|
|||||
|
|
Changes over three decades in outcome and the prognostic influence of age-at-diagnosis in young patients with neuroblastoma: a report from the International Neuroblastoma Risk Group Project |
|
| Authors: | Moroz Veronica Machin David Faldum Andreas Hero Barbara Iehara Tomoko Mosseri Veronique Ladenstein Ruth De Bernardi Bruno Rubie Hervé Berthold Frank Matthay Katherine K Monclair Tom Ambros Peter F Pearson Andrew D J Cohn Susan L London Wendy B |
| Affiliation: | a Children’s Cancer and Leukaemia Group Data Centre, University of Leicester, Leicester, UK b Institute of Medical Biostatistics, Epidemiology and Informatics, University of Mainz, Germany c Department of Pediatric Oncology and Hematology, Children’s Hospital, University of Cologne, Germany d Department of Pediatrics, Kyoto Prefectural University of Medicine, Kyoto, Japan e Service de Biostatistiques, Institut Curie, Paris, France f St. Anna Children’s Hospital, Kinderspitalgasse 6, 1090 Vienna, Austria g Department of Hematology-Oncology, Giannina Gaslini Children’s Hospital, Genova, Italy h Department of Hematology-Oncology, Hôpital des Enfants, Toulouse, France i Department of Pediatrics, and the University of California School of Medicine, San Francisco, CA, USA j Section for Paediatric Surgery, Division of Surgery, Rikshospitalet University Hospital, Oslo, Norway k Children’s Cancer Research Institute, St. Anna Kinderspital, Vienna, Austria l Section of Paediatrics, Institute of Cancer Research and Royal Marsden Hospital, Surrey, UK m Department of Pediatrics, The University of Chicago, Chicago, IL, USA n Children’s Oncology Group Statistics and Data Center, Dana Faber Children’s Hospital Cancer Center, Harvard Medical School, Boston, MA, USA |
| Abstract: | PurposeIncreasing age has been an adverse risk factor in children with neuroblastoma (NB) since the 1970’s, with a 12-month age-at-diagnosis cut-off for treatment stratification. Over the last 30 years, treatment intensity for children >12 months with advanced-stage disease has increased; to investigate if this strategy has improved outcome and/or reduced the prognostic influence of age, we analysed the International Neuroblastoma Risk Group (INRG) database.Patients and methodsData from 11,037 children with NB (1974-2002) from Australia, Europe, Japan, North America. Cox modelling of event-free survival (EFS) tested if the era and prognostic significance of age-of-diagnosis, adjusted for bone marrow (BM) metastases and MYCN status, effects on outcome had changed.ResultsOutcome improved over time: 3-year EFS 46% (1974-1989) and 71% (1997-2002). The risk for those >18 months against ?12 decreased: hazard ratio (HR); 4.61 and 3.94. For age 13-18 months, EFS increased from 42% to 77%. Outcome was worse if: >18 months (HR 4.47); BM metastases (HR 4.00); and MYCN amplified (HR 3.97). For 1997-2002, the EFS for >18 months with BM involvement and MYCN amplification was 18%, but 89% for 0-12 months with neither BM involvement nor MYCN amplification.ConclusionsThere is clear evidence for improving outcomes for children with NB over calendar time. The adverse influence of increasing age-at-diagnosis has declined but it remains a powerful indicator of unfavourable prognosis. These results support the age-of-diagnosis cut-off of greater than 18 months as a risk criterion in the INRG classification system. |
| Keywords: | Neuroblastoma Outcome Prognosis Age-at-diagnosis INRG |
| 本文献已被 ScienceDirect PubMed 等数据库收录！ | |