Artikel
Incidence patterns, survival probabilities, and second primary neoplasms after retinoblastoma among children in Germany, 1981–2018: A nationwide assessment based on data from the German Childhood Cancer Registry
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Autoren
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Desiree Grabow
- Abteilung Epidemiologie von Krebs im Kindesalter/Deutsches Kinderkrebsregister, Institut für Medizinische Biometrie, Epidemiologie und Informatik (IMBEI), Universitätsmedizin der Johannes Gutenberg-Universität Mainz, Mainz, Germany
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Meike Ressing
- Abteilung Epidemiologie von Krebs im Kindesalter/Deutsches Kinderkrebsregister, Institut für Medizinische Biometrie, Epidemiologie und Informatik (IMBEI), Universitätsmedizin der Johannes Gutenberg-Universität Mainz, Mainz, Germany
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Claudia Spix
- Abteilung Epidemiologie von Krebs im Kindesalter/Deutsches Kinderkrebsregister, Institut für Medizinische Biometrie, Epidemiologie und Informatik (IMBEI), Universitätsmedizin der Johannes Gutenberg-Universität Mainz, Mainz, Germany
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Irene Jung
- Abteilung Epidemiologie von Krebs im Kindesalter/Deutsches Kinderkrebsregister, Institut für Medizinische Biometrie, Epidemiologie und Informatik (IMBEI), Universitätsmedizin der Johannes Gutenberg-Universität Mainz, Mainz, Germany
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Claudia Trübenbach
- Abteilung Epidemiologie von Krebs im Kindesalter/Deutsches Kinderkrebsregister, Institut für Medizinische Biometrie, Epidemiologie und Informatik (IMBEI), Universitätsmedizin der Johannes Gutenberg-Universität Mainz, Mainz, Germany
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Maike Wellbrock
- Abteilung Epidemiologie von Krebs im Kindesalter/Forschungsgruppe: Ätiologie und Ungleichheiten bei Kinderkrebs, Institut für Medizinische Biometrie, Epidemiologie und Informatik (IMBEI), Universitätsmedizin der Johannes Gutenberg-Universität Mainz, Mainz, Germany
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Petra Ketteler
- Klinik für Kinderheilkunde III, Universitätsklinikum Essen, Universität Duisburg-Essen, Essen, Germany
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Angelika Eggert
- Klinik für Pädiatrie mit Schwerpunkt Onkologie und Hämatologie, Charité, Universitätsmedizin Berlin, Berlin, Germany
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Dirk Reinhardt
- Klinik für Kinderheilkunde III, Universitätsklinikum Essen, Universität Duisburg-Essen, Essen, Germany
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Dietmar Lohmann
- Klinische Forschergruppe Ophthalmologische Onkologie und Genetik, Institut für Humangenetik, Universität Duisburg-Essen, Essen, Germany
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Nikolaos Bechrakis
- Klinik für Augenheilkunde, Universitätsklinikum Essen, Universität Duisburg-Essen, Essen, Germany
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Eva Biewald
- Klinik für Augenheilkunde, Universitätsklinikum Essen, Universität Duisburg-Essen, Essen, Germany
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Cécile M. Ronckers
- Abteilung Epidemiologie von Krebs im Kindesalter/Deutsches Kinderkrebsregister, Institut für Medizinische Biometrie, Epidemiologie und Informatik (IMBEI), Universitätsmedizin der Johannes Gutenberg-Universität Mainz, Mainz, Germany
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Friederike Erdmann
- Abteilung Epidemiologie von Krebs im Kindesalter/Forschungsgruppe: Ätiologie und Ungleichheiten bei Kinderkrebs, Institut für Medizinische Biometrie, Epidemiologie und Informatik (IMBEI), Universitätsmedizin der Johannes Gutenberg-Universität Mainz, Mainz, Germany
| Veröffentlicht: | 6. September 2024 |
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Gliederung
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Introduction: Retinoblastoma (RB) is a rare malignant tumour of the retina, occurring primarily in early childhood. We sought to provide the first comprehensive description and interpretation of incidence patterns, survival probabilities and incidence of second primary neoplasms after a retinoblastoma among children in Germany, using the nationwide population-based data of the German Childhood Cancer Registry (GCCR).
Methods: We identified all incident RB cases diagnosed before the age of 15 years between 1981 and 2018 from the GCCR. For survival analyses, only children with a follow-up of at least five years (as of 30 November 2021) were included. We evaluated incidence patterns by calculating age-specific, age-standardized (ASR), and cumulative incidence rates. For time-trend analysis, we estimated average annual percentage changes (AAPC, by applying Poisson regression models).
We estimated 5-, 10-, and 15-year overall survival probabilities (by decade), using the life table method extension by Brenner and Spix. Moreover, we calculated frequencies of second primary neoplasms.
Results: During the 38-year study period, 1,521 childhood RB cases were reported to the GCCR, resulting in an average ASR of 4.3/million (95% confidence interval (CI) 4.1; 4.6). While incidence rates were similar between boys (ASR 4.4/million (95% CI 4.1; 4.7) and girls (4.3/million (95% CI 4.0; 4.6), we observed considerable differences by age. Approximately 43% of all RB cases occurred within the first year of life (24.2/million; 95% CI 22.3; 26.1) and more than 95% within the first five years of life. The ASR was markedly higher for unilateral RB (2.6/million (95% CI 2.5; 2.8) compared to bilateral RB (1.7/million (95% CI 1.5; 1.8)).
The ASR of RB remained rather stable over time during the period 1981-2010, followed by a pronounced increase in the ASR in recent years (8.6% on average per annum during 2011-2018 (95% CI 3.8; 13.6)). The 5-, 10- and 15-year survival probabilities exceeded 95% since the 1990ies. In all, 50 RB survivors developed a second primary neoplasm, four of them developed further primary neoplasms. The cumulative incidence of SPNs was 6.9% (95% CI 4.8; 9.1), with a higher risk observed after bilateral retinoblastoma (a cumulative incidence of 13.8% (95% CI 9.2; 18.4)).
Conclusions: Observed incidence patterns, survival probabilities and diagnoses of SPNs after RB in Germany were comparable to those of other European countries. Explanations for the recent increase in incidence rates remain speculative. It coincides with the establishment of the multicentre clinical RB database in Essen in 2012. Further monitoring and temporal as well as regional analyses by laterality may contribute to a better understanding of the recent incidence trend.
The authors declare that they have no competing interests.
The authors declare that an ethics committee vote is not required.
