Paraskevi Panagopoulou1, Marios K Georgakis1, Margarita Baka2, Maria Moschovi3, Vassilios Papadakis4, Sophia Polychronopoulou4, Maria Kourti5, Emmanuel Hatzipantelis6, Eftichia Stiakaki7, Helen Dana8, Athanasios Tragiannidis1, Evdoxia Bouka1, Luis Antunes9, Joana Bastos10, Daniela Coza11, Anna Demetriou12, Domenic Agius13, Sultan Eser14, Raluca Gheorghiu15, Mario Šekerija16, Maciej Trojanowski17, Tina Žagar18, Anna Zborovskaya19, Anton Ryzhov20, Nick Dessypris1, Daniel Morgenstern21, Eleni Th Petridou22. 1. Department of Hygiene, Epidemiology and Medical Statistics Medical School, National and Kapodistrian University of Athens, Athens Greece. 2. Department of Pediatric Hematology-Oncology, "Pan. & Agl. Kyriakou" Children's Hospital, Athens, Greece. 3. Pediatric Hematology/Oncology Unit, First Department of Pediatrics, University of Athens, "Agia Sofia" Children's Hospital, Athens, Greece. 4. Department of Pediatric Hematology-Oncology, "Agia Sofia" Children's Hospital, Athens, Greece. 5. Department of Pediatric Hematology and Oncology, Hippokration Hospital, Thessaloniki, Greece. 6. Hematology-Oncology Unit, 2nd Pediatric Department, Aristotle University of Thessaloniki, AHEPA Hospital, Thessaloniki, Greece. 7. Department of Pediatric Hematology-Oncology, University of Crete, Heraklion, Greece. 8. Pediatric Hematology-Oncology Department, "Mitera" Children's Hospital, Athens, Greece. 9. North Region Cancer Registry of Portugal (RORENO), Portuguese Institute of Oncology, Porto, Portugal. 10. Registo Oncológico Regional do Centro (ROR-Centro), Portuguese Institute of Oncology, Coimbra, Portugal. 11. The Oncology Institute "Prof. Dr. Ion Chiricuţă", Cluj-Napoca, Romania. 12. Health Monitoring Unit, Ministry of Health, Nicosia, Cyprus. 13. Malta National Cancer Registry, Department for Policy in Health - Health Information and Research, Pieta, Malta. 14. Izmir Cancer Registry, Izmir Hub, Izmir and Hacettepe, University Institute of Public Health, Ankara, Turkey. 15. Regional Cancer Registry, National Institute of Public Health, Iasi, Romania. 16. Croatian Institute of Public Health, Croatian National Cancer Registry, Zagreb, Croatia; Andrija Štampar School of Public Health, School of Medicine, University of Zagreb, Croatia. 17. Greater Poland Cancer Registry, Greater Poland Cancer Center, Poznań, Poland. 18. Cancer Registry of Slovenia, Institute of Oncology Ljubljana, Ljubljana, Slovenia. 19. Belarusian Research Center for Paediatric Oncology, Haematology and Immunology, Childhood Cancer Subregistry of Belarus, Minsk, Belarus. 20. National Cancer Registry of Ukraine, National Institute of Cancer, Kiev, Ukraine. 21. Division of Haematology/Oncology, The Hospital for Sick Children, Toronto, Ontario, M5G 1X8, Canada. 22. Department of Hygiene, Epidemiology and Medical Statistics Medical School, National and Kapodistrian University of Athens, Athens Greece; Clinical Epidemiology Unit, Department of Medicine, Karolinska Institute, Stockholm, Sweden. Electronic address: epetrid@med.uoa.gr.
Abstract
AIM: Neuroblastoma outcomes vary with disease characteristics, healthcare delivery and socio-economic indicators. We assessed survival patterns and prognostic factors for patients with neuroblastoma in 11 Southern and Eastern European (SEE) countries versus those in the US, including-for the first time-the Nationwide Registry for Childhood Hematological Malignancies and Solid Tumours (NARECHEM-ST)/Greece. METHODS: Overall survival (OS) was calculated in 13 collaborating SEE childhood cancer registries (1829 cases, ∼1990-2016) and Surveillance, Epidemiology, and End Results (SEER), US (3072 cases, 1990-2012); Kaplan-Meier curves were used along with multivariable Cox regression models assessing the effect of age, gender, primary tumour site, histology, Human Development Index (HDI) and place of residence (urban/rural) on survival. RESULTS: The 5-year OS rates varied widely among the SEE countries (Ukraine: 45%, Poland: 81%) with the overall SEE rate (59%) being significantly lower than in SEER (77%; p < 0.001). In the common registration period within SEE (2000-2008), no temporal trend was noted as opposed to a significant increase in SEER. Age >12 months (hazard ratio [HR]: 2.8-4.7 in subsequent age groups), male gender (HR: 1.1), residence in rural areas (HR: 1.3), living in high (HR: 2.2) or medium (HR: 2.4) HDI countries and specific primary tumour location were associated with worse outcome; conversely, ganglioneuroblastoma subtype (HR: 0.28) was associated with higher survival rate. CONCLUSIONS: Allowing for the disease profile, children with neuroblastoma in SEE, especially those in rural areas and lower HDI countries, fare worse than patients in the US, mainly during the early years after diagnosis; this may be attributed to presumably modifiable socio-economic and healthcare system performance differentials warranting further research.
AIM: Neuroblastoma outcomes vary with disease characteristics, healthcare delivery and socio-economic indicators. We assessed survival patterns and prognostic factors for patients with neuroblastoma in 11 Southern and Eastern European (SEE) countries versus those in the US, including-for the first time-the Nationwide Registry for Childhood Hematological Malignancies and Solid Tumours (NARECHEM-ST)/Greece. METHODS: Overall survival (OS) was calculated in 13 collaborating SEE childhood cancer registries (1829 cases, ∼1990-2016) and Surveillance, Epidemiology, and End Results (SEER), US (3072 cases, 1990-2012); Kaplan-Meier curves were used along with multivariable Cox regression models assessing the effect of age, gender, primary tumour site, histology, Human Development Index (HDI) and place of residence (urban/rural) on survival. RESULTS: The 5-year OS rates varied widely among the SEE countries (Ukraine: 45%, Poland: 81%) with the overall SEE rate (59%) being significantly lower than in SEER (77%; p < 0.001). In the common registration period within SEE (2000-2008), no temporal trend was noted as opposed to a significant increase in SEER. Age >12 months (hazard ratio [HR]: 2.8-4.7 in subsequent age groups), male gender (HR: 1.1), residence in rural areas (HR: 1.3), living in high (HR: 2.2) or medium (HR: 2.4) HDI countries and specific primary tumour location were associated with worse outcome; conversely, ganglioneuroblastoma subtype (HR: 0.28) was associated with higher survival rate. CONCLUSIONS: Allowing for the disease profile, children with neuroblastoma in SEE, especially those in rural areas and lower HDI countries, fare worse than patients in the US, mainly during the early years after diagnosis; this may be attributed to presumably modifiable socio-economic and healthcare system performance differentials warranting further research.