Outcomes of Infants and Young Children With Relapsed Medulloblastoma After Initial Craniospinal Irradiation-Sparing Approaches: An International Cohort Study
Jazyk angličtina Země Spojené státy americké Médium print-electronic
Typ dokumentu časopisecké články, práce podpořená grantem
Grantová podpora
DRCRPG-NOV22/100002
Cancer Research UK - United Kingdom
PubMed
36548930
DOI
10.1200/jco.21.02968
Knihovny.cz E-zdroje
- MeSH
- chronická nemoc MeSH
- dítě MeSH
- kohortové studie MeSH
- kojenec MeSH
- kraniospinální iradiace * škodlivé účinky MeSH
- lidé MeSH
- lokální recidiva nádoru MeSH
- meduloblastom * radioterapie MeSH
- nádory mozečku * radioterapie MeSH
- nádory mozku * terapie MeSH
- předškolní dítě MeSH
- prospektivní studie MeSH
- proteiny hedgehog MeSH
- Check Tag
- dítě MeSH
- kojenec MeSH
- lidé MeSH
- předškolní dítě MeSH
- Publikační typ
- časopisecké články MeSH
- práce podpořená grantem MeSH
- Názvy látek
- proteiny hedgehog MeSH
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.
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 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 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
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 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
Hospital of Pediatrics SAMIC Prof Dr Juan P Garrahan Buenos Aires Argentina
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
Research Methods Unit Nova Scotia Health Authority Halifax NS Canada
Citace poskytuje Crossref.org
Salvage therapies for first relapse of SHH medulloblastoma in early childhood
