M L Tas1, A M J Reedijk2, H E Karim-Kos3, L C M Kremer4, C P van de Ven2, M P Dierselhuis2, N K A van Eijkelenburg2, M van Grotel2, K C J M Kraal2, A M L Peek2, J W W Coebergh5, G O R Janssens6, B de Keizer7, R R de Krijger8, R Pieters2, G A M Tytgat9, M M van Noesel10. 1. Princess Máxima Center for Pediatric Oncology, Utrecht, the Netherlands. Electronic address: m.tas@prinsesmaximacentrum.nl. 2. Princess Máxima Center for Pediatric Oncology, Utrecht, the Netherlands. 3. Department of Research, Netherlands Comprehensive Cancer Organisation (IKNL), Utrecht, the Netherlands; Department of Public Health, Erasmus Medical Center, Rotterdam, the Netherlands. 4. Princess Máxima Center for Pediatric Oncology, Utrecht, the Netherlands; Department of Pediatric Oncology, Amsterdam UMC, University of Amsterdam, Amsterdam, the Netherlands. 5. Department of Public Health, Erasmus Medical Center, Rotterdam, the Netherlands. 6. Princess Máxima Center for Pediatric Oncology, Utrecht, the Netherlands; Department of Radiation Oncology, University Medical Center Utrecht, Utrecht, the Netherlands. 7. Princess Máxima Center for Pediatric Oncology, Utrecht, the Netherlands; Department of Radiology and Nuclear Medicine, University Medical Center Utrecht, Utrecht, the Netherlands. 8. Princess Máxima Center for Pediatric Oncology, Utrecht, the Netherlands; Department of Pathology, University Medical Center Utrecht, Utrecht, the Netherlands. 9. Princess Máxima Center for Pediatric Oncology, Utrecht, the Netherlands; Department of Pediatric Oncology, Amsterdam UMC, University of Amsterdam, Amsterdam, the Netherlands; Dutch Childhood Oncology Group, Utrecht, the Netherlands. 10. Princess Máxima Center for Pediatric Oncology, Utrecht, the Netherlands; Dutch Childhood Oncology Group, Utrecht, the Netherlands.
Abstract
PURPOSE: Long-term trends in neuroblastoma incidence and survival in unscreened populations are unknown. We explored trends in incidence, stage at diagnosis, treatment and survival of neuroblastoma in the Netherlands from 1990 to 2014. METHODS: The Netherlands Cancer Registry provided data on all patients aged <18 years diagnosed with a neuroblastoma. Trends in incidence and stage were evaluated by calculating the average annual percentage change (AAPC). Univariate and multivariable survival analyses were performed for stage 4 disease to test whether changes in treatment are associated with survival. RESULTS: Of the 593 newly diagnosed neuroblastoma cases, 45% was <18 months of age at diagnosis and 52% had stage 4 disease. The age-standardized incidence rate for stage 4 disease increased at all ages from 3.2 to 5.3 per million children per year (AAPC + 2.9%, p < .01). This increase was solely for patients ≥18 months old (3.0-5.4; AAPC +3.3%, p = .01). Five-year OS of all patients increased from 44 ± 5% to 61 ± 4% from 1990 to 2014 (p < .01) and from 19 ± 6% to 44 ± 6% (p < .01) for patients with stage 4 disease. Multivariable analysis revealed that high-dose chemotherapy followed by autologous stem cell rescue and anti-GD2-based immunotherapy were associated with this survival increase (HR 0.46, p < .01 and HR 0.37, p < .01, respectively). CONCLUSION: Incidence of stage 4 neuroblastoma increased exclusively in patients aged ≥18 months since 1990, whereas the incidence of other stages remained stable. The 5-year OS of stage 4 patients improved, mostly due to the introduction of high-dose chemotherapy followed by stem cell rescue and immunotherapy.
PURPOSE: Long-term trends in neuroblastoma incidence and survival in unscreened populations are unknown. We explored trends in incidence, stage at diagnosis, treatment and survival of neuroblastoma in the Netherlands from 1990 to 2014. METHODS: The Netherlands Cancer Registry provided data on all patients aged <18 years diagnosed with a neuroblastoma. Trends in incidence and stage were evaluated by calculating the average annual percentage change (AAPC). Univariate and multivariable survival analyses were performed for stage 4 disease to test whether changes in treatment are associated with survival. RESULTS: Of the 593 newly diagnosed neuroblastoma cases, 45% was <18 months of age at diagnosis and 52% had stage 4 disease. The age-standardized incidence rate for stage 4 disease increased at all ages from 3.2 to 5.3 per million children per year (AAPC + 2.9%, p < .01). This increase was solely for patients ≥18 months old (3.0-5.4; AAPC +3.3%, p = .01). Five-year OS of all patients increased from 44 ± 5% to 61 ± 4% from 1990 to 2014 (p < .01) and from 19 ± 6% to 44 ± 6% (p < .01) for patients with stage 4 disease. Multivariable analysis revealed that high-dose chemotherapy followed by autologous stem cell rescue and anti-GD2-based immunotherapy were associated with this survival increase (HR 0.46, p < .01 and HR 0.37, p < .01, respectively). CONCLUSION: Incidence of stage 4 neuroblastoma increased exclusively in patients aged ≥18 months since 1990, whereas the incidence of other stages remained stable. The 5-year OS of stage 4 patients improved, mostly due to the introduction of high-dose chemotherapy followed by stem cell rescue and immunotherapy.
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