期刊
PLOS ONE
卷 14, 期 8, 页码 -出版社
PUBLIC LIBRARY SCIENCE
DOI: 10.1371/journal.pone.0221373
关键词
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资金
- Princess Maxima Center for Pediatric Oncology, Foundation KiKa (Children Cancerfree) [278]
- Great Ormond Street Hospital Children's Charity [W1090]
- Cancer Research UK [C1188/A8687]
- UK National Cancer Research Network and Children's Cancer and Leukaemia Group
- Societe Francaise des Cancers de l'Enfant
- Association Leon Berard Enfant Cancereux
- Gesellschaft fur Padiatrische Onkologie und Hamatologie and Deutsche Krebshilfe, Grupo Cooperativo Brasileiro para o Tratamento do Tumour de Wilms'
- Sociedade Brasileira de Oncologia Pediatrica
- Spanish Society of Paediatric Haematology
- Spanish Association Against Cancer
- National Institute of Health Research (NIHR) Biomedical Research Centre at Great Ormond Street Hospital
Background To enhance risk stratification for Wilms tumour (WT) in a pre-operative chemotherapy setting, we explored the prognostic significance and optimal age cutoffs in patients treated according to International Society of Paediatric Oncology Renal Tumour Study Group (SIOP-RTSG) protocols. Methods Patients(6 months-18 years) with unilateral WT were selected from prospective STOP 93-01 and 2001 studies(1993-2016). Martingale residual analysis was used to explore optimal age cutoffs. Outcome according to age was analyzed by uni- and multivariable analysis, adjusted for sex, biopsy(yes/no), stage, histology and tumour volume at surgery. Results 5631 patients were included; median age was 3.4 years(IQR: 2-5.1). Estimated 5-year event-free survival (EFS) and overall survival (OS) were 85%(95%CI 83.5-85.5) and 93% (95%CI 92.0-93.4). Martingale residual plots detected no optimal age cutoffs. Multivariable analysis showed lower EFS with increasing age(linear trend P<0.001). Using previously described age categories, EFS was lower for patients aged 2-4(HR 1.34, P = 0.02), 4-10 (HR 1.83, P<0.0001) and 10-18 years(HR 1.74, P = 0.01) as compared to patients aged 6 months-2 years. OS was lower for patients 4-10 years(HR 1.67, P = 0.01) and 10-18 years (HR 1.87, P = 0.04), but not for 2-4 years(HR 1.29, P = 0.23). Higher stage, histological risk group and tumour volume were independent adverse prognostic factors. Conclusion Although optimal age cutoffs could not be identified, we demonstrated the prognostic significance of age as well as previously described cutoffs for EFS (2 and 4 years) and OS (4 years) in children with WT treated with pre-operative chemotherapy. These findings encourage the consideration of age in the design of future SIOP-RTSG protocols.
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