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  • 1. af Sandeberg, Margareta
    et al.
    Wettergren, Lena
    Bjork, Olle
    Arvidson, Johan
    Uppsala University, Disciplinary Domain of Medicine and Pharmacy, Faculty of Medicine, Department of Women's and Children's Health.
    Johansson, Eva
    Does school attendance during initial cancer treatment in childhood increase the risk of infection?2013In: Pediatric Blood & Cancer, ISSN 1545-5009, E-ISSN 1545-5017, Vol. 60, no 8, p. 1307-1312Article in journal (Refereed)
    Abstract [en]

    Background The present study aimed to investigate the relationship between school attendance and infection requiring antimicrobial treatment in children undergoing treatment for cancer. Procedure A national cohort of children aged 7-16 years undergoing cancer treatment was assessed during two observation periods of 19 days each, 1 month (n=89) and 2.5 months (n=89) poststart of treatment. Children free from infection at start of each observation period were included. Multivariable logistic regression analyses were performed including factors potentially associated with start of antimicrobial treatment. Results Twenty-seven (30%) children started antimicrobial treatment during the first observation period. Factors associated with an increased risk of starting antimicrobial treatment were diagnosed with sarcoma (OR=24.37, P=0.002) or non-Hodgkin lymphoma (OR=17.57, P=0.025), having neutropenia (OR=5.92, P=0.020) and age less than 13 years (OR=8.54, P=0.014). During the second observation period, when 20 (22%) children started antimicrobial treatment, the probability of starting treatment was increased in children with neutropenia (OR=4.25, P=0.007). There was no statistically significant association between starting treatment for infection and school attendance. Conclusions In this study, children attending school while undergoing cancer treatment did not run a higher risk of starting antimicrobial treatment than children absent from school. However, there is a need for further studies evaluating risk of infections in children with ongoing cancer treatment. 

  • 2. Af Sandeberg, Margareta
    et al.
    Wettergren, Lena
    Bjork, Olle
    Arvidson, Johan
    Uppsala University, Disciplinary Domain of Medicine and Pharmacy, Faculty of Medicine, Department of Women's and Children's Health, Pediatrics.
    Johansson, Eva
    Is school attendance during initial childhood cancer treatment associated with infection?2012In: Pediatric Blood & Cancer, ISSN 1545-5009, E-ISSN 1545-5017, Vol. 59, no 6, p. 1112-1112Article in journal (Other academic)
  • 3.
    Anastasopoulou, Stavroula
    et al.
    Karolinska Inst, Dept Womens & Childrens Hlth, Stockholm, Sweden;Karolinska Univ Hosp, Stockholm, Sweden.
    Eriksson, Mats A.
    Karolinska Inst, Dept Womens & Childrens Hlth, Stockholm, Sweden;Karolinska Univ Hosp, Stockholm, Sweden.
    Heyman, Mats
    Karolinska Inst, Dept Womens & Childrens Hlth, Stockholm, Sweden;Karolinska Univ Hosp, Stockholm, Sweden.
    Wang, Chen
    Karolinska Inst, Dept Womens & Childrens Hlth, Stockholm, Sweden;Karolinska Univ Hosp, Stockholm, Sweden.
    Niinimäki, Riitta
    Oulu Univ Hosp, Dept Children & Adolescents, Oulu, Finland;Univ Oulu, PEDEGO Res Unit, Oulu, Finland.
    Mikkel, Sirje
    Univ Tartu, Dept Hematol & Oncol, Tartu, Estonia.
    Vaitkeviciene, Goda E.
    Vilnius Univ Hosp Santaros Klin, Childrens Hosp, Vilnius, Lithuania;Vilnius Univ, Vilnius, Lithuania.
    Johannsdottir, Inga Maria
    Oslo Univ Hosp, Dept Pediat Hematol Oncol, Oslo, Norway.
    Myrberg, Ida Hed
    Karolinska Inst, Dept Womens & Childrens Hlth, Stockholm, Sweden.
    Jonsson, Olafur Gisli
    Univ Iceland, Dept Pediat, Reykjavik, Iceland.
    Als-Nielsen, Bodil
    Rigshosp, Univ Hosp, Dept Pediat & Adolescent Med, Copenhagen, Denmark;Univ Copenhagen, Fac Med, Inst Clin Med, Copenhagen, Denmark.
    Schmiegelow, Kjeld
    Rigshosp, Univ Hosp, Dept Pediat & Adolescent Med, Copenhagen, Denmark;Univ Copenhagen, Fac Med, Inst Clin Med, Copenhagen, Denmark.
    Banerjee, Joanna
    Univ Helsinki, Childrens Hosp, Dept Pediat Hematol Oncol & Stem Cell Transplanta, Helsinki, Finland;Helsinki Univ Hosp, Helsinki, Finland.
    Harila-Saari, Arja H.
    Uppsala University, Disciplinary Domain of Medicine and Pharmacy, Faculty of Medicine, Department of Women's and Children's Health, Research group (Dept. of women´s and children´s health), Neuropediatrics/Paediatric oncology.
    Ranta, Susanna
    Karolinska Inst, Dept Womens & Childrens Hlth, Stockholm, Sweden;Karolinska Univ Hosp, Stockholm, Sweden.
    Posterior reversible encephalopathy syndrome in children with acute lymphoblastic leukemia: Clinical characteristics, risk factors, course, and outcome of disease2019In: Pediatric Blood & Cancer, ISSN 1545-5009, E-ISSN 1545-5017, Vol. 66, no 5, article id e27594Article in journal (Refereed)
    Abstract [en]

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

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

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

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

  • 4.
    Bergmann, Anke K.
    et al.
    Christian Albrechts Univ Kiel, Univ Hosp Schleswig Holstein, Inst Human Genet, Campus Kiel,Schwanenweg 24, Kiel, Germany.;Christian Albrechts Univ Kiel, Univ Hosp Schleswig Holstein, Dept Pediat, Campus Kiel, Kiel, Germany..
    Castellano, Giancarlo
    Univ Barcelona, Inst Biomed August Pi Sunyer IDIBAPS, Dept Analomia Palol & Microbiol, Dept Farmacol & Microbiol, Barcelona, Spain..
    Alten, Julia
    Christian Albrechts Univ Kiel, Univ Hosp Schleswig Holstein, Dept Pediat, Campus Kiel, Kiel, Germany..
    Ammerpohl, Ole
    Christian Albrechts Univ Kiel, Univ Hosp Schleswig Holstein, Inst Human Genet, Campus Kiel,Schwanenweg 24, Kiel, Germany..
    Kolarova, Julia
    Christian Albrechts Univ Kiel, Univ Hosp Schleswig Holstein, Inst Human Genet, Campus Kiel,Schwanenweg 24, Kiel, Germany.;Univ Ulm, Univ Med Ctr Ulm, Inst Human Genet, Ulm, Germany..
    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.
    Martin-Subero, Jose Ignacio
    Univ Barcelona, Inst Biomed August Pi Sunyer IDIBAPS, Dept Analomia Palol & Microbiol, Dept Farmacol & Microbiol, Barcelona, Spain..
    Schrappe, Martin
    Christian Albrechts Univ Kiel, Univ Hosp Schleswig Holstein, Dept Pediat, Campus Kiel, Kiel, Germany..
    Siebert, Reiner
    Christian Albrechts Univ Kiel, Univ Hosp Schleswig Holstein, Inst Human Genet, Campus Kiel,Schwanenweg 24, Kiel, Germany.;Univ Ulm, Univ Med Ctr Ulm, Inst Human Genet, Ulm, Germany..
    DNA methylation profiling of pediatric B-cell lymphoblastic leukemia with KMT2A rearrangement identifies hypomethylation at enhancer sites2017In: Pediatric Blood & Cancer, ISSN 1545-5009, E-ISSN 1545-5017, Vol. 64, no 3, article id e26251Article in journal (Refereed)
    Abstract [en]

    Deregulation of the epigenome is an important pathogenetic mechanism in acute lymphoblastic leukemia (ALL) with lysine (K)-specific methyltransferase 2A rearrangement (KMT2Ar). We performed array-based DNA methylation profiling of KMT2Ar ALL cells from 26 children in comparison to normal B-cell precursors. Significant changes in DNA methylation in KMT2Ar ALL were identified in 2,545 CpG loci, influenced by age and the translocation partners AFF1 and MLLT1. In KMT2Ar ALL, DNA methylation loss was enriched at enhancers and for certain transcription factor binding sites such as BCL11A, EBF, and MEF2A. In summary, DNA methylation changes in KMT2Ar ALL target enhancers, genes involved in leukemogenesis and normal hematopoiesis, as well as transcription factor networks.

  • 5.
    Bochennek, K.
    et al.
    Univ Hosp Frankfurt, Pediat Hematol & Oncol, Frankfurt, Germany..
    Dantonello, T.
    Olgahosp Klinikum Stuttgart, Cooperat Weichteilsarkom Studie CWS Study Ctr, Stuttgart, Germany..
    Borkhardt, A.
    Univ Hosp Duesseldorf, Pediat Hematol & Oncol, Dusseldorf, Germany..
    Dirksen, U.
    Univ Hosp Muenster, Pediat Oncol & Hematol, Munster, Germany..
    Eggert, A.
    Charite, Pediat Hematol & Oncol, D-13353 Berlin, Germany..
    Greiner, J.
    Ostschweizer Sauglings & Kinderspital, Ctr Pediat Hematol & Oncol, CH-9007 St Gallen, Switzerland..
    Handgretinger, R.
    Univ Hosp, Tubingen, Germany.;Fac Med Tuebingen, Genereal Pediat Hematol & Oncol, Tubingen, Germany..
    Kazanowska, B.
    Univ Hosp Wroclaw, Dept Pediat Bone Marrow Transplantat Oncol & Hema, Wroclaw, Poland..
    Kratz, C.
    Hannover Med Sch, Pediat Hematol & Oncol, Hannover, Germany..
    Ladenstein, R.
    St Anna Childrens Hosp, CCRI, Vienna, Austria..
    Ljungman, Gustaf
    Childrens Univ Hosp Uppsala, Dept Pediat Hematol & Oncol, Uppsala, Sweden..
    Hallmen, E.
    Olgahosp Klinikum Stuttgart, Cooperat Weichteilsarkom Studie CWS Study Ctr, Stuttgart, Germany..
    Koscielniak, E.
    Olgahosp Klinikum Stuttgart, Cooperat Weichteilsarkom Studie CWS Study Ctr, Stuttgart, Germany..
    Klingebiel, T.
    Univ Hosp Frankfurt, Pediat Hematol & Oncol, Frankfurt, Germany..
    Better Outcome With Maintenance Therapy: Pediatric Patients With Stage Iv Soft Tissue Sarcoma Benefit From Long Term Therapy Compared To Sct Or No Further Therapy2015In: Pediatric Blood & Cancer, ISSN 1545-5009, E-ISSN 1545-5017, Vol. 62, p. S152-S152Article in journal (Other academic)
  • 6. Carlsson, G
    et al.
    Winiarski, J
    Ljungman, P
    Ringdén, O
    Mattsson, J
    Nordenskjöld, M
    Touw, I
    Henter, J-I
    Palmblad, J
    Fadeel, B
    Hägglund, Hans
    Hematology Center, Department of Medicine, Karolinska University Hospital, Karolinska Institutet, Stockholm, Sweden.
    Hematopoietic stem cell transplantation in severe congenital neutropenia2011In: Pediatric Blood & Cancer, ISSN 1545-5009, E-ISSN 1545-5017, Vol. 56, no 3, p. 444-451Article in journal (Refereed)
    Abstract [en]

    BACKGROUND:

    Severe congenital neutropenia (SCN) is an immunodeficiency characterized by disturbed myelopoiesis and an absolute neutrophil count (ANC) <0.5 × 10(9)/L. SCN is also a premalignant condition; a significant proportion of patients develop myelodysplastic syndrome or leukemia (MDS/L). Allogeneic hematopoietic stem cell transplantation (HSCT) is the only curative treatment for SCN.

    PROCEDURE:

    Since 2004, eight HSCT have been performed in seven patients at our center. The indications were transformation to MDS/L (n = 2), granulocyte colony-stimulating factor receptor (CSF3R) mutation(s) (n = 2), granulocyte colony-stimulating factor (G-CSF) resistance (n = 2), and at the patient's own request (n = 1).

    RESULTS:

    The mean age at transplantation was 13 years (2.8-28 years) (mean follow-up 32 months, range 21-60). Three patients harbored ELANE mutations, three HAX1 mutations, and in one patient no causative mutation was identified. Two of the ELANE mutations were novel mutations. Three patients initially received myeloablative conditioning and four had reduced intensity conditioning (RIC). Three grafts were from HLA-identical siblings, three from matched unrelated donors and two were cord blood units. Engraftment occurred in all patients. Two of seven (29%) patients died; both had MDS/L and both were among the three that underwent myeloablative conditioning. One patient has chronic GVHD 2 years post-transplant.

    CONCLUSIONS:

    The role of HSCT should be explored further in patients with SCN. In particular, the influence of the conditioning regime needs to be evaluated in a larger cohort of patients.

  • 7. Carlsson, Göran
    et al.
    Elinder, Göran
    Malmgren, Helena
    Trebinska, Alicja
    Grzybowska, Ewa
    Dahl, Niklas
    Uppsala University, Disciplinary Domain of Medicine and Pharmacy, Faculty of Medicine, Department of Genetics and Pathology.
    Nordenskjöld, Magnus
    Fadeel, Bengt
    Compound heterozygous HAX1 mutations in a Swedish patient with severe congenital neutropenia and no neurodevelopmental abnormalities2009In: Pediatric Blood & Cancer, ISSN 1545-5009, E-ISSN 1545-5017, Vol. 53, no 6, p. 1143-1146Article in journal (Refereed)
    Abstract [en]

    Kostmann disease or severe congenital neutropenia (SCN) is an autosomal recessive disorder of neutrophil production. Homozygous HAX1 mutations were recently identified in SCN patients belonging to the original family in northern Sweden described by Kostmann. Moreover, recent studies have suggested an association between neurological dysfunction and HAX1 deficiency. Here we describe a patient with a compound heterozygous HAX1 mutation consisting of a nonsense mutation (c.568C > T, p.Glu190X) and a frame-shift mutation (c.91delG, p.Glu31LysfsX54) resulting in a premature stop codon. The patient has a history of neutropenia and a propensity for infections, but has shown no signs of neurodevelopmental abnormalities.

  • 8. Dantonello, Tobias
    et al.
    Handgretinger, Rupert
    Kosztyla, Daniel
    Kube, Stefanie
    Leuschners, Ivo
    Boelling, Tobias
    Bielack, Stefan S.
    Ladenstein, Ruth
    Niggli, Felix
    Kazanowska, Bernarda
    Ljungman, Gustaf
    Uppsala University, Disciplinary Domain of Medicine and Pharmacy, Faculty of Medicine, Department of Women's and Children's Health.
    Treuner, Joern
    Klingebiel, Thomas
    Koscielniak, Ewa
    SURVIVAL AFTER RECURRENCE OF METASTATIC RHAFIDOMYOSARCOMA - AN UNPROBABLE, BUT NOT IMPOSSIBLE EVENT IN THE EXPERIENCE OF THE COOPERATIVE WEICHTEILSARKOM STUDIENGRUPPE (CWS)2010In: Pediatric Blood & Cancer, ISSN 1545-5009, E-ISSN 1545-5017, Vol. 55, no 5, p. 900-900Article in journal (Other academic)
  • 9. Dantonello, Tobias
    et al.
    Leuschner, Ivo
    Greulich, Michael
    Schuck, Andreas
    Ljungman, Gustaf
    Uppsala University, Disciplinary Domain of Medicine and Pharmacy, Faculty of Medicine, Department of Women's and Children's Health.
    Pal, Niklas
    Berthold, Frank
    Bielack, Stefan S.
    Treuner, Joern
    Klingebiel, Thomas
    Koscielniak, Ewa
    MALIGNANT ECTOMESENCHYMOMA TREATED IN THE COOPERATIVE WEICHTEILSARKOM STUDIENGRUPPE (CWS)2010In: Pediatric Blood & Cancer, ISSN 1545-5009, E-ISSN 1545-5017, Vol. 55, no 5, p. 900-901Article in journal (Other academic)
  • 10. Dantonello, Tobias M.
    et al.
    Int-Veen, Christoph
    Schuck, Andreas
    Seitz, Guido
    Leuschner, Ivo
    Nathrath, Michaela
    Schlegel, Paul-Gerhardt
    Kontny, Udo
    Behnisch, Wolfgang
    Veit-Friedrich, Iris
    Kube, Stefanie
    Hallmen, Erika
    Kazanowska, Bernarda
    Ladenstein, Ruth
    Paulussen, Michael
    Ljungman, Gustaf
    Uppsala University, Disciplinary Domain of Medicine and Pharmacy, Faculty of Medicine, Department of Women's and Children's Health, Pediatrics.
    Bielack, Stefan S.
    Klingebiel, T.
    Koscielniak, E.
    Survival following disease recurrence of primary localized alveolar rhabdomyosarcoma2013In: Pediatric Blood & Cancer, ISSN 1545-5009, E-ISSN 1545-5017, Vol. 60, no 8, p. 1267-1273Article in journal (Refereed)
    Abstract [en]

    Background Recurrences in primary localized alveolar rhabdomyosarcoma (RMA) are common. Post-relapse survival is poor. We evaluated prognostic factors including relapse treatment in patients with recurrent RMA. Methods Relapses occurred in 115/235 patients with nonmetastatic RMA treated in four consecutive CWS-trials after achievement of a complete remission. Sufficient information about post-relapse treatment and outcome could be obtained in 99 patients and was retrospectively analyzed. Results Nine of 99 patients received no salvage therapy and died after a median of 2 months. The remaining 90 patients received multimodal relapse treatment including mandatory chemotherapy. Recurrences were grossly resected in 39 patients; 57 patients received radiation. At a median follow-up from relapse of 8 years, 20 patients were alive and disease-free (5-year post-relapse survival [PROS] 21.3 +/- 8). All surviving patients apart from a single individual had an isolated, circumscribed recurrence. Sixteen of 20 survivors were treated with adequate local relapse therapy (ALRT, i.e., either complete resection or gross resection+radiation). Survival in the subgroup of 27 individuals with circumscribed recurrences and ALRT was significantly better (PROS 53.7 +/- 19) compared with disseminated recurrences and/or tumors treated without ALRT. Absence of primary lymph node involvement, circumscribed relapses, ALRT, and achievement of a second CR were identified as independent favorable risk factors. Conclusion Post-relapse survival for primary localized RMA is generally poor. However, certain patient groups differed significantly in their likelihood of survival and 50% of patients with circumscribed relapses treated with ALRT survived. These findings may form the basis for an evidence-based risk-stratification for recurrent disease including relapse treatment. 

  • 11. Dantonello, Tobias M.
    et al.
    Kube, Stefanie
    von Kalle, Thekla
    Carrle, Dorothe
    Feuchtgruber, Simone
    Kosztyla, Daniel
    Niggli, Felix
    Ljungman, Gustaf
    Uppsala University, Disciplinary Domain of Medicine and Pharmacy, Faculty of Medicine, Department of Women's and Children's Health, Pediatrics.
    Bielack, Stefan S.
    Klingebiel, Thomas
    Koscielniak, Ewa
    Winkler, Peter
    Improved radiologic assessment of children with soft tissue sarcoma in the framework of the cooperative weichteilsarkom studiengruppe (CWS)2012In: Pediatric Blood & Cancer, ISSN 1545-5009, E-ISSN 1545-5017, Vol. 59, no 6, p. 977-977Article in journal (Other academic)
  • 12.
    Dantonello, Tobias M
    et al.
    Olgahospital, Pediatrics 5, Klinikum Stuttgart, Germany.
    Leuschner, Ivo
    Vokuhl, Christian
    Gfroerer, Stefan
    Schuck, Andreas
    Kube, Stefanie
    Nathrath, Michaela
    Bernbeck, Benedikt
    Kaatsch, Peter
    Pal, Niklas
    Ljungman, Gustaf
    Uppsala University, Disciplinary Domain of Medicine and Pharmacy, Faculty of Medicine, Department of Women's and Children's Health, Pediatrics.
    Bielack, Stefan S
    Klingebiel, Thomas
    Koscielniak, Ewa
    Olgahospital, Pediatrics 5, Klinikum Stuttgart, Germany.
    Malignant ectomesenchymoma in children and adolescents: Report from the Cooperative Weichteilsarkom Studiengruppe (CWS)2013In: Pediatric Blood & Cancer, ISSN 1545-5009, E-ISSN 1545-5017, Vol. 60, no 2, p. 224-229Article in journal (Refereed)
    Abstract [en]

    BACKGROUND:

    Malignant ectomesenchymoma (MEM) is a soft tissue tumor with heterologous rhabdomyoblastic components believed to arise from pluripotent migratory neural crest cells. To date merely 50 cases have been published and the knowledge about the course of disease and optimal treatment is limited.

    METHODS:

    Six patients with MEM were registered 1996-2009. The diagnosis was confirmed according to current criteria. Their treatment and outcome was analyzed.

    RESULTS:

    The median age of the three females and three males was 0.6 years (range, 0.2-13.5). The mesenchymal component in all tumors was rhabdomyosarcoma (RMS), the neural component ganglioneuroblastoma/neuroblastoma (n = 5) and peripheral primitive neuroectodermal tumor in one case. Five patients presented with localized, one with metastatic disease. All but one patient received multiagent chemotherapy during their initial treatment. The tumors of 4/5 patients with localized MEM were at least grossly resected at best surgery; the patient without gross resection was additionally irradiated. Three of four evaluable tumors responded well to induction chemotherapy. All patients achieved a first complete remission (CR), but three recurrences (two local, one systemic) occurred. The individual with metastatic MEM did not survive, but all five patients with localized MEM are currently alive in CR with a median follow-up of 5 years (range: 2.1-13.7).

    CONCLUSIONS:

    Risk-factors and outcome of MEM appear to be comparable with other highly malignant pediatric soft tissue sarcoma when a multimodal treatment strategy including chemotherapy and adequate local treatment is pursued. We propose that treatment of patients with MEM be done according to pediatric protocols similar to other rhabdomyosarcoma-like soft tissue sarcoma.

  • 13. Dantonello, Tobias M.
    et al.
    Schuck, Andreas
    Kube, Stefanie
    Greulich, Michael
    Kuehnle, Ingrid
    Feuchtgruber, Simone
    Kosztyla, Daniel
    Leuschner, Ivo
    Kazanowska, Bernarda
    Godzinski, Jan
    Ladenstein, Ruth
    Niggli, Felix
    Ljungman, Gustaf
    Uppsala University, Disciplinary Domain of Medicine and Pharmacy, Faculty of Medicine, Department of Women's and Children's Health, Pediatrics.
    Bielack, Stefan S.
    Klingebiel, Thomas
    Koscielniak, Ewa
    Delayed resection can avoid irradiation in localized embryonal rhabdomyosarcoma2012In: Pediatric Blood & Cancer, ISSN 1545-5009, E-ISSN 1545-5017, Vol. 59, no 6, p. 977-977Article in journal (Other academic)
  • 14.
    Dantonello, Tobias M
    et al.
    Paediatrics 5 (oncology, hematology, immunology), Olgahospital Klinikum Stuttgart, Germany.
    Stark, Monika
    Paediatrics 5 (oncology, hematology, immunology), Olgahospital Klinikum Stuttgart, Germany.
    Timmermann, Beate
    Fuchs, Jörg
    Selle, Barbara
    Linderkamp, Christin
    Handgretinger, Rupert
    Hagen, Rudolf
    Feuchtgruber, Simone
    Kube, Stefanie
    Kosztyla, Daniel
    Kazanowska, Bernarda
    Ladenstein, Ruth
    Niggli, Felix
    Ljungman, Gustaf
    Uppsala University, Disciplinary Domain of Medicine and Pharmacy, Faculty of Medicine, Department of Women's and Children's Health, Pediatrics.
    Bielack, Stefan S
    Klingebiel, Thomas
    Koscielniak, Ewa
    Paediatrics 5 (oncology, hematology, immunology), Olgahospital Klinikum Stuttgart, Germany.
    Tumour volume reduction after neoadjuvant chemotherapy impacts outcome in localised embryonal rhabdomyosarcoma2015In: Pediatric Blood & Cancer, ISSN 1545-5009, E-ISSN 1545-5017, Vol. 62, no 1, p. 16-23Article in journal (Refereed)
    Abstract [en]

    BACKGROUND: Response (tumour volume reduction) to induction chemotherapy has been used to stratify secondary local and systemic treatment of Intergroup Rhabdomyosarcoma Study Group III (IRSG-III) embryonal rhabdomyosarcoma (RME) in consecutive CWS-trials. To evaluate its actual impact we studied response-related treatment and outcomes.

    PROCEDURE: Patients with IRSG-III RME <21 years and non-response (NR, <33% volume reduction) in five consecutive CWS-trials were analysed and compared with partial responders (PAR, ≥33% reduction). The NR was reviewed and sub-classified as Objective Response (OR, <0%-33% reduction) or Stable/Progressive Disease (SPD).

    RESULTS: Fifty-nine of 529 patients had NR (n = 34 OR, n = 25 SPD). Primary risk-factors including age, tumour size, and TN-classification did not differ between NR and PAR groups but NR had more patients with unfavourable sites comparatively (P = 0.04). There were no differences in primary risk-factors between OR and SPD. Significant factors associated with poor outcome in multivariate analysis were NR, TN-classification, age >10 years, tumour size >5 cm and therapy in older trials. After response assessment n = 24 NR continued to receive induction chemotherapy, n = 32 received other combinations and n = 3 no further chemotherapy. Forty-two non-responders were irradiated, and the tumours were completely resected in n = 20. After a median follow-up of 8 years, 34 NR are alive. Seventeen of 21 failures leading to disease-related deaths were locoregional. The five-year overall survival rate (OS) was 76 ± 4% for PAR, 79 ± 14% for OR, but only 40 ± 19% for SPD (P < 0.001).

    CONCLUSION: Response to induction chemotherapy appears to be an important surrogate marker of poor outcome in patients with SPD largely due to ineffective local control.

  • 15.
    Dantonello, Tobias M
    et al.
    Pediatrics 5 (Oncology, Hematology, Immunology), Olgahospital, Klinikum Stuttgart, Germany.
    Winkler, Peter
    Department of Pediatric Radiology, Olgahospital, Klinikum Stuttgart, Germany.
    Boelling, Tobias
    Department of Radiotherapy, University of Muenster, Muenster, Germany.
    Friedel, Godehard
    Department of Thoracic Surgery, Klinik Schillerhoehe, Gerlingen, Germany.
    Schmid, Irene
    Department of Pediatric Oncology, Dr. von Haunersches Kinderspital, University of Munich, Munich, Germany.
    Mattke, Adrian C
    Department for Pediatric Intensive Care Medicine, Royal Children’s Hospital, Parkville, Victoria, Australia.
    Ljungman, Gustaf
    Uppsala University, Disciplinary Domain of Medicine and Pharmacy, Faculty of Medicine, Department of Women's and Children's Health, Pediatrics.
    Bielack, Stefan S
    Pediatrics 5 (Oncology, Hematology, Immunology), Olgahospital, Klinikum Stuttgart, Germany.
    Klingebiel, Thomas
    Department of Pediatric Oncology, University of Frankfurt, Frankfurt (Main), Germany.
    Koscielniak, Ewa
    Pediatrics 5 (Oncology, Hematology, Immunology), Olgahospital, Klinikum Stuttgart, Germany.
    Embryonal rhabdomyosarcoma with metastases confined to the lungs: Report from the CWS Study Group.2011In: Pediatric Blood & Cancer, ISSN 1545-5009, E-ISSN 1545-5017, Vol. 56, no 5, p. 725-732Article in journal (Refereed)
    Abstract [en]

    BACKGROUND: Embryonal rhabdomyosarcoma [RME] is the most common pediatric soft tissue sarcoma. Whereas the prognosis of localized rhabdomyosarcoma has improved, it remains poor for metastatic disease. METHODS: We analyzed RME-patients with isolated pulmonary metastases [PRME] treated in four consecutive CWS-trials. Treatment included multiagent chemotherapy and local treatment of the primary tumor. Therapy of lung metastases after induction chemotherapy depended on response and individual decisions. RESULTS: Twenty-nine patients <21 years had PRME. Their median age was six years, the median follow-up nine years. Twenty-eight children had their primary tumor located in an unfavorable site and 22 of the primaries were >5 cm. In addition to conventional chemotherapy, seven patients received high-dose treatment and eight patients oral metronomic chemotherapy. The lung metastases were in remission after induction chemotherapy in 22 individuals. 19 patients received no local treatment of metastases; 3 patients had pulmonary metastasectomy and lung radiation was administered to 9 individuals. In total, 24/29 patients achieved a complete remission [CR]. Actuarial 5-year event-free and overall survival for all patients was 37.9 ± 18% and 48.7 ± 18%, respectively; it was 45.8 ± 20% and 58.3 ± 20% for the 24 patients who achieved a CR. Local treatment of metastases had no impact on the failure pattern. Younger age, good response, achievement of CR and maintenance-treatment were favorable prognostic factors in univariate analysis. CONCLUSIONS: Children with PRME have a fair prognosis. Local treatment of metastases did not improve outcome in our sample. Metronomic treatment may be an attractive option for PREM-patients. [correction made here after initial online publication].

  • 16.
    Fogelstrand, Linda
    et al.
    Department of Clinical Chemistry and Transfusion Medicine, Institute of Biomedicine, Sahlgrenska Academy at University of Gothenburg, Sweden.
    Staffas, Anna
    Department of Clinical Chemistry and Transfusion Medicine, Institute of Biomedicine, Sahlgrenska Academy at University of Gothenburg, Sweden.
    Wasslavik, Carina
    Department of Clinical Chemistry and Transfusion Medicine, Institute of Biomedicine, Sahlgrenska Academy at University of Gothenburg, Sweden.
    Sjögren, Helene
    Department of Clinical Chemistry and Transfusion Medicine, Institute of Biomedicine, Sahlgrenska Academy at University of Gothenburg, Sweden.
    Söderhäll, Stefan
    Childhood Cancer Research Unit, Department of Women and Child Health, Astrid Lindgren Children's Hospital, Stockholm, Sweden.
    Frost, Britt-Marie
    Uppsala University, Disciplinary Domain of Medicine and Pharmacy, Faculty of Medicine, Department of Women's and Children's Health.
    Forestier, Erik
    Department of Medical Biosciences, Clinical Genetics, Umeå University, Sweden.
    Degerman, Sofie
    Department of Medical Biosciences, Pathology, Umeå University, Sweden.
    Behrendtz, Mikael
    Department of Paediatrics, University Hospital Linköping, Sweden.
    Heldrup, Jesper
    Department of Pediatrics, Skåne University Hospital, Lund University, Sweden.
    Karrman, Kristina
    Department of Clinical Genetics, University and Regional Laboratories, Lund University, Sweden.
    Johansson, Bertil
    Department of Clinical Genetics, University and Regional Laboratories, Lund University, Sweden.
    Heyman, Mats
    Department of Woman and Child Health, Karolinska Institute, Stockholm, Sweden.
    Abrahamsson, Jonas
    Department of Pediatrics, Institute of Clinical Sciences, Sahlgrenska Academy at University of Gothenburg, Sweden.
    Palmqvist, Lars
    Department of Clinical Chemistry and Transfusion Medicine, Institute of Biomedicine, Sahlgrenska Academy at University of Gothenburg, Sweden.
    Prognostic implications of mutations in NOTCH1 and FBXW7 in childhood T-ALL treated according to the NOPHO ALL-1992 and ALL-2000 protocols.2014In: Pediatric Blood & Cancer, ISSN 1545-5009, E-ISSN 1545-5017, Vol. 61, no 3, p. 424-30Article in journal (Refereed)
    Abstract [en]

    BACKGROUND: In children, T-cell acute lymphoblastic leukemia (T-ALL) has inferior prognosis compared with B-cell precursor ALL. In order to improve survival, individualized treatment strategies and thus risk stratification algorithms are warranted, ideally already at the time of diagnosis.

    PROCEDURE: We analyzed the frequency and prognostic implication of mutations in NOTCH1 and FBXW7 in 79 cases of Swedish childhood T-ALL treated according to the Nordic Society of Pediatric Hematology and Oncology (NOPHO) ALL-1992 and ALL-2000 protocols. In a subgroup of patients, we also investigated the functional relevance of NOTCH1 mutations measured as expression of the HES1, MYB, and MYC genes.

    RESULTS: Forty-seven of the cases (59%) displayed mutations in NOTCH1 and/or FBXW7. There was no difference in overall (P = 0.14) or event-free survival (EFS) (P = 0.10) in patients with T-ALL with mutation(s) in NOTCH1/FBXW7 compared with patients with T-ALL without mutations in any of these genes. T-ALL carrying NOTCH1 mutations had increased HES1 and MYB mRNA expression (HES1 9.2 ± 1.9 (mean ± SEM), MYB 8.7 ± 0.8 (mean ± SEM)) compared to T-ALL with wild-type NOTCH1 (HES1 1.8 ± 0.7, MYB 5.1 ± 1.2, P = 0.02 and 0.008, respectively). In cases of T-ALL with high HES1 expression, improved overall (P = 0.02) and EFS (P = 0.028) was seen.

    CONCLUSIONS: Increased NOTCH activity, reflected by increased HES1 expression, is associated with improved outcome in pediatric T-ALL, but its role as a diagnostic tool or a therapeutic target in future clinical treatment protocols remains to be elucidated.

  • 17.
    Frisk, Per
    et al.
    Uppsala University, Disciplinary Domain of Medicine and Pharmacy, Faculty of Medicine, Department of Women's and Children's Health, Pediatrics.
    Arvidson, Johan
    Uppsala University, Disciplinary Domain of Medicine and Pharmacy, Faculty of Medicine, Department of Women's and Children's Health, Pediatrics.
    Hedenström, Hans
    Uppsala University, Disciplinary Domain of Medicine and Pharmacy, Faculty of Medicine, Department of Medical Sciences, Clinical Physiology.
    A longitudinal study of pulmonary function after stem cell transplantation, from childhood to young adulthood2012In: Pediatric Blood & Cancer, ISSN 1545-5009, E-ISSN 1545-5017, Vol. 58, no 5, p. 775-779Article in journal (Refereed)
    Abstract [en]

    Background Impairment of pulmonary function after stem cell transplantation (SCT) in childhood has been reported before. However, long-term longitudinal studies are scarce.

    Procedure. We measured lung volumes and performed dynamic spirometry serially in 18 patients after SCT. At the last investigation, a median of 18.2 years after SCT, the patients were compared with 18 matched controls. The diffusing capacity (DLCO) was only compared cross-sectionally.

    Results. There was a significant increase in the prevalence of restrictive lung disease (RLD, total lung capacity <80% of that predicted) from 7% (1/14) before SCT to 28% (5/18) 5 years after SCT, and 61% (11/18) a median of 18.2 years after SCT (P = 0.002). In comparison, none of the controls had RLD (61% vs. 0%, P = 0.001). Before SCT, no patient had obstructive lung disease (OLD, forced expiratory volume in 1 sec/vital capacity < 70). OLD was found in one of 18 patients (6%) 5 years after SCT but in none of the patients a median of 18.2 years after SCT. Three of the controls had OLD (P = 0.25). Eleven patients had diffusion impairment (DLCO < 80% of that predicted), as opposed to none of the controls (P = 0.001). The DLCO corrected for alveolar volume was decreased in only two patients.

    Conclusion. We documented an increase in the prevalence of RLD, but not of OLD, after SCT. At the last investigation, only two patients had diffusion impairment after correction for alveolar volume.

  • 18.
    Genberg, Margareta
    et al.
    Uppsala University, Disciplinary Domain of Medicine and Pharmacy, Faculty of Medicine, Department of Medical Sciences, Clinical Physiology.
    Öberg, Anders
    Uppsala University, Disciplinary Domain of Medicine and Pharmacy, Faculty of Medicine, Department of Women's and Children's Health, Pediatrics.
    Andrén, Bertil
    Uppsala University, Disciplinary Domain of Medicine and Pharmacy, Faculty of Medicine, Department of Medical Sciences, Clinical Physiology.
    Hedenström, Hans
    Uppsala University, Disciplinary Domain of Medicine and Pharmacy, Faculty of Medicine, Department of Medical Sciences, Clinical Physiology.
    Frisk, Per
    Uppsala University, Disciplinary Domain of Medicine and Pharmacy, Faculty of Medicine, Department of Women's and Children's Health, Pediatrics.
    Flachskampf, Frank A.
    Uppsala University, Disciplinary Domain of Medicine and Pharmacy, Faculty of Medicine, Department of Medical Sciences, Cardiology.
    Cardiac Function After Hematopoietic Cell Transplantation: An Echocardiographic Cross-Sectional Study in Young Adults Treated in Childhood2015In: Pediatric Blood & Cancer, ISSN 1545-5009, E-ISSN 1545-5017, Vol. 62, no 1, p. 143-147Article in journal (Refereed)
    Abstract [en]

    BackgroundHematopoietic cell transplantation (HCT) including preparative regimens with chemotherapy and total body irradiation (TBI) is an accepted treatment for many malignant disorders but may have side-effects for several organs, including the cardiovascular system. The aim of this study was to study very long-term consequences on cardiac function after childhood HCT. ProcedureCardiac function was evaluated using echocardiography and levels of NT-proBNP and growth hormone (GHmax) in 18 patients, at a median of 18 years after HCT including TBI, and in 18 matched controls. ResultsPatients after HCT had cardiac dimensions, volumes, and left ventricular ejection fractions within normal range after correction for body size. However, compared with the control group, patients after HCT had significantly lower E/A ratio, as a measure of left ventricular diastolic function, significantly lower fractional shortening and mitral annular plane systolic excursion, as measures of left ventricular systolic function, significantly lower tricuspid annular plane systolic excursion, as a measure of right ventricular function, and significantly higher NT-proBNP, as a measure of total cardiac function. Also, pulmonary flow acceleration time was shorter in the group after HCT, indicating possible pulmonary involvement. Heart rate was significantly higher and GHmax significantly lower in patients after HCT. ConclusionsAlmost two decades after HCT, including preparative regimens with TBI, cardiac function in patients was found to be within normal range. However, when compared with a healthy control group, patients after HCT showed lower systolic and diastolic left ventricular function as well as lower right ventricular function. Pediatr Blood Cancer 2015;62:143-147.

  • 19.
    Georgantzi, Clary
    et al.
    Uppsala University, Disciplinary Domain of Medicine and Pharmacy, Faculty of Medicine, Department of Women's and Children's Health.
    Sköldenberg, Erik
    Uppsala University, Disciplinary Domain of Medicine and Pharmacy, Faculty of Medicine, Department of Women's and Children's Health.
    Stridsberg, Mats
    Uppsala University, Disciplinary Domain of Medicine and Pharmacy, Faculty of Medicine, Department of Medical Sciences, Biochemical endocrinology.
    Jakobson, A.
    Christofferson, Rolf
    Uppsala University, Disciplinary Domain of Medicine and Pharmacy, Faculty of Medicine, Department of Women's and Children's Health, Paediatric Surgery.
    Chromogranin A In Neuroblastoma: Correlation To Stage And Prognostic Factors2013In: Pediatric Blood & Cancer, ISSN 1545-5009, E-ISSN 1545-5017, Vol. 60, no S3, p. 228-228Article in journal (Refereed)
  • 20.
    Georgantzi, Kleopatra
    et al.
    Uppsala University, Disciplinary Domain of Medicine and Pharmacy, Faculty of Medicine, Department of Women's and Children's Health, Pediatrics.
    Tsolakis, Apostolos V
    Uppsala University, Disciplinary Domain of Medicine and Pharmacy, Faculty of Medicine, Department of Medical Sciences, Endocrine Oncology.
    Stridsberg, Mats
    Uppsala University, Disciplinary Domain of Medicine and Pharmacy, Faculty of Medicine, Department of Medical Sciences, Biochemical endocrinology.
    Jakobson, Åke
    Department of Womeńs and Childreńs Health, Astrid Lindgren Childreńs Hospital, Karolinska Institute, Stockholm, Sweden.
    Christofferson, Rolf
    Uppsala University, Disciplinary Domain of Medicine and Pharmacy, Faculty of Medicine, Department of Women's and Children's Health, Paediatric Surgery.
    Janson, Eva Tiensuu
    Uppsala University, Disciplinary Domain of Medicine and Pharmacy, Faculty of Medicine, Department of Medical Sciences, Endocrine Oncology.
    Differentiated expression of somatostatin receptor subtypes in experimental models and clinical neuroblastoma2011In: Pediatric Blood & Cancer, ISSN 1545-5009, E-ISSN 1545-5017, Vol. 56, no 4, p. 584-589Article in journal (Refereed)
    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.

  • 21.
    Glosli, H.
    et al.
    Oslo Univ Hosp HF, Dept Paediat & Adolescent Med, Oslo, Norway..
    Bartholdson, C.
    Karolinska Inst, Dept Paediat, Stockholm, Sweden..
    Broner, T.
    Arhus Univ Hosp, Dept Paediat, Aarhus, Denmark..
    Hauge, H. F.
    Oslo Univ Hosp, Dept Paediat & Adolescent Med, Oslo, Norway..
    Karlsson, S.
    Uppsala University, Disciplinary Domain of Medicine and Pharmacy, Faculty of Medicine, Department of Women's and Children's Health, Pediatrics.
    Pergert, P.
    Karolinska Inst, Dept Paediat, Stockholm, Sweden..
    Poroddsdottir, S.
    Landspitalinn Isl, Dept Paediat, Reykjavik, Iceland..
    Tornudd, L.
    Linkoping Hosp, Dept Paediat, Linkoping, Sweden..
    Stigmar, J.
    Univ Lund Hosp, Dept Paediat, Lund, Sweden..
    Petersen, G.
    Copenhagen Univ Hosp, Dept Paediat, Copenhagen, Denmark..
    Enhancing Nurse/Physician Collaboration in Ethical Issues in Paediatric Oncology2016In: Pediatric Blood & Cancer, ISSN 1545-5009, E-ISSN 1545-5017, Vol. 63, p. S226-S226Article in journal (Refereed)
  • 22.
    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.

  • 23.
    Hedborg, Fredrik
    et al.
    Uppsala University, Disciplinary Domain of Medicine and Pharmacy, Faculty of Medicine, Department of Genetics and Pathology.
    Cetinkaya, Cihan
    Uppsala University, Disciplinary Domain of Medicine and Pharmacy, Faculty of Medicine, Department of Genetics and Pathology.
    Martinsson, Tommy
    Kogner, Per
    Dumanski, Jan
    Uppsala University, Disciplinary Domain of Medicine and Pharmacy, Faculty of Medicine, Department of Genetics and Pathology.
    Trager, Catarina
    de Stahl, Teresita Diaz
    Uppsala University, Disciplinary Domain of Medicine and Pharmacy, Faculty of Medicine, Department of Genetics and Pathology.
    AGE-DEPENDENT GENOTYPES IN HIGH-RISK NEUROBLASTOMA: MYCN AMPLIFICATION IS A FAST TRACK TO AGGRESSIVE DISEASE WHEREAS SEGMENTAL DELETION OF 11Q IMPLIES A MORE COMPLEX, MULTI-STEP TUMOR EVOLUTION2010In: Pediatric Blood & Cancer, ISSN 1545-5009, E-ISSN 1545-5017, Vol. 55, no 5, p. 896-896Article in journal (Other academic)
  • 24.
    Hedström, Mariann
    et al.
    Uppsala University, Disciplinary Domain of Medicine and Pharmacy, Faculty of Medicine, Department of Public Health and Caring Sciences.
    Kreuger, Anders
    Uppsala University, Disciplinary Domain of Medicine and Pharmacy, Faculty of Medicine, Department of Women's and Children's Health.
    Ljungman, Gustaf
    Uppsala University, Disciplinary Domain of Medicine and Pharmacy, Faculty of Medicine, Department of Women's and Children's Health.
    Nygren, Peter
    Uppsala University, Disciplinary Domain of Medicine and Pharmacy, Faculty of Medicine, Department of Oncology, Radiology and Clinical Immunology.
    von Essen, Louise
    Uppsala University, Disciplinary Domain of Medicine and Pharmacy, Faculty of Medicine, Department of Public Health and Caring Sciences.
    Accuracy of assessment of distress, anxiety, and depression by physicians and nurses in adolescents recently diagnosed with cancer2006In: Pediatric Blood & Cancer, ISSN 1545-5009, E-ISSN 1545-5017, Vol. 46, no 7, p. 773-9Article in journal (Refereed)
    Abstract [en]

    Background. As staff members prioritize medical resources forpatients, it is imperative to find out whether their assessments ofpatients’ health status agree with patients’ assessments. The degree towhich physicians and nurses can identify the distress, anxiety, anddepression experienced by adolescents recently diagnosed withcancer was examined here. Procedure. Adolescents undergoingchemotherapy (13–19 years, n¼53), physicians (n¼48), and nurses(n¼53) completed a structured telephone interview, 4–8 weeksafter diagnosis or relapse, investigating disease and treatment-relateddistress, anxiety, and depression. Results. The accuracy of staffratings of physical distress could be considered acceptable.However, problems of a psychosocial nature, which were frequentlyoverestimated, were difficult for staff to identify. Staff underestimatedthe distress caused by mucositis and worry about missing schoolmore than they overestimated distress. These aspects were some ofthe most prevalent and overall worst according to the adolescents.Both physicians and nurses overestimated levels of anxiety anddepression. Nurses tended to show higher sensitivity than physiciansfor distress related to psychosocial aspects of distress, whilephysicians tended to show higher accuracy than nurses for physicaldistress. Conclusions. Staff was reasonably accurate at identifyingphysical distress in adolescents recently diagnosed with cancerwhereas psychosocial problems were generally poorly identified.Thus, the use of staff ratings as a ‘‘test’’ to guide specific supportseems problematic. Considering that the accuracy of staff ratingsoutside a research study is probably lower, identification of andaction taken on adolescent problems in relation to cancer diagnosisand treatment need to rely on direct communication.

  • 25.
    Hedén, Lena
    et al.
    Uppsala University, Disciplinary Domain of Medicine and Pharmacy, Faculty of Medicine, Department of Women's and Children's Health, Pediatrics.
    Pöder, Ulrika
    Uppsala University, Disciplinary Domain of Medicine and Pharmacy, Faculty of Medicine, Department of Public Health and Caring Sciences, Caring Sciences.
    von Essen, Louise
    Uppsala University, Disciplinary Domain of Medicine and Pharmacy, Faculty of Medicine, Department of Public Health and Caring Sciences, Psychosocial oncology and supportive care.
    Ljungman, Gustaf
    Uppsala University, Disciplinary Domain of Medicine and Pharmacy, Faculty of Medicine, Department of Women's and Children's Health, Pediatrics.
    Parents' Perceptions of Their Child's Symptom Burden During and After Cancer Treatment2013In: Pediatric Blood & Cancer, ISSN 1545-5009, E-ISSN 1545-5017, Vol. 60, no S3, p. 42-42Article in journal (Other academic)
  • 26.
    Hedén, Lena
    et al.
    Uppsala University, Disciplinary Domain of Medicine and Pharmacy, Faculty of Medicine, Department of Women's and Children's Health.
    von Essen, Louise
    Uppsala University, Disciplinary Domain of Medicine and Pharmacy, Faculty of Medicine, Department of Public Health and Caring Sciences, Psychosocial oncology and supportive care.
    Frykholm, Peter
    Uppsala University, Disciplinary Domain of Medicine and Pharmacy, Faculty of Medicine, Department of Surgical Sciences, Anaesthesiology and Intensive Care.
    Ljungman, Gustaf
    Uppsala University, Disciplinary Domain of Medicine and Pharmacy, Faculty of Medicine, Department of Women's and Children's Health.
    Low-dose oral midazolam reduces fear and distress during needle procedures in children with cancer2009In: Pediatric Blood & Cancer, ISSN 1545-5009, E-ISSN 1545-5017, Vol. 53, no 7, p. 1200-1204Article in journal (Refereed)
    Abstract [en]

    BACKGROUND

    Children with cancer often mention needle procedures as the most frightening, distressing, and sometimes painful aspect of the disease and treatment. The aim was to investigate whether children experience less fear, distress, and/or pain according to parents, nurses, and children >or=7 years of age when they receive oral midazolam versus placebo before a needle is inserted in a subcutaneously implanted intravenous port.

    PROCEDURE

    Fifty children 1-18 years of age who were being treated in a pediatric oncology and hematology setting were included consecutively when undergoing routine needle insertion into an intravenous port. All children were subjected to one needle insertion in this randomized, triple-blind, placebo-controlled study in which orally administered midazolam (n = 24) 0.3 mg/kg body weight (maximum 10 mg) was compared with placebo (n = 26). Parents, nurses, and children >or=7 years reported the patients' fear, distress, and pain on 0-100 mm Visual Analogue Scales.

    RESULTS

    Fear was lower in the midazolam group according to parents (P = 0.001), nurses (P = 0.001), and children (P = 0.015). Parents and nurses also reported lower distress (P = 0.020 and 0.007, respectively). Post hoc analyses indicated that the effects were more pronounced in younger children (<7 years of age).

    CONCLUSION

    Low-dose oral midazolam was effective in reducing fear and distress in pediatric oncology patients, especially in younger children, undergoing subcutaneous port needle insertion.

  • 27.
    Jalmsell, Li
    et al.
    Uppsala University, Disciplinary Domain of Medicine and Pharmacy, Faculty of Medicine, Department of Public Health and Caring Sciences, Centre for Research Ethics and Bioethics.
    Kontio, T.
    Stein, M.
    Henter, J. I.
    Kreicbergs, U.
    On the child's own initiative: Parents communicate with their dying child about death2014In: Pediatric Blood & Cancer, ISSN 1545-5009, E-ISSN 1545-5017, Vol. 61, p. S250-S251Article in journal (Other academic)
  • 28. Jalmsell, Li
    et al.
    Kreicbergs, Ulrika
    Onelöv, Erik
    Steineck, Gunnar
    Henter, Jan-Inge
    Anxiety is contagious: symptoms of anxiety in the terminally ill chid affect long-term psychological well-being in bereaved parents2010In: Pediatric Blood & Cancer, ISSN 1545-5009, E-ISSN 1545-5017Article in journal (Refereed)
  • 29.
    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)
  • 30. Koscielniak, E.
    et al.
    Dantonello, T.
    Blank, B.
    Seitz, G.
    Leuschner, I.
    Kazanowska, B.
    Ladenstein, R.
    Ljungman, Gustaf
    Uppsala University, Disciplinary Domain of Medicine and Pharmacy, Faculty of Medicine, Department of Women's and Children's Health, Pediatrics.
    Niggli, F.
    Bielack, S.
    Hallmen, E.
    Klingebiel, T.
    Prognosis of Children and Adolescents with Soft Tissue Ewing Tumors (STET) Treated in 3 Consecutive, Prospective Studies of the Cooperative Weichsteilsarkom Studiengruppe (CWS)2014In: Pediatric Blood & Cancer, ISSN 1545-5009, E-ISSN 1545-5017, Vol. 61, p. S117-S117Article in journal (Other academic)
  • 31.
    Koscielniak, E.
    et al.
    Olgahosp Klinikum Stuttgart, Paediat Oncol Haematol, Stuttgart, Germany..
    Fuchs, J.
    Univ Klinikum Tubingen, Paediat Surg & Urol, Tubingen, Germany..
    Seitz, G.
    Univ Klinikum Tubingen, Paediat Surg & Urol, Tubingen, Germany..
    Leuschner, I.
    Univ Klinikum Schleswig Holstein, Paidopathol, Kiel, Germany..
    Scheer, M.
    Olgahosp Klinikum Stuttgart, Paediat Oncol Haematol, Stuttgart, Germany..
    Sparber-Sauer, M.
    Olgahosp Klinikum Stuttgart, Paediat Oncol Haematol, Stuttgart, Germany..
    Kazanowska, B.
    Suprareg Ctr Paediat Oncol Cape Hope, Paediat Haematol Oncol & BMT, Wroclaw, Poland..
    Ladenstein, R.
    St Anna Childrens Hosp, Childrens Canc Res Inst, Vienna, Austria..
    Ljungman, Gustaf
    Childrens Univ Hosp, Paediat Haematol & Oncol, Uppsala, Sweden..
    Niggli, F.
    Kinderspital, Paediat Oncol, Zurich, Switzerland..
    Bielack, S.
    Olgahosp Klinikum Stuttgart, Paediat Oncol Haematol, Stuttgart, Germany..
    Muenter, M.
    Katharinenhosp Klinikum Stuttgart, Radiooncol, Stuttgart, Germany..
    von Kalle, T.
    Olgahosp Klinikum Stuttgart, Paediat Radiol, Stuttgart, Germany..
    Hallmen, E.
    Olgahosp Klinikum Stuttgart, Paediat Oncol Haematol, Stuttgart, Germany..
    Klingebiel, T.
    Childrens Univ Hosp, Paediat Oncol Haematol & Hemostasis, Frankfurt, Germany..
    Prognosis of Children with Localized Rhabdomyosarcoma (RMS) of Genitourinary Regions (GU) Treated in 5 Prospective Studies of the Cooperative Weichsteilsarkom Studiengruppe (CWS)2016In: Pediatric Blood & Cancer, ISSN 1545-5009, E-ISSN 1545-5017, Vol. 63, p. S29-S30Article in journal (Refereed)
  • 32. Koscielniak, E.
    et al.
    Kosztyla, D.
    Dantonello, T.
    Kube, S.
    Kazanowska, B.
    Ladenstein, R.
    Niggli, F.
    Ljungman, Gustaf
    Uppsala University, Disciplinary Domain of Medicine and Pharmacy, Faculty of Medicine, Department of Women's and Children's Health, Pediatrics.
    Bielack, S.
    Leuschner, I.
    Schuck, A.
    Report of the CWS 2002P Study: Treatment Results for Soft Tissue Sarcomas (STS) in Childhood and Adolescence2013In: Pediatric Blood & Cancer, ISSN 1545-5009, E-ISSN 1545-5017, Vol. 60, no S3, p. 32-32Article in journal (Other academic)
  • 33.
    Ljungman, Gustaf
    et al.
    Uppsala University, Disciplinary Domain of Medicine and Pharmacy, Faculty of Medicine, Department of Women's and Children's Health.
    Hedén, Lena
    Uppsala University, Disciplinary Domain of Medicine and Pharmacy, Faculty of Medicine, Department of Women's and Children's Health.
    von Essen, Louise
    Uppsala University, Disciplinary Domain of Medicine and Pharmacy, Faculty of Medicine, Department of Public Health and Caring Sciences, Psychosocial oncology and supportive care.
    Effect of morphine on fear, distress and pain in needle procedures in children with cancer2010In: Pediatric Blood & Cancer, ISSN 1545-5009, E-ISSN 1545-5017, Vol. 55, no 5, p. 954-955Article in journal (Other academic)
    Abstract [en]

    Purpose: Children with cancer often mention needle procedures as the most frightening, distressing and sometimes painful aspect of the disease and treatment.The aim was to investigate whether children experience less fear, distress, and/or pain according to parents, nurses, and children>7 years of age when they receive oral morphine vs. placebo before a needle is inserted in a subcutaneously implanted intravenous port and if there is a difference in procedure time.

    Method: Fifty children 1–18 years of age who were being treated in a pediatric oncology and hematology setting were included consecutively when undergoing routine needle insertion into an intravenous port. All children were subjected to one needle insertion and recieved EMLA in this randomized, triple-blind, placebo controlled study in which orally administered morphine (n¼26) 0.25 mg/kg body weight was compared with placebo (n¼24). Parents, nurses and children>7 years reported the patients’ fear, distress, and pain on 0–100 mm Visual Analogue Scales. In addition behavioral observation with CHEOPS for pain and PBCL for procedure related distress was performed.

    Results: No differences between the morphine and the placebo group were found with respect to age, weight, height, physical status, sex, weeks from diagnosis, or weeks from latest needle insertion. According to parents, nurses, and children oral morphinein a dose of 0.25 mg/kg body weight did not reduce the primary outcome measure fear; neither did it reduce distress nor pain in the morphine group compared to placebo. No differences in behavioral observations with CHEOPS and PBCL or procedure time for morphine vs. placebo were found.

    Conclusion: Surprisingly, oral morphine in this dose does not add any value in reducing fear, distress or pain combined with topical anaesthesia in pediatric oncology patients undergoing subcutaneous port needle insertion, and it probably would not add any value for other similar procedures, e.g. venous cannulation.

  • 34.
    Ljungman, Gustaf
    et al.
    Uppsala University, Disciplinary Domain of Medicine and Pharmacy, Faculty of Medicine, Department of Women's and Children's Health.
    Jakobson, Åke
    Department of Paediatrics, Astrid Lindgren Children’s Hospital, Karolinska Institute, Stockholm, Sweden.
    Behrendtz, Mikael
    Department of Paediatrics, University Hospital, University of Linkoping, Linkoping, Sweden.
    Ek, Torben
    Department of Paediatrics, County Hospital, Halmstad, Sweden.
    Friberg, Lars-Göran
    Department of Paediatric Surgery, The Queen Silvia Children’s Hospital, University of Gothenburg, Gothenburg, Sweden.
    Hjalmars, Ulf
    Department of Paediatrics, University Hospital, University of Umea, Umea, Sweden.
    Hjorth, Lars
    Department of Paediatrics, University Hospital, University of Lund, Lund, Sweden.
    Lindh, Jack
    Department of Radiation Sciences, Oncology, Umea University, Umea, Sweden.
    Pal, Niklas
    Department of Paediatrics, Astrid Lindgren Children’s Hospital, Karolinska Institute, Stockholm, Sweden.
    Sandstedt, Bengt
    Department of Pathology, Karolinska Hospital, Karolinska Institute, Stockholm, Sweden.
    Österlundh, Gustaf
    Department of Paediatrics, The Queen Silvia Children’s Hospital, University of Gothenburg, Gothenburg, Sweden.
    Gustafsson, Göran
    Childhood Cancer Research Unit, Karolinska Institute, Stockholm, Sweden.
    INCIDENCE AND SURVIVAL ANALYSES IN CHILDREN WITH SOLID TUMOURS DIAGNOSED IN SWEDEN 1983-20072010In: Pediatric Blood & Cancer, ISSN 1545-5009, E-ISSN 1545-5017, Vol. 55, no 5, p. 935-935Article in journal (Other academic)
  • 35.
    Loeffen, Erik A. H.
    et al.
    Univ Groningen, Univ Med Ctr Groningen, Beatrix Childrens Hosp, Dept Pediat Oncol Hematol, Groningen, Netherlands.
    Kremer, Leontien C. M.
    Princess Maxima Ctr Pediat Oncol, Utrecht, Netherlands;Emma Childrens Hosp, Acad Med Ctr, Dept Pediat Oncol, Amsterdam, Netherlands.
    van de Wetering, Marianne D.
    Princess Maxima Ctr Pediat Oncol, Utrecht, Netherlands.
    Mulder, Renee L.
    Princess Maxima Ctr Pediat Oncol, Utrecht, Netherlands;Emma Childrens Hosp, Acad Med Ctr, Dept Pediat Oncol, Amsterdam, Netherlands.
    Font-Gonzalez, Anna
    Princess Maxima Ctr Pediat Oncol, Utrecht, Netherlands;Emma Childrens Hosp, Acad Med Ctr, Dept Pediat Oncol, Amsterdam, Netherlands.
    Dupuis, Lee L.
    Hosp Sick Children, Dept Pharm, Toronto, ON, Canada;Hosp Sick Children, Res Inst, Toronto, ON, Canada;Univ Toronto, Leslie Dan Fac Pharm, Toronto, ON, Canada.
    Campbell, Fiona
    Univ Toronto, Hosp Sick Children, Dept Anesthesia & Pain Med, Toronto, ON, Canada.
    Tissing, Wim J. E.
    Univ Groningen, Univ Med Ctr Groningen, Beatrix Childrens Hosp, Dept Pediat Oncol Hematol, Groningen, Netherlands;Princess Maxima Ctr Pediat Oncol, Utrecht, Netherlands.
    Anghelescu, Doralina L.
    St Jude Childrens Res Hosp, 332 N Lauderdale St, Memphis, TN 38105 USA.
    Birnie, Kathryn
    Hosp Sick Children, Toronto, ON, Canada.
    de Bont, Judith
    Univ Med Ctr Utrecht, Utrecht, Netherlands.
    Bredlau, Amy-Lee
    Med Univ South Carolina, Charleston, IL USA.
    Cullen, Patsy
    Regis Univ, Denver, CO USA.
    Daniels, Sarah
    St Jude Childrens Res Hosp, 332 N Lauderdale St, Memphis, TN 38105 USA.
    Dick, Bruce
    Univ Alberta, Edmonton, AB, Canada.
    van Dijk, Monique
    Erasmus MC Sophia Childrens Hosp, Rotterdam, Netherlands.
    Dingeman, R. Scott
    Johns Hopkins Univ Hosp, Baltimore, MD 21287 USA.
    Evan, Elena
    UCLA Hlth, Los Angeles, CA USA.
    Gegg, Julie
    Boston Childrens Hosp, Boston, MA USA.
    Gibson, Faith
    Great Ormond St Hosp Children NHS Fdn Trust, London, England;Univ Surrey, London, England.
    van Grotel, Marline
    Princess Maxima Ctr Pediat Oncol, Utrecht, Netherlands.
    Jibb, Lindsay
    Hosp Sick Children, Toronto, ON, Canada.
    Kao, Roy
    UCLA, David Geffen Sch Med, Los Angeles, CA 90095 USA.
    Knops, Rutger
    Princess Maxima Ctr Pediat Oncol, Utrecht, Netherlands.
    Kulkarni, Ketan
    IWK Hlth Ctr, Halifax, NS, Canada.
    Leroy, Piet
    Maastricht Univ, Med Ctr, Maastricht, Netherlands.
    Liossi, Christina
    Univ Southampton, Southampton, Hants, England.
    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.
    McLean, Jennifer
    Hosp Sick Children, Toronto, ON, Canada.
    Mensink, Maarten
    Univ Med Ctr Utrecht, Utrecht, Netherlands.
    Michiels, Erna
    Princess Maxima Ctr Pediat Oncol, Utrecht, Netherlands.
    Muckaden, Mary Ann
    Tata Mem Hosp, Mumbai, Maharashtra, India.
    Newman, Brittney
    St Jude Childrens Res Hosp, 332 N Lauderdale St, Memphis, TN 38105 USA.
    Positano, Karyn
    Hosp Sick Children, Toronto, ON, Canada.
    Rijsdijk, Mienke
    Univ Med Ctr Utrecht, Utrecht, Netherlands.
    Rowe, Emily
    Tufts Med Ctr, Boston, MA 02111 USA.
    Sangha, Gurjit
    Trillium Hlth Partners, Mississauga, ON, Canada.
    Stinson, Jennifer
    Hosp Sick Children, Toronto, ON, Canada.
    Taddio, Anna
    Univ Toronto, Toronto, ON, Canada.
    Taylor, Hannah
    St Jude Childrens Res Hosp, 332 N Lauderdale St, Memphis, TN 38105 USA.
    Tutelman, Perri
    Dalhousie Univ, Pediat, Ctr Pediat Pain Res, Halifax, NS, Canada.
    Twycross, Alison
    London South Bank Univ, London, England.
    Wijnen, Marc
    Princess Maxima Ctr Pediat Oncol, Utrecht, Netherlands.
    Zeltzer, Lonnie
    UCLA Hlth, Los Angeles, CA USA.
    Reducing pain in children with cancer: Methodology for the development of a clinical practice guideline2019In: Pediatric Blood & Cancer, ISSN 1545-5009, E-ISSN 1545-5017, Vol. 66, no 6, article id e27698Article in journal (Refereed)
    Abstract [en]

    Although pain is one of the most prevalent and bothersome symptoms children with cancer experience, evidence-based guidance regarding assessment and management is lacking. With 44 international, multidisciplinary healthcare professionals and nine patient representatives, we aimed to develop a clinical practice guideline (following GRADE methodology), addressing assessment and pharmacological, psychological, and physical management of tumor-, treatment-, and procedure-related pain in children with cancer. In this paper, we present our thorough methodology for this development, including the challenges we faced and how we approached these. This lays the foundation for our clinical practice guideline, for which there is a high clinical demand.

  • 36. Lohmann, D. J. A.
    et al.
    Abrahamsson, J.
    Ha, S. Y.
    Jonsson, O. G.
    Koskenvuo, M.
    Lausen, B.
    Palle, Josefine
    Uppsala University, Disciplinary Domain of Medicine and Pharmacy, Faculty of Medicine, Department of Medical Sciences, Molecular Medicine. Uppsala University, Disciplinary Domain of Medicine and Pharmacy, Faculty of Medicine, Department of Women's and Children's Health, Pediatrics.
    Zeller, B.
    Hasle, H.
    Toxicity is Associated with Age in Nopho-Aml 20042014In: Pediatric Blood & Cancer, ISSN 1545-5009, E-ISSN 1545-5017, Vol. 61, p. S127-S127Article in journal (Other academic)
  • 37.
    Lohmann, Ditte J. A.
    et al.
    Aarhus Univ Hosp, Dept Pediat & Adolescent Med, Palle Juul Jensens Blvd 99, DK-8200 Aarhus N, Denmark.
    Asdahl, Peter H.
    Aarhus Univ Hosp, Dept Hematol, Aarhus, Denmark.
    Abrahamsson, Jonas
    Queen Silvia Childrens Hosp, Inst Clin Sci, Dept Pediat, 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.
    Jonsson, Olafur G.
    Landspitali Univ Hosp, Dept Pediat, Reykjavik, Iceland.
    Kaspers, Gertjan J. L.
    Dutch Childhood Oncol Grp, The Hague, Netherlands;VU Univ Med Ctr Amsterdam, Dept Pediat, The Hague, Netherlands.
    Koskenvuo, Minna
    Univ Helsinki, Div Hematol Oncol & Stem Cell Transplantat, Childrens Hosp, Helsinki, Finland.
    Lausen, Birgitte
    Univ Copenhagen, Dept Pediat & Adolescent Med, Rigshosp, Copenhagen, Denmark.
    De Moerloose, Barbara
    Ghent Univ Hosp, Dept Pediat, Ghent, Belgium.
    Palle, Josefine
    Uppsala University, Disciplinary Domain of Medicine and Pharmacy, Faculty of Medicine, Department of Women's and Children's Health.
    Zeller, Bernward
    Oslo Univ Hosp, Div Pediat & Adolescent Med, Oslo, Norway.
    Hasle, Henrik
    Aarhus Univ Hosp, Dept Pediat & Adolescent Med, Palle Juul Jensens Blvd 99, DK-8200 Aarhus N, Denmark.
    Associations between neutrophil recovery time, infections and relapse in pediatric acute myeloid leukemia2018In: Pediatric Blood & Cancer, ISSN 1545-5009, E-ISSN 1545-5017, Vol. 65, no 9, article id e27231Article in journal (Refereed)
    Abstract [en]

    BackgroundChildren with acute myeloid leukemia (AML) treated similarly show different toxicity and leukemic responses. We investigated associations between neutrophil recovery time after the first induction course, infection and relapse in children treated according to NOPHO-AML 2004 and DB AML-01. ProcedureNewly diagnosed patients with AML with bone marrow blast<5% between day 15 after the start of the treatment and the start of second induction course, and in complete remission after the second induction course were included (n=279). Neutrophil recovery time was defined as the time from the start of the course to the last day with absolute neutrophil count<0.5x10(9)/l. Linear and Cox regressions were used to investigate associations. ResultsNeutrophil recovery time after the first induction course was positively associated with neutrophil recovery time after the remaining courses, and longer neutrophil recovery time (25 days) was associated with increased risk of grade 3-4 infections (hazard ratio 1.4, 95% confidence interval [CI], 1.1-1.8). Longer neutrophil recovery time after the first induction (>30 days) was associated with the increased risk of relapse (5-year cumulative incidence: 48% vs. 42%, hazard ratio 1.7, 95% CI, 1.1-2.6) for cases not treated with hematopoietic stem cell transplantation in first complete remission. ConclusionLonger neutrophil recovery time after the first induction course was associated with grade 3-4 infections and relapse. If confirmed, this knowledge could be incorporated into risk stratification strategies in pediatric AML.

  • 38.
    Lohnnann, Ditte J. A.
    et al.
    Aarhus Univ Hosp, Dept Pediat & Adolescent Med, Palle Juul Jensens Blvd 99, DK-8200 Aarhus N, Denmark.
    Asdahl, Peter H.
    Aarhus Univ Hosp, Dept Hematol, Aarhus, Denmark.
    Abrahamsson, Jonas
    Queen Silvia Childrens Hosp, Inst Clin Sci, Dept Pediat, Gothenburg, Sweden.
    Ha, Shau-Yin
    Queen Mary Hosp, Dept Pediat, Hong Kong, Peoples R China;HKPHOSG, Hong Kong, Peoples R China.
    Jonssons, Olafur G.
    Landspitali Univ Hosp, Dept Pediat, Reykjavik, Iceland.
    Kaspers, Gertjan J. L.
    Vrije Univ Amsterdam Med Ctr, Dept Pediat, Amsterdam, Netherlands;Princess Maxima Ctr Pediat Oncol, Utrecht, Netherlands;Dutch Childhood Oncol Grp, The Hague, Netherlands.
    Koskenvuo, Minna
    Univ Helsinki, Div Hematol Oncol & Stem Cell Transplantat, New Childrens Hosp, Helsinki, Finland;Helsinki Univ Hosp, Helsinki, Finland.
    Lausen, Birgitte
    Univ Copenhagen, Rigshosp, Dept Pediat & Adolescent Med, Copenhagen, Denmark.
    De Moerloose, Barbara
    Ghent Univ Hosp, Dept Pediat Hematol Oncol & Stem Cell Transplanta, Ghent, Belgium.
    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.
    Hasle, Henrik
    Aarhus Univ Hosp, Dept Pediat & Adolescent Med, Palle Juul Jensens Blvd 99, DK-8200 Aarhus N, Denmark.
    Use of granulocyte colony-stimulating factor and risk of relapse in pediatric patients treated for acute myeloid leukemia according to NOPHO-AML 2004 and DB AML-012019In: Pediatric Blood & Cancer, ISSN 1545-5009, E-ISSN 1545-5017, Vol. 66, no 6, article id e27701Article in journal (Refereed)
    Abstract [en]

    Background

    Supportive-care use of granulocyte colony-stimulating factor (G-CSF) in pediatric acute myeloid leukemia (AML) remains controversial due to a theoretical increased risk of relapse and limited impact on neutropenic complications. We describe the use of G-CSF in patients treated according to NOPHO-AML 2004 and DB AML-01 and investigated associations with relapse.

    Procedure

    Patients diagnosed with de novo AML completing the first week of therapy and not treated with hematopoietic stem cell transplantation in the first complete remission were included (n = 367). Information on G-CSF treatment after each course (yes/no) was registered prospectively in the study database and detailed information was gathered retrospectively from each center. Descriptive statistics were used to describe G-CSF use and Cox regression to assess the association between G-CSF and risk of relapse.

    Results

    G-CSF as supportive care was given to 128 (35%) patients after 268 (39%) courses, with a large variation between centers (0-93%). The use decreased with time-the country-adjusted odds ratio was 0.8/diagnostic year (95% confidence interval [CI] 0.7-0.9). The median daily dose was 5 mu g/kg (range 3-12 mu g/kg) and the median cumulative dose was 75 mu g/kg (range 7-1460 mu g/kg). Filgrastim was used in 82% of G-CSF administrations and infection was the indication in 44% of G-CSF administrations. G-CSF was associated with increased risk of relapse-the adjusted hazard ratio was 1.5 (95% CI 1.1-2.2).

    Conclusions

    G-CSF as supportive care was used in a third of patients, and use decreased with time. Our results indicate that the use of G-CSF may be associated with an increased risk of relapse.

  • 39.
    Lovgren, M.
    et al.
    Karolinska Inst, Dept Womens & Childrens Hlth, Stockholm, Sweden.;Dalarna Univ, Sch Educ Hlth & Soc, Falun, Sweden..
    Grenklo, T. Bylund
    Karolinska Inst, Dept Womens & Childrens Hlth, Stockholm, Sweden.;Linnaeus Univ, Dept Hlth & Caring Sci, Vaxjo, Sweden..
    Jalmsell, Li
    Uppsala University, Disciplinary Domain of Medicine and Pharmacy, Faculty of Medicine, Department of Public Health and Caring Sciences, Centre for Research Ethics and Bioethics.
    Wallin, A. Eilegard
    Dalarna Univ, Sch Educ Hlth & Soc, Falun, Sweden..
    Kreicbergs, U.
    Karolinska Inst, Dept Womens & Childrens Hlth, Stockholm, Sweden.;Ersta Skondal Univ Coll, Palliat Res Ctr, Stockholm, Sweden..
    Bereaved Siblings' Advice to Health Care Professionals Working with Children with Cancer and their Families2015In: Pediatric Blood & Cancer, ISSN 1545-5009, E-ISSN 1545-5017, Vol. 62, no Suppl. 4, p. S349-S349Article in journal (Other academic)
  • 40.
    Lovgren, M.
    et al.
    Karolinska Inst, Dept Childrens & Womens Hlth, Stockholm, Sweden.;Dalarna Univ, Sch Educ Hlth & Soc, Falun, Sweden..
    Jalmsell, Li
    Uppsala University, Disciplinary Domain of Medicine and Pharmacy, Faculty of Medicine, Department of Public Health and Caring Sciences, Centre for Research Ethics and Bioethics. Uppsala Univ, Ctr Res Eth Bioeth, Uppsala, Sweden..
    Wallin, A. Eilegrad
    Dalarna Univ, Sch Educ Hlth & Soc, Falun, Sweden..
    Steineck, G.
    Karolinska Inst, Dept Oncol Pathol, Stockholm, Sweden..
    Kreicbergs, U.
    Karolinska Inst, Dept Childrens & Womens Hlth, Stockholm, Sweden.;Ersta Skondal Univ Coll, Palliat Res Ctr, Stockholm, Sweden..
    Siblings' Experiences of the Brother'S or Sister'S Cancer Death: A Nationwide Follow-up 2-9 Years Later2015In: Pediatric Blood & Cancer, ISSN 1545-5009, E-ISSN 1545-5017, Vol. 62, no Suppl. 4, p. S206-S206Article in journal (Other academic)
  • 41.
    Lychou, Sara
    et al.
    Uppsala University, Disciplinary Domain of Medicine and Pharmacy, Faculty of Medicine, Department of Women's and Children's Health, Pediatrics.
    Gustafsson, Göran
    Karolinska Insitute, Pediat Oncol, Stockholm, Sweden..
    Ljungman, Gustaf
    Uppsala University, Disciplinary Domain of Medicine and Pharmacy, Faculty of Medicine, Department of Women's and Children's Health, Pediatrics.
    Decline In Survival In Pediatric Rhabdomyosarcoma In Sweden?: Characteristics And Outcomes In Children With Rhabdomyosarcoma Diagnosed In Sweden During The Years 1984-20102015In: Pediatric Blood & Cancer, ISSN 1545-5009, E-ISSN 1545-5017, Vol. 62, p. S233-S233Article in journal (Other academic)
  • 42.
    Lönnerholm, Gudmar
    et al.
    Uppsala University, Disciplinary Domain of Medicine and Pharmacy, Faculty of Medicine, Department of Women's and Children's Health.
    Frost, Britt-Marie
    Uppsala University, Disciplinary Domain of Medicine and Pharmacy, Faculty of Medicine, Department of Women's and Children's Health.
    Söderhäll, Stefan
    de Graaf, Siebold S. N.
    Vincristine pharmacokinetics in children with Down syndrome2009In: Pediatric Blood & Cancer, ISSN 1545-5009, E-ISSN 1545-5017, Vol. 52, no 1, p. 123-125Article in journal (Refereed)
    Abstract [en]

    Children with Down syndrome (DS), who represent about 2% of childhood acute lymphoblastic leukemia, have inferior prognosis compared to non-DS children. For vincristine (and many other anticancer agents) pharmacokinetic data are scant or missing, and there is considerable uncertainty about the optimal dosing of drugs to patients with DS. We studied vincristine pharmacokinetics on treatment day one in six children with DS and compared to 92 non-DS children. No differences were found. Thus, we found no rationale for dose reduction of vincristine in DS children from a strictly pharmacokinetic point of view.

  • 43.
    Lönnerholm, Gudmar
    et al.
    Uppsala University, Disciplinary Domain of Medicine and Pharmacy, Faculty of Medicine, Department of Women's and Children's Health.
    Valsecchi, Maria Grazia
    De Lorenzo, Paola
    Schrappe, Martin
    Hovi, Liisa
    Campbell, Myriam
    Mann, Georg
    Janka-Schaub, Gritta
    Li, Chi-Kong
    Stary, Jan
    Hann, Ian
    Pieters, Rob
    Pharmacokinetics of high-dose methotrexate in infants treated for acute lymphoblastic leukemia2009In: Pediatric Blood & Cancer, ISSN 1545-5009, E-ISSN 1545-5017, Vol. 52, no 5, p. 596-601Article in journal (Refereed)
    Abstract [en]

    BACKGROUND: Interfant-99 was an international collaborative treatment protocol for infants with acute lymphoblastic leukemia (ALL). PROCEDURE: We collected data on 103 infants at the time of their first treatment with high-dose methotrexate (HD MTX), 5 g/m(2). Children <6 months of age received two-third of the calculated dose based on body surface area (BSA), children 6-12 months three-fourth of the calculated dose, and children >12 months full dose. RESULTS: The median steady-state MTX concentration at the end of the 24-hr infusion was 57.8 microM (range 9.5-313). The median systemic clearance was 6.22 L/hr/m(2) BSA, and tended to increase with age (P = 0.099). Boys had higher clearance than girls, 6.77 and 5.28 L/hr/m(2) (P = 0.030), and tended to have lower median MTX concentration at 24 hr. Eight infants had MTX levels below 20 microM, a level judged to be sufficient in B-lineage ALL in children >1 year of age. All infants tolerated the dose well enough to receive a second dose of HD MTX without dose reduction. We found no significant effect on disease-free survival for MTX steady-state concentration, MTX clearance, or time to MTX below 0.2 microM. CONCLUSIONS: Our data provide no support for a change in the dosing rules for MTX used in Interfant-99. However, in view of the poor treatment results for infants, one might consider increase in the dose for patients who reach plasma levels below median after the first MTX dose.

  • 44.
    Maarbjerg, Sabine F.
    et al.
    Aarhus Univ Hosp, Dept Pediat & Adolescent Med, Palle Juul Jensens Blvd 99, DK-8200 Aarhus N, Denmark.
    Thorsted, Anders
    Uppsala University, Disciplinary Domain of Medicine and Pharmacy, Faculty of Pharmacy, Department of Pharmaceutical Biosciences.
    Kristoffersson, Anders
    Uppsala University, Disciplinary Domain of Medicine and Pharmacy, Faculty of Pharmacy, Department of Pharmaceutical Biosciences.
    Friberg, Lena E.
    Uppsala University, Disciplinary Domain of Medicine and Pharmacy, Faculty of Pharmacy, Department of Pharmaceutical Biosciences.
    Nielsen, Elisabet I.
    Uppsala University, Disciplinary Domain of Medicine and Pharmacy, Faculty of Pharmacy, Department of Pharmaceutical Biosciences.
    Wang, Mikala
    Aarhus Univ Hosp, Dept Clin Microbiol, Aarhus, Denmark.
    Brock, Birgitte
    Steno Diabet Ctr, Gentofte, Denmark.
    Schroder, Henrik
    Aarhus Univ Hosp, Dept Pediat & Adolescent Med, Palle Juul Jensens Blvd 99, DK-8200 Aarhus N, Denmark.
    Piperacillin pharmacokinetics and target attainment in children with cancer and fever: Can we optimize our dosing strategy?2019In: Pediatric Blood & Cancer, ISSN 1545-5009, E-ISSN 1545-5017, Vol. 66, no 6, article id e27654Article in journal (Refereed)
    Abstract [en]

    Background

    Data on piperacillin-tazobactam pharmacokinetics and optimal dosing in children with cancer and fever are limited. Our objective was to investigate piperacillin pharmacokinetics and the probability of target attainment (PTA) with standard intermittent administration (IA), and to simulate PTA in other dosing regimens.

    Procedure

    This prospective pharmacokinetic study was conducted from April 2016 to January 2018. Children with cancer receiving empiric piperacillin-tazobactam to treat infections were included. Piperacillin-tazobactam 100 mg/kg was infused over 5 min every 8 hours (IA). An optimized sample schedule provided six blood samples per subject for piperacillin concentration determination. The evaluated targets included: (1) 100% time of free piperacillin concentration above the minimum inhibitory concentration (fT > MIC) and (2) 50% fT > 4x MIC. MIC50 and MIC90 were defined based on an intrainstitutional MIC range.

    Results

    A total of 482 piperacillin concentrations were obtained from 43 children (aged 1-18 years) during 89 fever episodes. Standard IA resulted in insufficient target attainment, with significant differences in piperacillin pharmacokinetics for different body weights. Median fT > MIC was 61.2%, 53.5%, and 36.3% for MIC50 (2.0 mg/L), MIC90 (4.0 mg/L), and breakpoint for Pseudomonas aeruginosa (16.0 mg/L), respectively. Correspondingly, the median fT > 4x MIC was 43%, 36.3%, and 20.1%. Simulations showed that only continuous infusion reached a PTA of 95% for MIC = 16.0 mg/L, while extended infusion lasting half of the dosing interval reached a PTA of 95% for MIC <= 8 mg/L.

    Conclusions

    Our data revealed insufficient PTA with standard IA of piperacillin-tazobactam in children with cancer and fever. Alternative dosing strategies, preferably continuous infusion, are required to ensure adequate PTA.

  • 45.
    Machowcz, Rafal
    et al.
    Med Univ Warsaw, Warsaw, Poland..
    Suarez, Felipe
    Hop Necker Enfants Malad, Paris, France..
    Wiktor-Jedrzejczak, Wieslaw
    Med Univ Warsaw, Warsaw, Poland..
    Eikema, Diderik-Jan
    LUMC, Dept Med Stat & Bioinformat, Leiden, Netherlands..
    de Wreede, Liesbeth
    LUMC, Dept Med Stat & Bioinformat, Leiden, Netherlands..
    Blok, Henric-Jan
    LUMC, Dept Med Stat & Bioinformat, Leiden, Netherlands..
    Isaksson, Cecilia
    Umea Univ Hosp, Umea, Sweden..
    Poire, Xavier
    Clin Univ St Luc, Brussels, Belgium..
    Nikolousis, Manos
    Birmingham Heartlands Hosp, Birmingham, W Midlands, England..
    Kobbe, Guido
    Heinrich Heine Univ, Dusseldorf, Germany..
    Einsele, Herman
    Univ Klinikum Wurzburg, Wurzburg, Germany..
    Arnold, Renate
    Charite Univ Med Berlin, Berlin, Germany..
    Bonifazi, Francesca
    St Orsola Hosp, Bologna, Italy..
    McQuacker, Grant
    Gartnaval Gen Hosp, Glasgow, Lanark, Scotland..
    Lenhoff, Stig
    Skanes Univ Hosp, Lund, Sweden..
    Rohrlich, Pierre-Simon
    Hop ARCHET I, CHU Nice, Nice, France..
    Theobald, Matthias
    Univ Med Ctr Mainz, Mainz, Germany..
    Ljungman, Per
    Karolinska Univ Hosp, Stockholm, Sweden..
    Schaap, Nicolaas
    Radboud Univ Nijmegen, Med Ctr, Nijmegen, Netherlands..
    Collin, Matthew
    Freeman Rd Hosp, Newcastle Upon Tyne, Tyne & Wear, England..
    Albert, Michael
    Haunersches Kinderspital, Munich, Germany..
    Finke, Juergen
    Univ Freiburg, Freiburg, Germany..
    Ehninger, Gerhard
    Univ Klinikum Dresden, Dresden, Germany..
    Carlson, Kristina
    Uppsala University, Disciplinary Domain of Medicine and Pharmacy, Faculty of Medicine, Department of Medical Sciences, Haematology.
    Halaburda, Kazimierz
    Inst Hematol & Transfus Med, Warsaw, Poland..
    Johansson, Jan-Erik
    Sahlgrens Univ Hosp, Gothenburg, Sweden..
    Zecca, Marco
    Fdn IRCCS Policlin San Matteo, Pavia, Italy..
    Diez-Martin, J.
    Hosp Gregorio Maranon, Madrid, Spain..
    Lehmberg, Kai
    Univ Hosp Eppendorf, Hamburg, Germany..
    Schoenland, Stefan
    Heidelberg Univ, Heidelberg, Germany..
    Lankester, Arjan
    Leiden Univ Hosp, Leiden, Netherlands..
    Gennery, Andrew
    Newcastle Univ, Inst Cellular Med, Newcastle Upon Tyne, Tyne & Wear, England..
    Kroger, Nicolaus
    Univ Hosp Eppendorf, Hamburg, Germany..
    Allogeneic Hematopoietic Stem Cell Transplantation Provides Cure for Adult Patients with Hemophagocytic Lymphohistiocytosis (HLH): A Retrospective Study of The Chronic Malignancies and Inborn ErrorsWorking Parties (CMWP and IEWP) of The EBMT2017In: Pediatric Blood & Cancer, ISSN 1545-5009, E-ISSN 1545-5017, Vol. 64, no S2, p. S23-S24Article in journal (Other academic)
  • 46.
    Martinsson, Ulla
    et al.
    Uppsala University, Disciplinary Domain of Medicine and Pharmacy, Faculty of Medicine, Department of Radiology, Oncology and Radiation Science, Oncology.
    Arvidson, Johan
    Uppsala University, Disciplinary Domain of Medicine and Pharmacy, Faculty of Medicine, Department of Women's and Children's Health.
    Co-ordinating follow-up for adult survivors of childhood cancer treated in a tertiary cancer centre covering a large geographic region2013In: Pediatric Blood & Cancer, ISSN 1545-5009, E-ISSN 1545-5017, Vol. 60, no S3, p. 203-203Article in journal (Other academic)
  • 47.
    Mogensen, Signe Sloth
    et al.
    Rigshosp, Univ Hosp, Juliane Marie Ctr, Dept Pediat & Adolescent Med, JMC 5704,Blegdamsvej 9, DK-2100 Copenhagen, Denmark.
    Harila-Saari, Arja
    Karolinska Inst, Dept Womens & Childrens Hlth, Childhood Canc Res Unit, Stockholm, Sweden.
    Makitie, Outi
    Karolinska Inst, Dept Mol Med & Surg, Stockholm, Sweden;Karolinska Inst, Ctr Mol Med, Stockholm, Sweden;Karolinska Univ Hosp, Dept Clin Genet, Stockholm, Sweden.
    Myrberg, Ida Hed
    Karolinska Inst, Dept Womens & Childrens Hlth, Childhood Canc Res Unit, Stockholm, Sweden.
    Niinimaki, Riitta
    Oulu Univ Hosp, Dept Children & Adolescents, Oulu, Finland;Univ Oulu, PEDEGO Res Unit, Oulu, Finland.
    Vestli, Anne
    Oslo Univ Hosp, Div Pediat & Adolescent Med, Dept Pediat Oncol & Hematol, Oslo, Norway.
    Hafsteinsdottir, Solveig
    Landspitali Univ Hosp, Childrens Hosp, Reykjavik, Iceland.
    Griskevicius, Laimonas
    Vilnius Univ, Fac Med, Vilnius, Lithuania;Vilnius Univ Hosp Santariskiu Klinikos, Transfus Med Ctr, Vilnius, Lithuania;Vilnius Univ Hosp Santariskiu Klinikos, Dept Hematol, Oncol, Vilnius, Lithuania.
    Saks, Kadri
    Tallinn Childrens Hosp, Dept Hematol Oncol, Tallinn, Estonia.
    Hallböök, Helene
    Uppsala University, Disciplinary Domain of Medicine and Pharmacy, Faculty of Medicine, Department of Medical Sciences, Haematology. Uppsala Univ, Dept Med Sci, Uppsala, Sweden.
    Retpen, Jens
    Univ Hosp Herlev Gentofte, Dept Orthoped Surg, Copenhagen, Denmark.
    Helt, Louise Rold
    Rigshosp, Univ Hosp, Juliane Marie Ctr, Dept Pediat & Adolescent Med, JMC 5704,Blegdamsvej 9, DK-2100 Copenhagen, Denmark.
    Toft, Nina
    Rigshosp, Univ Hosp, Dept Hematol, Copenhagen, Denmark.
    Schmiegelow, Kjeld
    Univ Copenhagen, Fac Med, Inst Clin Med, Copenhagen, Denmark;Rigshosp, Univ Hosp, Juliane Marie Ctr, Dept Pediat & Adolescent Med, JMC 5704,Blegdamsvej 9, DK-2100 Copenhagen, Denmark.
    Frandsen, Thomas Leth
    Rigshosp, Univ Hosp, Juliane Marie Ctr, Dept Pediat & Adolescent Med, JMC 5704,Blegdamsvej 9, DK-2100 Copenhagen, Denmark.
    Comparing osteonecrosis clinical phenotype, timing, and risk factors in children and young adults treated for acute lymphoblastic leukemia2018In: Pediatric Blood & Cancer, ISSN 1545-5009, E-ISSN 1545-5017, Vol. 65, no 10, article id e27300Article in journal (Refereed)
    Abstract [en]

    Background: Treatment-related osteonecrosis (ON) is a serious complication of treatment of acute lymphoblastic leukemia (ALL).

    Procedure: This study included 1,489 patients with ALL, aged 1-45years, treated according to the Nordic Society of Paediatric Haematology and Oncology ALL2008 protocol, using alternate-week dexamethasone during delayed intensification, with prospective registration of symptomatic ON. We aimed at comparing risk factors, timing, and clinical characteristics of ON in children and young adults.

    Results: ON was diagnosed in 67 patients, yielding a 5-year cumulative incidence of 6.3%, but 28% in female adolescents. Median age at ALL diagnosis was 12.1years and 14.9years for females and males, respectively. At ON diagnosis, 59 patients had bone pain (91%) and 30 (46%) had multiple-joint involvement. The median interval between ALL and ON diagnosis was significantly shorter in children aged 1.0-9.9years (0.7years [range: 0.2-2.1]) compared with adolescents (1.8years [range: 0.3-3.7, P<0.001]) and adults (2.1years [range: 0.4-5.3, P=0.001]). Female sex was a risk factor in adolescent patients (hazard ratio [HR]=2.1, 95% confidence interval [CI]: 1.1-4.2) but not in children aged 1.1-9.9years (HR=2.4, 95% CI: 0.9-6.2, P=0.08) or adults aged 19-45years (HR=1.1, 95% CI: 0.3-4.0). Age above 10years at ALL diagnosis (odds ratio [OR]=3.7, P=0.026) and multiple joints affected at ON diagnosis (OR=3.4, P=0.027) were risk factors for developing severe ON.

    Conclusion: We provide a detailed phenotype of patients with ALL with symptomatic ON, including description of risk factors and timing of ON across age groups. This awareness is essential in exploring measures to prevent development of ON.

  • 48.
    Molgaard-Hansen, Lene
    et al.
    Department of Pediatrics, Aarhus University Hospital Skejby, Aarhus, Denmark.
    Glosli, Heidi
    Jahnukainen, Kirsi
    Jarfelt, Marianne
    Jónmundsson, Guðmundur K
    Malmros-Svennilson, Johan
    Nysom, Karsten
    Hasle, Henrik
    Department of Pediatrics, Aarhus University Hospital Skejby, Aarhus, Denmark.
    Quality of health in survivors of childhood acute myeloid leukemia treated with chemotherapy only: a NOPHO-AML study2011In: Pediatric Blood & Cancer, ISSN 1545-5009, E-ISSN 1545-5017, Vol. 57, no 7, p. 1222-1229Article in journal (Refereed)
    Abstract [en]

    BACKGROUND:

    More than 60% of children with acute myeloid leukemia (AML) become long-term survivors, and approximately 50% are cured with chemotherapy only. Limited data exist about their long-term morbidity and social outcomes. The aim of the study was to compare the self-reported use of health care services, health experience, social outcomes, and lifestyle behavior of AML survivors with that of their sibling controls.

    METHODS:

    This population-based study included 138 children treated for AML according to the Nordic Society of Pediatric Hematology and Oncology (NOPHO)-AML-84, -88, and -93 trials, and alive by June 30, 2007. Patients treated with hematopoietic stem cell transplantation (HSCT) or relapse were not included. Altogether, 102 (74%) survivors and 91% of their siblings completed a questionnaire.

    RESULTS:

    The median follow-up was 11 (range 4-25) years after diagnosis. AML survivors had no increased rate of hospitalization compared with sibling controls, but were more often receiving prescription drugs, especially for asthma (23% vs. 9%, P = 0.03). Self-reported health experience was excellent or very good in 77% and comparable with that of siblings. Educational achievement, employment, and marital status were comparable in the two groups. Among surviving AML patients, 23% were current smokers and 24% of their siblings were current smokers.

    CONCLUSIONS:

    The self-reported health of children treated on NOPHO-AML protocols without HSCT was good, and their use of health care services was limited. Reported health and social outcomes were comparable to those of their siblings. Many survivors were smoking which may increase the risk of late effects.

  • 49. Oskarsson, T.
    et al.
    Soderhall, S.
    Arvidson, Johan
    Uppsala University, Disciplinary Domain of Medicine and Pharmacy, Faculty of Medicine, Department of Women's and Children's Health, Pediatrics.
    Forestier, E.
    Frandsen, T.
    Carlsen, N.
    Hellebostad, M.
    Glomstein, A.
    Lahteenmaki, P.
    Heyman, M.
    Treatment Related Death in Relapsed Childhood Acute Lymphoblastic Leukemia2014In: Pediatric Blood & Cancer, ISSN 1545-5009, E-ISSN 1545-5017, Vol. 61, p. S264-S264Article in journal (Other academic)
  • 50. Oskarsson, Trausti
    et al.
    Soderhall, Stefan
    Arvidson, Johan
    Uppsala University, Disciplinary Domain of Medicine and Pharmacy, Faculty of Medicine, Department of Women's and Children's Health, Pediatrics.
    Forestier, Erik
    Montgomery, Scott
    Lausen, Birgitte
    Carlsen, Niels
    Hellebostad, Marit
    Glomstein, Anders
    Lahteenmaki, Paivi
    Pihkala, Ulla
    Jonsson, Olafur Gisli
    Heyman, Mats
    Relapsed acute lymphoblastic leukemia in the nordic countries: prognostic factors, treatment and outcome2012In: Pediatric Blood & Cancer, ISSN 1545-5009, E-ISSN 1545-5017, Vol. 59, no 6, p. 1007-1008Article in journal (Other academic)
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