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  • 1.
    Bento, Celeste
    et al.
    Department of Hematology, Centro Hospitalar e Universitário de Coimbra, Coimbra, Portugal .
    Percy, Melanie J.
    Department of Haematology, Belfast City Hospital, Belfast, United Kingdom .
    Gardie, Betty
    Unité Mixte de Recherche (UMR) 892 Inserm - 6299 CNRS, Université de Nantes, Nantes, France; Laboratoire de Génétique Oncologique de l'Ecole Pratique des Hautes Etudes (EPHE), INSERM U753, Institut de cancérologie Gustave Roussy, Villejuif, France .
    Maia, Tabita Magalhaes
    Department of Hematology, Centro Hospitalar e Universitário de Coimbra, Coimbra, Portugal .
    van Wijk, Richard
    Department of Clinical Chemistry and Haematology, University Medical Center Utrecht, Utrecht, Netherlands .
    Perrotta, Silverio
    Dipartimento della Donna, Del Bambino e di Chirurgia Generale e Specialistica, Second University of Naples, Naples, Italy .
    Della Ragione, Fulvio
    Department of Biochemistry, Biophysics and General Pathology, Second University of Naples, Naples, Italy .
    Almeida, Helena
    Department of Hematology, Centro Hospitalar e Universitário de Coimbra, Coimbra, Portugal .
    Rossi, Cedric
    Laboratoire d'Hématologie, Centre Hospitalier Universitaire Dijon, Dijon, France .
    Girodon, Francois
    Laboratoire d'Hématologie, Centre Hospitalier Universitaire Dijon, Dijon, France .
    Åström, Maria
    Örebro University Hospital. Departments of Medicine and Laboratory Medicine.
    Neumann, Drorit
    Department of Cell and Developmental Biology, Sackler Faculty of Medicine, Tel-Aviv University, Ramat-Aviv, Israel .
    Schnittger, Susanne
    Munich Leukemia Laboratory (MLL), Munich, Germany .
    Landin, Britta
    Department of Clinical Chemistry, Karolinska University Hospital, Stockholm, Sweden .
    Minkov, Milen
    Department of Hematology/Oncology, St. Anna Children's Hospital, Medical University of Vienna, Vienna, Austria .
    Randi, Maria Luigia
    Department of Medicine DIMED, University of Padua, Padua, Italy .
    Richard, Stephane
    Laboratoire de Génétique Oncologique de l'Ecole Pratique des Hautes Etudes (EPHE), INSERM U753, Institut de cancérologie Gustave Roussy, Villejuif, France .
    Casadevall, Nicole
    Hôpital Saint Antoine, Paris, France; Assistance Publique-Hôpitaux de Paris, Paris, France; Pierre et Marie Curie University, Paris, France; UMR1009 Institut Gustave Roussy Villejuif, Paris, France .
    Vainchenker, William
    UMR 1009 and GRex, INSERM, Université Paris-Sud, Institut Gustave Roussy, Villejuif, France .
    Rives, Susana
    Department of Pediatric Hematology, Hospital Sant Joan de Déu de Barcelona, University of Barcelona, Barcelona, Spain .
    Hermouet, Sylvie
    Unité Mixte de Recherche (UMR) 892 Inserm - 6299 CNRS, Université de Nantes, Nantes, France .
    Ribeiro, M. Leticia
    Department of Hematology, Centro Hospitalar e Universitário de Coimbra, Coimbra, Portugal .
    McMullin, Mary Frances
    Department of Haematology, CCRCB, Queen's University, Belfast, Northern Ireland, United Kingdom .
    Cario, Holger
    Department of Pediatrics and Adolescent Medicine, University Medical Center Ulm, Ulm, Germany .
    Genetic Basis of Congenital Erythrocytosis: Mutation Update and Online Databases2014In: Human Mutation, ISSN 1059-7794, E-ISSN 1098-1004, Vol. 35, no 1, p. 15-26Article in journal (Refereed)
    Abstract [en]

    Congenital erythrocytosis (CE), or congenital polycythemia, represents a rare and heterogeneous clinical entity. It is caused by deregulated red blood cell production where erythrocyte overproduction results in elevated hemoglobin and hematocrit levels. Primary congenital familial erythrocytosis is associated with low erythropoietin (Epo) levels and results from mutations in the Epo receptor gene (EPOR). Secondary CE arises from conditions causing tissue hypoxia and results in increased Epo production. These include hemoglobin variants with increased affinity for oxygen (HBB, HBA mutations), decreased production of 2,3-bisphosphoglycerate due to BPGM mutations, or mutations in the genes involved in the hypoxia sensing pathway (VHL, EPAS1, and EGLN1). Depending on the affected gene, CE can be inherited either in an autosomal dominant or recessive mode, with sporadic cases arising de novo. Despite recent important discoveries in the molecular pathogenesis of CE, the molecular causes remain to be identified in about 70% of the patients. With the objective of collecting all the published and unpublished cases of CE the COST action MPN&MPNr-Euronet developed a comprehensive Internet-based database focusing on the registration of clinical history, hematological, biochemical, and molecular data (http://www.erythrocytosis.org/). In addition, unreported mutations are also curated in the corresponding Leiden Open Variation Database.

  • 2.
    Bergfelt, E.
    et al.
    Haematology, Dept of Medical Sciences, Uppsala University, Uppsala, Sweden.
    Kozlowski, Piotr
    Örebro University, School of Health Sciences.
    Ahlberg, L.
    Dept of Haematology, University Hospital of Linköping, Linköping, Sweden.
    Bernell, P.
    Div of Haematology, Dept of Medicine, Karolinska Institutet, Karolinska University, Stockholm, Sweden.
    Hulegårdh, E.
    Dept of Haematology and Coagulation, Sahlgrenska University Hospital, Göteborg, Sweden.
    Karbach, H.
    Dept of Haematology, Cancer Centre, University Hospital of Umeå, Umeå, Sweden.
    Karlsson, K.
    Dept of Haematology, Skåne University Hospital, Lund, Sweden .
    Tomaszewska-Toporska, B.
    Dept of Haematology, Skåne University Hospital, Lund, Sweden .
    Åström, Maria
    Örebro University, School of Medical Sciences.
    Hallböök, H.
    Haematology, Dept of Medical Sciences, Uppsala University, Uppsala, Sweden.
    RELAPSE OF ACUTE LYMPHOBLASTIC LEUKAEMIA IN OLDER/ELDERLY PATIENTS: A SWEDISH POPULATION-BASED STUDY2016In: Haematologica, ISSN 0390-6078, E-ISSN 1592-8721, Vol. 101, no Suppl. 1, p. 34-35Article in journal (Other academic)
  • 3.
    Bergfelt, E.
    et al.
    Department of Medical Sciences, Haematology, Uppsala University, Uppsala, Sweden.
    Kozlowski, Piotr
    Örebro University, School of Health and Medical Sciences, Örebro University, Sweden.
    Åström, Maria
    Örebro University, School of Health and Medical Sciences, Örebro University, Sweden.
    Ahlberg, L.
    Department of Haematology, University Hospital of Linköping, Linköping, Sweden.
    Bernell, P.
    Dept Med, Div Haematol, Karolinska Univ Hosp, Karolinska Inst, Stockholm, Sweden.
    Hulegårdh, E.
    Department of Haematology and Coagulation, Sahlgrenska University Hospital, Göteborg, Sweden.
    Karbach, H.
    Ctr Canc, Dept Haematol, Umeå Univ Hosp, Umeå, Sweden.
    Karlsson, K.
    Department of Haematology, Skåne University Hospital, Lund, Sweden.
    Tomaszewska-Toporska, B.
    Department of Haematology, Skåne University Hospital, Lund, Sweden.
    Hallböök, H.
    Department of Medical Sciences, Haematology, Uppsala University, Uppsala, Sweden.
    Prognosis in older/elderly patients with acute lymphoblastic leukaemia diagnosed 2005-2012: results from a Swedish population-based study2015In: Haematologica, ISSN 0390-6078, E-ISSN 1592-8721, Vol. 100, no Suppl. 1, p. 202-202Article in journal (Other academic)
  • 4.
    Bergfelt, Emma
    et al.
    Department of Medical Sciences, Haematology, Uppsala University, Uppsala, Sweden.
    Kozlowski, Piotr
    Örebro University, School of Health and Medical Sciences, Örebro University, Sweden.
    Ahlberg, Lucia
    Department of Haematology, University Hospital of Linköping, Linköping, Sweden.
    Hulegårdh, Erik
    Department of Haematology and Coagulation, Sahlgrenska University Hospital, Göteborg, Sweden.
    Hägglund, Hans
    Division of Haematology, Department of Medicine Huddinge, Karolinska Institutet, Karolinska University Hospital, Stockholm, Sweden.
    Karlsson, Karin
    Department of Haematology, Skåne University Hospital, Lund, Sweden.
    Markuszewska-Kuczymska, Alicja
    Department of Haematology, Cancer Center, University Hospital of Umeå, Umeå , Sweden.
    Tomaszewska-Toporska, Beata
    Department of Haematology, Skåne University Hospital, Lund, Sweden.
    Smedmyr, Bengt
    Department of Medical Sciences, Haematology, Uppsala University, Uppsala, Sweden.
    Åström, Maria
    Örebro University, School of Health and Medical Sciences, Örebro University, Sweden.
    Amini, Rose-Marie
    Department of Immunology, Genetics and Pathology, Uppsala University, Uppsala, Sweden.
    Hallböök, Helene
    Department of Medical Sciences, Haematology, Uppsala University, Uppsala, Sweden.
    Satisfactory outcome after intensive chemotherapy with pragmatic use of minimal residual disease (MRD) monitoring in older patients with Philadelphia-negative B cell precursor acute lymphoblastic leukaemia: a Swedish registry-based study2015In: Medical Oncology, ISSN 1357-0560, E-ISSN 1559-131X, Vol. 32, no 4, article id 135Article in journal (Refereed)
    Abstract [en]

    The introduction of minimal residual disease (MRD) monitoring, in the Swedish national guidelines for acute lymphoblastic leukaemia, was evaluated in 35 patients aged 46-79 years (median 61), who were diagnosed from 2007 to 2011 and treated with high-intensity, block-based chemotherapy (ABCDV/VABA induction). Both a high complete remission rate (91 %) and acceptable overall survival (OS) rate (47 %) at 5 years were achieved. MRD by flow cytometry was measured in 73 % of the patients reaching complete remission after the first course, but was omitted by the clinicians for eight patients who were either over 70 years of age or already met conventional high-risk criteria. Factors negatively influencing OS were age over 65 years and WHO status >= 2. MRD < 0.1 % after induction had positive impact on continuous complete remission but not on OS. Only five patients were allocated to allogeneic haematopoietic stem cell transplantation in first remission, mainly due to conventional high risk factors. Thus, use of intensive remission induction therapy is effective in a selection of older patients. In a population for whom the possibilities of treatment escalation are limited, the optimal role of MRD monitoring remains to be determined.

  • 5.
    Cario, Holger
    et al.
    University Medical Center, Ulm, Germany.
    McMullin, Mary Frances
    Queens university, Belfast, Northern Ireland.
    Bento, Celeste
    University Hospital, Coimbra, Portugal.
    Pospisilova, Dagmar
    Palacky University, Olomouc, Czech Republic.
    Percy, Melanie J.
    Belfast City Hospital, Belfast, Northern Ireland.
    Hussein, Kais
    Hannover Medical School, Hannover, Germany.
    Schwarz, Jiri
    Institute of Hematology and Blood Transfusion, Prague, Czech Republic.
    Åström, Maria
    Örebro University, School of Health and Medical Sciences, Örebro University, Sweden. Örebro University Hospital.
    Hermouet, Sylvie
    Université de Nantes, Nantes, France.
    Erythrocytosis in Children and Adolescents: Classification, Characterization, and Consensus Recommendations for the Diagnostic Approach2013In: Pediatric Blood & Cancer, ISSN 1545-5009, E-ISSN 1545-5017, Vol. 60, no 11, p. 1734-1738Article in journal (Refereed)
    Abstract [en]

    During recent years, the increasing knowledge of genetic and physiological changes in polycythemia vera (PV) and of different types of congenital erythrocytosis has led to fundamental changes in recommendations for the diagnostic approach to patients with erythrocytosis. Although widely accepted for adult patients this approach may not be appropriate with regard to children and adolescents affected by erythrocytosis. The congenital erythrocytosis working group established within the framework of the MPN&MPNr-EuroNet (COST action BM0902) addressed this question in a consensus finding process and developed a specific algorithm for the diagnosis of erythrocytosis in childhood and adolescence which is presented here. Pediatr Blood Cancer 2013;60:1734-1738. (c) 2013 Wiley Periodicals, Inc.

  • 6.
    Danielsson, Signe
    et al.
    Medicinska kliniken, Universitetssjukhuset Örebro, Örebro, Sweden.
    Merup, Mats
    Hematologiskt centrum, Karolinska universitetssjukhuset, Huddinge, Sweden.
    Olsson, Lovisa A.
    Laboratoriemedicinska länskliniken/klinisk kemi, Universitetssjukhuset, Örebro, Sweden.
    Palmblad, Jan
    Hematologiskt centrum, Karolinska universitetssjukhuset, Huddinge, Sweden; Institutionen för medicin, Karolinska institutet, Stockholm, Sweden.
    Åström, Maria
    Medicinska kliniken, Universitetssjukhuset Örebro, Örebro, Sweden.
    X-bunden trombocytopeni med talassemi i två svenska familjer: Överväg hereditära orsaker till trombocytopeni och benmärgsfibros2012In: Läkartidningen, ISSN 0023-7205, E-ISSN 1652-7518, Vol. 109, no 34-35, p. 1474-7Article in journal (Other academic)
  • 7.
    Fergedal, May
    et al.
    Department of Pathology, Örebro Medical Center Hospital, Örebro, Sweden.
    Åström, Maria
    Department of Internal Medicine, Örebro Medical Center Hospital, Örebro, Sweden.
    Tidefelt, Ulf
    Department of Internal Medicine, Örebro Medical Center Hospital, Örebro, Sweden.
    Karlsson, Mats G.
    Department of Pathology, Örebro Medical Center Hospital, Örebro, Sweden.
    Differences in CD14 and alpha-naphthyl acetate esterase positivity and relation to prognosis in AML1998In: Leukemia research: a Forum for Studies on Leukemia and Normal Hemopoiesis, ISSN 0145-2126, E-ISSN 1873-5835, Vol. 22, no 1, p. 25-30Article in journal (Refereed)
    Abstract [en]

    Alpha-naphthyl acetate esterase (ANAE) and CD14 expression, used for determination of monocytic cells, were compared and related to prognosis in 65 AML patients. Bone marrow aspiration material from AML patients has been used for the cytochemistry as well as flow cytometry. All non-erythroid cells have been included in the evaluation in both methods. 17/65 cases showed at least 15% difference between the proportion CD14 and ANAE positive cells. Cases with 20% or more CD14 positivity had poorer prognosis. For FAB classes M0-M3, presence of 10% or more CD14 was negative for overall survival (P = 0.01). ANAE did not show significant prognostic influence.

  • 8.
    Hulegardh, E.
    et al.
    Dept Hematol & Coagulat, Sahlgrenska Univ Hosp, Gothenburg, Sweden.
    Hagglund, H.
    Hematol Ctr, Karolinska Univ Hosp, Stockholm, Sweden.
    Ahlberg, L.
    Dept Hematol, Linköping Univ Hosp, Linköping, Sweden.
    Karlsson, K.
    Dept Hematol & Vasc Disorders, Skåne Univ Hosp, Lund, Sweden.
    Karbach, H.
    Dept Hematol, Ctr Canc, Umeå Univ Hosp, Umeå, Sweden.
    Markuszewska, A.
    Dept Hematol, Ctr Canc, Umeå Univ Hosp, Umeå, Sweden.
    Persson, I.
    Dept Stat, Uppsala Univ, Uppsala, Sweden.
    Åström, Maria
    Örebro University Hospital. Department of Medicine, Hematology Section, Örebro University Hospital, Örebro, Sweden.
    Hallbook, H.
    Sect Hematol, Uppsala Univ, Uppsala, Sweden.
    Outcome after HSCT in Philadelphia chromosome positive acute lymphoblastic leukemia in Sweden: a population-based study2014In: Medical Oncology, ISSN 1357-0560, E-ISSN 1559-131X, Vol. 31, no 8, article id 66Article in journal (Refereed)
    Abstract [en]

    Even in the tyrosine kinase inhibitor era, allogeneic hematopoietic stem cell transplantation (HSCT) is regarded as standard care for adult Philadelphia (Ph) positive acute lymphoblastic leukemia (ALL). In this retrospective national study, we have reviewed the outcome after HSCT in Sweden for adult Ph-positive ALL between 2000 and 2009. In total, 51 patients with median age 42 (range 20-66) years underwent HSCT. Mainly allogeneic HSCT was performed (24 related donor, 24 unrelated donor and one cord blood), and only two patients were treated with an autologous HSCT. The 5-year OS was 51 (37-64) %. The probabilities of morphological relapse and non-relapse mortality (NRM) at 5 years were 36 (23-49) and 18 (9-29) %, respectively. For the allogeneic transplanted, the 5-year OS was for patients <40 years 70 (50-90) % and for patients >= 40 years 34 (16-52) %, p = 0.002. The 5-year probability of NRM was for patients <40 years 10 (2-28) % compared to 25 (11-42) % for patients >= 40 years (p = 0.04). Patients with chronic graft-versus-host disease (GVHD) had a 5-year morphological relapse probability of 20 (6-40) % compared to 59 (35-77) % for patients without chronic GVHD (p = 0.03). Age >= 40 years and the absence of chronic GVHD were confirmed as independent negative prognostic factors for relapse and non-relapse mortality in a multivariate analysis although the impact of chronic GVHD was significant only in the older age cohort.

  • 9.
    Kozlowski, Piotr
    et al.
    Örebro University, School of Health Sciences. Hematology Section, Department of Medicine.
    Bergfelt, Emma
    Hematology, Department of Medical Sciences, Uppsala University, Uppsala.
    Ahlberg, Lucia
    Department of Hematology, University Hospital of Linköping, Linköping.
    Bernell, Per
    Division of Hematology, Department of Medicine, Karolinska Institutet, Karolinska University Hospital, Stockholm.
    Hulegårdh, Erik
    Department of Hematology and Coagulation, Sahlgrenska University, Göteborg.
    Karbach, Holger
    Department of Hematology, Cancer Center, University Hospital of Umeå, Umeå .
    Karlsson, Karin
    Department of Hematology, Skåne University Hospital, Lund.
    Tomaszewska-Toporska, Beata
    Department of Hematology, Skåne University Hospital, Lund.
    Åström, Maria
    Örebro University, School of Health Sciences. Hematology Section, Department of Medicine.
    Hallböök, Helene
    Hematology, Department of Medical Sciences, Uppsala University, Uppsala.
    Age but not Philadelphia positivity impairs outcome in older/elderly patients with Acute Lymphoblastic Leukemia in the Swedish populationManuscript (preprint) (Other academic)
  • 10.
    Kozlowski, Piotr
    et al.
    Örebro University, School of Health Sciences.
    Lennmyr, Emma
    Dept of Medical Sciences, Uppsala University, Uppsala, Sweden.
    Ahlberg, Lucia
    University Hospital of Linköping, Linköping, Sweden.
    Bernell, Per
    Div of Hematology, Dept of Medicine, Karolinska University Hospital, Karolinska Institutet, Stockholm, Sweden.
    Hulegårdh, Erik
    Dept of Hematology and Coagulation, Sahlgrenska University Hospital, Göteborg, Sweden.
    Karbach, Holger
    Dept of Hematology, Cancer Center, University Hospital of Umeå, Umeå, Sweden.
    Karlsson, Karin
    Dept of Hematology and Oncology, Skåne University Hospital, Lund, Sweden.
    Tomaszewska-Toporska, Beata
    Dept of Hematology and Oncology, Skåne University Hospital, Lund, Sweden.
    Åström, Maria
    Örebro University, School of Medical Sciences. Örebro University Hospital.
    Hallböök, Heléne
    Dept of Medical Sciences, Uppsala University, Uppsala, Sweden.
    The Swedish Adult Acute Lymphoblastic Leukemia Group (SVALL), Group author
    Age but not Philadelphia positivity impairs outcome in older/elderly patients with acute lymphoblastic leukemia in Sweden2017In: European Journal of Haematology, ISSN 0902-4441, E-ISSN 1600-0609, Vol. 99, no 2, p. 141-149Article in journal (Refereed)
    Abstract [en]

    OBJECTIVES: Older/elderly patients with acute lymphoblastic leukemia (ALL) are poorly represented in clinical trials.

    METHODS: Using Swedish national leukemia registries, we investigated disease/patient characteristics, treatment choices, outcome, and the impact of an age-adapted protocol (introduced in 2009) in this population-based study of patients aged 55-85 years, diagnosed with ALL 2005-2012.

    RESULTS: Of 174 patients, 82% had B-phenotype, 11% Burkitt leukemia (excluded), and 7% T-phenotype. Philadelphia chromosome positivity (Ph+) occurred in 35%. Of the 155 B- and T- ALL patients, 80% were treated with intensive protocols, and 20% with a palliative approach. Higher age and WHO performance status ≥2 influenced the choice of palliation. Intensive, palliative, and both approaches, resulted in complete remission rate 83/16/70%, and 3 year overall survival (OS) 32/3/26%. The age-adapted protocol did not improve outcome. With intensive treatment, platelet count ≤35 × 10(9) /L, and age ≥75 years were adverse prognostic factors for OS, Ph+ was not. Male sex was an adverse prognostic factor in the 55-64 year group.

    CONCLUSIONS: We report a high frequency of Ph+ in older/elderly patients, with no evidence of poorer outcome compared to Ph negative disease. Overall prognosis for elderly patients with ALL remains dismal, despite the use of age-adapted treatment. This article is protected by copyright. All rights reserved.

  • 11.
    Kozlowski, Piotr
    et al.
    Örebro University Hospital. Hematology Section, Department of Medicine, Örebro University Hospital, Örebro, Sweden.
    Åström, Maria
    Örebro University Hospital. Hematology Section, Department of Medicine, Örebro University Hospital, Örebro, Sweden.
    Ahlberg, Lucia
    Department of Hematology, University Hospital of Linköping, Linköping, Sweden.
    Bernell, Per
    Karolinska University Hospital, Stockholm, Sweden.
    Hulegårdh, Erik
    Department of Hematology and Coagulation, Sahlgrenska University, Göteborg, Sweden.
    Hägglund, Hans
    Karolinska University Hospital, Stockholm, Sweden.
    Karlsson, Karin
    Department of Hematology, Skåne University Hospital, Lund, Sweden.
    Markuszewska-Kuczymska, Alicja
    Department of Hematology, Cancer Center, University Hospital, Umeå, Sweden.
    Tomaszewska-Toporska, Beata
    Department of Hematology, Skåne University Hospital, Lund, Sweden.
    Smedmyr, Bengt
    Department of Hematology, Uppsala University, Uppsala, Sweden.
    Amini, Rose-Marie
    Department of Immunology, Genetics and Pathology, Uppsala University, Uppsala, Sweden.
    Hallböök, Helene
    Department of Hematology, Uppsala University, Uppsala, Sweden.
    High relapse rate of T cell acute lymphoblastic leukemia in adults treated with Hyper-CVAD chemotherapy in Sweden2014In: European Journal of Haematology, ISSN 0902-4441, E-ISSN 1600-0609, Vol. 92, no 5, p. 377-381Article in journal (Refereed)
    Abstract [en]

    Background: Hyper-CVAD is widely used to treat acute lymphoblastic leukemia (ALL) and aggressive lymphomas. This multicenter, population-based study assessed the efficacy of Hyper-CVAD as first-line therapy in patients with T-cell ALL (T-ALL).

    Patients and methods: Between October 2002 and September 2006, 24 patients were diagnosed with T-ALL in Sweden; 19 were eligible for treatment with the protocol.

    Results: The median age was 32 yr (range 18-72 yr). Complete remission (CR) was obtained in 17 of 19 (89%) patients, and the treatment was relatively well tolerated. Allogeneic stem cell transplantation (SCT) was recommended in high-risk disease and was performed in four patients upfront. Two- and 5-yr leukemia-free survivals (LFS) in 17 patients with CR achievement were identical, at 29% (95% confidence interval [CI]: 8-51). Two- and 5-yr overall survival (OS) in whole cohort was 63% (95% CI: 42-85) and 47% (95% CI: 26-69), respectively. The 5-yr LFS for 15 patients who did not receive allogeneic SCT upfront were 20% (95% CI: 0-40), although 14 of 15 completed the protocol (eight cycles). Relapse occurred in 2 of 4 upfront-transplanted patients and in 12 of 15 patients treated with chemotherapy alone, six of whom received allogeneic SCT in CR2. Age ≥35 yr influenced OS negatively in univariate analysis (HR 5.1, 95% CI: 1.55-16.7).

    Conclusions: Hyper-CVAD treatment resulted in a high CR rate and appeared safe, but it showed poor efficacy at preventing relapse. Therefore, this treatment is no longer recommended for adults with T-ALL in Sweden.

  • 12.
    Kozlowski, Piotr
    et al.
    Hematology Section, Department of Medicine, Örebro University Hospital, Örebro, Sweden.
    Åström, Maria
    Hematology Section, Department of Medicine, Örebro University Hospital, Örebro, Sweden.
    Ahlberg, Lucia
    Department of Hematology, University Hospital of Linköping, Linköping, Sweden.
    Bernell, Per
    Karolinska University Hospital, Stockholm, Sweden.
    Hulegårdh, Erik
    Department of Hematology and Coagulation, Sahlgrenska Hospital, Gothenburg, Sweden.
    Hägglund, Hans
    Karolinska University Hospital, Stockholm, Sweden.
    Karlsson, Karin
    Department of Hematology, Skåne University Hospital, Lund, Sweden.
    Markuszewska-Kuczymska, Alicja
    Department of Hematology, Cancer Center, University Hospital, Umeå, Sweden.
    Tomaszewska-Toporska, Beata
    Department of Hematology, Skåne University Hospital, Lund, Sweden.
    Smedmyr, Bengt
    Department of Hematology, Uppsala University, Uppsala, Sweden.
    Hallböök, Helene
    Department of Hematology, Uppsala University, Uppsala, Sweden.
    High curability via intensive reinduction chemotherapy and stem cell transplantation in young adults with relapsed acute lymphoblastic leukemia in Sweden 2003-20072012In: Haematologica, ISSN 0390-6078, E-ISSN 1592-8721, Vol. 97, no 9, p. 1414-1421Article in journal (Refereed)
    Abstract [en]

    BACKGROUND: A minority of patients with adult acute lymphoblastic leukemia who relapse are rescued. The aim of this population-based study was to assess the results of reinduction treatment and allogeneic stem cell transplantation in patients in second complete remission.

    DESIGN AND METHODS: Between 2003-2007, 76 adults (<66 years) with relapsed acute lymphoblastic leukemia (Burkitt's leukemia excluded) were prospectively reported to The Swedish Adult Acute Leukemia Registry and later evaluated.

    RESULTS: Reinduction with: (i) mitoxantrone, etoposide, and cytarabine (MEA); (ii) fludarabine, cytarabine, pegylated-asparaginase plus granulocyte colony-stimulating factor (FLAG-Asp); and (iii) cytarabine, betamethasone, cyclophosphamide, daunorubicin, and vincristine (ABCDV) resulted in complete remission in 6/9 (67%), 10/16 (63%) and 9/21 (43%) of the patients, respectively. Allogeneic stem cell transplantation was performed during second complete remission in 29 patients. Multivariate analysis regarding overall survival after relapse revealed that age over 35 years at diagnosis and relapse within 18 months were negative prognostic factors. Overall survival rates at 3 and 5 years were 22% (95% CI: 13-32) and 15% (95% CI: 7-24). Of 19 patients less than 35 years at diagnosis who underwent allogeneic stem cell transplantation in second remission, ten (53%) are still alive at a median of 5.5 years (range, 4.2-8.3) after relapse, whereas all patients over 35 years old at diagnosis have died.

    CONCLUSIONS: Allogeneic stem cell transplantation remains the treatment of choice for young adults with relapsed acute lymphoblastic leukemia. Both (i) mitoxantrone, etoposide, and cytarabine and (ii) fludarabine, cytarabine, pegylated-asparaginase plus granulocyte colony-stimulating factor seem effective as reinduction treatments and should be further evaluated. New salvage strategies are needed, especially for patients over 35 years old at diagnosis.

  • 13.
    Lennmyr, Emma Bergfelt
    et al.
    Department of Medical Sciences, Uppsala University Hospital, Uppsala, Sweden.
    Kozlowski, Piotr
    Department of Medicine.
    Ahlberg, Lucia
    Department of Hematology, University Hospital of Linköping, Linköping, Sweden.
    Bernell, Per
    Division of Hematology, Department of Medicine, Karolinska University Hospital, Karolinska Institutet, Stockholm, Sweden.
    Hulegårdh, Erik
    Department of Hematology and Coagulation, Sahlgrenska University Hospital, Göteborg, Sweden.
    Izarra, Antonio Santamaria
    Department of Hematology, Cancer Center, University Hospital of Umeå, Umeå, Sweden.
    Karlsson, Karin
    Department of Hematology and Oncology, Skåne University Hospital, Lund, Sweden.
    Tomaszewska-Toporska, Beata
    Department of Hematology and Oncology, Skåne University Hospital, Lund, Sweden.
    Åström, Maria
    Örebro University, School of Medical Sciences. Örebro University Hospital. Department of Medicine.
    Hallböök, Helene
    Department of Medical Sciences, Uppsala University Hospital, Uppsala, Sweden.
    Swedish Adult Acute Lymphoblastic Leukemia Group (SVALL), Group Author
    Real-world data on first relapse of acute lymphoblastic leukemia in patients >55 years2018In: Leukemia and Lymphoma, ISSN 1042-8194, E-ISSN 1029-2403, Vol. 59, no 10, p. 2470-2473Article in journal (Refereed)
  • 14. Welander, Edward
    et al.
    Åström, Maria
    Örebro University, School of Medical Sciences. Örebro University, School of Health Sciences.
    Enonge Fotabe, Leslie
    Kardeby, Caroline
    Örebro University, School of Medical Sciences.
    Tina, Elisabet
    Örebro University, School of Medical Sciences.
    Elgbratt, Kristina
    Örebro University, School of Health Sciences.
    Pourlofti, Arvid
    Abawi, Akram
    Romild, Alma
    Kruse, Robert
    Örebro University, School of Medical Sciences.
    Repsilber, Dirk
    Örebro University, School of Medical Sciences.
    Crafoord, Jakob
    Ahlstrand, Erik
    Ivarsson, Mikael
    Örebro University, School of Health Sciences.
    Integrated analysis indicates reciprocal immune response dysregulations between bone marrow multipotent stromal cells and granulocytes at the mRNA but not at the protein level in myelofibrosis2018Conference paper (Refereed)
  • 15.
    Åström, Maria
    Dpt of medicine and laboratory medicine, Örebro University Hospital, Örebro, Sweden.
    Clinical cases: Presentation, diagnosis, treatment and follow-up: Case 2 - Chuvash polycythemia2015In: Congenital Erythrocytosis and Hereditary Thrombocytosis: Clinical presentation, diagnosis, treatment and follow-up. A practical guide with clinical cases. / [ed] Sylvie Hermouet, Portugal: European cooperation in science and technology , 2015Chapter in book (Other academic)
  • 16.
    Åström, Maria
    Örebro University, School of Medical Sciences. Örebro University Hospital.
    Undersökningar av X-bunden trombocytopeni med talassemi (XLTT) och jämförelser med andra former av myelofibros2010In: Oss hematologer emellan, Vol. 4, no 22, p. 39-40Article in journal (Other academic)
  • 17.
    Åström, Maria
    et al.
    Örebro University, School of Health and Medical Sciences, Örebro University, Sweden.
    Hahn-Strömberg, Victoria
    Örebro University, School of Health and Medical Sciences, Örebro University, Sweden.
    Zetterberg, Eva
    Departments of Medicine and Hematology, Karolinska Institutet, Karolinska University Hospital Huddinge, Stockholm, Sweden .
    Vedin, Inger
    Departments of Medicine and Hematology, Karolinska Institutet, Karolinska University Hospital Huddinge, Stockholm, Sweden .
    Merup, Mats
    Departments of Medicine and Hematology, Karolinska Institutet, Karolinska University Hospital Huddinge, Stockholm, Sweden .
    Palmblad, Jan
    Departments of Medicine and Hematology, Karolinska Institutet, Karolinska University Hospital Huddinge, Stockholm, Sweden .
    X-linked thrombocytopenia with thalassemia displays bone marrow reticulin fibrosis and enhanced angiogenesis: comparisons with primary myelofibrosis2015In: American Journal of Hematology, ISSN 0361-8609, E-ISSN 1096-8652, Vol. 90, no 3, p. E44-E48Article in journal (Refereed)
    Abstract [en]

    X-linked thrombocytopenia with thalassemia (XLTT) is caused by the mutation 216R > Q in exon 4 of the GATA1 gene. Male hemizygous patients display macrothrombocytopenia, splenomegaly, and a β-thalassemia trait. We describe two XLTT families where three males were initially misdiagnosed as having primary myelofibrosis (PMF) and all five investigated males showed mild-moderate bone marrow (BM) reticulin fibrosis. Comparative investigations were performed on blood samples and BM biopsies from males with XLTT, PMF patients and healthy controls. Like PMF, XLTT presented with high BM microvessel density, low GATA1 protein levels in megakaryocytes, and elevated blood CD34+ cell counts. But unlike PMF, the BM microvessel pericyte coverage was low in XLTT, and no collagen fibrosis was found. Further, as evaluated by immunohistochemistry, expressions of the growth factors VEGF, AGGF1, and CTGF were low in XLTT megakaryocytes and microvessels but high in PMF. Thus, although the reticulin fibrosis in XLTT might simulate PMF, opposing stromal and megakaryocyte features may facilitate differential diagnosis. Additional comparisons between these disorders may increase the understanding of mechanisms behind BM fibrosis in relation to pathological megakaryopoiesis.

  • 18.
    Åström, Maria
    et al.
    Örebro University, School of Medical Sciences. Örebro University Hospital. Division of Hematology, Department of Medicine, Örebro University Hospital, Örebro, Sweden; Department of Laboratory Medicine, Faculty of Medicine and Health, Örebro University, Örebro, Sweden; iRiSC – Inflammatory Response and Infection Susceptibility Centre, Faculty of Medicine and Health, Örebro University, Örebro, Sweden.
    Tajeddinn, Walid
    iRiSC – Inflammatory Response and Infection Susceptibility Centre, Faculty of Medicine and Health, Örebro University, Örebro, Sweden.
    Karlsson, Mats G.
    Örebro University, School of Medical Sciences. Department of Laboratory Medicine.
    Linder, Olle
    Division of Hematology, Department of Medicine, Örebro University Hospital, Örebro, Sweden.
    Palmblad, Jan
    Division of Hematology, Department of Medicine, Karolinska University Hospital Huddinge, Karolinska Institutet, Stockholm, Sweden.
    Lindblad, Per
    Örebro University, School of Medical Sciences. Department of Urology.
    Cytokine Measurements for Diagnosing and Characterizing Leukemoid Reactions and Immunohistochemical Validation of a Granulocyte Colony-Stimulating Factor and CXCL8-Producing Renal Cell Carcinoma2018In: Biomarker Insights, ISSN 1177-2719, E-ISSN 1177-2719, Vol. 13, article id UNSP 1177271918792246Article in journal (Refereed)
    Abstract [en]

    BACKGROUND: Various paraneoplastic syndromes are encountered in renal cell carcinomas. This case report illustrates that a paraneoplastic leukemoid reaction may precede the diagnosis of renal cell carcinoma and be explained by cytokine production from the cancer cells.

    CASE PRESENTATIONS: A 64-year-old man was referred for hematology workup due to pronounced leukocytosis. While being evaluated for a possible hematologic malignancy as the cause, he was found to have a metastasized renal cell carcinoma, and hyperleukocytosis was classified as a leukemoid reaction. A multiplex panel for measurement of 25 serum cytokines/chemokines showed highly elevated levels of granulocyte colony-stimulating factor (G-CSF) and CXCL8 (C-X-C-motif chemokine ligand 8, previously known as interleukin [IL]-8). By immunohistochemistry it was shown that the renal carcinoma cells expressed both these cytokines. Two additional, consecutive patients with renal cell carcinoma with paraneoplastic leukocytosis also showed elevated serum levels of CXCL8, but not of G-CSF. Nonparametric statistical evaluation showed significantly higher serum concentrations of CXCL8, IL-6, IL-10, monocyte chemoattractant protein 1 (MCP-1), and tumor necrosis factor, but lower interferon gamma (IFN-gamma) and IL-1 alpha, for the 3 renal cell carcinoma cases compared with healthy blood donors.

    CONCLUSIONS: In suspected paraneoplastic leukocytosis, multiplex serum cytokine analyses may facilitate diagnosis and provide an understanding of the mechanisms for the reaction. In the index patient, combined G-CSF and CXCL8 protein expression by renal carcinoma cells was uniquely documented. A rapidly fatal course was detected in all 3 cases, congruent with the concept that autocrine/paracrine growth signaling in renal carcinoma cells may induce an aggressive tumor phenotype. Immune profiling studies could improve our understanding for possible targets when choosing therapies for patients with metastatic renal cell carcinoma.

  • 19.
    Åström, Maria
    et al.
    Örebro University, School of Medical Sciences. Örebro University Hospital.
    Welander, Edvard
    Pourlotfi, Arvid
    Abawi, Akram
    Ahlstrand, Erik
    Ivarsson, Mikael
    Örebro University, School of Health Sciences.
    Activated interferon signaling in cultured BMSC from myelofibrosis patients: core finding of a proteomic study2018Conference paper (Refereed)
1 - 19 of 19
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