The Global Retinoblastoma Outcome Study: a prospective, cluster-based analysis of 4064 patients from 149 countries
Makaleler > Tam Makale · 2022
Yazarlar: Ido Didi Fabian, Elhassan Abdallah, Shehu U. Abdullahi, Rula A. Abdulqader, Aminatu A. Abdulrahaman, Sherif Abouelnaga, Dupe S. Ademola-Popoola, Adedayo Adio, Mahmoud A. Afifi, Armin R. Afshar, Priyanka Aggarwal, Ada E. Aghaji, Alia Ahmad, Marliyanti NR Akib, Marliyanti NR Akib, Marliyanti NR Akib, Adeseye Akinsete, Lamis Al Harby, Saleh Al Mesfer, Mouroge H. Al Ani, Silvia Alarcón Portabella, Safaa AF Al-Badri, Ana Patricia A. Alcasabas, Saad A. Al-Dahmash, Amanda Alejos, Ernesto Alemany-Rubio, Amadou I. Alfa Bio, Yvania Alfonso Carreras, Christiane E. Al-Haddad, Hamoud HY Al-Hussaini, Amany M. Ali, Donjeta B. Alia, Donjeta B. Alia, Mazin F. Al-Jadiry, Usama Al-Jumaily, Hind M. Alkatan, Charlotta All-Eriksson, Ali ARM Al-Mafrachi, Argentino A. Almeida, Khalifa M. Alsawidi, Athar ASM Al-Shaheen, Entissar H. Al-Shammary, Doreen Amankwaa-Frempong, Primawita O. Amiruddin, Inggar Armytasari, Nicholas J. Astbury, Hatice T. Atalay, Eda Ataseven, La ongsri Atchaneeyasakul, Rose Atsiaya, Rudolf Autrata, Julia Balaguer, Ruhengiz Balayeva, Honorio Barranco, Paulina Bartoszek, Paulina Bartoszek, Katarina Bartuma, Covadonga Bascaran, Nikolaos E. Bechrakis, Maja Beck Popovic, Ainura S. Begimkulova, Sarra Benmiloud, Rokia C. Berete, Jesse L. Berry, Anirban Bhaduri, Sunil Bhat, Arpita Bhattacharyya, Eva M. Biewald, Elaine Binkley, Sharon Blum, Nadia Bobrova, Nadia Bobrova, H. C. Boldt, H. C. Boldt, Maria Teresa BC Bonanomi, Maria Teresa BC Bonanomi, Gabrielle C. Bouda, Hédi Bouguila, Rachel C. Brennan, Bénédicte G. Brichard, Jassada Buaboonnam, Aléine Budiongo, Matthew J. Burton, Patricia Calderón-Sotelo, Doris A. Calle Jara, Jayne E. Camuglia, Miriam R. Cano, Michael Capra, Shani Caspi, Nathalie Cassoux, Guilherme Castela, Luis Castillo, Jaume Català-Mora, Isabel Cavieres, Arthika Chandramohan, Guillermo L. Chantada, Shabana Chaudhry, Bhavna Chawla, Wensi Chen, Wensi Chen, Mehmet KANTAR
LANCET GLOBAL HEALTH, SCI Expanded İndekslerine Giren Dergi, Uluslararası, Scopus, 2022, cilt 10, ss e1128-e1140
DOI: 10.1016/S2214-109X(22)00250-9
Özet
Background: 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. Funding: Queen Elizabeth Diamond Jubilee Trust.