Hematopoietic Stem Cell Transplantation Positively Affects the Natural History of Cancer in Nijmegen Breakage Syndrome.


Journal

Clinical cancer research : an official journal of the American Association for Cancer Research
ISSN: 1557-3265
Titre abrégé: Clin Cancer Res
Pays: United States
ID NLM: 9502500

Informations de publication

Date de publication:
15 01 2021
Historique:
received: 03 07 2020
revised: 26 08 2020
accepted: 16 10 2020
pubmed: 22 10 2020
medline: 11 1 2022
entrez: 21 10 2020
Statut: ppublish

Résumé

Nijmegen breakage syndrome (NBS) is a DNA repair disorder with a high predisposition to hematologic malignancies. We describe the natural history of NBS, including cancer incidence, risk of death, and the potential effectiveness of hematopoietic stem cell transplantation (HSCT) in preventing both pathologies: malignancy and immunodeficiency. Among 241 patients with NBS enrolled in the study from 11 countries, 151 (63.0%) patients were diagnosed with cancer. Incidence rates for primary and secondary cancer, tumor characteristics, and risk factors affecting overall survival (OS) were estimated. The cumulative cancer incidence was 40.21% ± 3.5% and 77.78% ± 3.4% at 10 years and 20 years of follow-up, respectively. Most of the tumors There is a beneficial effect of HSCT on the long-term survival of patients with NBS transplanted in their first complete remission of cancer.

Identifiants

pubmed: 33082212
pii: 1078-0432.CCR-20-2574
doi: 10.1158/1078-0432.CCR-20-2574
doi:

Types de publication

Journal Article Multicenter Study Research Support, Non-U.S. Gov't

Langues

eng

Sous-ensembles de citation

IM

Pagination

575-584

Informations de copyright

©2020 American Association for Cancer Research.

Références

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Auteurs

Beata Wolska-Kusnierz (B)

Department of Immunology, Children's Memorial Health Institute, Warsaw, Poland.

Agata Pastorczak (A)

Department Pediatrics, Oncology and Hematology, Medical University of Lodz, Lodz, Poland.

Wojciech Fendler (W)

Department of Biostatistics and Translational Medicine, Medical University of Lodz, Lodz, Poland.
Department of Radiation Oncology, Dana-Farber Cancer Institute, Boston, Massachusetts.

Anna Wakulinska (A)

Department of Oncology, Children's Memorial Health Institute, Warsaw, Poland.

Bozena Dembowska-Baginska (B)

Department of Oncology, Children's Memorial Health Institute, Warsaw, Poland.

Edyta Heropolitanska-Pliszka (E)

Department of Immunology, Children's Memorial Health Institute, Warsaw, Poland.

Barbara Piątosa (B)

Histocompatibility Laboratory, Children's Memorial Health Institute, Warsaw, Poland.

Barbara Pietrucha (B)

Department of Immunology, Children's Memorial Health Institute, Warsaw, Poland.

Krzysztof Kałwak (K)

Department of Pediatric Hematology, Oncology and Bone Marrow Transplantation, Wroclaw Medical University, Wroclaw, Poland.

Marek Ussowicz (M)

Department of Pediatric Hematology, Oncology and Bone Marrow Transplantation, Wroclaw Medical University, Wroclaw, Poland.

Anna Pieczonka (A)

Department of Pediatric Oncology, Poznan University of Medical Sciences, Poznan, Poland.

Katarzyna Drabko (K)

Department of Pediatric Hematology, Oncology and Transplantology, Medical University of Lublin, Poland.

Monika Lejman (M)

Department of Pediatric Hematology, Oncology and Transplantology, Medical University of Lublin, Poland.

Sylwia Koltan (S)

Department of Pediatric Hematology and Oncology, Collegium Medicum, Nicolaus Copernicus University Torun, Bydgoszcz, Poland.

Jolanta Gozdzik (J)

Department of Transplantation, Institute of Pediatrics, Jagiellonian University Medical College, Krakow, Poland.

Jan Styczynski (J)

Department of Pediatric Hematology and Oncology, Collegium Medicum, Nicolaus Copernicus University Torun, Bydgoszcz, Poland.

Alina Fedorova (A)

Belarusian Research Center for Pediatric Oncology and Hematology, Minsk, Belarus.

Natalia Miakova (N)

Department of Pediatric Oncology and Hematology, Federal Research Center for Pediatric Hematology, Oncology and Immunology, Moscow, Russia.

Elena Deripapa (E)

Department of Immunology and Hematopoietic Stem Cell Transplantation, Federal Research Center for Pediatric Hematology, Oncology and Immunology, Moscow, Russia.

Larysa Kostyuchenko (L)

Department of Pediatric Immunology, Western Ukrainian Specialized Children's Medical Centre, Lviv, Ukraine.

Zdenka Krenova (Z)

Department of Pediatric Oncology, University Hospital and Faculty of Medicine, Masaryk University, Brno, Czech Republic.

Eva Hlavackova (E)

Department of Pediatric Oncology, University Hospital and Faculty of Medicine, Masaryk University, Brno, Czech Republic.
Department of Clinical Immunology and Allergology, St. Anne's University Hospital in Brno and Faculty of Medicine, Masaryk University, Brno, Czech Republic.

Andrew R Gennery (AR)

Translational and Clinical Research Institute, Newcastle University and Newcastle upon Tyne Hospitals NHS Foundation Trust, Newcastle, United Kingdom.

Karl-Walter Sykora (KW)

Department of Pediatrics, Hannover Medical School (MHH), Hannover, Germany.

Sujal Ghosh (S)

Department of Pediatric Oncology, Hematology and Clinical Immunology, Medical Faculty, Center of Child and Adolescent Health, Heinrich-Heine-University, Düsseldorf, Germany.

Michael H Albert (MH)

Dr. von Hauner University Children's Hospital, Ludwig-Maximilians-University, Munich, Germany.

Dmitry Balashov (D)

Department of Hematopoietic Stem Cell Transplantation, Dmitriy Rogachev National Center for Pediatric Hematology, Oncology, and Immunology, Moscow, Russia.

Mary Eapen (M)

Center for International Blood and Marrow Transplant, Department of Medicine, Medical College of Wisconsin, Milwaukee, Wisconsin.

Peter Svec (P)

Department of Pediatric Hematology and Oncology, Comenius University and National Institute of Children's Diseases, Bratislava, Slovakia.

Markus G Seidel (MG)

Research Unit Pediatric Hematology and Immunology, Division of Pediatric Hematology-Oncology, Department of Pediatrics and Adolescent Medicine, Medical University Graz, Graz, Austria.

Sara S Kilic (SS)

Pediatric Immunology Division, Department of Pediatrics, Uludag University Medical Faculty, Bursa, Turkey.

Agnieszka Tomaszewska (A)

Department of Hematology, Oncology and Internal Medicine, Medical University of Warsaw, Warsaw, Poland.

Ewa Wiesik-Szewczyk (E)

Department of Internal Medicine, Pneumonology, Allergology and Clinical Immunology, Central Clinical Hospital of the Ministry of National Defense, Military Institute of Medicine, Warsaw, Poland.

Alexandra Kreins (A)

Great Ormond Street Hospital for Children NHS Foundation Trust, London, United Kingdom.

Johann Greil (J)

Department of Pediatric Hematology and Oncology, University Hospital, Heidelberg, Germany.

Jochen Buechner (J)

Department of Pediatric Hematology and Oncology, Oslo University Hospital, Oslo, Norway.

Bendik Lund (B)

Pediatric Department, St Olav University Hospital, Trondheim, Norway.

Hanna Gregorek (H)

Department of Microbiology and Clinical Immunology, The Children's Memorial Health Institute, Warsaw, Poland.

Krystyna Chrzanowska (K)

Department of Medical Genetics, The Children's Memorial Health Institute, Warsaw, Poland.

Wojciech Mlynarski (W)

Department Pediatrics, Oncology and Hematology, Medical University of Lodz, Lodz, Poland. wojciech.mlynarski@umed.lodz.pl.

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