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  • 1.
    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.
    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.
    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.

  • 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.
    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)
  • 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.
    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.

  • 5.
    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.

  • 6.
    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.

  • 7.
    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, 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.

  • 8.
    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)
  • 9.
    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.

  • 10.
    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.

  • 11.
    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.

  • 12.
    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)
  • 13.
    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.

  • 14.
    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.

  • 15.
    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.

  • 16.
    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
  • 17.
    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)
  • 18.
    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.

  • 19.
    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)
  • 20.
    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.

  • 21.
    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.

  • 22.
    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.

  • 23.
    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.

  • 24.
    Kamsvåg-Magnusson, Tove
    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.
    von Essen, Louise
    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), Clinical Psychology in Healthcare.
    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.
    Svanberg, Anncarin
    Uppsala University, Disciplinary Domain of Medicine and Pharmacy, Faculty of Medicine, Department of Medical Sciences, Haematology.
    Mellgren, K.
    Sahlgrens Acad, Dept Clin Sci Pediat, Gothenburg, Sweden..
    Garming-Legert, K.
    Karolinska Inst, Dept Dent Med, Stockholm, Sweden..
    Toporski, J.
    Skane Univ Hosp, Dept Clin Sci, Sect Pediat Oncol Hematol, Lund, Sweden..
    Winiarski, J.
    Karolinska Inst, Dept Clin Sci Intervent & Technol, Stockholm, Sweden..
    Ljungman, G.
    Uppsala Univ, Dept Womens & Childrens Hlth, Uppsala, Sweden..
    Oral Cryotherapy to Reduce the Incidence of Severe Oral Mucositis in Children Undergoing Hematopoietic Stem Cell Transplantation: Results of a Randomized Clinical Trial2017In: Pediatric Blood & Cancer, ISSN 1545-5009, E-ISSN 1545-5017, Vol. 64, no S3, p. S360-S361Article in journal (Other academic)
  • 25.
    Karlsson, Lene
    et al.
    Univ Gothenburg, Sahlgrenska Acad, Inst Clin Sci, Dept Paediat, Gothenburg, Sweden..
    Forestier, Erik
    Umea Univ, Dept Clin Sci, Paediat, Umea, Sweden..
    Hasle, Henrik
    Aarhus Univ, Hosp Skejby, Dept Paediat, Aarhus, Denmark..
    Jahnukainen, Kirsi
    Childrens Hosp, Div Haematol Oncol & Stem Cell Transplantat, Helsinki, Finland..
    Jonsson, Olafur G.
    Landspitali Univ Hosp, Childrens Hosp, Hringbraut, Reykjavik, Iceland..
    Lausen, Birgitte
    Univ Copenhagen, Rigshosp, Dept Paediat & Adolescent Med, Copenhagen, Denmark..
    Nyström, Ulrika Noren
    Umea Univ, Dept Clin Sci, Paediat, Umea, Sweden..
    Palle, J
    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.
    Tierens, Anne
    Toronto Gen Hosp, Univ Hlth Network, Dept Pathobiol & Lab Med, Toronto, ON, Canada..
    Zeller, Bernward
    Oslo Univ Hosp, Dept Paediat, Oslo, Norway..
    Abrahamsson, Jonas
    Univ Gothenburg, Sahlgrenska Acad, Inst Clin Sci, Dept Paediat, Gothenburg, Sweden..
    Outcome after intensive reinduction therapy and allogeneic stem cell transplant in paediatric relapsed acute myeloid leukaemia2017In: British Journal of Haematology, ISSN 0007-1048, E-ISSN 1365-2141, Vol. 178, no 4, p. 592-602Article in journal (Refereed)
    Abstract [en]

    Given that 30-40% of children with acute myeloid leukaemia (AML) relapse after primary therapy it is important to define prognostic factors and identify optimal therapy. From 1993 to 2012, 543 children from the Nordic countries were treated according to two consecutive protocols: 208 children relapsed. The influence of disease characteristics, first line treatment, relapse therapy and duration of first remission on outcome was analysed. Second complete remission (CR2) was achieved in 146 (70%) patients. Estimated 5-year overall survival (OS5y) was 39 +/- 4% for the whole group and 43 +/- 4% for the 190 patients given re-induction therapy, of whom 76% received regimens that included fludarabine, cytarabine (FLA) +/- anthracyclines, 18% received Nordic Society for Paediatric Haematology and Oncology (NOPHO) upfront blocks and 5% received other regimens. Late relapse >= 1 year from diagnosis, no allogeneic stem cell transplantation (SCT) in first remission and core binding factor AML were independent favourable prognostic factors for survival. For the 128 children (124 in CR2) that received SCT as consolidation therapy after relapse, OS5y was 61 +/- 5%. Four of 19 children (21%) survived without receiving SCT as part of relapse therapy. Our data show that intensive re-induction followed by SCT can give cure rates of 40% in children with relapsed AML.

  • 26.
    Kassa, Ann-Marie
    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), Pediatric Surgery. Univ Childrens Hosp, Dept Paediat Surg, SE-75185 Uppsala, Sweden..
    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.
    Engstrand Lilja, Helene
    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. Univ Childrens Hosp, Dept Paediat Surg, SE-75185 Uppsala, Sweden..
    Young children with severe congenital malformations (VACTERL) expressed mixed feelings about their condition and worries about needles and anaesthesia2017In: Acta Paediatrica, ISSN 0803-5253, E-ISSN 1651-2227, Vol. 106, no 10, p. 1694-1701Article in journal (Refereed)
    Abstract [en]

    Aim: Our knowledge of the perceptions that children with severe congenital malformations have of their health, treatment and how to improve hospital care is limited. This study focused on patients with vertebral defects, anal atresia, cardiac defects, tracheo-oesophageal fistula, renal anomalies and limb abnormalities (VACTERL).

    Methods: We interviewed 10 children aged five to eight years with VACTERL association who were treated in a Swedish tertiary paediatric surgical centre, using a computer-assisted technique called In My Shoes. The interviews were analysed by qualitative content analysis.

    Results: The children described their awareness of their health history and said they felt proud but different due to their physical dysfunction. They were happy to visit the hospital to meet familiar staff, but expressed negative feelings about missing normal life. They were afraid of needle-related procedures and not wakening up after anaesthesia. Various ways of coping with difficult situations were expressed, and suggestions to improve hospital care were voiced.

    Conclusion: Careful follow-up of these children by multidisciplinary teams is crucial to optimise their health and functional status. Fear of medical procedures may be reduced by carefully delivered information, listening to the children, providing continuity of care and creating individual care strategies.

  • 27.
    Kinch, Amelie
    et al.
    Uppsala University, Disciplinary Domain of Medicine and Pharmacy, Faculty of Medicine, Department of Medical Sciences, Infectious Diseases.
    Hallböök, Helene
    Uppsala University, Disciplinary Domain of Medicine and Pharmacy, Faculty of Medicine, Department of Medical Sciences, Haematology.
    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.
    Sällström, K.
    Uppsala University, Disciplinary Domain of Medicine and Pharmacy, Faculty of Medicine, Department of Medical Sciences, Infectious Diseases.
    Bondeson, Kåre
    Uppsala University, Disciplinary Domain of Medicine and Pharmacy, Faculty of Medicine, Department of Medical Sciences, Infection medicine.
    Pauksen, Karlis
    Uppsala University, Disciplinary Domain of Medicine and Pharmacy, Faculty of Medicine, Department of Medical Sciences, Infectious Diseases.
    Epstein-Barr virus-related disease after allogeneic HSCT and use of pre-emptive rituximab: Clinical Features And Outcome2017In: Bone Marrow Transplantation, ISSN 0268-3369, E-ISSN 1476-5365, Vol. 52, no Supplement: 1, p. S88-S88Article in journal (Other academic)
  • 28.
    Kinch, Amelie
    et al.
    Uppsala University, Disciplinary Domain of Medicine and Pharmacy, Faculty of Medicine, Department of Medical Sciences, Infectious Diseases.
    Hallböök, Helene
    Uppsala University, Disciplinary Domain of Medicine and Pharmacy, Faculty of Medicine, Department of Medical Sciences, Haematology.
    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.
    Sällström, Kalle
    Uppsala University, Disciplinary Domain of Medicine and Pharmacy, Faculty of Medicine, Department of Medical Sciences, Infectious Diseases.
    Bondeson, Kåre
    Uppsala University, Disciplinary Domain of Medicine and Pharmacy, Faculty of Medicine, Department of Medical Sciences, Clinical Virology.
    Pauksen, Karlis
    Uppsala University, Disciplinary Domain of Medicine and Pharmacy, Faculty of Medicine, Department of Medical Sciences, Infectious Diseases.
    Long-term outcome of Epstein-Barr virus DNAemia and PTLD with the use of preemptive rituximab following allogeneic HSCT2018In: Leukemia and Lymphoma, ISSN 1042-8194, E-ISSN 1029-2403, Vol. 59, no 5, p. 1172-1179Article in journal (Refereed)
    Abstract [en]

    We studied retrospectively the outcome of Epstein-Barr virus (EBV)-related disease with EBV monitoring and preemptive rituximab to prevent post-transplant lymphoproliferative disorder (PTLD) in 319 consecutive allogeneic stem cell transplantations 2004-2012. Patients who received anti-thymocyte globulin (ATG) or alemtuzumab were regarded as high-risk for PTLD (n = 214). EBV DNAemia ≥1000 copies/mL plasma was observed in 50 (23%) of the high-risk patients. Thirty-three of the high-risk (15%) and one of the low-risk (1%) patients received rituximab, in combination with reduction of immunosuppression (n = 24) or chemotherapy (n = 4). Although rituximab was initiated only 5 d after first EBV load ≥1000 copies/mL, 85% of the rituximab-treated patients developed symptoms (lymphadenopathy 50%, fever 76%, and encephalitis/meningitis 12%). Response-rate to EBV treatment was 88%. Overall survival at 1- and 5-year was 71 and 52% for rituximab-treated patients, which was not inferior to all other patients post-transplant. In conclusion, rituximab therapy for EBV DNAemia does not affect long-term survival negatively.

  • 29.
    Lindqvist, Carl Mårten
    et al.
    Uppsala University, Disciplinary Domain of Medicine and Pharmacy, Faculty of Medicine, Department of Medical Sciences, Molecular Medicine. Uppsala University, Science for Life Laboratory, SciLifeLab.
    Dahlberg, Johan
    Uppsala University, Disciplinary Domain of Medicine and Pharmacy, Faculty of Medicine, Department of Medical Sciences, Molecular Medicine. Uppsala University, Science for Life Laboratory, SciLifeLab.
    Raine, Amanda
    Uppsala University, Disciplinary Domain of Medicine and Pharmacy, Faculty of Medicine, Department of Medical Sciences, Molecular Medicine. Uppsala University, Science for Life Laboratory, SciLifeLab.
    Övernäs, Elin
    Uppsala University, Science for Life Laboratory, SciLifeLab. Uppsala University, Disciplinary Domain of Medicine and Pharmacy, Faculty of Medicine, Department of Medical Sciences, Molecular Medicine.
    Ekman, Diana
    Science for Life Laboratory, Department of Biochemistry and Biophysics, Stockholm University, Stockholm, 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.
    Frost, B M
    Uppsala University, Disciplinary Domain of Medicine and Pharmacy, Faculty of Medicine, Department of Women's and Children's Health.
    Grandér, Dan
    Karolinska Institutet, Dept. Oncology and Pathology, Stockholm, Sweden.
    Forestier, Erik
    Dept. of Medical Biosciences, University of Umeå, Umeå, Sweden.
    Lönnerholm, 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), Neuropediatrics/Paediatric oncology.
    Syvänen, Ann-Christine
    Uppsala University, Disciplinary Domain of Medicine and Pharmacy, Faculty of Medicine, Department of Medical Sciences, Molecular Medicine. Uppsala University, Science for Life Laboratory, SciLifeLab.
    Berglund, Eva Caroline
    Uppsala University, Disciplinary Domain of Medicine and Pharmacy, Faculty of Medicine, Department of Medical Sciences, Molecular Medicine. Uppsala University, Disciplinary Domain of Science and Technology, Biology, Department of Cell and Molecular Biology, Molecular Evolution. Uppsala University, Science for Life Laboratory, SciLifeLab.
    Identification of somatic single nucleotide variants inleukemia by targeted sequencing of non-indexed overlapping poolsManuscript (preprint) (Other academic)
  • 30.
    Ljungman, Lisa
    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), Clinical Psychology in Healthcare.
    Cernvall, Martin
    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), Clinical Psychology in Healthcare.
    Ghaderi, Ata
    Division of Psychology, Department of Clinical Neuroscience, Karolinska Institutet, Stockholm, Sweden.
    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.
    von Essen, Louise
    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), Clinical Psychology in Healthcare.
    Ljótsson, Brjánn
    Division of Psychology, Department of Clinical Neuroscience, Karolinska Institutet, Stockholm, Sweden.
    An open trial of individualized face-to-face cognitive behavior therapy for psychological distress in parents of children after end of treatment for childhood cancer including a cognitive behavioral conceptualization2018In: PeerJ, ISSN 2167-8359, E-ISSN 2167-8359, Vol. 6, article id e4570Article in journal (Refereed)
    Abstract [en]

    Objective

    A subgroup of parents of children who have been treated for childhood cancer report high levels of psychological distress. To date there is no empirically supported psychological treatment targeting cancer-related psychological distress in this population. The aim of the current study was to test the feasibility and preliminarily evaluate the effect of individualized face-to-face cognitive behavior therapy (CBT) for parents of children after the end of treatment for childhood cancer. A secondary aim was to present a cognitive behavioral conceptualization of cancer-related distress for these parents.

    Methods

    An open trial was conducted where 15 parents of children who had completed successful treatment for cancer three months to five years earlier and who reported psychological distress related to a child’s previous cancer disease were provided CBT at a maximum of 15 sessions. Participants were assessed at baseline, post-intervention, and three-month follow-up using self-reported psychological distress (including posttraumatic stress symptoms (PTSS), depression, and anxiety) and the diagnostic Mini-International Neuropsychiatric Interview. Feasibility outcomes relating to recruitment, data collection, and delivery of the treatment were also examined. Individual case formulations for each participant guided the intervention and these were aggregated and presented in a conceptualization detailing core symptoms and their suggested maintenance mechanisms.

    Results

    A total of 93% of the participants completed the treatment and all of them completed the follow-up assessment. From baseline to post-assessment, parents reported significant improvements in PTSS, depression, and anxiety with medium to large effect sizes (Cohen’s d = 0.65–0.92). Results were maintained or improved at a three-month follow-up. At baseline, seven (47%) participants fulfilled the diagnostic criteria for major depressive disorder and four (29%) fulfilled the criteria for posttraumatic stress disorder, compared to none at a post-assessment and a follow-up assessment. The resulting cognitive behavioral conceptualization suggests traumatic stress and depression as the core features of distress, and avoidance and inactivity is suggested as the core maintenance mechanisms.

    Conclusion

    The treatment was feasible and acceptable to the participants. Significant improvements in distress were observed during the study. Overall, results suggest that the psychological treatment for parents of children after end of treatment for childhood cancer used in the current study is promising and should be tested and evaluated in future studies.

  • 31.
    Marincevic-Zuniga, Yanara
    et al.
    Uppsala University, Disciplinary Domain of Medicine and Pharmacy, Faculty of Medicine, Department of Medical Sciences, Molecular Medicine. Uppsala University, Science for Life Laboratory, SciLifeLab.
    Dahlberg, Johan
    Uppsala University, Disciplinary Domain of Medicine and Pharmacy, Faculty of Medicine, Department of Medical Sciences, Molecular Medicine. Uppsala University, Science for Life Laboratory, SciLifeLab.
    Nilsson, Sara
    Uppsala University, Disciplinary Domain of Medicine and Pharmacy, Faculty of Medicine, Department of Medical Sciences, Molecular Medicine. Uppsala University, Science for Life Laboratory, SciLifeLab.
    Raine, Amanda
    Uppsala University, Disciplinary Domain of Medicine and Pharmacy, Faculty of Medicine, Department of Medical Sciences, Molecular Medicine. Uppsala University, Science for Life Laboratory, SciLifeLab.
    Nystedt, Sara
    Uppsala University, Disciplinary Domain of Medicine and Pharmacy, Faculty of Medicine, Department of Medical Sciences, Molecular Medicine. Uppsala University, Science for Life Laboratory, SciLifeLab.
    Lindqvist, Carl Mårten
    Uppsala University, Disciplinary Domain of Medicine and Pharmacy, Faculty of Medicine, Department of Medical Sciences, Molecular Medicine. Uppsala University, Science for Life Laboratory, SciLifeLab.
    Berglund, Eva C.
    Uppsala University, Disciplinary Domain of Medicine and Pharmacy, Faculty of Medicine, Department of Medical Sciences, Molecular Medicine. Uppsala University, Science for Life Laboratory, SciLifeLab.
    Abrahamsson, Jonas
    Univ Gothenburg, Sahlgrenska Acad, Dept Pediat, Inst Clin Sci, Gothenburg, Sweden..
    Cavelier, Lucia
    Uppsala University, Disciplinary Domain of Medicine and Pharmacy, Faculty of Medicine, Department of Immunology, Genetics and Pathology, Medicinsk genetik och genomik.
    Forestier, Erik
    Univ Umea, Dept Med Biosci, Umea, Sweden..
    Heyman, Mats
    Karolinska Univ Hosp, Astrid Lindgren Childrens Hosp, Childhood Canc Res Unit, Karolinska Inst, Stockholm, Sweden..
    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.
    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.
    Syvänen, Ann-Christine
    Uppsala University, Disciplinary Domain of Medicine and Pharmacy, Faculty of Medicine, Department of Medical Sciences, Molecular Medicine. Uppsala University, Science for Life Laboratory, SciLifeLab.
    Transcriptome sequencing in pediatric acute lymphoblastic leukemia identifies fusion genes associated with distinct DNA methylation profiles2017In: Journal of Hematology & Oncology, ISSN 1756-8722, E-ISSN 1756-8722, Vol. 10, article id 148Article in journal (Refereed)
    Abstract [en]

    Background: Structural chromosomal rearrangements that lead to expressed fusion genes are a hallmark of acute lymphoblastic leukemia (ALL). In this study, we performed transcriptome sequencing of 134 primary ALL patient samples to comprehensively detect fusion transcripts. Methods: We combined fusion gene detection with genome-wide DNA methylation analysis, gene expression profiling, and targeted sequencing to determine molecular signatures of emerging ALL subtypes. Results: We identified 64 unique fusion events distributed among 80 individual patients, of which over 50% have not previously been reported in ALL. Although the majority of the fusion genes were found only in a single patient, we identified several recurrent fusion gene families defined by promiscuous fusion gene partners, such as ETV6, RUNX1, PAX5, and ZNF384, or recurrent fusion genes, such as DUX4-IGH. Our data show that patients harboring these fusion genes displayed characteristic genome-wide DNA methylation and gene expression signatures in addition to distinct patterns in single nucleotide variants and recurrent copy number alterations. Conclusion: Our study delineates the fusion gene landscape in pediatric ALL, including both known and novel fusion genes, and highlights fusion gene families with shared molecular etiologies, which may provide additional information for prognosis and therapeutic options in the future.

  • 32.
    Marincevic-Zuniga, Yanara
    et al.
    Uppsala University, Disciplinary Domain of Medicine and Pharmacy, Faculty of Medicine, Department of Medical Sciences, Molecular Medicine. Uppsala University, Science for Life Laboratory, SciLifeLab.
    Nystedt, Sara
    Uppsala University, Disciplinary Domain of Medicine and Pharmacy, Faculty of Medicine, Department of Medical Sciences, Molecular Medicine. Uppsala University, Science for Life Laboratory, SciLifeLab.
    Nilsson, Sara
    Uppsala University, Disciplinary Domain of Medicine and Pharmacy, Faculty of Medicine, Department of Medical Sciences, Molecular Medicine. Uppsala University, Science for Life Laboratory, SciLifeLab.
    Almlöf, Jonas Carlsson
    Uppsala University, Disciplinary Domain of Medicine and Pharmacy, Faculty of Medicine, Department of Medical Sciences, Molecular Medicine. Uppsala University, Science for Life Laboratory, SciLifeLab.
    Lilljebjörn, Henrik
    Fioretos, Thoas
    Flaegstad, Trond
    Forestier, Erik
    Heyman, Mats
    Kanerva, Jukka
    Schmiegelow, Kjeld
    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.
    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.
    DNA methylation classification in combination with RNA-sequencing for subtype discovery in pediatric B-cell precursor acute lymphoblastic leukemiaManuscript (preprint) (Other academic)
  • 33.
    Montgomery, Cecilia
    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), Perinatal, Neonatal and Pediatric Cardiology Research.
    Johansen, Kine
    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.
    Lucas, Steven
    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.
    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.
    Persson, Kristina
    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), Perinatal, Neonatal and Pediatric Cardiology Research.
    The Structured Observation of Motor Performance in Infants can detect cerebral palsy early in neonatal intensive care recipients2017In: Early Human Development, ISSN 0378-3782, E-ISSN 1872-6232, Vol. 113, p. 31-39Article in journal (Refereed)
    Abstract [en]

    Background

    The detection of motor problems in infancy requires a detailed assessment method that measures both the infants' level of motor development and movement quality.

    Aim

    To evaluate the ability of the Structured Observation of Motor Performance in Infants (SOMP-I) to detect cerebral palsy (CP) in neonatal intensive care recipients.

    Study design

    Prospective cohort study analyzed retrospectively.

    Subjects

    212 (girls: 96) neonatal intensive care recipients (mean gestational age 34 weeks, range: 23–43). Twenty infants were diagnosed with CP.

    Outcome measures

    The infants were assessed using SOMP-I at 2, 4, 6 and 10 months' corrected age. Accuracy measures were calculated for level of motor development, quality of motor performance and a combination of the two to detect CP at single and repeated assessments.

    Results

    At 2 months, 17 of 20 infants with CP were detected, giving a sensitivity of 85% (95% CI 62–97%) and a specificity of 48% (95% CI 40–55%), while the negative likelihood ratio was 0.3 (95% CI 0.1–0.9) and the positive likelihood ratio was 1.6 (95% CI 1.3–2.0). At 6 months all infants with CP were detected using SOMP-I, and all infants had repeatedly been assessed outside the cut-offs. Specificity was generally lower for all assessment ages, however, for repeated assessments sensitivity reached 90% (95% CI 68–99%) and specificity 85% (95% CI 79–90%).

    Conclusions

    SOMP-I is sensitive for detecting CP early, but using the chosen cut-off can lead to false positives for CP. Assessing level and quality in combination and at repeated assessments improved predictive ability.

  • 34.
    Mårtensson, Thomas
    et al.
    Karolinska Institutet, Department of Clinical Science, Intervention and Technology (CLINTEC), Stockholm, Sweden.
    Szakos, Attila
    Karolinska University Hospital, Department of Clinical Pathology and Cytology, Stockholm, Sweden.
    Mellgren, Karin
    University of Gothenburg, Department of Pediatrics, Institute of Clinical Sciences Sahlgrenska Academy, Sweden.
    Toporski, Jacek
    Skåne University Hospital, Department of Pediatrics, Lund, 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.
    Casswall, Thomas H
    Karolinska Institutet, Department of Clinical Science, Intervention and Technology (CLINTEC), Stockholm, Sweden.
    Gustafsson, Britt
    Karolinska Institutet, Department of Clinical Science, Intervention and Technology (CLINTEC), Stockholm, Sweden.
    Choice of Endoscopic Procedure in Children With Clinically Suspected Gastrointestinal Graft-Versus-Host Disease.2018In: Journal of Pediatric Gastroenterology and Nutrition - JPGN, ISSN 0277-2116, E-ISSN 1536-4801, Vol. 66, no 5, p. 744-750Article in journal (Refereed)
    Abstract [en]

    Objectives: Gastrointestinal graft-versus-host disease (GI-GVHD) is a potentially life-threatening complication after hematopoietic stem cell transplantation. Symptoms indicating GI-GVHD motivates endoscopy with biopsy sampling and histopathological confirmation. Optimal extent of endoscopy in children is, however, presently unknown. Therefore, we aimed to evaluate whether biopsies from the rectosigmoid area versus the rest of the colon/ileocolon with or without biopsies from simultaneous upper endoscopy, were equally reliable for detection of GI-GVHD and relevant differential diagnoses.

    Methods: Retrospective multicenter study based on histopathological re-evaluation of biopsies and hospital record data, collected from children with suspected GI-GVHD.

    Results: Forty-four children with 51 endoscopic occasions (81 procedures) were included. Thirty-nine of 51 (76.5%) were diagnosed as GI-GVHD, 14 (27.4%) received a differential diagnosis and 7 (13.7%) had normal histology findings. Comorbidity, that is, simultaneous detection of a differential diagnosis and GI-GVHD, was observed in 9 (23.1%) cases. Cytomegalovirus infection was the most frequent differential diagnosis, 6 of 7 were detected in biopsies from rectosigmoid and esophagogastroduodenal areas. Sensitivity for detection of GI-GVHD in biopsies collected from rectosigmoid-ileocolonic-, rectosigmoid-, or esophagogastroduodenal areas were 97.4%, 84.6%, 83.3%, respectively, and 97.4% when the latter 2 were merged. The difference, nondetected GI-GVHD in the rectosigmoid area versus detected elsewhere in the GI tract, was statistically significant (P = 0.03).

    Conclusions: Biopsies collected from the rectosigmoid area solely were not optimal for detection of pediatric GI-GVHD. When biopsy sampling from rectosigmoid and upper GI tract areas was combined, the sensitivity for GI-GVHD was, however, equally high as for ileocolonoscopy or full upper and lower endoscopy.

  • 35.
    Nordlund, Jessica
    et al.
    Uppsala University, Disciplinary Domain of Medicine and Pharmacy, Faculty of Medicine, Department of Medical Sciences, Molecular Medicine. Uppsala University, Science for Life Laboratory, SciLifeLab.
    Marincevic-Zuniga, Yanara
    Uppsala University, Disciplinary Domain of Medicine and Pharmacy, Faculty of Medicine, Department of Medical Sciences, Molecular Medicine. Uppsala University, Science for Life Laboratory, SciLifeLab.
    Cavelier, Lucia
    Uppsala University, Science for Life Laboratory, SciLifeLab. Uppsala University, Disciplinary Domain of Medicine and Pharmacy, Faculty of Medicine, Department of Immunology, Genetics and Pathology, Medicinsk genetik och genomik.
    Raine, Amanda
    Uppsala University, Disciplinary Domain of Medicine and Pharmacy, Faculty of Medicine, Department of Medical Sciences, Molecular Medicine. Uppsala University, Science for Life Laboratory, SciLifeLab.
    Martin, Tom
    Uppsala University, Disciplinary Domain of Medicine and Pharmacy, Faculty of Medicine, Department of Medical Sciences, Molecular Medicine. Uppsala University, Science for Life Laboratory, SciLifeLab.
    Lundmark, Anders
    Uppsala University, Disciplinary Domain of Medicine and Pharmacy, Faculty of Medicine, Department of Medical Sciences, Molecular Medicine. Uppsala University, Science for Life Laboratory, SciLifeLab.
    Abrahamsson, Jonas
    Norén-Nyström, Ulrika
    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.
    Syvänen, Ann-Christine
    Uppsala University, Disciplinary Domain of Medicine and Pharmacy, Faculty of Medicine, Department of Medical Sciences, Molecular Medicine. Uppsala University, Science for Life Laboratory, SciLifeLab.
    Refined detection and phasing of structural aberrations in pediatric acute lymphoblastic leukemia with linked-read whole genome sequencingManuscript (preprint) (Other academic)
  • 36.
    Nowinski, Daniel
    et al.
    Uppsala University, Disciplinary Domain of Medicine and Pharmacy, Faculty of Medicine, Department of Surgical Sciences.