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Sinusoidal Obstruction Syndrome/Veno-Occlusive Disease after Autologous or Allogeneic Hematopoietic Stem Cell Transplantation in Children: a retrospective study of the Italian Hematology-Oncology Association-Hematopoietic Stem Cell Transplantation Group.
Biol Blood Marrow Transplant. 2019 02; 25(2):313-320.BB

Abstract

Sinusoidal obstruction syndrome (SOS), also known as veno-occlusive disease (VOD), is a potentially life-threatening complication that may develop after hematopoietic stem cell transplantation (HSCT). The aims of this retrospective multicenter study were to evaluate the incidence of SOS/VOD in a large cohort of children transplanted in centers across Italy by applying the new European Society for Blood and Marrow Transplantation (EBMT) criteria and to analyze the risk factors underlying this complication. We retrospectively reviewed data of pediatric HSCTs performed in 13 AIEOP (Associazione Italiana di Ematologia e Oncologia Pediatrica)-affiliated centers between January 2000 and April 2016. The new pediatric EBMT criteria were retrospectively applied for diagnoses of SOS/VOD and severity grading. Among 5072 transplants considered at risk for SOS/VOD during the study period, 103 children (2%) developed SOS/VOD, and the grade was severe or very severe in all patients. The median time of SOS/VOD occurrence was 17 days after HSCT (range, 1 to 104). Sixty-nine patients (67%) were treated with defibrotide for a median time of 16 days (range, 4 to 104). In multivariable analysis age < 2 years, use of busulfan during the conditioning regimen, female gender, and hemophagocytic lymphohistiocytosis were risk factors statistically associated with the development of SOS/VOD. The overall mortality directly related to SOS/VOD was 15.5%. Overall survival at 1 year was worse in patients with SOS/VOD (P = .0033), and this difference disappeared 5 years after HSCT. Nonrelapse mortality was significantly higher 1 and 5 years after transplantation in patients who developed SOS/VOD (P < .001). Based on the application of new EBMT criteria, the overall incidence of SOS/VOD recorded in this large Italian pediatric retrospective study was 2%. Nonrelapse mortality was significantly higher in patients who developed SOS/VOD. Identifying the risk factors associated with SOS/VOD can lead to more effective early treatment strategies of this potentially fatal HSCT complication in childhood.

Authors+Show Affiliations

Hematopoietic Stem Cell Unit, Department of Hematology-Oncology, IRCSS-Istituto G. Gaslini, Genova, Italy. Electronic address: maurafaraci@alice.it.Department of Pediatric Hematology and Oncology, IRCCS, Ospedale Bambino Gesù, Rome, Italy.Department of Pediatric Oncology, Fondazione IRCCS Istituto Nazionale dei Tumori, Milan, Italy.Pediatric Hematology-Oncology Unit, Department of Women's and Children's Health, Azienda Ospedaliera-University of Padova, Padova, Italy.Hematopoietic Stem Cell Unit, Department of Hematology-Oncology, IRCSS-Istituto G. Gaslini, Genova, Italy.Pediatric Onco-Hematology, Stem Cell Transplantation and Cellular Therapy Division, Regina Margherita Children's Hospital, Torino, Italy.Oncology, Hematology and Hematopoietic Stem Cell Transplant Program, U.O. Pediatrics- S. Orsola-Malpighi University of Bologna, Bologna, Italy.Haematopoietic Stem Cell Transplantation Unit, Pediatric Clinic of University of Pisa, Pisa, Italy.Department of Hemato-Oncology, Santobono-Pausilipon Hospital, BMT Unit, Napoli, Italy.Transplantation Unit, Department of Pediatric Oncology, Meyer Children's Hospital, Florence, Italy.Pediatric Hematology Oncology, Azienda Ospedaliera Universitaria Integrata, Verona, Italy.Ospedale Civile, Dipartimento di Ematologia, Medicina Trasfusionale e Biotecnologie, Pescara, Italy.Bone Marrow Transplant Center, Hospital Binaghi and Microcitemico, Cagliari, Italy.Department of Pediatric Hematology and Oncology, IRCCS, Ospedale Bambino Gesù, Rome, Italy.Pediatric Hematology/Oncology, Fondazione IRCCS Policlinico San Matteo, Pavia, Italy.

Pub Type(s)

Clinical Trial
Journal Article
Multicenter Study

Language

eng

PubMed ID

30266674

Citation

Faraci, Maura, et al. "Sinusoidal Obstruction Syndrome/Veno-Occlusive Disease After Autologous or Allogeneic Hematopoietic Stem Cell Transplantation in Children: a Retrospective Study of the Italian Hematology-Oncology Association-Hematopoietic Stem Cell Transplantation Group." Biology of Blood and Marrow Transplantation : Journal of the American Society for Blood and Marrow Transplantation, vol. 25, no. 2, 2019, pp. 313-320.
Faraci M, Bertaina A, Luksch R, et al. Sinusoidal Obstruction Syndrome/Veno-Occlusive Disease after Autologous or Allogeneic Hematopoietic Stem Cell Transplantation in Children: a retrospective study of the Italian Hematology-Oncology Association-Hematopoietic Stem Cell Transplantation Group. Biol Blood Marrow Transplant. 2019;25(2):313-320.
Faraci, M., Bertaina, A., Luksch, R., Calore, E., Lanino, E., Saglio, F., Prete, A., Menconi, M., De Simone, G., Tintori, V., Cesaro, S., Santarone, S., Orofino, M. G., Locatelli, F., & Zecca, M. (2019). Sinusoidal Obstruction Syndrome/Veno-Occlusive Disease after Autologous or Allogeneic Hematopoietic Stem Cell Transplantation in Children: a retrospective study of the Italian Hematology-Oncology Association-Hematopoietic Stem Cell Transplantation Group. Biology of Blood and Marrow Transplantation : Journal of the American Society for Blood and Marrow Transplantation, 25(2), 313-320. https://doi.org/10.1016/j.bbmt.2018.09.027
Faraci M, et al. Sinusoidal Obstruction Syndrome/Veno-Occlusive Disease After Autologous or Allogeneic Hematopoietic Stem Cell Transplantation in Children: a Retrospective Study of the Italian Hematology-Oncology Association-Hematopoietic Stem Cell Transplantation Group. Biol Blood Marrow Transplant. 2019;25(2):313-320. PubMed PMID: 30266674.
* Article titles in AMA citation format should be in sentence-case
TY - JOUR T1 - Sinusoidal Obstruction Syndrome/Veno-Occlusive Disease after Autologous or Allogeneic Hematopoietic Stem Cell Transplantation in Children: a retrospective study of the Italian Hematology-Oncology Association-Hematopoietic Stem Cell Transplantation Group. AU - Faraci,Maura, AU - Bertaina,Alice, AU - Luksch,Roberto, AU - Calore,Elisabetta, AU - Lanino,Edoardo, AU - Saglio,Francesco, AU - Prete,Arcangelo, AU - Menconi,Mariacristina, AU - De Simone,Giusy, AU - Tintori,Veronica, AU - Cesaro,Simone, AU - Santarone,Stella, AU - Orofino,Maria Grazia, AU - Locatelli,Franco, AU - Zecca,Marco, Y1 - 2018/09/26/ PY - 2018/06/11/received PY - 2018/09/19/accepted PY - 2018/9/30/pubmed PY - 2019/12/25/medline PY - 2018/9/30/entrez KW - Children KW - Diagnostic criteria KW - Severity grading KW - Sinusoidal obstruction syndrome SP - 313 EP - 320 JF - Biology of blood and marrow transplantation : journal of the American Society for Blood and Marrow Transplantation JO - Biol. Blood Marrow Transplant. VL - 25 IS - 2 N2 - Sinusoidal obstruction syndrome (SOS), also known as veno-occlusive disease (VOD), is a potentially life-threatening complication that may develop after hematopoietic stem cell transplantation (HSCT). The aims of this retrospective multicenter study were to evaluate the incidence of SOS/VOD in a large cohort of children transplanted in centers across Italy by applying the new European Society for Blood and Marrow Transplantation (EBMT) criteria and to analyze the risk factors underlying this complication. We retrospectively reviewed data of pediatric HSCTs performed in 13 AIEOP (Associazione Italiana di Ematologia e Oncologia Pediatrica)-affiliated centers between January 2000 and April 2016. The new pediatric EBMT criteria were retrospectively applied for diagnoses of SOS/VOD and severity grading. Among 5072 transplants considered at risk for SOS/VOD during the study period, 103 children (2%) developed SOS/VOD, and the grade was severe or very severe in all patients. The median time of SOS/VOD occurrence was 17 days after HSCT (range, 1 to 104). Sixty-nine patients (67%) were treated with defibrotide for a median time of 16 days (range, 4 to 104). In multivariable analysis age < 2 years, use of busulfan during the conditioning regimen, female gender, and hemophagocytic lymphohistiocytosis were risk factors statistically associated with the development of SOS/VOD. The overall mortality directly related to SOS/VOD was 15.5%. Overall survival at 1 year was worse in patients with SOS/VOD (P = .0033), and this difference disappeared 5 years after HSCT. Nonrelapse mortality was significantly higher 1 and 5 years after transplantation in patients who developed SOS/VOD (P < .001). Based on the application of new EBMT criteria, the overall incidence of SOS/VOD recorded in this large Italian pediatric retrospective study was 2%. Nonrelapse mortality was significantly higher in patients who developed SOS/VOD. Identifying the risk factors associated with SOS/VOD can lead to more effective early treatment strategies of this potentially fatal HSCT complication in childhood. SN - 1523-6536 UR - https://www.unboundmedicine.com/medline/citation/30266674/Sinusoidal_Obstruction_Syndrome/Veno_Occlusive_Disease_after_Autologous_or_Allogeneic_Hematopoietic_Stem_Cell_Transplantation_in_Children:_a_retrospective_study_of_the_Italian_Hematology_Oncology_Association_Hematopoietic_Stem_Cell_Transplantation_Group_ L2 - https://linkinghub.elsevier.com/retrieve/pii/S1083-8791(18)30594-9 DB - PRIME DP - Unbound Medicine ER -