The Global Retinoblastoma Outcome Study: a prospective, cluster-based analysis of 4064 patients from 149 countries. Issue 8 (August 2022)

• Record Type: Journal Article
• Authors: Fabian, Ido Didi; Abdallah, Elhassan; Abdullahi, Shehu U; Abdulqader, Rula A; Abdulrahaman, Aminatu A; Abouelnaga, Sherif; Ademola-Popoola, Dupe S; Adio, Adedayo; Afifi, Mahmoud A; Afshar, Armin R; Aggarwal, Priyanka; Aghaji, Ada E; Ahmad, Alia; Akib, Marliyanti NR; Akinsete, Adeseye; Al Harby, Lamis; Al Mesfer, Saleh; Al Ani, Mouroge H; Alarcón Portabella, Silvia; Al-Badri, Safaa AF; Alcasabas, Ana Patricia A; Al-Dahmash, Saad A; Alejos, Amanda; Alemany-Rubio, Ernesto; Alfa Bio, Amadou I; Alfonso Carreras, Yvania; Al-Haddad, Christiane E; Al-Hussaini, Hamoud HY; Ali, Amany M; Alia, Donjeta B; Al-Jadiry, Mazin F; Al-Jumaily, Usama; Alkatan, Hind M; All-Eriksson, Charlotta; Al-Mafrachi, Ali ARM; Almeida, Argentino A; Alsawidi, Khalifa M; Al-Shaheen, Athar ASM; Al-Shammary, Entissar H; Amankwaa-Frempong, Doreen; Amiruddin, Primawita O; Armytasari, Inggar; Astbury, Nicholas J; Atalay, Hatice T; Ataseven, Eda; Atchaneeyasakul, La-ongsri; Atsiaya, Rose; Autrata, Rudolf; Balaguer, Julia; Balayeva, Ruhengiz; Barranco, Honorio; Bartoszek, Paulina; Bartuma, Katarina; Bascaran, Covadonga; Bechrakis, Nikolaos E; Beck Popovic, Maja; Begimkulova, Ainura S; Benmiloud, Sarra; Berete, Rokia C; Berry, Jesse L; Bhaduri, Anirban; Bhat, Sunil; Bhattacharyya, Arpita; Biewald, Eva M; Binkley, Elaine; Blum, Sharon; Bobrova, Nadia; Boldt, H.C.; Bonanomi, Maria Teresa BC; Bouda, Gabrielle C; Bouguila, Hédi; Brennan, Rachel C; Brichard, Bénédicte G; Buaboonnam, Jassada; Budiongo, Aléine; Burton, Matthew J; Calderón-Sotelo, Patricia; Calle Jara, Doris A; Camuglia, Jayne E; Cano, Miriam R; Capra, Michael; Caspi, Shani; Cassoux, Nathalie; Castela, Guilherme; Castillo, Luis; Català-Mora, Jaume; Cavieres, Isabel; Chandramohan, Arthika; Chantada, Guillermo L; Chaudhry, Shabana; Chawla, Bhavna; Chen, Wensi; Chiwanga, Faraja S; Chuluunbat, Tsengelmaa; Cieslik, Krzysztof; Clark, Antony; Cockcroft, Ruellyn L; Comsa, Codruta; Correa Llano, Maria G; Corson, Timothy W; Couitchere, Line; Cowan-Lyn, Kristin E; Csóka, Monika; Dangboon, Wantanee; Das, Anirban; Das, Pranab; Das, Sima; Davanzo, Jacquelyn M; Davidson, Alan; De Francesco, Sonia; De Potter, Patrick; Quintero D, Karina; Demirci, Hakan; Desjardins, Laurence; Díaz Coronado, Rosdali Y; Dimaras, Helen; Dodgshun, Andrew J; Donato Macedo, Carla R; Dragomir, Monica D; Du, Yi; Du Bruyn, Magritha; Du Plessis, Johannes; Dudeja, Gagan; Eerme, Katrin; Eka Sutyawan, I Wayan; El Kettani, Asmaa; Elbahi, Amal M; Elder, James E; Elhaddad, Alaa M; Elhassan, Moawia MA; Elzembely, Mahmoud M; Ericksen, Connor; Essuman, Vera A; Evina, Ted Grimbert A; Ezegwui, Ifeoma R; Fadoo, Zehra; Fandiño, Adriana C; Faranoush, Mohammad; Fasina, Oluyemi; Fernández, Delia DPG; Fernández-Teijeiro, Ana; Foster, Allen; Frenkel, Shahar; Fu, Ligia D; Fuentes-Alabi, Soad L; Garcia, Juan L; García Aldana, David; Garcia Pacheco, Henry N; Geel, Jennifer A; Ghassemi, Fariba; Girón, Ana V; Goenz, Marco A; Gold, Aaron S; Goldberg, Hila; Gole, Glen A; Gomel, Nir; Gonzalez, Efren; Gonzalez Perez, Graciela; González-Rodríguez, Liudmira; Gorfine, Malka; Graells, Jaime; Gregersen, Pernille A; Grigorovski, Nathalia DAK; Guedenon, Koffi M; Gunasekera, D Sanjeeva; Gündüz, Ahmet K; Gupta, Himika; Gupta, Sanjiv; Gupta, Vineeta; Hadjistilianou, Theodora; Hamel, Patrick; Hamid, Syed A; Hamzah, Norhafizah; Hansen, Eric D; Harbour, J William; Hartnett, M. Elizabeth; Hasanreisoglu, Murat; Muhammad, Hassan; Hassan, Sadiq; Hassan, Shadab; Hautz, Wojciech; Haydar, Huda; Hederova, Stanislava; Hessissen, Laila; Hongeng, Suradej; Hordofa, Diriba F; Hubbard, G. Baker; Hummelen, Marlies; Husakova, Kristina; Hussein Al-Janabi, Allawi N; Ibanga, Affiong; Ida, Russo; Ilic, Vesna R; Islamov, Ziyavuddin; Jairaj, Vivekaraj; Janjua, Teyyeb; Jeeva, Irfan; Ji, Xunda; Jo, Dong Hyun; Jones, Michael M; Kabesha Amani, Theophile B; Kabore, Rolande L; Kaliki, Swathi; Kalinaki, Abubakar; Kamsang, Pius; Kantar, Mehmet; Kapelushnik, Noa; Kardava, Tamar; Kebudi, Rejin; Keomisy, Jonny; Kepak, Tomas; Ketteler, Petra; Khan, Zohora J; Khaqan, Hussain A; Khetan, Vikas; Khodabande, Alireza; Khotenashvili, Zaza; Kim, Jonathan W; Kim, Jeong Hun; Kiratli, Hayyam; Kivela, Tero T.; Klett, Artur; Koç, Irem; Kosh Komba Palet, Jess Elio; Krivaitiene, Dalia; Kruger, Mariana; Kulvichit, Kittisak; Kuntorini, Mayasari W; Kyara, Alice; Lam, Geoffrey C; Larson, Scott A; Latinović, Slobodanka; Laurenti, Kelly D; Lavy, Yotam; Lavric Groznik, Alenka; Leverant, Amy A; Li, Cairui; Li, Kaijun; Limbu, Ben; Liu, Chun-Hsiu; Quah, BoonLong; López, Juan P; Lukamba, Robert M; Luna-Fineman, Sandra; Lutfi, Delfitri; Lysytsia, Lesia; Madgar, Shiran; Magrath, George N; Mahajan, Amita; Maitra, Puja; Maka, Erika; Makimbetov, Emil K; Maktabi, Azza; Maldonado, Carlos; Mallipatna, Ashwin; Manudhane, Rebecca; Manzhuova, Lyazat; Martín-Begue, Nieves; Masud, Sidra; Matende, Ibrahim O; Mattosinho, Clarissa CDS; Matua, Marchelo; Mayet, Ismail; Mbumba, Freddy B; McKenzie, John D; Mehrvar, Azim; Mengesha, Aemero A; Menon, Vikas; Mercado, Gary John V; Mets, Marilyn B; Midena, Edoardo; Miller, Audra; Mishra, Divyansh KC; Mndeme, Furahini G; Mohamedani, Ahmed A; Mohammad, Mona T; Moll, Annette C; Montero, Margarita M; Moreira, Claude; Mruthyunjaya, Prithvi; Msina, Mchikirwa S; Msukwa, Gerald; Mudaliar, Sangeeta S; Muma, Kangwa I M; Munier, Francis L; Murray, Timothy G; Musa, Kareem O; Mushtaq, Asma; Musika, Anne A; Mustak, Hamzah; Mustapha, Tajudeen; Muyen, Okwen M; Myezo, Khumo H; Naidu, Gita; Naidu, Natasha; Nair, Akshay Gopinathan; Natarajan, Sundaram; Naumenko, Larisa; Ndoye Roth, Paule Aïda; Nency, Yetty M; Neroev, Vladimir; Ng, Yvonne; Nikitovic, Marina; Nkanga, Elizabeth D; Nkumbe, Henry E; Numbi, Marcel N; Nummi, Kalle; Nuruddin, Murtuza; Nyaywa, Mutale; Nyirenda, Chinsisi; Obono-Obiang, Ghislaine; Oliver, Scott CN; Oporto, Joaquin; Ortega-Hernández, Miriam; Oscar, Alexander H; Ossandon, Diego; Pagarra, Halimah; Paintsil, Vivian; Paiva, Luisa; Palanivelu, Mahesh Shanmugam; Papyan, Ruzanna; Parrozzani, Raffaele; Pascual Morales, Claudia R; Paton, Katherine E; Pe'er, Jacob; Peralta Calvo, Jesús; Perić, Sanja; Pham, Chau TM; Philbert, Remezo; Plager, David A; Pochop, Pavel; Polania, Rodrigo A.; Polyakov, Vladimir; Ponce, Jimena; Qadir, Ali O; Qayyum, Seema; Qian, Jiang; Refaeli, David; Rahman, Ardizal; Rajkarnikar, Purnima; Ramanjulu, Rajesh; Ramasubramanian, Aparna; Ramirez-Ortiz, Marco A; Randhawa, Jasmeen K; Randrianarisoa, Hoby Lalaina; Raobela, Léa; Rashid, Riffat; Reddy, M.A.; Renner, Lorna A; Reynders, David; Ribadu, Dahiru; Ritter-Sovinz, Petra; Rogowska, Anna; Rojanaporn, Duangnate; Romero, Livia; Roy, Soma R; Saab, Raya H; Saakyan, Svetlana; Sabhan, Ahmed H; Sagoo, Mandeep S; Said, Azza MA; Saiju, Rohit; Salas, Beatriz; San Román Pacheco, Sonsoles; Sánchez, Gissela L; Sanchez Orozco, Alma Janeth; Sayalith, Phayvanh; Scanlan, Trish A; Schlüter, Sabrina; Schwab, Christoph; Sedaghat, Ahad; Seth, Rachna; Sgroi, Mariana; Shah, Ankoor S; Shakoor, Shawkat A; Sharma, Manoj K; Sherief, Sadik T; Shields, Carol L; Sia, David; Siddiqui, Sorath Noorani; Sidi cheikh, Sidi; Silva, Sónia; Singh, Arun D; Singh, Usha; Singha, Penny; Sitorus, Rita S; Skalet, Alison H; Soebagjo, Hendrian D; Sorochynska, Tetyana; Ssali, Grace; Stacey, Andrew W; Staffieri, Sandra E; Stahl, Erin D; Steinberg, David M; Stones, David K; Strahlendorf, Caron; Suarez, Maria Estela Coleoni; Sultana, Sadia; Sun, Xiantao; Superstein, Rosanne; Supriyadi, Eddy; Surukrattanaskul, Supawan; Suzuki, Shigenobu; Svojgr, Karel; Sylla, Fatoumata; Tamamyan, Gevorg; Tan, Deborah; Tandili, Alketa; Tang, Jing; Tarrillo Leiva, Fanny F; Tashvighi, Maryam; Tateshi, Bekim; Teh, Kok Hoi; Tehuteru, Edi S; Teixeira, Luiz F; Tekavcic Pompe, Manca; Thawaba, Abdullah Dahan M; Theophile, Tuyisabe; Toledano, Helen; Trang, Doan L; Traoré, Fousseyni; Tripathy, Devjyoti; Tuncer, Samuray; Tyau-Tyau, Harba; Umar, Ali B; Unal, Emel; Uner, Ogul E; Urbak, Steen F; Ushakova, Tatiana L; Usmanov, Rustam H; Valeina, Sandra; Valente, Paola; van Hoefen Wijsard, Milo; Vasquez Anchaya, Jacqueline Karina; Vaughan, Leon O; Veleva-Krasteva, Nevyana V; Verma, Nishant; Victor, Andi A; Viksnins, Maris; Villacís Chafla, Edwin G; Villegas, Victor M; Vishnevskia-Dai, Victoria; Waddell, Keith; Wali, Amina H; Wang, Yi-Zhuo; Wangtiraumnuay, Nutsuchar; Wetter, Julie; Widiarti, Widiarti; Wilson, Matthew W; Wime, Amelia DC; Wiwatwongwana, Atchareeya; Wiwatwongwana, Damrong; Wolley Dod, Charlotte; Wong, Emily S; Wongwai, Phanthipha; Wu, Si-qi; Xiang, Daoman; Xiao, Yishuang; Xu, Bing; Xue, Kang; Yaghy, Antonio; Yam, Jason C; Yang, Huasheng; Yanga, Jenny M; Yaqub, Muhammad A; Yarovaya, Vera A; Yarovoy, Andrey A; Ye, Huijing; Yee, Roberto I; Yousef, Yacoub A; Yuliawati, Putu; Zapata López, Arturo M; Zein, Ekhtelbenina; Zhang, Yi; Zhilyaeva, Katsiaryna; Zia, Nida; Ziko, Othman AO; Zondervan, Marcia; Bowman, Richard;
• Is Part Of: Lancet. Volume 10:Issue 8(2022)
• Page Start: e1128
• Page End: e1140
• Publication Date: 2022-08
• Subjects: World health -- Periodicals; 362.105
• DOI: 10.1016/S2214-109X(22)00250-9
• Languages: English
• ISSNs: 2214-109X

Abstract

Summary: 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.