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Outcomes of Infants and Young Children With Relapsed Medulloblastoma After Initial Craniospinal Irradiation-Sparing Approaches: An International Cohort Study

C. Erker, M. Mynarek, S. Bailey, CM. Mazewski, L. Baroni, M. Massimino, J. Hukin, D. Aguilera, AM. Cappellano, V. Ramaswamy, A. Lassaletta, S. Perreault, CN. Kline, R. Rajagopal, G. Michaiel, M. Zapotocky, V. Santa-Maria Lopez, AM. La Madrid, C....

. 2023 ; 41 (10) : 1921-1932. [pub] 20221222

Jazyk angličtina Země Spojené státy americké

Typ dokumentu časopisecké články, práce podpořená grantem

Perzistentní odkaz   https://www.medvik.cz/link/bmc23003706

Grantová podpora
Cancer Research UK - United Kingdom

PURPOSE: Infant and young childhood medulloblastoma (iMB) is usually treated without craniospinal irradiation (CSI) to avoid neurocognitive late effects. Unfortunately, many children relapse. The purpose of this study was to assess salvage strategies and prognostic features of patients with iMB who relapse after CSI-sparing therapy. METHODS: We assembled a large international cohort of 380 patients with relapsed iMB, age younger than 6 years, and initially treated without CSI. Univariable and multivariable Cox models of postrelapse survival (PRS) were conducted for those treated with curative intent using propensity score analyses to account for confounding factors. RESULTS: The 3-year PRS, for 294 patients treated with curative intent, was 52.4% (95% CI, 46.4 to 58.3) with a median time to relapse from diagnosis of 11 months. Molecular subgrouping was available for 150 patients treated with curative intent, and 3-year PRS for sonic hedgehog (SHH), group 4, and group 3 were 60%, 84%, and 18% (P = .0187), respectively. In multivariable analysis, localized relapse (P = .0073), SHH molecular subgroup (P = .0103), CSI use after relapse (P = .0161), and age ≥ 36 months at initial diagnosis (P = .0494) were associated with improved survival. Most patients (73%) received salvage CSI, and although salvage chemotherapy was not significant in multivariable analysis, its use might be beneficial for a subset of children receiving salvage CSI < 35 Gy (P = .007). CONCLUSION: A substantial proportion of patients with relapsed iMB are salvaged after initial CSI-sparing approaches. Patients with SHH subgroup, localized relapse, older age at initial diagnosis, and those receiving salvage CSI show improved PRS. Future prospective studies should investigate optimal CSI doses and the role of salvage chemotherapy in this population.

Cancer and Blood Disease Institute Children's Hospital Los Angeles Keck School of Medicine of University of Southern California Los Angeles CA

Center for Cancer and Blood Disorders Phoenix Children's Hospital Phoenix AZ

Centre Hospitalier Universitaire Sainte Justine Université de Montreal Montreal QC Canada

Children's Cancer Centre Royal Children's Hospital

Children's Healthcare of Atlanta and Emory University Atlanta GA

Children's Hospital of Colorado and University of Colorado School of Medicine Denver CO

Dana Farber Boston Children's Cancer and Blood Disorder Center Boston MA

Department of Developmental Neurobiology St Jude Children's Research Hospital Memphis TN

Department of Oncology Hospital Sant Joan de Déu Barcelona Spain

Department of Oncology St Jude Children's Research Hospital Memphis TN

Department of Paediatric Haematology and Oncology 2nd Faculty of Medicine Charles University and University Hospital Motol Prague Czech Republic

Department of Pediatric Hematology and Oncology Hospital Infantil Universitario Niño Jesús Madrid Spain

Department of Pediatric Hematology and Oncology University Medical Center Hamburg Eppendorf Hamburg Germany

Department of Pediatrics Centre Mère enfant Soleil du CHU de Québec CRCHU de Québec Université Laval Quebec City QC Canada

Department of Pediatrics Division of Hematology Oncology University of Alabama at Birmingham Birmingham AL

Department of Pediatrics Memorial Sloan Kettering Cancer Center New York NY

Department of Pediatrics Stollery Children's Hospital University of Alberta Edmonton AB Canada

Department of Pediatrics University of California San Francisco San Francisco CA

Departments of Pediatric Hematology and Oncology Dell Children's Medical Center of Central Texas and University of Texas Austin TX

Division of Haematology Oncology Department of Paediatrics IWK Health Centre and Dalhousie University Halifax NS Canada

Division of Haematology Oncology Department of Paediatrics The Hospital for Sick Children and University of Toronto Toronto ON Canada

Division of Hematology and Oncology Children's Hospital of Richmond and Virginia Commonwealth University Richmond VA

Division of Hematology Oncology and Blood and Marrow Transplant Nationwide Children's Hospital and The Ohio State University Columbus OH

Division of Hematology Oncology and Bone Marrow Transplant Nationwide Children's Hospital The Ohio State University Columbus OH

Division of Hematology Oncology Department of Paediatrics IWK Health Centre and Dalhousie University Halifax NS Canada

Division of Hematology Oncology Department of Pediatrics University Malaya Medical Center Kuala Lumpur Malaysia

Division of Oncology The Children's Hospital of Philadelphia Philadelphia PA

Division of Pediatric Hematology and Oncology Children's Mercy Hospital Kansas City MO

Division of Pediatric Hematology Oncology and Bone Marrow Transplant Washington University School of Medicine in St Louis St Louis MO

Division of Pediatric Hematology Oncology BMT Medical College of Wisconsin Milwaukee WI

Division of Pediatric Hematology Oncology Cook Children's Medical Center Fort Worth TX

Division of Pediatric Hematology Oncology Western University London ON Canada

Division of Pediatric Oncology BMT Instituto de Oncologia Pediátrica GRAACC UNIFESP São Paulo Brazil

Divisions of Neurology and Hematology Oncology Bone Marrow Transplant Department of Pediatrics British Columbia Children's Hospital and University of British Columbia Vancouver BC Canada

Fondazione Istituto Di Ricovero e Cura a Carattere Scientifico Istituto Nazionale dei Tumori Milan Italy

Hospital of Pediatrics SAMIC Prof Dr Juan P Garrahan Buenos Aires Argentina

Mildred Scheel Cancer Career Center HaTriCS4 University Medical Center Hamburg Eppendorf Hamburg Germany

Monash Children's Cancer Centre Monash Children's Hospital Monash Health Center for Cancer Research Hudson Institute of Medical Research and Department of Molecular and Translational Science School of Medicine Nursing and Health Science Monash University Melbourne Australia

Murdoch Children's Research Institute

Nemours Children's Health Wolfson's Children's Hospital and University of Florida Jacksonville FL

Pediatric Hemato Oncology Department Sheba Medical Center at Tel HaShomer Ramat Gan Israel

Pediatric Hematology and Oncology Pediatrics 3 University Hospital of Essen Essen Germany

Pediatric Oncology The Sidney Kimmel Comprehensive Cancer Center at Johns Hopkins Baltimore MD

Radiation Medicine Program Princess Margaret Cancer Centre University Health Network Toronto ON Canada

Research Methods Unit Nova Scotia Health Authority Halifax NS Canada

Schneider Children's Medical Center of Israel Petah Tikva and Sackler faculty of Medicine Tel Aviv University Israel

Section of Pediatric Hematology and Bone Marrow Transplantation Alberta Children's Hospital Calgary AB Canada

The University of Texas Health Science Center Department of Pediatric Hematology Oncology San Antonio TX

University of Melbourne Melbourne Australia

Wolfson Childhood Cancer Research Centre Newcastle University Centre for Cancer Newcastle upon Tyne United Kingdom

Citace poskytuje Crossref.org

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