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Survival and Health Care Burden of Children With Retinoblastoma in Europe

2024; American Medical Association; Volume: 142; Issue: 11 Linguagem: Inglês

10.1001/jamaophthalmol.2024.4140

2168-6173

Gianni Virgili, Riccardo Capocaccia, Laura Botta, Damien Bennett, Theodora Hadjistilianou, Kaire Innos, Henrike E. Karim‐Kos, Claudia E. Kuehni, Ursula Kühnel, Cinzia Mazzini, Adela Cañete, Keiu Paapsi, Mariacristina Parravano, Cécile M. Ronckers, Silvia Rossi, Charles Stiller, Giulio Vicini, Otto Visser, Gemma Gatta, M. Hackl, Elisabeth van Eycken, Nancy Van Damme, Z. Valerianova, Mario Šekerija, Ioanna Gregoriu, Anna Demetriou, Ladislav Dušek, Denisa Krejici, Hans H. Storm, Margit Mägi, Janne Pitkäniemi, Jacqueline Clavel, Claire Poulalhon, Brigitte Lacour, Emmanuel Désandes, Valérie Jooste, Alain Monnereau, Meike Ressing, Alexander Katalinic, Eleni Petridou, Georgios Markozannes, Miklós Garami, Zsusanna Jakab, Helgi Birgisson, Deirdre M. Murray, Guido Mazzoleni, Fabio Vittadello, Francesco Cuccaro, Rocco Galasso, Giuseppe Sampietro, Stefano Rosso, Cinzia Gasparotti, Giovanni Maifredi, Margherita Ferrante, Antonina Torrisi, Antonella Sutera Sardo, Maria Letizia Gambino, Monica Lanzoni, Paola Ballotari, Erica Giacomazzi, Stefano Ferretti, Adele Caldarella, Gianfranco Manneschi, Milena Sant, Paolo Baili, Franco Berrino, Annalisa Trama, Roberto Lillini, Alice Bernasconi, S. Bonfarnuzzo, Claudia Vener, Fabio Didonè, Paolo Lasalvia, Lucia Buratti, Giovanna Tagliabue, Diego Serraino, Martina Taborelli, Roberta De Angelis, Elena Demuru, Francesco Cerza, Fabrizio Di Mari, Corrado Di Benedetto, Mariano Santaquilani, Serenella Venanzi, Marco Tallon, Luca Boni, Silvia Iacovacci, Antonio Russo, Federico Gervasi, Gianbattista Spagnoli, Luca Cavalieri d’Oro, Mario Fusco, Maria Francesca Vitale, P. Pinna, Walter Mazzucco, Maria Michiara, Giorgio Chiranda, Caelotta Sacerdote, Milena Maule, Giorgio Cascone, Eugenia Spata, Lucìa Mangone, Fabio Falcini, Rossella Cavallo, Daniela Piras, Ylenia Y. Dinaro, Marine Castaing, Anna Clara Fanetti, Sante Minerba, Giuseppina Candela, T. Scuderi, William Mantovani, Maria Gentilini, Fabirizio Stracci, Manuel Zorzi, Nicola Ferrarini, Eliana Liepina, Giedrė Smailytė, Mirima Azzopardi, Neville Calleja, Tom Børge Johannesen, Joanna Didkowska, Urszula Wojciechowska, Magdalena Bielska‐Lasota, Ana Pais, Maria Josè Bento, Rita Calisto, Antonio Lourenço, Alexandra Mayer, Chakameh Safaei Diba, Vesna Zadnik, Tina Žagar, Arantza Lopez de Munain, Marta De-La-Cruz, Montserrat Garrido, Ana Vizcaíno, Fernando Almela, Rafael Marcos‐Gragera, Arantza Sanvisens, Maria Josè Sanchez, Daniel Redondo‐Sánchez, María Dolores Chirlaque López, Antonia Sánchez-Gil, Marcela Guevara, Eva Ardanáz, Rafael Peris‐Bonet, Jaume Galceran, M. Carulla, Saarah Stevens, Anna Gavin, David Morrison, Dyfed Huws, Stephanie Smits,

Hematopoietic Stem Cell Transplantation

Importance Studies on the epidemiology of retinoblastoma (RB) could lead to improvement in management. Objective To estimate the incidence and survival of RB in European children and the occurrence of second primary tumors (other than RB) in these patients. Design, Setting, and Participants This cohort study used population-based data from 81 cancer registries in 31 European countries adhering to the European Cancer Registries (EUROCARE-6) project. Data collection took place between January 2000 and December 2013. European children aged 0 to 14 years diagnosed with RB were included. Data were analyzed from May to November 2023. Exposures Diagnosis of RB with International Classification of Diseases for Oncology , Third Edition ( ICD-O-3 ), morphology coded 9510-9514 (retinoblastoma) and malignant behavior (fifth digit of morphology code, 3). Main Outcome and Measures Annual incidence (per million children aged 0-14 years), 5-year survival (%), and the standardized incidence ratio (SIR) of subsequent malignant neoplasms. Results The study included 3262 patients (mean [SD] age, 1.27 [1.63] years; 1706 [52%] male and 1556 [48%] female) from 81 registries. Of these, 3098 patients were considered in trend analysis after excluding registries with incomplete time coverage: 940 in 2000 to 2003, 703 in 2004 to 2006, 744 in 2007 to 2009, and 856 in 2010 to 2013. The estimated overall European incidence rate was 4.0 (95% CI, 3.9-4.1). Rates among countries varied from less than 2 million to greater than 6 million per year. No time trend of incidence was observed in any area. The overall European 5-year survival was 97.8% (95% CI, 95.5-98.9; 3180 cases). Five-year survival was lower in Estonia and Bulgaria (<80%) and 100% in several countries. Twenty-five subsequent malignant neoplasms were recorded during follow-up (up to 14 years), with an SIR of 8.2 and with cases occurring at mean ages between 1.3 and 8.9 years across different sites. An increased risk was found for hematological tumors (SIR, 5) and bone and soft tissue sarcomas (SIR, 29). Conclusions and Relevance This study showed RB incidence remained stable at 4.0 per 1 000 000 European children aged 0 to 14 years from 2000 to 2013, but estimates varied among countries and differences in survival across countries persist. These data might be used to monitor RB management and occurrences of second tumors. The findings suggest future registry studies should aim to collect standardized RB stage at diagnosis and treatment to interpret disparities and potentially improve surveillance.