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The Global Retinoblastoma Outcome Study: a prospective, cluster-based analysis of 4064 patients from 149 countries

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dc.contributor.authorGlobal Retinoblastoma Study Group
dc.contributor.authorAlia, Donjeta
dc.contributor.authorTandili, Alketa
dc.contributor.authorPaiva, Luisa
dc.contributor.authorWime, Amelia
dc.contributor.authorChantada, Guillermo
dc.contributor.authorFandiño, Adriana
dc.contributor.authorSgroi, Mariana
dc.contributor.authorPapyan, Ruzanna
dc.contributor.authorTamamyan, Gevorg
dc.contributor.authorCamuglia, Jayne
dc.contributor.authorGlen, Gole
dc.contributor.authorElder, James
dc.contributor.authorMcKenzie, John
dc.contributor.authorStaffieri, Sandra
dc.contributor.authorElder, James
dc.contributor.authorJones, Michael
dc.contributor.authorManudhane, Rebecca
dc.contributor.authorSia, David
dc.contributor.authorRitter, Petra
dc.contributor.authorSchwab, Christoph
dc.contributor.authorBalayeva, Ruhengiz
dc.contributor.authorKhan, Zohora
dc.contributor.authorNuruddin, Murtuza
dc.contributor.authorRoy, Soma
dc.contributor.authorRashid, Riffat
dc.contributor.authorSultana, Sadia
dc.contributor.authorShakoor, Shawkat
dc.contributor.authorNaumenko, Larisa
dc.contributor.authorZhilyaeva, Katsiaryna
dc.contributor.authorBartoszek, Paulina
dc.contributor.authorBrichard, Bénédicte
dc.contributor.authorDe Potter, Patrick
dc.contributor.authorAlfa, Amadou
dc.contributor.authorSalas,Beatriz
dc.contributor.authorColeoni, Maria
dc.contributor.authorMbumba, Freddy
dc.contributor.authorDonato, Carla
dc.contributor.authorBonanomi, Maria
dc.contributor.authorGrigorovski, Nathalia
dc.contributor.authorOssandón Villaseca, Diego
dc.date.accessioned2024-01-05T15:03:17Z
dc.date.available2024-01-05T15:03:17Z
dc.date.issued2022
dc.descriptionGlobal Retinoblastoma Study Group: Mattosinho, Clarissa; Teixeira, Luiz; Oscar, Alexander; Veleva, Nevyana; Bouda, Gabrielle; Kabore, Rolande; Philbert, Remezo; Evina, Ted; Nkumbe, Henry; Kamsang, Pius; Muyen, Okwen; Dimaras, Helen; Mallipatna, Ashwin; Hamel, Patrick; Superstein, Rosanne; Paton, Katherine; Strahlendorf, Caron; Komba, Jess; Tyau-Tyau, Harba; Cavieres, Isabel; López, Juan; Oporto, Joaquin; et.al.
dc.description.abstractBackground: Retinoblastoma is the most common intraocular cancer worldwide. There is some evidence to suggest that major differences exist in treatment outcomes for children with retinoblastoma from different regions, but these differences have not been assessed on a global scale. We aimed to report 3-year outcomes for children with retinoblastoma globally and to investigate factors associated with survival. Methods: We did a prospective cluster-based analysis of treatment-naive patients with retinoblastoma who were diagnosed between Jan 1, 2017, and Dec 31, 2017, then treated and followed up for 3 years. Patients were recruited from 260 specialised treatment centres worldwide. Data were obtained from participating centres on primary and additional treatments, duration of follow-up, metastasis, eye globe salvage, and survival outcome. We analysed time to death and time to enucleation with Cox regression models. Findings: The cohort included 4064 children from 149 countries. The median age at diagnosis was 23·2 months (IQR 11·0-36·5). Extraocular tumour spread (cT4 of the cTNMH classification) at diagnosis was reported in five (0·8%) of 636 children from high-income countries, 55 (5·4%) of 1027 children from upper-middle-income countries, 342 (19·7%) of 1738 children from lower-middle-income countries, and 196 (42·9%) of 457 children from low-income countries. Enucleation surgery was available for all children and intravenous chemotherapy was available for 4014 (98·8%) of 4064 children. The 3-year survival rate was 99·5% (95% CI 98·8-100·0) for children from high-income countries, 91·2% (89·5-93·0) for children from upper-middle-income countries, 80·3% (78·3-82·3) for children from lower-middle-income countries, and 57·3% (52·1-63·0) for children from low-income countries. On analysis, independent factors for worse survival were residence in low-income countries compared to high-income countries (hazard ratio 16·67; 95% CI 4·76-50·00), cT4 advanced tumour compared to cT1 (8·98; 4·44-18·18), and older age at diagnosis in children up to 3 years (1·38 per year; 1·23-1·56). For children aged 3-7 years, the mortality risk decreased slightly (p=0·0104 for the change in slope). Interpretation: This study, estimated to include approximately half of all new retinoblastoma cases worldwide in 2017, shows profound inequity in survival of children depending on the national income level of their country of residence. In high-income countries, death from retinoblastoma is rare, whereas in low-income countries estimated 3-year survival is just over 50%. Although essential treatments are available in nearly all countries, early diagnosis and treatment in low-income countries are key to improving survival outcomes.
dc.description.versionVersión Publicada
dc.identifier.citationGlobal Retinoblastoma Study Group. The Global Retinoblastoma Outcome Study: a prospective, cluster-based analysis of 4064 patients from 149 countries. Lancet Glob Health. 2022 Aug;10(8):e1128-e1140. doi: 10.1016/S2214-109X(22)00250-9
dc.identifier.doihttps://doi.org/10.1016/S2214-109X(22)00250-9
dc.identifier.urihttps://repositorio.udd.cl/handle/11447/8276
dc.language.isoen
dc.subjectChild
dc.subjectRetinal Neoplasms / drug therapy
dc.subjectHumans
dc.subjectProspective Studies
dc.subjectInfant
dc.subjectRetinal Neoplasms / therapy
dc.subjectRetinoblastoma / diagnosis
dc.subjectRetinoblastoma / epidemiology
dc.subjectRetinoblastoma / therapy
dc.subjectTreatment Outcome
dc.titleThe Global Retinoblastoma Outcome Study: a prospective, cluster-based analysis of 4064 patients from 149 countries
dc.typeArticle
dcterms.accessRightsAcceso Abierto
dcterms.sourceThe Lancet Global health
dspace.entity.typePublication
relation.isAuthorOfPublication80199dfb-63aa-4965-8e0e-15dfed288656
relation.isAuthorOfPublication.latestForDiscovery80199dfb-63aa-4965-8e0e-15dfed288656

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