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  • 1.
    Ahlsten, G
    et al.
    Uppsala University, Disciplinary Domain of Medicine and Pharmacy, Faculty of Medicine, Department of Women's and Children's Health, Research group (Dept. of women´s and children´s health), Neuropediatrics/Paediatric oncology.
    Nowinski, Daniel
    Uppsala University, Disciplinary Domain of Medicine and Pharmacy, Faculty of Medicine, Department of Surgical Sciences.
    Mikrocefali och makrocefali2017In: Barnneurologi / [ed] Martin Jägervall och Johan Lundgren, Studentlitteratur AB, 2017, 1, p. 223-228Chapter in book (Other academic)
  • 2.
    Ahlsten, Gunnar
    et al.
    Uppsala University, Disciplinary Domain of Medicine and Pharmacy, Faculty of Medicine, Department of Women's and Children's Health, Research group (Dept. of women´s and children´s health), Neuropediatrics/Paediatric oncology. Dept of Pediatrics, Uppsala Hospital, Uppsala, Sweden.
    Nilsson, P
    Uppsala University, Disciplinary Domain of Medicine and Pharmacy, Faculty of Medicine, Department of Neuroscience, Neurosurgery. Dept of Neuroscience, Neurosurgery, University Hospital, Uppsala, Sweden.
    Wesslén, N
    Uppsala University, Disciplinary Domain of Medicine and Pharmacy, Faculty of Medicine, Department of Neuroscience, Neurosurgery. Dept of Neuroscience, Neurosurgery, University Hospital, Uppsala, Sweden.
    Axelsson, H
    Dept of Neuroscience, Neurophysiology, University Hospital, Uppsala, Sweden.
    Westbom, L
    Dept of Pediatrics, Univeristy Hospital, Lund, Sweden.
    Dorsal Rhizotomy for Spasticity Management in Cerbral Palsy2018In: Cerebral Palsy / [ed] F. Miller et al, Springer International Publishing AG , 2018Chapter in book (Refereed)
  • 3.
    Almblad, Ann-Charlotte
    et al.
    Uppsala University, Disciplinary Domain of Medicine and Pharmacy, Faculty of Medicine, Department of Women's and Children's Health, International Maternal and Child Health (IMCH), International Child Health and Nutrition.
    Brylid, Anna
    Uppsala University, Disciplinary Domain of Medicine and Pharmacy, Faculty of Medicine, Department of Women's and Children's Health.
    Engvall, Gunn
    Uppsala University, Disciplinary Domain of Medicine and Pharmacy, Faculty of Medicine, Department of Women's and Children's Health, Research group (Dept. of women´s and children´s health), Neuropediatrics/Paediatric oncology.
    Målqvist, Mats
    Uppsala University, Disciplinary Domain of Medicine and Pharmacy, Faculty of Medicine, Department of Women's and Children's Health, International Maternal and Child Health (IMCH), International Child Health and Nutrition.
    Increased intensive care admission rate after introduction of Early Detection and Treatment program for Children and the establishment of a pediatric intensive care unit at a tertiary hospital in SwedenIn: Article in journal (Refereed)
    Abstract [en]

    Objective: To evaluate the introduction of an Early Detection and Treatment program- Children (EDT-C) including a paediatric early warning score (PEWS) in relation to admission and length of stay at intensive care unit (ICU). Design: Before-after study utilizing data from the Electronic Patient Record (EPR) system, comparing outcomes over a total time period of 60 months between April 2010 and September 2015. Setting: A Swedish tertiary hospital. Patients: A total of 16,283 paediatric patients were included over the study period. Interventions: EDT-C including PEWS Measurements and Main Results: The following variables were extracted from the EPR data: 1) Admissions to paediatric wards 2) Length of stay at paediatric wards 3) Admissions to intensive care units 4) Length of stay at intensive care unit 5) Diagnosis. Intensive care unit admission increased from 5.0% (440/8746) before to 10.2 % (772/7537) after the introduction of the EDT-C (p<0.01). Mean treatment time at ICU did not change (41.0 vs 48.3 hours, p=0.23). Conclusion: The introduction of EDT-C including PEWS, in conjunction with the establishment of a paediatric intensive care unit at the hospital, resulted in an increased intensive care admittance rate among paediatric in-patients.

  • 4.
    Almblad, Ann-Charlotte
    et al.
    Uppsala University, Disciplinary Domain of Medicine and Pharmacy, Faculty of Medicine, Department of Women's and Children's Health, International Maternal and Child Health (IMCH), International Child Health and Nutrition.
    Målqvist, Mats
    Uppsala University, Disciplinary Domain of Medicine and Pharmacy, Faculty of Medicine, Department of Women's and Children's Health, International Maternal and Child Health (IMCH), International Child Health and Nutrition.
    Engvall, Gunn
    Uppsala University, Disciplinary Domain of Medicine and Pharmacy, Faculty of Medicine, Department of Women's and Children's Health, Research group (Dept. of women´s and children´s health), Neuropediatrics/Paediatric oncology.
    From skepticism to assurance and control: Implementation of a patient safety system at a pediatric hospital in Sweden2018In: PLoS ONE, ISSN 1932-6203, E-ISSN 1932-6203, Vol. 13, no 11, article id e0207744Article in journal (Refereed)
    Abstract [en]

    BACKGROUND: The use of evidence-based practice among healthcare professionals directly correlates to better outcomes for patients and higher professional satisfaction. Translating knowledge in practice and mobilizing evidence-based clinical care remains a continuing challenge in healthcare systems across the world.

    PURPOSE: To describe experiences from the implementation of an Early Detection and Treatment Program for Children (EDT-C) among health care professionals at a pediatric hospital in Sweden.

    DESIGN AND METHODS: Sixteen individual interviews were conducted with physicians, nurses and nurse assistants, which of five were instructors. Data were analyzed with qualitative content analysis.

    RESULTS: An overarching theme was created: From uncertainty and skepticism towards assurance and control. The theme was based on the content of eight categories: An innovation suitable for clinical practice, Differing conditions for change, Lack of organizational slack, Complex situations, A pragmatic implementation strategy, Delegated responsibility, Experiences of control and Successful implementation.

    CONCLUSIONS: Successful implementation was achieved when initial skepticism among staff was changed into acceptance and using EDT-C had become routine in their daily work. Inter-professional education including material from authentic patient cases promotes knowledge about different professions and can strengthen teamwork. EDT-C with evidenced-based material adapted to the context can give healthcare professionals a structured and objective tool with which to assess and treat patients, giving them a sense of control and assurance.

  • 5.
    Almblad, Ann-Charlotte
    et al.
    Uppsala University, Disciplinary Domain of Medicine and Pharmacy, Faculty of Medicine, Department of Women's and Children's Health, International Maternal and Child Health (IMCH), International Child Health and Nutrition.
    Målqvist, Mats
    Uppsala University, Disciplinary Domain of Medicine and Pharmacy, Faculty of Medicine, Department of Women's and Children's Health, International Maternal and Child Health (IMCH), International Child Health and Nutrition.
    Engvall, Gunn
    Uppsala University, Disciplinary Domain of Medicine and Pharmacy, Faculty of Medicine, Department of Women's and Children's Health, Research group (Dept. of women´s and children´s health), Neuropediatrics/Paediatric oncology.
    From skepticism to assurance and control: implementation of a patient safety system at a pediatric hospital in SwedenIn: Article in journal (Refereed)
  • 6.
    Almblad, Ann-Charlotte
    et al.
    Uppsala University, Disciplinary Domain of Medicine and Pharmacy, Faculty of Medicine, Department of Women's and Children's Health, International Maternal and Child Health (IMCH), International Child Health and Nutrition.
    Siltberg, Petra
    Uppsala University, Disciplinary Domain of Medicine and Pharmacy, Faculty of Medicine, Department of Women's and Children's Health, Research group (Dept. of women´s and children´s health), Neuropediatrics/Paediatric oncology.
    Engvall, Gunn
    Uppsala University, Disciplinary Domain of Medicine and Pharmacy, Faculty of Medicine, Department of Women's and Children's Health, Research group (Dept. of women´s and children´s health), Neuropediatrics/Paediatric oncology.
    Målqvist, Mats
    Uppsala University, Disciplinary Domain of Medicine and Pharmacy, Faculty of Medicine, Department of Women's and Children's Health, International Maternal and Child Health (IMCH), International Child Health and Nutrition.
    Implementation of Pediatric Early Warning Score: Adherence to Guidelines and Influence of Context2018In: Journal of Pediatric Nursing: Nursing Care of Children and Families, ISSN 0882-5963, E-ISSN 1532-8449, Vol. 38, p. 33-39Article in journal (Refereed)
    Abstract [en]

    PURPOSE: To describe data of Pediatric Early Warning Score (PEWS) registrations and to evaluate the implementation of PEWS by examining adherence to clinical guidelines based on measured PEWS, and to relate findings to work context.

    DESIGN AND METHODS: PEWS, as a part of a concept called Early Detection and Treatment-Children (EDT-C) was implemented at three wards at a Children's Hospital in Sweden. Data were collected from the Electronic Patient Record (EPR) retrospectively to assess adherence to guidelines. The Alberta Context Tool (ACT) was used to assess work context among healthcare professionals (n=110) before implementation of EDT-C.

    RESULTS: The majority of PEWS registrations in EPR were low whereas 10% were moderate to high. Adherences to ward-specific guidelines at admission and for saturation in respiratory distress were high whereas adherence to pain assessment was low. There were significant differences in documented recommended actions between wards. Some differences in leadership and evaluation between wards were identified.

    CONCLUSIONS: Evaluation of PEWS implementation indicated frequent use of the tool despite most scores being low. High scores (5-9) occurred 28 times, which may indicate that patients with a high risk of clinical deterioration were identified. Documentation of the consequent recommended actions was however incomplete and there was a large variation in adherence to guidelines. Contextual factors may have an impact on adherence.

    PRACTICE IMPLICATIONS: EDT-C can lead to increased knowledge about early detection of deterioration, strengthen nurses as professionals, optimize treatment and teamwork and thereby increase patient safety for children treated in hospitals.

  • 7.
    Anastasopoulou, Stavroula
    et al.
    Karolinska Inst, Dept Womens & Childrens Hlth, Stockholm, Sweden;Karolinska Univ Hosp, Stockholm, Sweden.
    Eriksson, Mats A.
    Karolinska Inst, Dept Womens & Childrens Hlth, Stockholm, Sweden;Karolinska Univ Hosp, Stockholm, Sweden.
    Heyman, Mats
    Karolinska Inst, Dept Womens & Childrens Hlth, Stockholm, Sweden;Karolinska Univ Hosp, Stockholm, Sweden.
    Wang, Chen
    Karolinska Inst, Dept Womens & Childrens Hlth, Stockholm, Sweden;Karolinska Univ Hosp, Stockholm, Sweden.
    Niinimäki, Riitta
    Oulu Univ Hosp, Dept Children & Adolescents, Oulu, Finland;Univ Oulu, PEDEGO Res Unit, Oulu, Finland.
    Mikkel, Sirje
    Univ Tartu, Dept Hematol & Oncol, Tartu, Estonia.
    Vaitkeviciene, Goda E.
    Vilnius Univ Hosp Santaros Klin, Childrens Hosp, Vilnius, Lithuania;Vilnius Univ, Vilnius, Lithuania.
    Johannsdottir, Inga Maria
    Oslo Univ Hosp, Dept Pediat Hematol Oncol, Oslo, Norway.
    Myrberg, Ida Hed
    Karolinska Inst, Dept Womens & Childrens Hlth, Stockholm, Sweden.
    Jonsson, Olafur Gisli
    Univ Iceland, Dept Pediat, Reykjavik, Iceland.
    Als-Nielsen, Bodil
    Rigshosp, Univ Hosp, Dept Pediat & Adolescent Med, Copenhagen, Denmark;Univ Copenhagen, Fac Med, Inst Clin Med, Copenhagen, Denmark.
    Schmiegelow, Kjeld
    Rigshosp, Univ Hosp, Dept Pediat & Adolescent Med, Copenhagen, Denmark;Univ Copenhagen, Fac Med, Inst Clin Med, Copenhagen, Denmark.
    Banerjee, Joanna
    Univ Helsinki, Childrens Hosp, Dept Pediat Hematol Oncol & Stem Cell Transplanta, Helsinki, Finland;Helsinki Univ Hosp, Helsinki, Finland.
    Harila-Saari, Arja H.
    Uppsala University, Disciplinary Domain of Medicine and Pharmacy, Faculty of Medicine, Department of Women's and Children's Health, Research group (Dept. of women´s and children´s health), Neuropediatrics/Paediatric oncology.
    Ranta, Susanna
    Karolinska Inst, Dept Womens & Childrens Hlth, Stockholm, Sweden;Karolinska Univ Hosp, Stockholm, Sweden.
    Posterior reversible encephalopathy syndrome in children with acute lymphoblastic leukemia: Clinical characteristics, risk factors, course, and outcome of disease2019In: Pediatric Blood & Cancer, ISSN 1545-5009, E-ISSN 1545-5017, Vol. 66, no 5, article id e27594Article in journal (Refereed)
    Abstract [en]

    Background: Posterior reversible encephalopathy syndrome (PRES) is a distinct entity with incompletely known predisposing factors. The aim of this study is to describe the incidence, risk factors, clinical course, and outcome of PRES in childhood acute lymphoblastic leukemia (ALL).

    Procedure: Patients aged 1.0 to 17.9 years diagnosed with ALL from July 2008 to December 2015 and treated according to the Nordic Society of Pediatric Hematology and Oncology (NOPHO) ALL2008 protocol were included. Patients with PRES were identified in the prospective NOPHO leukemia toxicity registry, and clinical data were collected from the medical records.

    Results: The study group included 1378 patients, of whom 52 met the criteria for PRES. The cumulative incidence of PRES at one month was 1.7% (95% CI, 1.1-2.5) and at one year 3.7% (95% CI, 2.9-4.9). Older age (hazard ratios [HR] for each one-year increase in age 1.1; 95% CI, 1.0-1.2, P = 0.001) and T-cell immunophenotype (HR, 2.9; 95% CI, 1.6-5.3, P = 0.0005) were associated with PRES. Central nervous system (CNS) involvement (odds ratios [OR] = 2.8; 95% CI, 1.2-6.5, P = 0.015) was associated with early PRES and high-risk block treatment (HR = 2.63; 95% CI, 1.1-6.4, P = 0.033) with late PRES. At follow-up of the PRES patients, seven patients had epilepsy and seven had neurocognitive difficulties.

    Conclusion: PRES is a neurotoxicity in the treatment of childhood ALL with both acute and long-term morbidity. Older age, T-cell leukemia, CNS involvement and high-risk block treatment are risk factors for PRES.

  • 8.
    Armuand, G. M.
    et al.
    Karolinska Inst, Dept Neurobiol Care Sci & Soc, Stockholm, Sweden..
    Nilsson, J.
    Karolinska Inst, Dept Neurobiol Care Sci & Soc, Stockholm, Sweden..
    Rodriguez-Wallberg, K. A.
    Karolinska Inst, Dept Oncol Pathol, Stockholm, Sweden.;Karolinska Univ Hosp, Reprod Med, Stockholm, Sweden..
    Malmros, J.
    Karolinska Univ Hosp, Astrid Lindgren Childrens Hosp, Paediat Oncol Unit, Stockholm, Sweden.;Karolinska Inst, Dept Womens & Childrens Hlth, Stockholm, Sweden..
    Arvidson, Johan
    Uppsala University, Disciplinary Domain of Medicine and Pharmacy, Faculty of Medicine, Department of Women's and Children's Health, Research group (Dept. of women´s and children´s health), Neuropediatrics/Paediatric oncology.
    Lampic, C.
    Karolinska Inst, Dept Neurobiol Care Sci & Soc, Stockholm, Sweden..
    Wettergren, L.
    Karolinska Inst, Dept Neurobiol Care Sci & Soc, Stockholm, Sweden..
    Physicians' self-reported practice behaviour regarding fertility-related discussions in paediatric oncology in Sweden2017In: Psycho-Oncology, ISSN 1057-9249, E-ISSN 1099-1611, Vol. 26, no 10, p. 1684-1690Article in journal (Refereed)
    Abstract [en]

    Objective: The aim of this study was to investigate practice behaviours of Swedish physicians with regard to discussing the impact of cancer treatment on fertility with paediatric oncology patients and their parents, and to identify factors associated with such discussions.

    Methods: A cross-sectional survey study was conducted targeting all physicians in Sweden working in paediatric oncology care settings. Participants responded to a questionnaire measuring practice behaviour, attitudes, barriers, and confidence in knowledge. Multivariable logistic regression was used to determine factors associated with seldom discussing fertility.

    Results: More than half of the physicians routinely talked with their patients/parents about the treatment's potential impact on fertility (male patients: 62%; female patients: 57%; P = 0.570). Factors associated with less frequently discussing fertility with patients/parents were working at a non-university hospital (male patients: OR 11.49, CI 1.98-66.67; female patients: OR 33.18, CI 4.06-271.07), concerns that the topic would cause worry (male patients: OR 8.23, CI 1.48-45.89; female patients: OR 12.38, CI 1.90-80.70), and perceiving the parents as anxious (male patients: OR 7.18, CI 1.20-42.85; female patients: OR 11.65, CI 1.32-103.17).

    Conclusions: Based on our findings, we recommend structured training in how to communicate about fertility issues in stressful situations, which in turn might increase fertility-related discussions in paediatric oncology.

  • 9.
    Bager, Ninna
    et al.
    Aarhus Univ Hosp, Dept Paediat & Adolescent Med, Palle Juul Jensens Blvd 99, DK-8200 Aarhus N, Denmark.
    Juul-Dam, Kristian L.
    Aarhus Univ Hosp, Dept Paediat & Adolescent Med, Palle Juul Jensens Blvd 99, DK-8200 Aarhus N, Denmark.
    Sandahl, Julie D.
    Aarhus Univ Hosp, Dept Paediat & Adolescent Med, Palle Juul Jensens Blvd 99, DK-8200 Aarhus N, Denmark.
    Abrahamsson, Jonas
    Queen Silvia Childrens Hosp, Inst Clin Sci, Dept Paediat, Gothenburg, Sweden.
    Beverloo, Berna
    Erasmus MC Sophia Childrens Hosp, Dept Cytogenet, Rotterdam, Netherlands.
    de Bont, Eveline S. J. M.
    Univ Med Ctr Groningen, Dept Paediat, Groningen, Netherlands.
    Ha, Shau-Yin
    Queen Mary Hosp, Hong Kong Paediat Haematol & Oncol Study Grp, Dept Paediat, Hong Kong, Hong Kong, Peoples R China.
    Jahnukainen, Kirsi
    Univ Helsinki, Childrens Hosp, Helsinki, Finland;Helsinki Univ Hosp, Helsinki, Finland.
    Jonsson, Olafur G.
    Landspital Inn, Dept Paediat, Reykjavik, Iceland.
    Kaspers, Gertjan L.
    Vrije Univ Amsterdam Med Ctr, Paediat Oncol Haematol, Amsterdam, Netherlands;Acad Princess Maxima Ctr Paediat Oncol, Utrecht, Netherlands.
    Kovalova, Zhanna
    Childrens Clin Univ Hosp, Dept Paediat, Riga, Latvia.
    Lausen, Birgitte
    Univ Copenhagen, Rigshosp, Dept Paediat & Adolescent Med, Copenhagen, Denmark.
    De Moerloose, Barbara
    Ghent Univ Hosp, Dept Paediat, Ghent, Belgium.
    Noren-Nystroem, Ulrika
    Umea Univ Hosp, Dept Med Biosci, Genet, Umea, Sweden.
    Palle, Josefine
    Uppsala University, Disciplinary Domain of Medicine and Pharmacy, Faculty of Medicine, Department of Women's and Children's Health, Research group (Dept. of women´s and children´s health), Neuropediatrics/Paediatric oncology.
    Saks, Kadri
    SA Tallinna Lastehaigla, Dept Paediat, Tallinn, Estonia.
    Zeller, Bernward
    Oslo Univ Hosp, Div Paediat & Adolescent Med, Oslo, Norway.
    Kjeldsen, Eigil
    Aarhus Univ Hosp, Dept Cytogenet, Aarhus, Denmark.
    Hasle, Henrik
    Aarhus Univ Hosp, Dept Paediat & Adolescent Med, Palle Juul Jensens Blvd 99, DK-8200 Aarhus N, Denmark.
    Complex and monosomal karyotype are distinct cytogenetic entities with an adverse prognostic impact in paediatric acute myeloid leukaemia: A NOPHO-DBH-AML study2018In: British Journal of Haematology, ISSN 0007-1048, E-ISSN 1365-2141, Vol. 183, no 4, p. 618-628Article in journal (Refereed)
    Abstract [en]

    Data on occurrence, genetic characteristics and prognostic impact of complex and monosomal karyotype (CK/MK) in children with acute myeloid leukaemia (AML) are scarce. We studied CK and MK in a large unselected cohort of childhood AML patients diagnosed and treated according to Nordic Society for Paediatric Haematology and Oncology (NOPHO)-AML protocols 1993-2015. In total, 800 patients with de novo AML were included. CK was found in 122 (15%) and MK in 41 (5%) patients. CK and MK patients were young (median age 2.1 and 3.3 years, respectively) and frequently had FAB M7 morphology (24% and 22%, respectively). Refractory disease was more common in MK patients (15% vs. 4%) and stem cell transplantation in first complete remission was more frequent (32% vs. 19%) compared with non-CK/non-MK patients. CK showed no association with refractory disease but was an independent predictor of an inferior event-free survival (EFS; hazard ratio [HR] 1.43, P = 0.03) and overall survival (OS; HR 1.48, P = 0.01). MK was associated with a poor EFS (HR 1.57, P = 0.03) but did not show an inferior OS compared to non-MK patients (HR 1.14, P = 0.62). In a large paediatric cohort, we characterized AML with non-recurrent abnormal karyotype and unravelled the adverse impact of CK and MK on prognosis.

  • 10.
    Baygan, Arjang
    et al.
    Translational Cell Therapy Research Group (TCR), Division of Therapeutic Immunology, Department of LabMed, Karolinska Institutet, Stockholm, Sweden.
    Aronsson-Kurttila, Wictor
    Translational Cell Therapy Research Group (TCR), Division of Therapeutic Immunology, Department of LabMed, Karolinska Institutet, Stockholm, Sweden.
    Moretti, Gianluca
    Translational Cell Therapy Research Group (TCR), Division of Therapeutic Immunology, Department of LabMed, Karolinska Institutet, Stockholm, Sweden.
    Tibert, Babylonia
    Translational Cell Therapy Research Group (TCR), Division of Therapeutic Immunology, Department of LabMed, Karolinska Institutet, Stockholm, Sweden.
    Dahllöf, Göran
    Department of Dental Medicine, Karolinska Institutet, Stockholm, Sweden.
    Klingspor, Lena
    Uppsala University, Disciplinary Domain of Medicine and Pharmacy, Faculty of Medicine, Department of Medical Sciences, Clinical Microbiology. Department of Microbiology, Uppsala University Hospital, Uppsala, Sweden.
    Gustafsson, Britt
    Uppsala University, Disciplinary Domain of Medicine and Pharmacy, Faculty of Medicine, Department of Women's and Children's Health, Research group (Dept. of women´s and children´s health), Neuropediatrics/Paediatric oncology.
    Khoein, Bita
    Translational Cell Therapy Research Group (TCR), Division of Therapeutic Immunology, Department of LabMed, Karolinska Institutet, Stockholm, Sweden.
    Moll, Guido
    Translational Cell Therapy Research Group (TCR), Division of Therapeutic Immunology, Department of LabMed, Karolinska Institutet, Stockholm, Sweden.
    Hausmann, Charlotta
    Center for Allogeneic Stem Cell Transplantation, Department of Pathology/Oncology, Karolinska University Hospital, Stockholm, Sweden.
    Svahn, Britt-Marie
    Translational Cell Therapy Research Group (TCR), Division of Therapeutic Immunology, Department of LabMed, Karolinska Institutet, Stockholm, Sweden.
    Westgren, Magnus
    Department of Obstetrics and Gynecology, Karolinska University Hospital, Stockholm, Sweden.
    Remberger, Mats
    Center for Allogeneic Stem Cell Transplantation, Department of Pathology/Oncology, Karolinska University Hospital, Stockholm, Sweden.
    Sadeghi, Behnam
    Translational Cell Therapy Research Group (TCR), Division of Therapeutic Immunology, Department of LabMed, Karolinska Institutet, Stockholm, Sweden.
    Ringden, Olle
    Translational Cell Therapy Research Group (TCR), Division of Therapeutic Immunology, Department of LabMed, Karolinska Institutet, Stockholm, Sweden.
    Safety and Side Effects of Using Placenta-Derived Decidual Stromal Cells for Graft-versus-Host Disease and Hemorrhagic Cystitis2017In: Frontiers in Immunology, ISSN 1664-3224, E-ISSN 1664-3224, Vol. 8, article id 795Article in journal (Refereed)
    Abstract [en]

    Mesenchymal stromal cells (MSCs) are increasingly used in regenerate medicine. Placenta-derived decidual stromal cells (DSCs) are a novel therapy for acute graft-versus-host-disease (GVHD) and hemorrhagic cystitis (HC) after allogeneic hematopoietic stem cell transplantation (HSCT). DSCs are more immunosuppressive than MSCs. We assessed adverse events and safety using DSCs among 44 treated patients and 40 controls. The median dose of infused cells was 1.5 (range 0.9–2.9) × 106 DSCs/kg. The patients were given 2 (1–5) doses, with a total of 82 infusions. Monitoring ended 3 months after the last DSC infusion. Three patients had transient reactions during DSC infusion. Laboratory values, hemorrhages, and transfusions were similar in the two groups. The frequency of leukemic relapse (2/2, DSC/controls) and invasive fungal infections (6/6) were the same in the two groups. Causes of death were those seen in HSCT patients: infections (5/3), respiratory failure (1/1), circulatory failure (3/1), thromboembolism (1/0), multiorgan failure (0/1), and GVHD and others (2/7). One-year survival for the DSC patients with GVHD was 67%, which was significantly better than achieved previously at our center. One-year survival was 90% in the DSC-treated HC group. DSC infusions appear safe. Randomized studies are required to prove efficacy.

  • 11.
    Byrne, Julianne
    et al.
    Boyne Res Inst, 5 Bolton Sq East, Drogheda A92 RY6K, Louth, Ireland..
    Alessi, Daniela
    Univ Turin, Citta Salute & Sci Hosp, Canc Epidemiol Unit, Childhood Canc Registry Piedmont, Via Santena 7, I-10126 Turin, Italy.;Ctr Canc Prevent CPO, Via Santena 7, I-10126 Turin, Italy..
    Allodji, Rodrigue S.
    Univ Paris Saclay, Gustave Roussy, Canc & Radiat, Unit INSERM 1018, 39 Rue Camille Desmoulins, F-94805 Villejuif, France..
    Bagnasco, Francesca
    Ist Giannina Gaslini, Epidemiol & Biostat Unit, Italian Off Therapy Registry OTR, Via Gerolamo Gaslini 5, I-16148 Genoa, Italy..
    Bardi, Edit
    Semmelweis Univ, Dept Pediat 2, Budapest, Hungary.;Kepler Univ Klinikum, Linz, Austria..
    Bautz, Andrea
    Danish Canc Soc, Res Ctr, Strandboulevarden 49, DK-2100 Copenhagen, Denmark..
    Bright, Chloe J.
    Univ Birmingham, Inst Appl Hlth Res, Ctr Childhood Canc Survivor Studies, Birmingham B15 2TT, W Midlands, England..
    Brown, Morven
    Newcastle Univ, Inst Hlth & Soc, Newcastle Upon Tyne NE4 5PL, Tyne & Wear, England..
    Diallo, Ibrahima
    Univ Paris Saclay, Gustave Roussy, Canc & Radiat, Unit INSERM 1018, 39 Rue Camille Desmoulins, F-94805 Villejuif, France..
    Feijen, Elizabeth A. M. (Lieke)
    Acad Med Ctr Amsterdam, Emma Childrens Hosp, Dept Pediat Oncol, Meibergdreef 9, NL-1105 AZ Amsterdam, Netherlands.;Princess Maxima Ctr Paediat Oncol, Utrecht, Netherlands..
    Fidler, Miranda M.
    WHO, Sect Canc Surveillance, Int Agcy Res Canc, F-69372 Lyon 08, France..
    Frey, Eva
    St Anna Childrens Hosp, Kinderspitalgasse 6, A-1090 Vienna, Austria..
    Garwicz, Stanislaw
    Lund Univ, Dept Clin Sci, Pediat, Skane Univ Hosp, Lund, Sweden..
    Grabow, Desiree
    Univ Med Ctr, Inst Med Biostat Epidemiol & Informat, GCCR, Mainz, Germany..
    Gudmundsdottir, Thorgerdur
    Danish Canc Soc, Res Ctr, Strandboulevarden 49, DK-2100 Copenhagen, Denmark..
    Hagberg, Oskar
    Reg Canc Ctr South, Lund, Sweden..
    Harila-Saari, Arja H.
    Uppsala University, Disciplinary Domain of Medicine and Pharmacy, Faculty of Medicine, Department of Women's and Children's Health, Research group (Dept. of women´s and children´s health), Neuropediatrics/Paediatric oncology.
    Hau, Eva M.
    Univ Bern, Inst Social & Prevent Med, Swiss Childhood Canc Registry, Bern, Switzerland.;Univ Bern, Univ Childrens Hosp Bern, Dept Paediat, Bern, Switzerland..
    Haupt, Riccardo
    Ist Giannina Gaslini, Epidemiol & Biostat Unit, Italian Off Therapy Registry OTR, Via Gerolamo Gaslini 5, I-16148 Genoa, Italy..
    Hawkins, Mike M.
    Univ Birmingham, Inst Appl Hlth Res, Ctr Childhood Canc Survivor Studies, Birmingham B15 2TT, W Midlands, England..
    Jakab, Zsuzsanna
    Semmelweis Univ, Dept Pediat 2, Hungarian Childhood Canc Registry, Budapest, Hungary..
    Jankovic, Momcilo
    Univ Milano Bicocca, Fdn MBBM Monza, Pediat Clin, Monza, Italy.;Italian Off Therapy Register OTR, Monza, Italy..
    Kaatsch, Peter
    Univ Med Ctr, Inst Med Biostat Epidemiol & Informat, GCCR, Mainz, Germany..
    Kaiser, Melanie
    Univ Med Ctr, Inst Med Biostat Epidemiol & Informat, GCCR, Mainz, Germany..
    Kremer, Leontien C. M.
    Acad Med Ctr Amsterdam, Emma Childrens Hosp, Dept Pediat Oncol, Meibergdreef 9, NL-1105 AZ Amsterdam, Netherlands.;Princess Maxima Ctr Paediat Oncol, Utrecht, Netherlands..
    Kuehni, Claudia E.
    Univ Bern, Inst Social & Prevent Med, Swiss Childhood Canc Registry, Bern, Switzerland.;Univ Bern, Univ Childrens Hosp Bern, Dept Paediat, Bern, Switzerland..
    Kuonen, Rahel
    Univ Bern, Inst Social & Prevent Med, Swiss Childhood Canc Registry, Bern, Switzerland..
    Ladenstein, Ruth
    Med Univ, St Anna Childrens Canc Res Inst, Dept Paediat, Vienna, Austria..
    Lahteenmaki, Paivi Maria
    Turku Univ, Dept Pediat & Adolescent Med, Turku, Finland.;Turku Univ Hosp, Turku, Finland..
    Levitt, Gill
    Great Ormond St Hosp Children NHS Fdn Trust, Dept Oncol, London WCIN3JH, England..
    Linge, Helena
    Lund Univ, Dept Clin Sci, Pediat, Skane Univ Hosp, Lund, Sweden..
    LLanas, Damien
    Univ Paris Saclay, Gustave Roussy, Canc & Radiat, Unit INSERM 1018, 39 Rue Camille Desmoulins, F-94805 Villejuif, France..
    Michel, Gisela
    Univ Lucerne, Dept Hlth Sci & Hlth Policy, Luzern, Switzerland..
    Morsellino, Vera
    Ist Giannina Gaslini, Epidemiol & Biostat Unit, Italian Off Therapy Registry OTR, Via Gerolamo Gaslini 5, I-16148 Genoa, Italy..
    Mulder, Renee L.
    Acad Med Ctr Amsterdam, Emma Childrens Hosp, Dept Pediat Oncol, Meibergdreef 9, NL-1105 AZ Amsterdam, Netherlands.;Princess Maxima Ctr Paediat Oncol, Utrecht, Netherlands..
    Reulen, Raoul C.
    Univ Birmingham, Inst Appl Hlth Res, Ctr Childhood Canc Survivor Studies, Birmingham B15 2TT, W Midlands, England..
    Ronckers, Cecile M.
    Acad Med Ctr Amsterdam, Emma Childrens Hosp, Dept Pediat Oncol, Meibergdreef 9, NL-1105 AZ Amsterdam, Netherlands.;Princess Maxima Ctr Paediat Oncol, Utrecht, Netherlands..
    Sacerdote, Carlotta
    Univ Turin, Citta Salute & Sci Hosp, Canc Epidemiol Unit, Childhood Canc Registry Piedmont, Via Santena 7, I-10126 Turin, Italy.;Ctr Canc Prevent CPO, Via Santena 7, I-10126 Turin, Italy..
    Skinner, Roderick
    Royal Victoria Infirm, Great North Childrens Hosp, Dept Paediat & Adolescent Haematol & Oncol, Childrens BMT Unit, Newcastle Upon Tyne NE1 4LP, Tyne & Wear, England.;Newcastle Univ, Northern Inst Canc Res, Newcastle Upon Tyne NE4 5PL, Tyne & Wear, England..
    Steliarova-Foucher, Eva
    WHO, Sect Canc Surveillance, Int Agcy Res Canc, F-69372 Lyon 08, France..
    van der Pal, Helena J.
    Princess Maxima Ctr Paediat Oncol, Utrecht, Netherlands..
    de Vathaire, Florent
    Univ Paris Saclay, Gustave Roussy, Canc & Radiat, Unit INSERM 1018, 39 Rue Camille Desmoulins, F-94805 Villejuif, France..
    Bezin, Giao Vu
    Univ Paris Saclay, Gustave Roussy, Canc & Radiat, Unit INSERM 1018, 39 Rue Camille Desmoulins, F-94805 Villejuif, France..
    Wesenberg, Finn
    Norwegian Canc Register, Oslo, Norway.;Oslo Univ Hosp, Dept Pediat Med, Oslo, Norway.;Univ Oslo, Fac Med, Inst Clin Med, Oslo, Norway..
    Wiebe, Thomas
    Lund Univ, Dept Clin Sci, Pediat, Skane Univ Hosp, Lund, Sweden..
    Winter, David L.
    Univ Birmingham, Inst Appl Hlth Res, Ctr Childhood Canc Survivor Studies, Birmingham B15 2TT, W Midlands, England..
    Winther, Jeanette Falck
    Danish Canc Soc, Res Ctr, Strandboulevarden 49, DK-2100 Copenhagen, Denmark.;Aarhus Univ, Fac Hlth, Dept Clin Med, Palle Juul Jensens Blvd 82, DK-8200 Aarhus, Denmark..
    Witthoff, Elise
    Lund Univ, Dept Clin Sci, Pediat, Skane Univ Hosp, Lund, Sweden..
    Zaletel, Lorna Zadravec
    Inst Oncol, Div Radiotherapy, Zaloska Cesta 2, SI-1000 Ljubljana, Slovenia..
    Hjorth, Lars
    Lund Univ, Dept Clin Sci, Pediat, Skane Univ Hosp, Lund, Sweden..
    The PanCareSurFup consortium: research and guidelines to improve lives for survivors of childhood cancer2018In: European Journal of Cancer, ISSN 0959-8049, E-ISSN 1879-0852, Vol. 103, no Nov, p. 238-248Article in journal (Refereed)
    Abstract [en]

    Background: Second malignant neoplasms and cardiotoxicity are among the most serious and frequent adverse health outcomes experienced by childhood and adolescent cancer survivors (CCSs) and contribute significantly to their increased risk of premature mortality. Owing to differences in health-care systems, language and culture across the continent, Europe has had limited success in establishing multi-country collaborations needed to assemble the numbers of survivors required to clarify the health issues arising after successful cancer treatment. PanCareSurFup (PCSF) is the first pan-European project to evaluate some of the serious long-term health risks faced by survivors. This article sets out the overall rationale, methods and preliminary results of PCSF. Methods: The PCSF consortium pooled data from 13 cancer registries and hospitals in 12 European countries to evaluate subsequent primary malignancies, cardiac disease and late mortality in survivors diagnosed between ages 0 and 20 years. In addition, PCSF integrated radiation dosimetry to sites of second malignancies and to the heart, developed evidence-based guidelines for long-term care and for transition services, and disseminated results to survivors and the public. Results: We identified 115,596 individuals diagnosed with cancer, of whom 83,333 were 5-year survivors and diagnosed from 1940 to 2011. This single data set forms the basis for cohort analyses of subsequent malignancies, cardiac disease and late mortality and case-control studies of subsequent malignancies and cardiac disease in 5-year survivors. Conclusions: PCSF delivered specific estimates of risk and comprehensive guidelines to help survivors and care-givers. The expected benefit is to provide every European CCS with improved access to care and better long-term health.

  • 12.
    Chen, D.
    et al.
    Univ Gothenburg, Dept Rheumatol & Inflammat Res, Gothenburg, Sweden..
    Gerasimcik, N.
    Univ Gothenburg, Dept Rheumatol & Inflammat Res, Gothenburg, Sweden..
    Camponeschi, A.
    Univ Gothenburg, Dept Rheumatol & Inflammat Res, Gothenburg, Sweden..
    Tan, Y.
    Guizhou Med Univ, Affiliated Hosp, Cent Lab, Guiyang, Peoples R China..
    Wu, Q.
    Guizhou Med Univ, Affiliated Hosp, Cent Lab, Guiyang, Peoples R China..
    Brynjolfsson, S.
    Univ Gothenburg, Dept Microbiol & Immunol, Gothenburg, Sweden..
    Zheng, J.
    Univ Gothenburg, Dept Rheumatol & Inflammat Res, Gothenburg, Sweden..
    Abrahamsson, J.
    Univ Gothenburg, Dept Pediat, Gothenburg, Sweden..
    Nordlund, Jessica
    Uppsala University, Disciplinary Domain of Medicine and Pharmacy, Faculty of Medicine, Department of Medical Sciences, Molecular Medicine. Uppsala University, Science for Life Laboratory, SciLifeLab.
    Lönnerholm, Gudmar
    Uppsala University, Disciplinary Domain of Medicine and Pharmacy, Faculty of Medicine, Department of Women's and Children's Health, Research group (Dept. of women´s and children´s health), Neuropediatrics/Paediatric oncology.
    Fogelstrand, L.
    Sahlgrens Univ Hosp, Dept Clin Chem, Gothenburg, Sweden.;Univ Gothenburg, Dept Clin Chem & Transfus Med, Gothenburg, Sweden..
    Mårtensson, I-L
    Univ Gothenburg, Dept Rheumatol & Inflammat Res, Gothenburg, Sweden..
    CD27 expression and its association with clinical outcome in children and adults with pro-B acute lymphoblastic leukemia2017In: Blood Cancer Journal, ISSN 2044-5385, E-ISSN 2044-5385, Vol. 7, article id e575Article in journal (Other academic)
  • 13.
    Cooper, Tess E
    et al.
    Cochrane Pain, Palliative and Supportive Care Group, Pain Research Unit, Churchill Hospital, Oxford, UK.
    Heathcote, Lauren C
    Department of Anesthesiology, Perioperative and Pain Medicine, Stanford University, Palo Alto, California, USA.
    Anderson, Brian
    Paediatric Intensive Care Unit, Starship Childrens Hospital, Auckland, New Zealand.
    Grégoire, Marie-Claude
    Paediatric Palliative Care, Department of Paediatrics, IWK Health Centre, Dalhousie University, Halifax, Canada.
    Ljungman, Gustaf
    Uppsala University, Disciplinary Domain of Medicine and Pharmacy, Faculty of Medicine, Department of Women's and Children's Health, Research group (Dept. of women´s and children´s health), Neuropediatrics/Paediatric oncology.
    Eccleston, Christopher
    Cochrane Pain, Palliative and Supportive Care Group, Pain Research Unit, Churchill Hospital, Oxford, UK.
    Non-steroidal anti-inflammatory drugs (NSAIDs) for cancer-related pain in children and adolescents2017In: Cochrane Database of Systematic Reviews, ISSN 1469-493X, E-ISSN 1469-493X, Vol. 7, article id CD012563Article, review/survey (Refereed)
    Abstract [en]

    BACKGROUND: Pain is a common feature of childhood and adolescence around the world, and for many young people, that pain is chronic. The World Health Organization (WHO) guidelines for pharmacological treatments for persisting pain in children acknowledge that pain in children is a major public health concern of high significance in most parts of the world. Views on children's pain have changed over time and relief of pain is now seen as important. In the past, pain was largely dismissed and was frequently left untreated, and it was assumed that children quickly forgot about painful experiences.We designed a suite of seven reviews in chronic non-cancer pain and cancer pain (looking at antidepressants, antiepileptic drugs, non-steroidal anti-inflammatory drugs, opioids, and paracetamol as priority areas) to review the evidence for children's pain using pharmacological interventions.As one of the leading causes of mortality and morbidity for children and adolescents in the world today, childhood cancer (and its associated pain) is a major health concern. Specific mortality and morbidity data relating to children are not currently identified. All childhood cancer rates are on the rise; for example, in the USA approximately 10,380 children aged under 15 years were expected to be diagnosed with cancer by the end of 2016. However, with survival rates also increasing, over 80% of paediatric cancer patients are expected to survive for five years or more, thus identifying the need to address pain management in this population.Cancer pain in infants, children, and adolescents is primarily nociceptive pain with negative long term effects. Cancer-related pain is generally caused directly by the tumour itself such as compressing on the nerve or inflammation of the organs. Cancer-related pain generally occurs as a result of perioperative procedures, nerve damage caused by radiation or chemotherapy treatments, or mucositis. However, this review focused on pain caused directly by the tumour itself such as nerve infiltration, external nerve compression, and other inflammatory events.Non-steroidal anti-inflammatory drugs (NSAIDs) are used to treat pain, reduce fever, and for their anti-inflammatory properties. They are commonly used within paediatric pain management. NSAIDs are currently licensed for use in western countries, however not approved for infants aged under three months. Primary adverse effects include gastrointestinal issues and possible renal impairment with long term use. Other adverse effects in children include diarrhoea, headache, nausea, constipation, rash, dizziness, and abdominal pain.

    OBJECTIVES: To assess the analgesic efficacy, and adverse events, of non-steroidal anti-inflammatory drugs (NSAIDs) used to treat cancer-related pain in children and adolescents aged from birth and 17 years, in any setting.

    SEARCH METHODS: We searched the Cochrane Central Register of Controlled Trials (CENTRAL) via the Cochrane Register of Studies Online, MEDLINE via Ovid, and Embase via Ovid from inception to 21 February 2017. We also searched the reference lists of retrieved studies and reviews, and searched online clinical trial registries.

    SELECTION CRITERIA: Randomised, double-blind trials of any dose, and any route, treating cancer-related pain in children and adolescents, comparing NSAIDs with placebo or an active comparator.

    DATA COLLECTION AND ANALYSIS: Two review authors independently assessed studies for eligibility. We planned to use dichotomous data to calculate risk ratio and number needed to treat for one additional event, using standard methods. We assessed GRADE (Grading of Recommendations Assessment, Development and Evaluation) and planned to create a 'Summary of findings' table.

    MAIN RESULTS: No studies were eligible for inclusion in this review (very low quality evidence). We downgraded the quality of evidence by three levels due to the lack of data reported for any outcome.

    AUTHORS' CONCLUSIONS: There is no evidence from randomised controlled trials that non-steroidal anti-inflammatory drugs (NSAIDs) reduce cancer-related pain in children and adolescents. This means that no reliance or conclusions can be made about efficacy or harm in the use of NSAIDs to treat chronic cancer-related pain in children and adolescents.

  • 14.
    Ehrstedt, Christoffer
    et al.
    Uppsala University, Disciplinary Domain of Medicine and Pharmacy, Faculty of Medicine, Department of Women's and Children's Health, Research group (Dept. of women´s and children´s health), Neuropediatrics/Paediatric oncology.
    Ahlsten, Gunnar
    Uppsala University, Disciplinary Domain of Medicine and Pharmacy, Faculty of Medicine, Department of Women's and Children's Health, Research group (Dept. of women´s and children´s health), Neuropediatrics/Paediatric oncology.
    Strömberg, Bo
    Uppsala University, Disciplinary Domain of Medicine and Pharmacy, Faculty of Medicine, Department of Women's and Children's Health, Research group (Dept. of women´s and children´s health), Neuropediatrics/Paediatric oncology.
    Lindskog, C
    Uppsala University, Disciplinary Domain of Medicine and Pharmacy, Faculty of Medicine, Department of Immunology, Genetics and Pathology, Clinical and experimental pathology.
    Casar-Borata, Olivera
    Uppsala University, Disciplinary Domain of Medicine and Pharmacy, Faculty of Medicine, Department of Immunology, Genetics and Pathology, Clinical and experimental pathology.
    Somatostatin receptor expression, mTOR pathway activation and BRAFV600E mutational status in glioneuronal tumours of childhoodManuscript (preprint) (Other academic)
  • 15.
    Ehrstedt, Christoffer
    et al.
    Uppsala University, Disciplinary Domain of Medicine and Pharmacy, Faculty of Medicine, Department of Women's and Children's Health.
    Ahlsten, Gunnar
    Uppsala University, Disciplinary Domain of Medicine and Pharmacy, Faculty of Medicine, Department of Women's and Children's Health, Research group (Dept. of women´s and children´s health), Neuropediatrics/Paediatric oncology.
    Strömberg, Bo
    Uppsala University, Disciplinary Domain of Medicine and Pharmacy, Faculty of Medicine, Department of Women's and Children's Health, Research group (Dept. of women´s and children´s health), Neuropediatrics/Paediatric oncology.
    Lindskog, Cecilia
    Uppsala University, Science for Life Laboratory, SciLifeLab. Uppsala University, Disciplinary Domain of Medicine and Pharmacy, Faculty of Medicine, Department of Immunology, Genetics and Pathology, Clinical and experimental pathology.
    Casar Borota, Olivera
    Uppsala University, Science for Life Laboratory, SciLifeLab. Uppsala University, Disciplinary Domain of Medicine and Pharmacy, Faculty of Medicine, Department of Immunology, Genetics and Pathology, Clinical and experimental pathology.
    Somatostatin receptor expression and mTOR pathway activation in glioneuronal tumours of childhoodManuscript (preprint) (Other academic)
  • 16.
    Ehrstedt, Christoffer
    et al.
    Uppsala University, Disciplinary Domain of Medicine and Pharmacy, Faculty of Medicine, Department of Women's and Children's Health, Research group (Dept. of women´s and children´s health), Neuropediatrics/Paediatric oncology.
    Canto Moreira, Nuno
    Uppsala University, Disciplinary Domain of Medicine and Pharmacy, Faculty of Medicine, Department of Surgical Sciences, Radiology.
    Casar Borota, Olivera
    Uppsala University, Disciplinary Domain of Medicine and Pharmacy, Faculty of Medicine, Department of Immunology, Genetics and Pathology, Clinical and experimental pathology.
    Strömberg, Bo
    Uppsala University, Disciplinary Domain of Medicine and Pharmacy, Faculty of Medicine, Department of Women's and Children's Health, Research group (Dept. of women´s and children´s health), Neuropediatrics/Paediatric oncology.
    Ahlsten, Gunnar
    Uppsala University, Disciplinary Domain of Medicine and Pharmacy, Faculty of Medicine, Department of Women's and Children's Health, Research group (Dept. of women´s and children´s health), Neuropediatrics/Paediatric oncology.
    Glioneuronal tumors in childhood - Before and after surgery. A long-term follow-up study2017In: Epilepsy & Behavior, ISSN 1525-5050, E-ISSN 1525-5069, Vol. 72, p. 82-88Article in journal (Refereed)
    Abstract [en]

    Aim: To give a detailed description of the long-term outcome of a cohort of children with glioneuronal tumors regarding pre-and postsurgical factors, including "dual" and "double" pathology, seizure freedom, and psychosocial outcome.

    Methods: During a fifteen-year period (1995-2009), all patients (age 0-17.99 years) with a glioneuronal brain tumor diagnosed and treated at Uppsala University Children's Hospital were identified from the National Brain Tumor Registry and the National Epilepsy Surgery Registry. Hospital medical records were reviewed and neuroradiological and neuropathological findings were re-evaluated. A cross-sectional long-term follow-up prospective evaluation, including an interview, neurologic examination, and electroencephalogram, was accomplished in patients accepting participants in the study.

    Results: A total of 25 out of 28 (89%) eligible patientswere included. The M: F ratiowas 1.5: 1. Mean follow-up time after surgery was 12.1 years (range 5.0-19.3). Twenty patients were adults (N18 years) at follow-up. Seizure freedomwas achieved in 64%. Gross total resection (GTR) was the only preoperative factor significantly correlating to seizure freedom (p= 0.027). Thirty-eight percent were at some time postoperatively admitted for a psychiatric evaluation. There was a trend towards both higher educational level and employment status in adults who became seizure free.

    Conclusion: Long-termoutcome is good regarding seizure freedom if GTR can be achieved, but late seizure recurrence can occur. "Dual" and "double" pathology is uncommon and does not influence seizure outcome. Obtaining seizure freedomseems to be important for psychosocial outcome, but there is a risk for psychiatric comorbidities and long-term follow-up by a multi-professional team is advisable.

  • 17.
    Ehrstedt, Christoffer
    et al.
    Uppsala University, Disciplinary Domain of Medicine and Pharmacy, Faculty of Medicine, Department of Women's and Children's Health, Research group (Dept. of women´s and children´s health), Neuropediatrics/Paediatric oncology.
    Rydell, Ann-Margret
    Uppsala University, Disciplinary Domain of Humanities and Social Sciences, Faculty of Social Sciences, Department of Psychology.
    Hallsten, Marina Gabert
    Uppsala University, Disciplinary Domain of Medicine and Pharmacy, Faculty of Medicine, Department of Women's and Children's Health, Research group (Dept. of women´s and children´s health), Neuropediatrics/Paediatric oncology.
    Strömberg, Bo
    Uppsala University, Disciplinary Domain of Medicine and Pharmacy, Faculty of Medicine, Department of Women's and Children's Health, Research group (Dept. of women´s and children´s health), Neuropediatrics/Paediatric oncology.
    Ahlsten, Gunnar
    Uppsala University, Disciplinary Domain of Medicine and Pharmacy, Faculty of Medicine, Department of Women's and Children's Health, Research group (Dept. of women´s and children´s health), Neuropediatrics/Paediatric oncology.
    Cognition, Health-Related Quality Of Life and Mood in Children and Young Adults Diagnosed with a Glioneuronal Tumor in Childhood2018In: Neuro-Oncology, ISSN 1522-8517, E-ISSN 1523-5866, Vol. 20, p. 161-161Article in journal (Other academic)
  • 18.
    Ehrstedt, Christoffer
    et al.
    Uppsala University, Disciplinary Domain of Medicine and Pharmacy, Faculty of Medicine, Department of Women's and Children's Health, Research group (Dept. of women´s and children´s health), Neuropediatrics/Paediatric oncology. Uppsala Univ, Childrens Hosp, Uppsala, Sweden.
    Rydell, Ann-Margret
    Uppsala University, Disciplinary Domain of Humanities and Social Sciences, Faculty of Social Sciences, Department of Psychology.
    Hallsten, Marina Gabert
    Uppsala University, Disciplinary Domain of Medicine and Pharmacy, Faculty of Medicine, Department of Women's and Children's Health, Research group (Dept. of women´s and children´s health), Neuropediatrics/Paediatric oncology. Uppsala Univ, Childrens Hosp, Uppsala, Sweden.
    Strömberg, Bo
    Uppsala University, Disciplinary Domain of Medicine and Pharmacy, Faculty of Medicine, Department of Women's and Children's Health, Research group (Dept. of women´s and children´s health), Neuropediatrics/Paediatric oncology. Uppsala Univ, Childrens Hosp, Uppsala, Sweden.
    Ahlsten, Gunnar
    Uppsala University, Disciplinary Domain of Medicine and Pharmacy, Faculty of Medicine, Department of Women's and Children's Health, Research group (Dept. of women´s and children´s health), Neuropediatrics/Paediatric oncology. Uppsala Univ, Childrens Hosp, Uppsala, Sweden.
    Cognition, health-related quality of life, and mood in children and young adults diagnosed with a glioneuronal tumor in childhood2018In: Epilepsy & Behavior, ISSN 1525-5050, E-ISSN 1525-5069, Vol. 83, p. 59-66Article in journal (Refereed)
    Abstract [en]

    Aims: The aim of this study was to investigate long-term cognitive outcome, health-related quality of life (HRQoL), and psychiatric symptoms in children and young adults diagnosed with a glioneuronal tumor in childhood.

    Methods: Twenty-eight children and adolescents (0-17.99 years) with a minimum postoperative follow-up time of five years were eligible for the study; four persons declined participation. A cross-sectional long-term follow-up evaluation was performed using the following study measures: Wechsler Intelligence Scale for Children (WISC-IV) or Wechsler Adult Intelligence Scale (WAIS-IV), Reys Complex Figure Test (RCFT), Short Form 36 version 2 (SF-36v2), Short Form 10 (SF-10), Quality of Life in Epilepsy 31 (QOLIE-31), Hospital Anxiety Depression Scale (HADS) or Beck Youth Inventory Scales (BYI), and Rosenberg Self-Esteem Scale. Historical WISC-III and RCFT data were used to compare cognitive longitudinal data.

    Results: Mean follow-up time after surgery was 12.1 years. Sixty-three percent (15/24) were seizure-free. Despite a successive postoperative gain in cognitive function, a significant reduction relative to norms was seen in the seizure-free group with respect to perceptual reasoning index (PRI), working memory index (WMI), and full-scale intelligence quotient (FSIQ). Seizure freedom resulted in acceptable HRQoL. Thirty-two percent and 16% exceeded the threshold level of possible anxiety and depression, respectively, despite seizure freedom.

    Conclusion: Although lower than in corresponding reference groups, cognitive outcome and HRQoL are good provided that seizure freedom or at least a low seizure severity can be achieved. There is a risk of elevated levels of psychiatric symptoms. Long-term clinical follow-up is advisable.

  • 19.
    Englund, Annika
    et al.
    Uppsala University, Disciplinary Domain of Medicine and Pharmacy, Faculty of Medicine, Department of Women's and Children's Health.
    Glimelius, Ingrid
    Uppsala University, Disciplinary Domain of Medicine and Pharmacy, Faculty of Medicine, Department of Immunology, Genetics and Pathology, Experimental and Clinical Oncology. Karolinska Inst, Dept Med Solna, Clin Epidemiol Unit, Stockholm.
    Rostgaard, Klaus
    Statens Serum Inst, Dept Epidemiol Res, Copenhagen.
    Smedby, Karin E
    Karolinska Inst, Dept Med Solna, Clin Epidemiol Unit, Stockholm; Karolinska Univ Hosp, Hematol Ctr, Stockholm.
    Eloranta, Sandra
    Karolinska Inst, Dept Med Solna, Clin Epidemiol Unit, Stockholm.
    Molin, Daniel
    Uppsala University, Disciplinary Domain of Medicine and Pharmacy, Faculty of Medicine, Department of Immunology, Genetics and Pathology, Experimental and Clinical Oncology.
    Kuusk, Thomas
    Uppsala University, Disciplinary Domain of Medicine and Pharmacy, Faculty of Medicine, Department of Women's and Children's Health.
    Brown, Peter de Nully
    Rigshosp, Dept Haematol, Copenhagen.
    Kamper, Peter
    Aarhus Univ Hosp, Dept Haematol, Aarhus C.
    Hjalgrim, Henrik
    Statens Serum Inst, Dept Epidemiol Res, Copenhagen; Rigshosp, Dept Haematol, Copenhagen.
    Ljungman, Gustaf
    Uppsala University, Disciplinary Domain of Medicine and Pharmacy, Faculty of Medicine, Department of Women's and Children's Health, Research group (Dept. of women´s and children´s health), Neuropediatrics/Paediatric oncology.
    Hjalgrim, Lisa Lyngsie
    Statens Serum Inst, Dept Epidemiol Res, Copenhagen; Rigshosp, Dept Paediat & Adolescent Med, Copenhagen.
    Hodgkin lymphoma in children, adolescents and young adults - a comparative study of clinical presentation and treatment outcome.2018In: Acta Oncologica, ISSN 0284-186X, E-ISSN 1651-226X, Vol. 57, no 2, p. 276-282Article in journal (Refereed)
    Abstract [en]

    Background: Hodgkin lymphoma (HL) treatment protocols for children, adolescents and young adults traditionally differ, but the biological and clinical justification for this remains uncertain.

    Material and methods: We compared age-dependent clinical presentation and treatment and outcome for 1072 classical HL patients 0–24 years diagnosed in Denmark (1990–2010) and Sweden (1992–2009) in pediatric (n = 315, Denmark <15 years, Sweden <18 years) or adult departments (n = 757). Distribution of clinical characteristics was assessed with Pearson’s chi2-test and Mantel–Haenszel trend test. The Kaplan–Meier method was used for survival analyses. Hazard ratios (HR) were used to compare the different treatment groups and calculated using Cox regression.

    Results: Children (0–9 years) less often presented with advanced disease than adolescents (10–17 years) and young adults (18–24 years) (stage IIB-IV: children 32% vs. adolescents 50%, and adults 55%; p < .005). No variation in overall survival (OS) was seen between pediatric and adult departments or by country. Danish pediatric patients received radiotherapy (36%) less frequently than Swedish pediatric patients (71%) (p < .0001). Ten-year event-free survival (EFS) was lower among Danish pediatric patients (0–14 years) (0.79; 95% confidence interval (CI) 0.70–0.86) than among Swedish pediatric patients (0–17 years) (0.88; 95% CI 0.83–0.92), HR (1.93; 95% CI 1.08–3.46). A similar pattern was seen between adult patients in the two countries: Denmark 10-year EFS 0.85 (95% CI 0.81–0.88), Sweden 0.88 (95% CI 0.84–0.91), adjusted HR 1.51 (95% CI 1.03–2.22).

    Conclusion: Adolescents and young adults shared similar clinical presentation suggesting a rationale of harmonized treatment for these groups. Both adult and pediatric protocols provided high OS with no significant difference between the departments. The less frequent use of radiotherapy in Danish pediatric patients corresponded to a lower EFS, but comparable OS in all groups confirmed effective rescue strategies for the relapsing patients.

  • 20.
    Engvall, Gunn
    et al.
    Uppsala University, Disciplinary Domain of Medicine and Pharmacy, Faculty of Medicine, Department of Women's and Children's Health, Research group (Dept. of women´s and children´s health), Neuropediatrics/Paediatric oncology.
    Lindh, Viveca
    Department of Nursing, Umea University, Umea, Sweden.
    Mullaney, Tara
    Veryday, Stockholm, Sweden.
    Nyholm, Tufve
    Department of Radiation Sciences, Umea University, Umea, Sweden.
    Lindh, Jack
    Department of Radiation Sciences, Umea University, Umea, Sweden.
    Ångström-Brännström, Charlotte
    Department of Nursing, Umea University, Umea, Sweden.
    Children's experiences and responses towards an intervention for psychological preparation for radiotherapy.2018In: Radiation Oncology, ISSN 1748-717X, E-ISSN 1748-717X, Vol. 13, article id 9Article in journal (Refereed)
    Abstract [en]

    BACKGROUND: Children can experience distress when undergoing radiotherapy as a reaction to being scared of and unfamiliar with the procedure. The aim was to evaluate children's experiences and responses towards an intervention for psychological preparation for radiotherapy.

    METHODS: A case control design with qualitative content analysis of semi-structured interviews and statistical analysis of anxiety ratings were used for evaluating a strategy for psychological preparation and distraction. Fifty-seven children aged 2 to 18 years and their parents participated - 30 children in the baseline group and 27 in the intervention group. Child interviews were performed and the child and their parents rated the child's anxiety.

    RESULTS: The intervention was most appropriate for the younger children, who enjoyed the digital story, the stuffed animal and training with their parents. There were some technical problems and the digital story was not detailed enough to fit exactly with various cancer diagnoses. Children described suggestions for improvement of the intervention. The ratings of the child's anxiety during radiation treatment showed no differences between the baseline group and the intervention group.

    CONCLUSIONS: The children of all the age groups experienced their interventions as positive. The strength of the intervention was that it encouraged interaction within the family and provided an opportunity for siblings and peers to take part in what the child was going through. Future research on children's experiences to interventions should be encouraged. The intervention and the technical solutions could improve by further development.

  • 21.
    Espersen, Anne Dorte Lerche
    et al.
    Aarhus Univ Hosp Skejby, Dept Pediat, Aarhus, Denmark..
    Noren-Nystrom, Ulrika
    Umea Univ Hosp, Dept Pediat, Umea, Sweden..
    Abrahamsson, Jonas
    Queen Silvia Childrens Hosp, Dept Pediat, Inst Clin Sci, Gothenburg, Sweden..
    Ha, Shau-Yin
    Queen Mary Hosp, Dept Pediat, Hong Kong, Hong Kong, Peoples R China.;HKPHOSG, Hong Kong, Hong Kong, Peoples R China..
    Pronk, Cornelis Jan
    Univ Hosp, Dept Pediat, Lund, Sweden..
    Jahnukainen, Kirsi
    Univ Helsinki, Childrens Hosp, Helsinki, Finland.;Univ Helsinki, Cent Hosp, Helsinki, Finland..
    Jonsson, Olafur G.
    Landspitalinn, Dept Pediat, Reykjavik, Iceland..
    Lausen, Birgitte
    Univ Copenhagen, Rigshosp, Dept Pediat & Adolescent Med, Copenhagen, Denmark..
    Palle, Josefine
    Uppsala University, Disciplinary Domain of Medicine and Pharmacy, Faculty of Medicine, Department of Women's and Children's Health, Research group (Dept. of women´s and children´s health), Neuropediatrics/Paediatric oncology.
    Zeller, Bernward
    Oslo Univ Hosp, Div Pediat & Adolescent Med, Oslo, Norway..
    Palmqvist, Lars
    Univ Gothenburg, Sahlgrenska Acad, Inst Biomed, Dept Clin Chem & Transfus Med, Gothenburg, Sweden..
    Hasle, Henrik
    Aarhus Univ Hosp Skejby, Dept Pediat, Aarhus, Denmark..
    Acute myeloid leukemia (AML) with t(7;12)(q36;p13) is associated with infancy and trisomy 19: Data from Nordic Society for Pediatric Hematology and Oncology (NOPHO-AML) and review of the literature2018In: Genes, Chromosomes and Cancer, ISSN 1045-2257, E-ISSN 1098-2264, Vol. 57, no 7, p. 359-365Article, review/survey (Refereed)
    Abstract [en]

    The t(7;12)(q36;p13) (MNX1/ETV6) is not included in the WHO classification but has been described in up to 30% of acute myeloid leukemia (AML) in children <2 years and associated with a poor prognosis. We present the clinical and cytogenetics characteristics of AML cases with t(7;12)(p36;p13). A literature review identified 35 patients with this translocation, published between 2000 and 2015. Outcome data were available in 22 cases. The NOPHO-AML (Nordic Society for Pediatric Hematology and Oncology) database contained 651 patients with AML from 1993 to 2014 and seven (1.1%) had the translocation. The t(7;12) was only present in patients <2 years of age (median age 6 months) but none was diagnosed as newborn. These patients constituted 4.3% of the patients <2 years of age. There was a strong association with trisomy 19 (literature: 86%, NOPHO: 100%) and +8 (literature: 19%, NOPHO: 14%). Seventeen of 22 patients from the literature with t(7;12) and four of seven patients from the NOPHO database suffered from relapse. The patients with t(7;12) had a 3-year event free survival of 24% (literature) vs. 43% (NOPHO) and a 3-year overall survival of 42% (literature) vs. 100% (NOPHO). None of the NOPHO patients was treated with hematopoietic stem cell transplantation (HSCT) in first complete remission. Relapse was frequent but the salvage rate using HSCT was high. We conclude that t(7;12)(q36;13) is a unique subgroup of childhood AML with presentation before 2 years of age with most cases being associated with +19.

  • 22.
    Georgantzi, Kleopatra
    Uppsala University, Disciplinary Domain of Medicine and Pharmacy, Faculty of Medicine, Department of Women's and Children's Health, Research group (Dept. of women´s and children´s health), Neuropediatrics/Paediatric oncology.
    On the Diagnostics of Neuroblastoma: Clinical and Experimental Studies2018Doctoral thesis, comprehensive summary (Other academic)
    Abstract [en]

    Neuroblastoma (NB) is one of the most common childhood cancers. Patients with low stage tumor have high survival rate, while those with advanced stage and/or unfavorable molecular biology have poor prognosis. A correct histopathological diagnosis, clinical stage, and identified genetic aberrations are crucial for treatment stratification according to current protocol. The tumor sample is obtained either by fine needle aspiration, cutting needle biopsy or open biopsy. NB exhibits neuroendocrine differentiation by showing immunoreactivity for chromogranin A (CgA), synaptophysin (syn), and neuron specific enolase (NSE) and 90% of the patients have increased levels of urine catecholamine metabolites.

    Experimental and clinical NB tumor samples were immunostained for somatostatin receptors (SSTRs) 1-5, somatostatin and CgA. Clinical tumor samples were also immunostained for syn, synaptic vesicle protein 2 (SV2), and vesicle monoamine transporter 1 (VMAT1) and 2 (VMAT 2). Blood samples from 92 patients were analyzed for level of CgA, NSE, and chromogranin B and compared with control groups. The urinary excretion of catecholamine metabolites was analyzed in samples collected at diagnosis. Clinical and laboratory data were extracted from patient records, including information on the diagnostic accuracy of ultrasound guided cutting needle biopsies (UCNB) and potential complications.

    We found that NB expressed the different SSTRs and that receptor 2 was the most frequently expressed before chemotherapy. Furthermore, NB tumors showed immunoreactivity for SV2, VMAT 1 and VMAT2 alongside CgA and syn. The immunoreactivity of SV2 was comparable to CgA and superior to syn. Patients with NB had higher blood concentrations of CgA and NSE compared with controls. Patients with advanced stage disease, MYCN amplification and 1 p deletion had higher concentrations of both CgA and NSE while only NSE was correlated to outcome with higher concentrations in the deceased patients.

    A high urinary excretion of homovanillic acid and dopamine were correlated to inferior outcome. UCNB were found to be safe and may provide all necessary diagnostic requirements for adequate therapy stratification according to current treatment protocols.

    List of papers
    1. Differentiated expression of somatostatin receptor subtypes in experimental models and clinical neuroblastoma
    Open this publication in new window or tab >>Differentiated expression of somatostatin receptor subtypes in experimental models and clinical neuroblastoma
    Show others...
    2011 (English)In: Pediatric Blood & Cancer, ISSN 1545-5009, E-ISSN 1545-5017, Vol. 56, no 4, p. 584-589Article in journal (Refereed) Published
    Abstract [en]

    BACKGROUND:

    Neuroblastoma (NB) is a solid tumor of childhood originating from the adrenal medulla or sympathetic nervous system. Somatostatin (SS) is an important regulator of neural and neuroendocrine function, its actions being mediated through five specific membrane receptors. The aim of this study was to investigate the expression of the different somatostatin receptors (SSTRs) in NB tumor cells that may form targets for future therapeutic development.

    PROCEDURE:

    Tumor specimens from 11 children with stage II-IV disease were collected before and/or after chemotherapy. Experimental tumors derived from five human NB cell lines were grown subcutaneously in nude mice. Expression of SSRTs, the neuroendocrine marker chromogranin A (CgA) and SS was detected by immunohistochemistry using specific antibodies.

    RESULTS:

    SSTR2 was detected in 90%, SSTR5 in 79%, SSTR1 in 74%, SSTR3 in 68% whereas SSTR4 was expressed in 21% of the clinical tumors. The experimental tumors expressed SSTRs in a high but variable frequency. All clinical tumors showed immunoreactivity for CgA but not for SS.

    CONCLUSION:

    The frequent expression of SSTRs indicates that treatment with unlabeled or radiolabeled SS analogs should be further explored in NB.

    National Category
    Medical and Health Sciences
    Identifiers
    urn:nbn:se:uu:diva-140509 (URN)10.1002/pbc.22913 (DOI)000287986700013 ()21120894 (PubMedID)
    Available from: 2011-01-05 Created: 2011-01-05 Last updated: 2018-10-31
    2. Chromogranin A and neuron-specific enolase in neuroblastoma: Correlation to stage and prognostic factors.
    Open this publication in new window or tab >>Chromogranin A and neuron-specific enolase in neuroblastoma: Correlation to stage and prognostic factors.
    Show others...
    2018 (English)In: Pediatric Hematology & Oncology, ISSN 0888-0018, E-ISSN 1521-0669, Vol. 35, no 2, p. 156-165Article in journal (Refereed) Published
    Abstract [en]

    Chromogranin A (CgA) and neuron specific enolase (NSE) are important markers in adult neuroendocrine tumors (NET). Neuroblastoma (NB) has certain neuroendocrine properties. The aim of this study was to correlate blood concentrations of CgA, chromogranin B (CgB), and NSE to prognostic factors and outcome in children with NB. Blood samples from 92 patients with NB, 12 patients with benign ganglioneuroma (GN), 21 patients with non-NB solid tumors, 10 patients with acute leukemias, and 69 healthy children, were analyzed. CgA concentrations were higher in neonates vs. children older than one month in the control group (p < 0.0001), and in neonates with NB vs. the control group (p < 0.01). CgA and NSE concentrations were higher in patients with stages 3 and 4 disease (p < 0.05 and p < 0.05), in patients having tumors with amplification of MYCN (p < 0.05 and p < 0.001), or chromosome 1 p deletion (p < 0.05 and p < 0.05). NSE correlated to the tumor size at diagnosis (p < 0.001) and to tumor related death (p < 0.01) in NB. CgA and NSE concentrations were elevated in patients with NB and especially in those with advanced disease. Both CgA and NSE correlated to genetic markers, while only NSE correlated to primary tumor size and outcome in NB. We found that CgA and NSE are clinically valuable tumor markers in NB and they merit prospective clinical evaluations as such.

    Keywords
    Chromogranin A, neuroblastoma, neuron-specific enolase, prognosis, tumor markers
    National Category
    Cancer and Oncology
    Identifiers
    urn:nbn:se:uu:diva-360970 (URN)10.1080/08880018.2018.1464087 (DOI)000446356300007 ()29737901 (PubMedID)
    Funder
    Swedish Childhood Cancer Foundation
    Available from: 2018-09-20 Created: 2018-09-20 Last updated: 2018-12-07Bibliographically approved
    3. Synaptic Vesicle Protein 2 and Vesicular Monoamine Transporter 1 and 2 are expressed in Neuroblastoma
    Open this publication in new window or tab >>Synaptic Vesicle Protein 2 and Vesicular Monoamine Transporter 1 and 2 are expressed in Neuroblastoma
    Show others...
    (English)Manuscript (preprint) (Other academic)
    Keywords
    neuroblastoma, neuroendocrine, immunohistochemistry, urine-dopamine, urine-HVA, urine-VMA
    National Category
    Medical and Health Sciences
    Identifiers
    urn:nbn:se:uu:diva-364674 (URN)
    Available from: 2018-10-31 Created: 2018-10-31 Last updated: 2018-10-31
    4. Diagnostic Ultrasound-Guided Cutting Needle Biopsies in Neuroblastoma: a safe and efficient procedure
    Open this publication in new window or tab >>Diagnostic Ultrasound-Guided Cutting Needle Biopsies in Neuroblastoma: a safe and efficient procedure
    Show others...
    (English)Manuscript (preprint) (Other academic)
    Keywords
    ultrasound, biopsy, neuroblastoma, complications, child
    National Category
    Medical and Health Sciences
    Identifiers
    urn:nbn:se:uu:diva-364673 (URN)
    Available from: 2018-10-31 Created: 2018-10-31 Last updated: 2018-10-31
  • 23.
    Georgantzi, Kleopatra
    et al.
    Uppsala University, Disciplinary Domain of Medicine and Pharmacy, Faculty of Medicine, Department of Women's and Children's Health, Research group (Dept. of women´s and children´s health), Neuropediatrics/Paediatric oncology.
    Sköldenberg, Erik G
    Uppsala University, Disciplinary Domain of Medicine and Pharmacy, Faculty of Medicine, Department of Women's and Children's Health, Research group (Dept. of women´s and children´s health), Pediatric Surgery.
    Janson, Eva Tiensuu
    Uppsala University, Disciplinary Domain of Medicine and Pharmacy, Faculty of Medicine, Department of Medical Sciences.
    Jakobson, Åke
    Uppsala University, Disciplinary Domain of Medicine and Pharmacy, Faculty of Medicine, Department of Women's and Children's Health, Research group (Dept. of women´s and children´s health), Neuropediatrics/Paediatric oncology.
    Christofferson, Rolf H B
    Uppsala University, Disciplinary Domain of Medicine and Pharmacy, Faculty of Medicine, Department of Women's and Children's Health, Research group (Dept. of women´s and children´s health), Pediatric Surgery.
    Diagnostic ultrasound-guided cutting needle biopsies in neuroblastoma: A safe and efficient procedure.2019In: Journal of Pediatric Surgery, ISSN 0022-3468, E-ISSN 1531-5037, article id S0022-3468(18)30888-1Article in journal (Refereed)
    Abstract [en]

    BACKGROUND: Neuroblastoma (NB) is the most common extracranial solid tumor of childhood and accounts for 15% of deaths in pediatric oncology. Apart from the clinical stage at diagnosis, molecular factors are important for the characterization of the tumor and for decision on adequate treatment. Pretreatment diagnosis and molecular profiling are based on analysis of a tumor sample, obtained either by fine needle aspiration cytology (FNAC), cutting needle biopsy or open surgical biopsy. The method used depends on local tradition and routines. Ultrasound-guided cutting needle biopsy (UCNB) has been used at the Uppsala University Hospital since 1988 for diagnosis of pediatric solid tumors.

    PROCEDURES: Medical records of 29 patients with NB who underwent pretreatment, diagnostic, ultrasound-guided needle biopsy were reviewed. Information extracted from the patients' records included: age at diagnosis, gender, tumor site, clinical stage, molecular profiling made on biopsies (e.g. MYCN status, ploidy and chromosomal aberrations), and UCNB complications (i.e. bleeding, pain, or anesthesiologic complications).

    RESULTS: A total of 34 UCNBs were performed in the 29 patients. Repeated biopsies were done in three patients. UCNB was diagnostic in 90% (26/29). A complete molecular profiling was obtained in all UCNBs after 2008. Two patients (7%) developed a significant bleeding and two (7%) needed analgesics following UCNB. Neither infection nor tumor growth in the needle tract was observed. There were no anesthesiologic complications.

    CONCLUSIONS: UCNB is reasonably safe in patients with NB and usually gives a sufficient amount of tumor tissue for a histological diagnosis, molecular profiling, and biobank storage.

  • 24.
    Georgantzi, Kleopatra
    et al.
    Uppsala University, Disciplinary Domain of Medicine and Pharmacy, Faculty of Medicine, Department of Women's and Children's Health, Research group (Dept. of women´s and children´s health), Neuropediatrics/Paediatric oncology.
    Sköldenberg, Erik G
    Uppsala University, Disciplinary Domain of Medicine and Pharmacy, Faculty of Medicine, Department of Women's and Children's Health, Research group (Dept. of women´s and children´s health), Pediatric Surgery.
    Tiensuu Janson, Eva
    Uppsala University, Disciplinary Domain of Medicine and Pharmacy, Faculty of Medicine, Department of Medical Sciences, Endocrin Oncology.
    Jakobson, Åke
    Uppsala University, Disciplinary Domain of Medicine and Pharmacy, Faculty of Medicine, Department of Women's and Children's Health, Research group (Dept. of women´s and children´s health), Neuropediatrics/Paediatric oncology.
    Christofferson, Rolf H B
    Uppsala University, Disciplinary Domain of Medicine and Pharmacy, Faculty of Medicine, Department of Women's and Children's Health, Research group (Dept. of women´s and children´s health), Pediatric Surgery.
    Diagnostic Ultrasound-Guided Cutting Needle Biopsies in Neuroblastoma: a safe and efficient procedureManuscript (preprint) (Other academic)
  • 25.
    Georgantzi, Kleopatra
    et al.
    Uppsala University, Disciplinary Domain of Medicine and Pharmacy, Faculty of Medicine, Department of Women's and Children's Health, Research group (Dept. of women´s and children´s health), Neuropediatrics/Paediatric oncology. University Children's Hospital, Uppsala, Sweden.
    Sköldenberg, Erik
    Uppsala University, Disciplinary Domain of Medicine and Pharmacy, Faculty of Medicine, Department of Women's and Children's Health, Research group (Dept. of women´s and children´s health), Pediatric Surgery. University Children's Hospital, Uppsala, Sweden.
    Stridsberg, Mats
    Uppsala University, Disciplinary Domain of Medicine and Pharmacy, Faculty of Medicine, Department of Medical Sciences, Biochemical endocrinology. University Hospital, Uppsala, Sweden.
    Kogner, Per
    Department of Women´s and Children´s Health, Karolinska University Hospital, Solna, Stockholm, Sweden.
    Jakobson, Åke
    Uppsala University, Disciplinary Domain of Medicine and Pharmacy, Faculty of Medicine, Department of Women's and Children's Health, Research group (Dept. of women´s and children´s health), Neuropediatrics/Paediatric oncology. University Children's Hospital, Uppsala, Sweden.
    Tiensuu Janson, Eva
    Uppsala University, Disciplinary Domain of Medicine and Pharmacy, Faculty of Medicine, Department of Medical Sciences, Endocrin Oncology.
    Christofferson, Rolf. H.B.
    Uppsala University, Disciplinary Domain of Medicine and Pharmacy, Faculty of Medicine, Department of Women's and Children's Health, Research group (Dept. of women´s and children´s health), Pediatric Surgery. University Children's Hospital, Uppsala, Sweden.
    Chromogranin A and neuron-specific enolase in neuroblastoma: Correlation to stage and prognostic factors.2018In: Pediatric Hematology & Oncology, ISSN 0888-0018, E-ISSN 1521-0669, Vol. 35, no 2, p. 156-165Article in journal (Refereed)
    Abstract [en]

    Chromogranin A (CgA) and neuron specific enolase (NSE) are important markers in adult neuroendocrine tumors (NET). Neuroblastoma (NB) has certain neuroendocrine properties. The aim of this study was to correlate blood concentrations of CgA, chromogranin B (CgB), and NSE to prognostic factors and outcome in children with NB. Blood samples from 92 patients with NB, 12 patients with benign ganglioneuroma (GN), 21 patients with non-NB solid tumors, 10 patients with acute leukemias, and 69 healthy children, were analyzed. CgA concentrations were higher in neonates vs. children older than one month in the control group (p < 0.0001), and in neonates with NB vs. the control group (p < 0.01). CgA and NSE concentrations were higher in patients with stages 3 and 4 disease (p < 0.05 and p < 0.05), in patients having tumors with amplification of MYCN (p < 0.05 and p < 0.001), or chromosome 1 p deletion (p < 0.05 and p < 0.05). NSE correlated to the tumor size at diagnosis (p < 0.001) and to tumor related death (p < 0.01) in NB. CgA and NSE concentrations were elevated in patients with NB and especially in those with advanced disease. Both CgA and NSE correlated to genetic markers, while only NSE correlated to primary tumor size and outcome in NB. We found that CgA and NSE are clinically valuable tumor markers in NB and they merit prospective clinical evaluations as such.

  • 26.
    Georgantzi, Kleopatra
    et al.
    Uppsala University, Disciplinary Domain of Medicine and Pharmacy, Faculty of Medicine, Department of Women's and Children's Health, Research group (Dept. of women´s and children´s health), Neuropediatrics/Paediatric oncology.
    Tsolakis, Apostolos V
    Uppsala University, Disciplinary Domain of Medicine and Pharmacy, Faculty of Medicine, Department of Medical Sciences.
    Jakobson, Åke
    Uppsala University, Disciplinary Domain of Medicine and Pharmacy, Faculty of Medicine, Department of Women's and Children's Health, Research group (Dept. of women´s and children´s health), Neuropediatrics/Paediatric oncology.
    Christofferson, Rolf H B
    Uppsala University, Disciplinary Domain of Medicine and Pharmacy, Faculty of Medicine, Department of Women's and Children's Health, Research group (Dept. of women´s and children´s health), Pediatric Surgery.
    Tiensuu Janson, Eva
    Uppsala University, Disciplinary Domain of Medicine and Pharmacy, Faculty of Medicine, Department of Medical Sciences.
    Grimelius, Lars
    Uppsala University, Disciplinary Domain of Medicine and Pharmacy, Faculty of Medicine, Department of Immunology, Genetics and Pathology.
    Synaptic Vesicle Protein 2 and Vesicular Monoamine Transporter 1 and 2 are expressed in NeuroblastomaManuscript (preprint) (Other academic)
  • 27.
    Gustafsson, Britt
    et al.
    Uppsala University, Disciplinary Domain of Medicine and Pharmacy, Faculty of Medicine, Department of Women's and Children's Health, Research group (Dept. of women´s and children´s health), Neuropediatrics/Paediatric oncology. Karolinska Inst, Dept Clin Intervent & Technol CLINTEC, Stockholm, Sweden.;Childrens Univ Hosp, Dept Womens & Childrens Hlth, Pediat Oncol, Uppsala, Sweden..
    Frisk, Per
    Uppsala University, Disciplinary Domain of Medicine and Pharmacy, Faculty of Medicine, Department of Women's and Children's Health, Research group (Dept. of women´s and children´s health), Neuropediatrics/Paediatric oncology. Childrens Univ Hosp, Dept Womens & Childrens Hlth, Pediat Oncol, Uppsala, Sweden.
    Szakos, Attilla
    Karolinska Univ Hosp, Dept Pathol & Cytol, Stockholm, Sweden..
    Sadeghi, Behnam
    Karolinska Inst, Dept Lab Med, Div Therapeut Immunol, Stockholm, Sweden..
    Ringden, Olle
    Karolinska Inst, Dept Lab Med, Div Therapeut Immunol, Stockholm, Sweden..
    Frost, B M
    Uppsala University, Disciplinary Domain of Medicine and Pharmacy, Faculty of Medicine, Department of Women's and Children's Health, Research group (Dept. of women´s and children´s health), Neuropediatrics/Paediatric oncology. Childrens Univ Hosp, Dept Womens & Childrens Hlth, Pediat Oncol, Uppsala, Sweden.
    Successful treatment with placenta-derived decidual stromal cells in a pediatric patient with life-threatening acute gastrointestinal graft-versus-host disease2017In: Pediatric Transplantation, ISSN 1397-3142, E-ISSN 1399-3046, Vol. 21, no 5, article id e12990Article in journal (Refereed)
    Abstract [en]

    Severe aGvHD is a life-threatening complication after allogeneic HSCT. The GI tract is considered to play a key role in aGvHD, where the disease process can start and is one of the major target organs. Here, we present a case of a one-year-old child with a life-threatening GI-aGvHD stage IV, post-HSCT, resistant to steroids and MMF for 4 weeks. He was successfully treated with placenta-derived DSC.

  • 28.
    Gustafsson, Britt M.
    et al.
    Karolinska Inst, CLINTEC, Dept Clin Sci Intervent & Technol, SE-14186 Stockholm, Sweden.
    Mattsson, Kristin
    Karolinska Inst, CLINTEC, Dept Clin Sci Intervent & Technol, SE-14186 Stockholm, Sweden.
    Bogdanovic, Gordana
    Karolinska Inst, Dept Microbiol Tumor & Cell Biol, Stockholm, Sweden;Karolinska Univ Hosp, Dept Clin Microbiol, Stockholm, Sweden.
    Leijonhufvud, Gustaf
    Uppsala University, Disciplinary Domain of Medicine and Pharmacy, Faculty of Medicine, Department of Women's and Children's Health, Research group (Dept. of women´s and children´s health), Neuropediatrics/Paediatric oncology.
    Honkaniemi, Emma
    Karolinska Inst, CLINTEC, Dept Clin Sci Intervent & Technol, SE-14186 Stockholm, Sweden;Karolinska Univ Hosp, Liljeholmen Child & Adolescent Med Facil, Dept Paediat, Stockholm, Sweden.
    Ramme, Kim
    Karolinska Inst, CLINTEC, Dept Clin Sci Intervent & Technol, SE-14186 Stockholm, Sweden;Karolinska Univ Hosp, Astrid Lindgrens Children s Hosp, Dept Pediat Hematol Immunol & Stem Cell Transplan, Stockholm, Sweden.
    Ford, Anthony M.
    Inst Canc Res, Div Mol Pathol, Ctr Evolut & Canc, London, England.
    Origins of STIL-TAL1 fusion genes in children who later developed paediatric T-cell acute lymphoblastic leukaemia: An investigation of neonatal blood spots2018In: Pediatric Blood & Cancer, ISSN 1545-5009, E-ISSN 1545-5017, Vol. 65, no 11, article id e27310Article in journal (Refereed)
    Abstract [en]

    SCL/TAL1 interrupting locus (STIL)-T-cell acute leukaemia (TAL1) fusion genes are present in approximately 11-27% of children with paediatric T-cell acute lymphoblastic leukaemia (T-ALL), but the developmental timing of the rearrangement is still unknown. To investigate whether the fusion gene can be detected in neonatal blood spots (NBSs) from paediatric patients diagnosed with T-cell ALL, we analysed DNA from 38 paediatric patients with T-ALL by nested polymerase chain reaction and electrophoresis. The STIL-TAL1 fusion gene was not detected in NBSs from any of the 38 patients with T-ALL, suggesting that STIL-TAL1 fusion genes are most probably postnatal events in paediatric T-ALL.

  • 29.
    Hemmingsson, Helena
    et al.
    Stockholm Univ, Dept Special Educ, S-11418 Stockholm, Sweden.;Linkoping Univ, Dept Social & Welf Studies, S-58183 Norrkoping, Sweden..
    Ahlsten, Gunnar
    Uppsala University, Disciplinary Domain of Medicine and Pharmacy, Faculty of Medicine, Department of Women's and Children's Health, Research group (Dept. of women´s and children´s health), Neuropediatrics/Paediatric oncology.
    Wandin, Helena
    Uppsala University, Disciplinary Domain of Medicine and Pharmacy, Faculty of Medicine, Department of Public Health and Caring Sciences, Research in Disability and Habilitation. Swedish Natl Ctr Rett Syndrome & Related Disorder, S-83223 Froson, Sweden.
    Rytterström, Patrik
    Linkoping Univ, Dept Social & Welf Studies, S-58183 Norrkoping, Sweden..
    Borgestig, Maria
    Linkoping Univ, Dept Social & Welf Studies, S-58183 Norrkoping, Sweden.;Orebro Univ, Fac Med & Hlth, Sch Hlth Sci, S-70281 Orebro, Sweden..
    Eye-Gaze Control Technology as Early Intervention for a Non-Verbal Young Child with High Spinal Cord Injury: A Case Report2018In: TECHNOLOGIES, ISSN 2227-7080, Vol. 6, no 1, article id 12Article in journal (Refereed)
    Abstract [en]

    Assistive technology (AT) can be used as early intervention in order to reduce activity limitations in play and communication. This longitudinal case study examines eye-gaze control technology as early intervention for a young child with high spinal cord injury without the ability to make sounds. The young child was followed by repeated measures concerning performance and communication from baseline at 9 months to 26 months, and finalized at 36 months by field observations in the home setting. The results showed eye-gaze performance and frequency of use of eye-gaze control technology increased over time. Goals set at 15 months concerning learning and using the AT; naming objects and interactions with family was successfully completed at 26 months. Communicative functions regarding obtaining objects and social interaction increased from unintentional actions to purposeful choices and interactions. At 36 months, the toddler was partly independent in eye gazing, used all activities provided, and made independent choices. In conclusion, the results show that a 9-month-old child with profound motor disabilities can benefit from eye-gaze control technology in order to gradually perform activities, socially interact with family members, and make choices.

  • 30.
    Hjartarson, Helgi Thor
    et al.
    Astrid Lindgren Childrens Hosp, Dept Neuropediat, Q302, S-17176 Stockholm, Sweden..
    Ehrstedt, Christoffer
    Uppsala University, Disciplinary Domain of Medicine and Pharmacy, Faculty of Medicine, Department of Women's and Children's Health, Research group (Dept. of women´s and children´s health), Neuropediatrics/Paediatric oncology.
    Tedroff, Kristina
    Astrid Lindgren Childrens Hosp, Dept Neuropediat, Q302, S-17176 Stockholm, Sweden.;Karolinska Inst, Dept Womens & Childrens Hlth, Stockholm, Sweden..
    Intrathecal baclofen treatment an option in X-linked adrenoleukodystrophy2018In: European journal of paediatric neurology, ISSN 1090-3798, E-ISSN 1532-2130, Vol. 22, no 1, p. 178-181Article in journal (Refereed)
    Abstract [en]

    Background: X-linked adrenoleukodystrophy (X-ALD) is a genetic peroxisomal disorder associated with tissue accumulation of very long chain fatty acids (VLCFAs). In approximately one third of affected males, this causes progressive and irreversible damage to the brain white matter. Progress is often rapid with upper motor neuron damage leading to severe spasticity and dystonia. The increased muscle tone is frequently difficult to alleviate with oral drugs. Here, we describe two patients with X-ALD who have received treatment with intrathecal baclofen pumps (ITB). Case study: Both boys had a rapidly progressive cerebral form of the disorder resulting, among other things, in escalating spasticity and dystonia causing severe pain, dramatically reducing their quality of life. Both were treated with a variety of oral medications without adequate relief. Both patients tolerated ITB surgery without complications and the positive clinical effects of treatment with ITB became clear in the following weeks and months, with significantly reduced muscle tone, less pain and better sleep. Moreover, general caretaking became easier. Conclusion: The treatment of spasticity and dystonia in these patients is difficult partly due to the relentless nature of this progressive disorder. In our two patients, ITB has been effective from both a symptomatic and palliative perspective. We recommend that such treatment be considered as an early option for increased muscle tone in boys with the cerebral form of X-ALD. (C) 2017 European Paediatric Neurology Society. Published by Elsevier Ltd. All rights reserved.

  • 31.
    Højfeldt, Sofie G.
    et al.
    Aarhus Univ Hosp, Child & Adolescent Hlth, Aarhus, Denmark.
    Wolthers, Benjamin O
    Univ Hosp Rigshosp, Dept Paediat & Adolescent Med, Copenhagen, Denmark.
    Tulstrup, Morten
    Univ Hosp Rigshosp, Dept Paediat & Adolescent Med, Copenhagen, Denmark.
    Abrahamsson, Jonas
    Univ Gothenburg, Sahlgrenska Acad, Inst Clin Sci, Dept Paediat, Gothenburg, Sweden.
    Gupta, Ramneek
    Tech Univ Denmark, Dept Bio & Hlth Informat, Lyngby, Denmark.
    Harila-Saari, Arja H.
    Uppsala University, Disciplinary Domain of Medicine and Pharmacy, Faculty of Medicine, Department of Women's and Children's Health, Research group (Dept. of women´s and children´s health), Neuropediatrics/Paediatric oncology.
    Heyman, Mats
    Karolinska Univ Hosp, Childhood Canc Res Unit, Dept Womens & Childrens Hlth, Stockholm, Sweden; Karolinska Inst, Stockholm, Sweden.
    Henriksen, Louise T.
    Aarhus Univ Hosp, Child & Adolescent Hlth, Aarhus, Denmark.
    Jónsson, Òlafur G.
    Landspitali Univ Hosp, Childrens Hosp, Reykjavik, Iceland.
    Lähteenmäki, Päivi M
    Turku Univ Hosp, Dept Paediat & Adolescent Med, Turku, Finland; Turku Univ, Turku, Finland.
    Lund, Bendik
    St Olavs Hosp, Dept Paediat, Trondheim, Norway.
    Pruunsild, Kaie
    Univ Childrens Hosp, Dept Oncol & Haematol, Tallinn, Estonia.
    Vaitkeviciene, Goda
    Vilnius Univ, Fac Med, Clin Childrens Dis, Vilnius, Lithuania.
    Schmiegelow, Kjeld
    Univ Hosp Rigshosp, Dept Paediat & Adolescent Med, Copenhagen, Denmark.
    Albertsen, Birgitte K
    Aarhus Univ Hosp, Child & Adolescent Hlth, Aarhus, Denmark.
    Genetic predisposition to PEG-asparaginase hypersensitivity in children treated according to NOPHO ALL20082019In: British Journal of Haematology, ISSN 0007-1048, E-ISSN 1365-2141, Vol. 184, no 3, p. 405-417Article in journal (Refereed)
    Abstract [en]

    Asparaginase is essential in childhood acute lymphoblastic leukaemia (ALL) treatment, however hypersensitivity reactions to pegylated asparaginase (PEG-asparaginase) hampers anti-neoplastic efficacy. Patients with PEG-asparaginase hypersensitivity have been shown to possess zero asparaginase enzyme activity. Using this measurement to define the phenotype, we investigated genetic predisposition to PEG-asparaginase hypersensitivity in a genome-wide association study (GWAS). From July 2008 to March 2016, 1494 children were treated on the Nordic Society of Paediatric Haematology and Oncology ALL2008 protocol. Cases were defined by clinical hypersensitivity and no enzyme activity, controls had enzyme activity ≥ 100 iu/l and no hypersensitivity symptoms. PEG-asparaginase hypersensitivity was reported in 13·8% (206/1494) of patients. Fifty-nine cases and 772 controls fulfilled GWAS inclusion criteria. The CNOT3 variant rs73062673 on 19q13.42, was associated with PEG-asparaginase allergy (P = 4·68 × 10-8 ). We further identified two signals on chromosome 6 in relation to HLA-DQA1 (P = 9·37 × 10-6 ) and TAP2 (P = 1·59 × 10-5 ). This study associated variants in CNOT3 and in the human leucocyte antigen (HLA) region with PEG-asparaginase hypersensitivity, suggesting that not only genetic variations in the HLA region, but also regulation of these genes are of importance in the biology of this toxicity. Furthermore, our study emphasizes the importance of using asparaginase enzyme activity measurements to identify PEG-asparaginase hypersensitivity.

  • 32.
    Ifversen, Marianne
    et al.
    Copenhagen Univ Hosp, Rigshosp, Dept Paediat & Adolescent Med, Blegdamsvej 9, DK-2100 Copenhagen, Denmark.
    Turkiewicz, Dominik
    Lund Univ, Lund, Sweden.
    Marquart, Hanne V.
    Rigshosp, Copenhagen Univ Hosp, Dept Clin Immunol, Tissue Typing Lab, Copenhagen, Denmark.
    Winiarski, Jacek
    Karolinska Univ Hosp, Astrid Lindgren Childrens Hosp & Clintec, Stockholm, Sweden.
    Buechner, Jochen
    Oslo Univ Hosp, Dept Paediat Haematol & Oncol, Oslo, Norway.
    Mellgren, Karin
    Queen Silvia Childrens Hosp, Dept Paediat, Inst Clin Sci, Gothenburg, Sweden.
    Arvidson, Johan
    Uppsala University, Disciplinary Domain of Medicine and Pharmacy, Faculty of Medicine, Department of Women's and Children's Health, Research group (Dept. of women´s and children´s health), Neuropediatrics/Paediatric oncology.
    Rascon, Jelena
    Vilnius Univ Hosp, Childrens Hosp, Ctr Paediat Oncol & Haematol, Vilnius, Lithuania.
    Korgvee, Lenne-Triin
    Tallin Childrens Hosp, Tallinn, Estonia.
    Madsen, Hans O.
    Rigshosp, Copenhagen Univ Hosp, Dept Clin Immunol, Tissue Typing Lab, Copenhagen, Denmark.
    Abrahamsson, Jonas
    Queen Silvia Childrens Hosp, Dept Paediat, Inst Clin Sci, Gothenburg, Sweden.
    Lund, Bendik
    St Olavs Univ Hosp Trondheim, Dept Paediat, Trondheim, Norway;NTNU, Dept Clin & Mol Med, Trondheim, Norway.
    Jonsson, Olafur G.
    Landspitali Univ Hosp, Reykjavik, Iceland.
    Heilmann, Carsten
    Copenhagen Univ Hosp, Rigshosp, Dept Paediat & Adolescent Med, Blegdamsvej 9, DK-2100 Copenhagen, Denmark.
    Heyman, Mats
    Karolinska Inst, Childhood Canc Res Unit, Astrid Lindgrens Childrens Hosp, Karolinska Univ Hosp, Stockholm, Sweden.
    Schmiegelow, Kjeld
    Copenhagen Univ Hosp, Rigshosp, Dept Paediat & Adolescent Med, Blegdamsvej 9, DK-2100 Copenhagen, Denmark;Univ Copenhagen, Inst Clin Med, Copenhagen, Denmark.
    Vettenranta, Kim
    Univ Helsinki, Dept Paediat, Helsinki, Finland.
    Low burden of minimal residual disease prior to transplantation in children with very high risk acute lymphoblastic leukaemia: The NOPHO ALL2008 experience2019In: British Journal of Haematology, ISSN 0007-1048, E-ISSN 1365-2141, Vol. 184, no 6, p. 982-993Article in journal (Refereed)
    Abstract [en]

    The population-based Nordic/Baltic acute lymphoblastic leukaemia (ALL) Nordic Society for Paediatric Haematology and Oncology (NOPHO) ALL2008 protocol combined minimal residual disease (MRD)-driven treatment stratification with very intense first line chemotherapy for patients with high risk ALL. Patients with MRD >= 5% at end of induction or >= 10(-3) at end of consolidation or following two high risk blocks were eligible for haematopoietic cell transplantation (HCT) in first remission. After at least three high risk blocks a total of 71 children received HCT, of which 46 had MRD >= 5% at end of induction. Ten patients stratified to HCT were not transplanted; 12 received HCT without protocol indication. Among 69 patients with evaluable pre-HCT MRD results, 22 were MRD-positive, one with MRD >= 10(-3). After a median follow-up of 5 center dot 5 years, the cumulative incidence of relapse was 23 center dot 5% (95% confidence interval [CI]: 10 center dot 5-47 center dot 7) for MRD-positive versus 5 center dot 1% (95% CI: 1 center dot 3-19 center dot 2), P = 0 center dot 02) for MRD-negative patients. MRD was the only variable significantly associated with relapse (hazard ratio 9 center dot 1, 95% CI: 1 center dot 6-51 center dot 0, P = 0 center dot 012). Non-relapse mortality did not differ between the two groups, resulting in disease-free survival of 85 center dot 6% (95% CI: 75 center dot 4-97 center dot 2) and 67 center dot 4% (95% CI: 50 center dot 2-90 center dot 5), respectively. In conclusion, NOPHO block treatment efficiently reduced residual leukaemia which, combined with modern transplant procedures, provided high survival rates, also among pre-HCT MRD-positive patients.

  • 33.
    Jackmann, Natalja
    et al.
    Uppsala University, Disciplinary Domain of Medicine and Pharmacy, Faculty of Medicine, Department of Women's and Children's Health, Research group (Dept. of women´s and children´s health), Neuropediatrics/Paediatric oncology.
    Harila-Saari, Arja H.
    Uppsala University, Disciplinary Domain of Medicine and Pharmacy, Faculty of Medicine, Department of Women's and Children's Health, Research group (Dept. of women´s and children´s health), Neuropediatrics/Paediatric oncology.
    Mäkitie, Outi
    Karolinska Inst, Dept Mol Med & Surg, Stockholm, Sweden.
    Gustafsson, Jan
    Uppsala University, Disciplinary Domain of Medicine and Pharmacy, Faculty of Medicine, Department of Women's and Children's Health, Research group (Dept. of women´s and children´s health), Paediatric Inflammation Research.
    Dernroth, Dzeneta Nezirevich
    Dept Clin Chem & Expt Med, Linkoping, Sweden.
    Frisk, Per
    Uppsala University, Disciplinary Domain of Medicine and Pharmacy, Faculty of Medicine, Department of Women's and Children's Health, Research group (Dept. of women´s and children´s health), Neuropediatrics/Paediatric oncology.
    Vitamin D Status in Children with Leukemia2018In: Blood, ISSN 0006-4971, E-ISSN 1528-0020, Vol. 132Article in journal (Other academic)
  • 34.
    Jussila, Miro-Pekka
    et al.
    Univ Oulu, Res Unit Med Imaging Phys & Technol, Fac Med, Oulu, Finland;Univ Oulu, Med Res Ctr, Oulu, Finland.
    Remes, Tiina
    Oulu Univ Hosp, Dept Pediat & Adolescence, Oulu, Finland;Oulu Univ Hosp, PEDEGO Res Unit, Oulu, Finland;Univ Oulu, Oulu, Finland.
    Anttonen, Julia
    Oulu Univ Hosp, Dept Pediat & Adolescence, Oulu, Finland;Oulu Univ Hosp, PEDEGO Res Unit, Oulu, Finland;Univ Oulu, Oulu, Finland.
    Harila-Saari, Arja H.
    Uppsala University, Disciplinary Domain of Medicine and Pharmacy, Faculty of Medicine, Department of Women's and Children's Health, Research group (Dept. of women´s and children´s health), Neuropediatrics/Paediatric oncology.
    Niinimaki, Jaakko
    Univ Oulu, Res Unit Med Imaging Phys & Technol, Fac Med, Oulu, Finland;Univ Oulu, Med Res Ctr, Oulu, Finland;Univ Oulu, Oulu, Finland;Oulu Univ Hosp, Dept Diagnost Radiol, Oulu, Finland.
    Pokka, Tytti
    Oulu Univ Hosp, Dept Pediat & Adolescence, Oulu, Finland;Oulu Univ Hosp, PEDEGO Res Unit, Oulu, Finland;Univ Oulu, Oulu, Finland.
    Koskenkorva, Paivi
    Kuopio Univ Hosp, Dept Clin Radiol, Kuopio, Finland.
    Sutela, Anna
    Kuopio Univ Hosp, Dept Clin Radiol, Kuopio, Finland.
    Toiviainen-Sale, Sanna
    Univ Helsinki, Dept Pediat Radiol, HUS Med Imaging Ctr, Radiol, Helsinki, Finland;Helsinki Univ Hosp, Helsinki, Finland.
    Arikoski, Pekka
    Kuopio Univ Hosp, Dept Pediat & Adolescence, Kuopio, Finland;Univ Eastern Finland, Kuopio, Finland.
    Riikonen, Pekka
    Kuopio Univ Hosp, Dept Pediat & Adolescence, Kuopio, Finland;Univ Eastern Finland, Kuopio, Finland.
    Arola, Mikko
    Tampere Univ Hosp, Dept Pediat, Tampere, Finland;Univ Tampere, Fac Med & Life Sci, Tampere, Finland.
    Lahteenmaki, Paivi
    Turku Univ Hosp, Dept Pediat & Adolescent Med, Turku, Finland;Turku Univ, Turku, Finland.
    Sirkia, Kirsti
    Helsinki Univ Hosp, Dept Pediat & Adolescence, Helsinki, Finland.
    Rantala, Heikki
    Oulu Univ Hosp, Dept Pediat & Adolescence, Oulu, Finland;Oulu Univ Hosp, PEDEGO Res Unit, Oulu, Finland;Univ Oulu, Oulu, Finland.
    Suo-Palosaari, Maria
    Univ Oulu, Res Unit Med Imaging Phys & Technol, Fac Med, Oulu, Finland;Univ Oulu, Med Res Ctr, Oulu, Finland;Univ Oulu, Oulu, Finland;Oulu Univ Hosp, Dept Diagnost Radiol, Oulu, Finland.
    Ojaniemi, Marja
    Univ Oulu, Med Res Ctr, Oulu, Finland;Oulu Univ Hosp, Dept Pediat & Adolescence, Oulu, Finland;Oulu Univ Hosp, PEDEGO Res Unit, Oulu, Finland;Univ Oulu, Oulu, Finland.
    Late vertebral side effects in long-term survivors of irradiated childhood brain tumor2018In: PLoS ONE, ISSN 1932-6203, E-ISSN 1932-6203, Vol. 13, no 12, article id e0209193Article in journal (Refereed)
    Abstract [en]

    Purpose: Long-term side effects of the treatments are common in survivors of irradiated pediatric brain tumors. Ionizing radiation in combination with surgery and chemotherapy during childhood may reduce vertebral height and bone mineral density (BMD), and cause growth failure. The aim of this study was to evaluate the late consequences of tumor treatments on vertebrae in survivors of childhood brain tumors.

    Methods: 72 adult survivors (mean age 27.8 years, standard deviation 6.7) of irradiated childhood brain tumor were studied by spinal magnetic resonance imaging (MRI) for vertebral abnormalities from the national cohort of Finland. Patients were treated in five university hospitals in Finland between the years 1970 and 2008. Subject height and weight were measured and body mass index (BMI) was calculated. The morphology and height/ depth ratio of the vertebrae in the middle of the kyphotic thoracic curvature (Th8) and lumbar lordosis (L3) were examined. Vertebrae were analyzed by Genant's semiquantative (SQ) method and spinal deformity index (SDI) was calculated. BMD was measured by using dual X-ray absorptiometry.

    Results: 4.2% (3/72) of the patients had undiagnosed asymptomatic vertebral fracture and 5.6% (4/72) of patients had radiation- induced decreased vertebral body height. Male patients had flatter vertebrae compared with females. Patient age at the time of irradiation, BMI and irradiation area correlated to vertebral morphology differentially in males and females. BMD had no association with the vertebral shape. Patients who had received craniospinal irradiation were shorter than the general population.

    Conclusion: Childhood brain tumor survivors had a high number of vertebral abnormalities in young adulthood. Irradiation was associated with abnormal vertebral morphology and compromised final height. Male gender may predispose vertebrae to the side effects of irradiation.

  • 35. Järviaho, Tekla
    et al.
    Zachariadis, Vasilios
    Tesi, Bianca
    Chiang, Samuel
    Bryceson, Yenan T
    Möttönen, Merja
    Niinimäki, Riitta
    Bang, Benedicte
    Rahikkala, Elisa
    Taylan, Fulya
    Uusimaa, Johanna
    Harila-Saari, Arja H.
    Uppsala University, Disciplinary Domain of Medicine and Pharmacy, Faculty of Medicine, Department of Women's and Children's Health, Research group (Dept. of women´s and children´s health), Neuropediatrics/Paediatric oncology.
    Nordgren, Ann