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      Reporting Incidences of Neuroblastoma in Various Resource Settings

      research-article
      , MBChB, MMed, PhD 1 , 2 , , MSc 3 , , MSc 4 , 5 , , MBChB 4 , 5 , , MBChB, MMed 3 , 6 , , PhD, MPhil 1
      JCO Global Oncology
      Wolters Kluwer Health

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          PURPOSE

          The incidences of neuroblastoma (NB) differ significantly between various resource settings because of varying quality of cancer registries and underdiagnoses. This study aimed to evaluate current regional variations as reported by international cancer registries and the theoretical and reported differences in international NB incidences and to evaluate South Africa (SA) as a case for variable reporting.

          METHODS

          A comprehensive literature review on registries reporting on NB was performed to construct incidence tables. The SEER Program incidence of 10.5/million children was used to calculate the expected number of NB cases for each country. Registry data of NB cases between 2000 and 2016 were requested from The South African National Cancer registry (SA-NCR) and the South African Children's Tumour Registry (SACTR) for comparison and to perform a probabilistic linkage study.

          RESULTS

          Internationally, incidences varied between –97.1% and +80% compared with the SEER program. SA under-reported NB cases by an estimated 74.2%. Between 2000 and 2016, the SA-NCR reported between 23 and 51 cases/year, whereas the SACTR reported between 18 and 57 cases/year for the same period. The incidence reported by the SA-NCR varied between 1.5 and 2.8/million children under 15-year per year, whereas the SACTR reported 1.74-2.6 cases/million children. Both registries reported incidences less than high-income country. A probabilistic record linkage of the two registries resulted in a combined incidence of 2.9 cases/million children.

          CONCLUSION

          As with most low- and middle-income countries, SA has either a lower incidence or underdiagnoses of NB cases. The reasons for under-reporting are not clear, but can be due to undiagnosed NB cases with spontaneous regression, missed possible cases because of lack of autopsies, and diagnosed cases not recorded in registries.

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          Most cited references29

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          International incidence of childhood cancer, 2001–10: a population-based registry study

          Summary Background Cancer is a major cause of death in children worldwide, and the recorded incidence tends to increase with time. Internationally comparable data on childhood cancer incidence in the past two decades are scarce. This study aimed to provide internationally comparable local data on the incidence of childhood cancer to promote research of causes and implementation of childhood cancer control. Methods This population-based registry study, devised by the International Agency for Research on Cancer in collaboration with the International Association of Cancer Registries, collected data on all malignancies and non-malignant neoplasms of the CNS diagnosed before age 20 years in populations covered by high-quality cancer registries with complete data for 2001–10. Incidence rates per million person-years for the 0–14 years and 0–19 years age groups were age-adjusted using the world standard population to provide age-standardised incidence rates (WSRs), using the age-specific incidence rates (ASR) for individual age groups (0–4 years, 5–9 years, 10–14 years, and 15–19 years). All rates were reported for 19 geographical areas or ethnicities by sex, age group, and cancer type. The regional WSRs for children aged 0–14 years were compared with comparable data obtained in the 1980s. Findings Of 532 invited cancer registries, 153 registries from 62 countries, departments, and territories met quality standards, and contributed data for the entire decade of 2001–10. 385 509 incident cases in children aged 0–19 years occurring in 2·64 billion person-years were included. The overall WSR was 140·6 per million person-years in children aged 0–14 years (based on 284 649 cases), and the most common cancers were leukaemia (WSR 46·4), followed by CNS tumours (WSR 28·2), and lymphomas (WSR 15·2). In children aged 15–19 years (based on 100 860 cases), the ASR was 185·3 per million person-years, the most common being lymphomas (ASR 41·8) and the group of epithelial tumours and melanoma (ASR 39·5). Incidence varied considerably between and within the described regions, and by cancer type, sex, age, and racial and ethnic group. Since the 1980s, the global WSR of registered cancers in children aged 0–14 years has increased from 124·0 (95% CI 123·3–124·7) to 140·6 (140·1–141·1) per million person-years. Interpretation This unique global source of childhood cancer incidence will be used for aetiological research and to inform public health policy, potentially contributing towards attaining several targets of the Sustainable Development Goals. The observed geographical, racial and ethnic, age, sex, and temporal variations require constant monitoring and research. Funding International Agency for Research on Cancer and the Union for International Cancer Control.
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            Overview and recent advances in the treatment of neuroblastoma.

            Children with neuroblastoma have widely divergent outcomes, ranging from cure in >90% of patients with low risk disease to <50% for those with high risk disease. Recent research has shed light on the biology of neuroblastoma, allowing for more accurate risk stratification and treatment reduction in many cases, although newer treatment strategies for children with high-risk and relapsed neuroblastoma are needed to improve outcomes. Areas covered: Neuroblastoma epidemiology, diagnosis, risk stratification, and recent advances in treatment of both newly diagnosed and relapsed neuroblastoma. Expert commentary: The identification of newer tumor targets and of novel cell-mediated immunotherapy agents may lead to novel therapeutic approaches, and clinical trials for regimens designed to target individual genetic aberrations in tumors are underway. A combination of therapeutic modalities will likely be required to improve survival and cure rates for patients with high-risk neuroblastoma.
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              Estimating the total incidence of global childhood cancer: a simulation-based analysis

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                Author and article information

                Journal
                JCO Glob Oncol
                JCO Glob Oncol
                go
                go
                GO
                JCO Global Oncology
                Wolters Kluwer Health
                2687-8941
                2021
                17 June 2021
                : 7
                : GO.21.00054
                Affiliations
                [ 1 ]Department of Paediatrics and Child Health, Faculty of Medicine and Health Sciences, Stellenbosch University, Cape Town, South Africa
                [ 2 ]Paediatric Haematology and Oncology, Department of Paediatrics Antwerp University Hospital, Antwerp, Belgium
                [ 3 ]National Cancer Registry, National Health Laboratory Service, Johannesburg, South Africa
                [ 4 ]Paediatric Haematology and Oncology, Department of Paediatrics University of Pretoria, Steve Biko Academic Hospital, Pretoria, South Africa
                [ 5 ]South African Children's Tumour Registry, Pretoria, South Africa
                [ 6 ]School of Public Health, University of the Witwatersrand, Johannesburg, South Africa
                Author notes
                Jaques van Heerden, MBChB, MMed (Paeds), PhD, Department of Paediatric Haematology and Oncology, Antwerp University Hospital, Drie Eiken St 655, Edegem 2650, Belgium; e-mail: Jaques.vanheerden@ 123456uza.be .
                Author information
                https://orcid.org/0000-0002-4502-1169
                https://orcid.org/0000-0002-6154-3653
                https://orcid.org/0000-0002-6812-5446
                https://orcid.org/0000-0003-1259-2122
                https://orcid.org/0000-0002-6838-0180
                Article
                GO.21.00054
                10.1200/GO.21.00054
                8457875
                34138644
                2fe933a8-58bf-4b65-ae2e-659700192412
                © 2021 by American Society of Clinical Oncology

                Creative Commons Attribution Non-Commercial No Derivatives 4.0 License: https://creativecommons.org/licenses/by-nc-nd/4.0/

                History
                : 4 February 2021
                : 8 April 2021
                : 19 May 2021
                Page count
                Figures: 4, Tables: 5, Equations: 0, References: 40, Pages: 0
                Categories
                ORIGINAL REPORTS
                Pediatric Oncology

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