The met and unmet health care needs of adult survivors of childhood central nervous system tumors : A double-informant, population-based study 2011 Ingår i: Cancer. - : Wiley. - 0008-543X .- 1097-0142. ; 117:18, s. 4294-4303 Tidskriftsartikel (refereegranskat)abstract BACKGROUND:The purpose of the current study was to examine the persistent health care needs (HCNs) of adult survivors of childhood central nervous system tumors.METHODS:In this population-based study, 526 of 679 eligible survivors and 550 parents provided data. Survivors' HCNs were assessed using a questionnaire covering 4 domains: Medical Care, care coordination and communication (Care Coordination), Illness Education, and Psychosocial Services. Needs were categorized as no need, met need, and unmet need. Outcomes were analyzed specifically in relation to survivors' functional late effects as assessed using the Health Utilities Index Mark 2/3.RESULTS:Approximately 40% of survivors experienced their HCNs as exceeding the supposed general population average, and 41% had a current HCN that was unmet. The most common unmet need concerned the Psychosocial Services domain (reported by 40%), followed by a lack of Illness Education (35%), Care Coordination (22%), and Medical Care (15%). Survivors experiencing functional late effects had greater HCNs, and a greater percentage of unmet needs. Agreement between survivor-reported and parent proxy-reported HCNs was satisfactory, whereas agreement for survivors' unmet HCNs ranged from poor to satisfactory.CONCLUSIONS:Findings based on reliable double-informant data demonstrated that a considerable percentage of adult survivors report unmet HCNs, with female sex, younger age at diagnosis, and indications of disability and poor health status comprising significant risk factors. Issues critical for improved, comprehensive, long-term follow-up care were identified. Addressing these issues adequately in clinical follow-up extending into adulthood would likely improve the quality of comprehensive care for this patient group.
30.	Hörnquist, Lina, et al. (författare) Altered self-perception in adult survivors treated for a CNS tumor in childhood or adolescence : population-based outcomes compared with the general population 2015 Ingår i: Neuro-Oncology. - : Oxford University Press (OUP). - 1522-8517 .- 1523-5866. ; 17:5, s. 733-40 Tidskriftsartikel (refereegranskat)abstract BACKGROUND: Survivors of pediatric CNS tumors are at risk for persistent tumor/treatment-related morbidity, physical disability and social consequences that may alter self-perception, vital for self-identity, mental health and quality of survival. We studied the long-term impact of childhood CNS tumors and their treatment on the self-perception of adult survivors and compared outcomes with those of the general population.METHODS: The cohort included 697 Swedish survivors diagnosed with a primary CNS tumor during 1982-2001. Comparison data were randomly collected from a stratified general population sample. Survivors and general population individuals were compared as regards self-perception in 5 domains: body image, sports/physical activities, peers, work, and family, and with a global self-esteem index. Within the survivor group, determinants of impact on self-perception were identified.RESULTS: The final analyzed sample included 528 survivors, 75.8% of the entire national cohort. The control sample consisted of 995, 41% of 2500 addressed. Survivors had significantly poorer self-perception outcomes in domains of peers, work, body image, and sports/physical activities, and in the global self-perception measure, compared with those of the general population (all P < .001). Within the survivor group, female gender and persistent visible physical sequelae predicted poorer outcomes in several of the studied domains. Tumor type and a history of cranial radiation therapy were associated with outcomes.CONCLUSION: An altered self-perception is a potential late effect in adult survivors of pediatric CNS tumors. Self-perception and self-esteem are significant elements of identity, mental health and quality of survival. Therefore, care and psychosocial follow-up of survivors should include measures for identifying disturbances and for assessing the need for psychosocial intervention.
31.	Jarfelt, Marianne, 1962, et al. (författare) Body composition in young adult survivors of childhood acute lymphoblastic leukaemia. 2005 Ingår i: European journal of endocrinology / European Federation of Endocrine Societies. - : Oxford University Press (OUP). - 0804-4643. ; 153:1, s. 81-9 Tidskriftsartikel (refereegranskat)abstract OBJECTIVE: Obesity is frequently reported in patients treated for childhood leukaemia. Obesity, particularly abdominal obesity, is one of the main characteristics of the metabolic syndrome and a risk factor for cardiovascular disease and non-insulin-dependent diabetes mellitus (NIDDM). DESIGN: All patients treated for acute lymphoblastic leukaemia (ALL) before the onset of puberty in the region of western Sweden, between 1973 and 1985, and in first remission, were included. 35 out of 47 patients aged 20-32 years participated. 19 patients had received cranial radiotherapy, and the median follow-up time was 20 years. The focus of this report was to study body composition and signs of the metabolic syndrome and correlate the findings to spontaneous growth hormone (GH) secretion. METHODS: Body composition was assessed using dual-energy X-ray absorbtiometry (DEXA). We analyzed serum concentrations of insulin, glucose, leptin and lipids. RESULTS: No patient was obese according to World Health Organization criteria (body mass index, BMI > or = 30 kg/m2) but one-third were overweight (BMI 25-29.9 kg/m2). The maximal GH peak during 24 h (GHmax) was correlated to percentage of total body fat (r = -0.42; P = 0.017), trunk fat (r = -0.5; P = 0.005) and fat-free mass (r = 0.42; P = 0.017). GHmax was also correlated to s-triglycerides (r = -0.54; P = 0.001), low-density lipoprotein-cholesterol (r = -0.382; P = 0.024) and high-density lipoprotein-cholesterol (r = 0.45; P = 0.007). CONCLUSIONS: We found little effect on BMI but an increased percentage of total body fat, especially trunk fat, and a tendency for an unfavourable lipid profile in adult survivors of childhood leukaemia. These findings were related to low endogenous GH secretion due to cranial irradiation.
32.	Jarfelt, Marianne, 1962, et al. (författare) Bone mineral density and bone turnover in young adult survivors of childhood acute lymphoblastic leukaemia 2006 Ingår i: Eur J Endocrinol. ; 154:2, s. 303-9 Tidskriftsartikel (refereegranskat)abstract OBJECTIVE: Treatment for childhood leukaemia induces many risk factors for development of decreased bone mineral density (BMD). Physical activity is also known to affect BMD. The aim was to study BMD and markers of bone turnover in a well-defined group of survivors of acute lymphoblastic leukaemia (ALL) who had all reached final height as well as peak bone mass, taking both previous treatment and physical activity into consideration. DESIGN: All patients treated for ALL before the onset of puberty in the region of western Sweden, between 1973 and 1985, in first remission were included. Thirty-five out of forty-seven patients aged 20-32 years participated. Nineteen patients had received cranial radiotherapy, and the median follow-up time was 20 years. METHODS: BMD was assessed using dual-energy X-ray absorbtiometry (DEXA). Serum concentrations of markers of bone turnover were analysed. Physical performance was measured using a performance exercise capacity stress test. RESULTS: BMD was slightly reduced in lumbar spine (-0.4 SD), but not in femoral neck or total body. BMD in femoral neck was correlated to physical performance and dose of corticosteroid, but no correlation was found with spontaneous growth hormone (GH) secretion. Markers of bone turnover were also correlated to physical performance, but not to GH secretion. CONCLUSIONS: Physical fitness seems to be the most important factor in developing and preserving normal bone mineral density in ALL patients. We propose that lifestyle education promoting physical activity is encouraged from an early point in time for these patients.
33.	Jarfelt, Marianne, 1962, et al. (författare) Exercise echocardiography reveals subclinical cardiac dysfunction in young adult survivors of childhood acute lymphoblastic leukemia 2007 Ingår i: Pediatr Blood Cancer. - : Wiley. - 1545-5009 .- 1545-5017. ; 49:6, s. 835-40 Tidskriftsartikel (refereegranskat)abstract OBJECTIVE: Anthracyclines (AC) have contributed significantly to increased survival rate in acute lymphoblastic leukemia (ALL), although the use of these drugs is limited due to cardiotoxicity. The aim was to evaluate heart muscle function in asymptomatic adult survivors of ALL treated in early childhood in relation to the combined effects of AC and other potential cardiotoxic factors. PROCEDURE: Twenty-three young adult ALL survivors who had all received treatment with median 120 (120-400) mg AC/m(2) before the onset of puberty were examined median 21 years after remission and compared with 12 healthy controls. Basal echocardiography including two-dimensional (2D) M-mode and Doppler examination was performed, followed by a maximal exercise stress test and stress echocardiography immediately after stress test and after 5 min recovery. RESULTS: We found significant differences in systolic function between patients and controls at maximal exercise despite absence of reported symptoms from the patients. The most marked difference was in ejection fraction at stress 59.5% (32.6-81.1) and 77.3% (66.2-85.3), respectively (P < 0.00006). Ten out of 23 patients reduced their ejection fraction at stress compared with at rest; this was not found in any of the controls. Cardiovascular risk factors such as GH deficiency and a high proportion of trunk fat did not have an impact on cardiac function. CONCLUSIONS: With very long follow up in a homogenous cohort of ALL survivors, we found subclinical cardiac dysfunction with exercise stress echocardiography even after low doses of AC. Pediatr Blood Cancer 2007;49:835-840. (c) 2007 Wiley-Liss, Inc.
34.	Jarfelt, Marianne, 1962, et al. (författare) Young adult survivors of childhood acute lymphoblastic leukemia: spontaneous GH secretion in relation to CNS radiation 2004 Ingår i: Pediatr Blood Cancer. - : Wiley. - 1545-5009 .- 1545-5017. ; 42:7, s. 582-8 Tidskriftsartikel (refereegranskat)abstract BACKGROUND: Young adults who are long-term survivors of acute lymphoblastic leukaemia (ALL) in early childhood usually do well and do not have to go to regular medical checkups. Many of these survivors did receive prophylactic cranial radiotherapy during their oncological treatment. The effect of cranial irradiation on the hypothalamus is considered to be progressive. Therefore, late effects, such as reduced growth hormone (GH) secretion, may remain undetected until adulthood. PROCEDURE: Records from all patients treated for ALL before the onset of puberty in the region of West Sweden, between 1 January 1973 and 31 December 1985 were included, provided they were in first remission with a minimum follow-up time of 15 years, and a minimum age of 20. These criteria were met by 47 young adults aged 20-32 years, of whom 35 agreed to participate. We studied spontaneous GH secretion over 24 hr, IGF-I and IGFBP-3, final height and BMI. The patients had been treated according to three consecutive Swedish childhood leukaemia group protocols. The median follow-up time was 20 years, and 19 of the patients had been treated with cranial irradiation (CRT+), 16 had not (CRT-). RESULTS: CRT+ patients had significantly lower maximal peaks of GH than CRT- patients. Fifty percent of the CRT+ patients had a GH(max) below the cut-off level (3.3 microg/l), for GH treatment. CRT- patients all had GH(max) levels considered within the normal range. Final height of all the patients, except one CRT+ women, was in the range of expected midparental height, the median loss in final height in the CRT+ patients was 0.8 standard deviation (SD). No patient in this study was obese by definition (BMI <30 kg/m(2)). IGF-I and IGFBP-3 concentrations did not correlate to variations in spontaneous GH secretion in these patients. CONCLUSIONS: In spite of the little effect on final height, we found impaired spontaneous GH secretion in 79% of young adults 20-32 years of age, and GH deficiency (GHD) in 47% after low-dose cranial irradiation in early childhood. The consequences of this low-GH secretion need to be investigated.
35.	Kalm, Marie, 1981, et al. (författare) Irradiation-induced loss of microglia in the young brain. 2009 Ingår i: Journal of neuroimmunology. - : Elsevier BV. - 0165-5728. ; 206:1-2, s. 70-5 Tidskriftsartikel (refereegranskat)abstract Irradiation-induced loss of neural stem and progenitor cells may contribute to cognitive deficits. Furthermore, subsequent inflammation inhibits neural progenitor cell differentiation. Here we have characterized the microglia response after a single dose of 8 Gy to the brains of postnatal day 9 or 21 rats. The number of Iba-1-positive microglia increased 6 h after IR but had decreased 7 days later, below control levels, and this decrease was more pronounced in P9 rats. Active caspase-3 and TUNEL staining revealed irradiation-induced microglia death. This age-dependent IR-induced loss of microglia likely affects both the response to IR and further brain development.
36.	Kalm, Marie, 1981, et al. (författare) Transient inflammation in neurogenic regions after irradiation of the developing brain. 2009 Ingår i: Radiation research. - 0033-7587. ; 171:1, s. 66-76 Tidskriftsartikel (refereegranskat)abstract Kalm, M., Fukuda, A., Fukuda, H., Ohrfelt, A., Lannering, B., Björk-Eriksson, T., Blennow, K., Márky, I. and Blomgren, K. Transient Inflammation in Neurogenic Regions after Irradiation of the Developing Brain. Radiat. Res. 171, 66-76 (2009).We characterized the inflammatory response after a single dose of 8 Gy to the brains of postnatal day 9 rats. Affymetrix gene chips revealed activation of multiple inflammatory mechanisms in the acute phase, 6 h after irradiation. In the subacute phase, 7 days after irradiation, genes related to neurogenesis and cell cycle were down-regulated, but glial fibrillary acidic protein (GFAP) was up-regulated. The concentrations of 14 different cytokines and chemokines were measured using a microsphere-based xMAPtrade mark technology. CCL2, Gro/KC and IL-1alpha were the most strongly up-regulated 6 h after irradiation. CCL2 was expressed in astrocytes and microglia in the dentate gyrus and the subventricular zone (SVZ). Hypertrophy, but not hyperplasia, of astrocytes was demonstrated 7 days after irradiation. In summary, we found transient activation of multiple inflammatory mechanisms in the acute phase (6 h) after irradiation and activation of astrocytes in the subacute phase (7 days) after irradiation. It remains to be elucidated whether these transient changes are involved in the persistent effects of radiation observed on neurogenesis and cognition in rodents.
37.	Kennedy, Colin, et al. (författare) Quality of Survival and Growth in Children and Young Adults in the PNET4 European Controlled Trial of Hyperfractionated Versus Conventional Radiation Therapy for Standard-Risk Medulloblastoma. 2014 Ingår i: International journal of radiation oncology, biology, physics. - : Elsevier BV. - 1879-355X .- 0360-3016. ; 88:2, s. 292-300 Tidskriftsartikel (refereegranskat)abstract To compare quality of survival in "standard-risk" medulloblastoma after hyperfractionated radiation therapy of the central nervous system with that after standard radiation therapy, combined with a chemotherapy regimen common to both treatment arms, in the PNET4 randomised controlled trial.
38.	Lannering, Birgitta, 1948, et al. (författare) Classification, incidence and survival analyses of children with CNS tumours diagnosed in Sweden 1984-2005. 2009 Ingår i: Acta paediatrica (Oslo, Norway : 1992). - : Wiley. - 1651-2227 .- 0803-5253. ; 98:10, s. 1620-7 Tidskriftsartikel (refereegranskat)abstract AIM: Primary tumours in the central nervous system (CNS) are the second most common malignancy in childhood after leukaemia. Sweden has a high incidence and a high-survival rate in international comparative studies. This has raised the question about the type of tumours included in the Swedish Cancer registry. We therefore compared international data to the Swedish Childhood Cancer registry. METHODS: Central nervous system tumours registered in the Swedish Childhood Cancer Registry were reclassified according to ICCC-3. Incidence and survival analyses were performed in the study population. RESULTS: There were 1479 children (<15 years) in Sweden diagnosed with CNS tumours 1984-2005. The distribution of diagnoses was similar to that reported in other studies. The annual incidence was 4.2/100,000 children. The survival rates have not improved significantly between the two time periods before/after 1995 (70% vs. 74%; p = 0.10). CONCLUSIONS: The mean annual incidence of children with CNS tumours was 4.2/100,000 and has not increased during the study period. Survival rate for brain tumours at 10 years follow-up was 72%.
39.	Lannering, Birgitta, 1948, et al. (författare) Early onset group B streptococcal disease. Seven year experience and clinical scoring system. 1983 Ingår i: Acta paediatrica Scandinavica. - 0001-656X. ; 72:4, s. 597-602 Tidskriftsartikel (refereegranskat)abstract Early onset group B streptococcal disease was reviewed for the seven year period between 1975 to 1981 at Vanderbilt University Medical Center. One hundred and twenty cases were identified. The disease varied from asymptomatic bacteremia to fatal cardiopulmonary collapse. Factors associated with a poor outcome were prematurity, low Apgar score at 5 min, the presence of shock, leukopenia, rupture of membranes for more than 12 hours, and a delay in treatment after the onset of symptoms. A scoring system for probability of death based on these 6 factors was then developed. Over the seven year period mortality decreased from 50% to 10%. The only factor identified with the decrease in mortality was a significant decrease in the number of hours between the onset of symptoms and the beginning of treatment. Early recognition and prompt treatment seem to be the major causes of the decreasing mortality over the seven years of this report.
40.	Lannering, Birgitta, 1948, et al. (författare) Growth hormone release in children after cranial irradiation. 1987 Ingår i: Hormone research. - 0301-0163. ; 27:1, s. 13-22 Tidskriftsartikel (refereegranskat)abstract Growth retardation due to growth hormone (GH) deficiency is common in children after radiotherapy to the brain. Different methods for assessment of GH secretion were compared in 19 children who had received radiotherapy to the brain as part of treatment for a tumor of the brain, eye or epipharynx. GH was measured over a 24-hour period (72 sampling periods of 20 min each), as well as after administration of growth hormone-releasing hormone (GHRH) and arginine-insulin (AITT) tests. We found the 24-hour GH profile to be disturbed in all children; there was a low overall secretion with few peaks of low amplitude but a diurnal rhythm still discernable. In 16 children a prompt rise in GHRH after GHRH1-40 was seen indicating hypothalamic damage. The GH response after GHRH was not found to be correlated to the spontaneous secretion over 24 h. The results of the AITT showed discrepancies to the 24-hour GH profile in individual cases making this test unreliable in spite of a good overall correlation between the tests. Therefore, we suggest measurement of spontaneous secretion when GH-secretory capability is to be evaluated after cranial irradiation for a brain tumor.
41.	Lannering, Birgitta, 1948, et al. (författare) Hyperfractionated Versus Conventional Radiotherapy Followed by Chemotherapy in Standard-Risk Medulloblastoma: Results From the Randomized Multicenter HIT-SIOP PNET 4 Trial. 2012 Ingår i: Journal of clinical oncology : official journal of the American Society of Clinical Oncology. - 1527-7755. ; 30:26, s. 3187-93 Tidskriftsartikel (refereegranskat)abstract PURPOSE To compare event-free survival (EFS), overall survival (OS), pattern of relapse, and hearing loss in children with standard-risk medulloblastoma treated by postoperative hyperfractionated or conventionally fractionated radiotherapy followed by maintenance chemotherapy. PATIENTS AND METHODS In all, 340 children age 4 to 21 years from 122 European centers were postoperatively staged and randomly assigned to treatment with hyperfractionated radiotherapy (HFRT) or standard (conventional) fractionated radiotherapy (STRT) followed by a common chemotherapy regimen consisting of eight cycles of cisplatin, lomustine, and vincristine. Results After a median follow-up of 4.8 years (range, 0.1 to 8.3 years), survival rates were not significantly different between the two treatment arms: 5-year EFS was 77% ± 4% in the STRT group and 78% ± 4% in the HFRT group; corresponding 5-year OS was 87% ± 3% and 85% ± 3%, respectively. A postoperative residual tumor of more than 1.5 cm(2) was the strongest negative prognostic factor. EFS of children with all reference assessments and no large residual tumor was 82% ± 2% at 5 years. Patients with a delay of more than 7 weeks to the start of RT had a worse prognosis. Severe hearing loss was not significantly different for the two treatment arms at follow-up. CONCLUSION In this large randomized European study, which enrolled patients with standard-risk medulloblastoma from more than 100 centers, excellent survival rates were achieved in patients without a large postoperative residual tumor and without RT treatment delays. EFS and OS for HFRT was not superior to STRT, which therefore remains standard of care in this disease.
42.	Larsson, Susanna, et al. (författare) Stability of patient-derived in vitro cultures of stem-like cells from high-grade paediatric brain tumours. 2016 Ingår i: Neuro-Oncology. - : Osford Academic Press. - 1522-8517 .- 1523-5866. Konferensbidrag (refereegranskat)
43.	Porkholm, Mikaela, et al. (författare) Radiation therapy and concurrent topotecan followed by maintenance triple anti-angiogenic therapy with thalidomide, etoposide, and celecoxib for pediatric diffuse intrinsic pontine glioma. 2014 Ingår i: Pediatric blood & cancer. - : Wiley. - 1545-5017 .- 1545-5009. ; 61:9, s. 1603-1609 Tidskriftsartikel (refereegranskat)abstract Despite major treatment attempts, the prognosis for pediatric diffuse intrinsic pontine gliomas (DIPGs) remains dismal. Gliomas are highly vascularized tumors, suggesting that the prevention of vessel formation by anti-angiogenic treatment might be effective.
44.	Rydén, Isabelle, et al. (författare) Neuropsychological functioning in childhood cancer survivors following cranial radiotherapy - results from a long-term follow-up clinic 2022 Ingår i: Neurocase. - : Informa UK Limited. - 1355-4794 .- 1465-3656. ; 28:2, s. 163-172 Tidskriftsartikel (refereegranskat)abstract Treatment of malignant childhood posterior fossa tumors (CPFT) often includes surgical resection and craniospinal radiotherapy (CSI). Nasopharyngeal tumors in childhood (CNPHT) are often treated with surgery and radiotherapy (RT), leading to incidental brain irradiation. RT to the developing brain is associated with risks for cognitive impairments. We studied cognitive functioning, health-related quality of life (HRQOL), fatigue, and psychological distress, in adult survivors of CPFT and CNPHT, representing two groups, which had received high and low radiation dose-exposure to the brain, respectively. Cognitive tests were used to compare CPFT (n = 12) and CNPHT (n = 7) survivors to matched healthy controls (n = 28). HRQOL data was compared to the general population (GP) (n = 1415-1459). Average follow-up was 23 (CPFT) and 19 years (CNPHT). CPFT survivors had significant deficits in all cognitive domains. CNPHT survivors showed results below the control group but differed statistically only on one executive test. HRQOL-ratings indicated that both groups had similar self-reported cognitive problems. CPFT survivors reported more emotional problems and fatigue. Anxiety was seen in both CPFT and CNPHT survivors. This study confirmed long-term cognitive sequelae after RT in adult survivors of CPFT,and possible RT-induced cognitive deficits in adult CNPHT survivors.
45.	Sabel, Magnus, 1966, et al. (författare) Active video gaming improves body coordination in survivors of childhood brain tumours. 2016 Ingår i: Disability and rehabilitation. - : Informa UK Limited. - 1464-5165 .- 0963-8288. ; 38:21, s. 2073-2084 Tidskriftsartikel (refereegranskat)abstract Purpose We investigated whether active video gaming (AVG) could bring about regular, enjoyable, physical exercise in children treated for brain tumours, what level of physical activity could be reached and if the children's physical functioning improved. Methods Thirteen children, aged 7-17 years, were randomised to either AVG or waiting-list. After 10-12 weeks they crossed-over. Weekly Internet coaching sessions were used to sustain motivation and evaluate enjoyment. Energy expenditure (EE) levels were measured as Metabolic Equivalent of Task (MET), using a multisensory activity monitor. Single-blinded assessments of physical functioning were done, using the Bruininks-Osteretsky Test of Motor Performance, second edition, evaluating participants before and after the intervention period, as well as comparing the randomisation groups after the first period. Results All patients completed the study. AVG sessions (mean duration 47minutes) were performed on 72% of all days. Mean EE level during AVG sessions was 3.0 MET, corresponding to moderate physical activity. The Body Coordination score improved by 15% (p=0.021) over the intervention period. Conclusions In this group of childhood brain tumour survivors, home-based AVG, supported by a coach, was a feasible, enjoyable and moderately intense form of exercise that improved Body Coordination. Implications for Rehabilitation Childhood brain tumour survivors frequently have cognitive problems, inferior physical functioning and are less physically active compared to their healthy peers. Active video gaming (AVG), supported by Internet coaching, is a feasible home-based intervention in children treated for brain tumours, promoting enjoyable, regular physical exercise of moderate intensity. In this pilot study, AVG with Nintendo Wii improved Body Coordination.
46.	Sabel, Magnus, 1966, et al. (författare) Effects of physically active video gaming on cognition and activities of daily living in childhood brain tumor survivors: a randomized pilot study 2017 Ingår i: Neuro Oncology Practice. - : Oxford University Press (OUP). - 2054-2577 .- 2054-2585. ; 4:2, s. 98-110 Tidskriftsartikel (refereegranskat)abstract Background. Physical activity can enhance cognitive functions in both animals and humans. We hypothesized that physically active video gaming could: i) improve cognitive functions and ii) improve the execution of activities of daily living among survivors of childhood brain tumors.Methods. Children 7 to 17 years old who completed treatment, including radiotherapy, for a brain tumor 1 to 5 years earlier were randomized to either intervention or waiting list. After 10 to 12 weeks the groups crossed over. The intervention consisted of active video gaming, using a motion-controlled video console (Nintendo Wii), for a minimum of 30 minutes a day, 5 days a week and weekly Internet-based coaching sessions. Evaluations before and after each period included tests of the execution of activities of daily living, using the Assessment of Motor and Process Skills (AMPS) and cognitive tests. Test scores before and after the intervention were compared. A parallel group comparison was performed as a sensitivity analysis.Results. All 13 children enrolled completed the program. Compared to baseline, the motor (P= .012) and process (P=.002) parts of AMPS improved significantly after active video gaming. In the parallel group analysis the improvement in the process part of AMPS remained statistically significant (P= .029), but not the change in AMPS motor score (P= .059). No significant change was found in cognitive tests although there were trends for improvement in sustained attention (P = .090) and selective attention (P = .078).Conclusion. In this pilot study, active video gaming used as a home-based intervention for childhood brain tumor survivors improved motor and process skills in activities of daily living.
47.	Sabel, Magnus, 1966, et al. (författare) Hypothermia after cranial irradiation protects neural progenitor cells in the subventricular zone but not in the hippocampus. 2017 Ingår i: International journal of radiation biology. - : Informa UK Limited. - 1362-3095 .- 0955-3002. ; 93:8, s. 771-783 Tidskriftsartikel (refereegranskat)abstract To explore if hypothermia can reduce the harmful effects of ionizing radiation on the neurogenic regions of the brain in young rats.Postnatal day 9 rats were randomized into two treatment groups, hypo- and normothermia, or a control group. Treatment groups were placed in chambers submerged in temperature-controlled water baths (30˚C and 36˚C) for 8h, after receiving a single fraction of 8Gy to the left hemisphere. Seven days post-irradiation, we measured the sizes of the subventricular zone (SVZ) and the granule cell layer (GCL) of the hippocampus, and counted the number of proliferating (phospho-histone H3+) cells and microglia (Iba1+ cells).Irradiation caused a 53% reduction in SVZ size in the normothermia group compared to controls, as well as a reduction of proliferating cell numbers by >50%. These effects were abrogated in the hypothermia group. Irradiation reduced the number of microglia in both treatment groups, but resulted in a lower cell density of Iba1+ cells in the SVZs of the hypothermia group. In the GCL, irradiation decreased both GCL size and the proliferating cell numbers, but with no difference between the treatment groups. The number of microglia in the GCL did not change.Hypothermia immediately after irradiation protects the SVZ and its proliferative cell population but the GCL is not protected, one week post-irradiation.
48.	Sabel, Magnus, 1966, et al. (författare) Relapse patterns and outcome after relapse in standard risk medulloblastoma: a report from the HIT-SIOP-PNET4 study 2016 Ingår i: Journal of Neuro-Oncology. - : Springer Science and Business Media LLC. - 0167-594X .- 1573-7373. ; 129:3, s. 515-524 Tidskriftsartikel (refereegranskat)abstract © 2016 The Author(s)The HIT-SIOP-PNET4 randomised trial for standard risk medulloblastoma (MB) (2001–2006) included 338 patients and compared hyperfractionated and conventional radiotherapy. We here report the long-term outcome after a median follow up of 7.8 years, including detailed information on relapse and the treatment of relapse. Data were extracted from the HIT Group Relapsed MB database and by way of a specific case report form. The event-free and overall (OS) survival at 10 years were 76 ± 2 % and 78 ± 2 % respectively with no significant difference between the treatment arms. Seventy-two relapses and three second malignant neoplasms were reported. Thirteen relapses (18 %) were isolated local relapses in the posterior fossa (PF) and 59 (82 %) were craniospinal, metastatic relapses (isolated or multiple) with or without concurrent PF disease. Isolated PF relapse vs all other relapses occurred at mean/median of 38/35 and 28/26 months respectively (p = 0.24). Late relapse, i.e. >5 years from diagnosis, occurred in six patients (8 %). Relapse treatment consisted of combinations of surgery (25 %), focal radiotherapy (RT 22 %), high dose chemotherapy with stem cell rescue (HDSCR 21 %) and conventional chemotherapy (90 %). OS at 5 years after relapse was 6.0 ± 4 %. In multivariate analysis; isolated relapse in PF, and surgery were significantly associated with prolonged survival whereas RT and HDSCR were not. Survival after relapse was not related to biological factors and was very poor despite several patients receiving intensive treatments. Exploration of new drugs is warranted, preferably based on tumour biology from biopsy of the relapsed tumour.
49.	Sand, Peter, et al. (författare) The reliability of the health related quality of life questionnaire PedsQL 3.0 cancer module in a sample of Swedish children. 2020 Ingår i: BMC pediatrics. - : Springer Science and Business Media LLC. - 1471-2431. ; 20:1 Tidskriftsartikel (refereegranskat)abstract The Pediatric Quality of Life Inventory (PedsQL) is a modular instrument, designed to integrate generic and disease specific measures, and includes both self- and proxy-reports. The aim of the study was to assess the reliability and limited validity of the Swedish version of the disease specific Pediatric Quality of Life Inventory 3.0 Cancer Module Scales (PedsQL 3.0), in a sample of Swedish children diagnosed with cancer.A total of 94 families at The Queen Silvia Children's Hospital, Sahlgrenska University participated in the study. The Pediatric Quality of Life Inventory 4.0 Generic Core Scales (PedsQL 4.0) and the PedsQL 3.0 were administered to 63 children (aged 5-18years) with cancer and to 94 parents of children with cancer aged 2-18years.The internal consistency of the PedsQL 3.0, reached or exceeded Cronbach's alpha values of 0.70 for both -self- and proxy-reports. The PedsQL 4.0 and PedsQL 3.0 were highly correlated (r=0.94 for proxy-reports and r=0.91 for self-reports), indicating convergent validity.PedsQL 3.0 Cancer Module Scalescan be used as a valuable tool for measuring cancer-specific HRQOL in child populations, both in research and in clinical practice.
50.	Schepke, Elizabeth, et al. (författare) DNA methylation profiling improves routine diagnosis of paediatric central nervous system tumours: A prospective population-based study 2022 Ingår i: Neuropathology and Applied Neurobiology. - : Wiley. - 0305-1846 .- 1365-2990. ; 48:6 Tidskriftsartikel (refereegranskat)abstract Aims: Paediatric brain tumours are rare, and establishing a precise diagnosis can be challenging. Analysis of DNA methylation profiles has been shown to be a reliable method to classify central nervous system (CNS) tumours with high accuracy. We aimed to prospectively analyse CNS tumours diagnosed in Sweden, to assess the clinical impact of adding DNA methylation-based classification to standard paediatric brain tumour diagnostics in an unselected cohort. Methods: All CNS tumours diagnosed in children (0-18 years) during 2017-2020 were eligible for inclusion provided sufficient tumour material was available. Tumours were analysed using genome-wide DNA methylation profiling and classified by the MNP brain tumour classifier. The initial histopathological diagnosis was compared with the DNA methylation-based classification. For incongruent results, a blinded re-evaluation was performed by an experienced neuropathologist. Results: Two hundred forty tumours with a histopathology-based diagnosis were profiled. A high-confidence methylation score of 0.84 or more was reached in 78% of the cases. In 69%, the histopathological diagnosis was confirmed, and for some of these also refined, 6% were incongruent, and the re-evaluation favoured the methylation-based classification. In the remaining 3% of cases, the methylation class was non-contributory. The change in diagnosis would have had a direct impact on the clinical management in 5% of all patients. Conclusions: Integrating DNA methylation-based tumour classification into routine clinical analysis improves diagnostics and provides molecular information that is important for treatment decisions. The results from methylation profiling should be interpreted in the context of clinical and histopathological information.

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