Publication details

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

Authors	ALIA Donjeta B TANDILI Alketa PAIVA Luisa WIME Amelia Dc CHANTADA Guillermo L FANDINO Adriana C SGROI Mariana PAPYAN Ruzanna TAMAMYAN Gevorg CAMUGLIA Jayne E GOLE Glen A CLARK Antony LAM Geoffrey C ELDER James E MCKENZIE John D STAFFIERI Sandra E JONES Michael M MANUDHANE Rebecca SIA David RITTER-SOVINZ Petra SCHWAB Christoph BALAYEVA Ruhengiz KHAN Zohora NURUDDIN Murtuza ROY Soma R RASHID Riffat SULTANA Sadia SHAKOOR Shawkat A NAUMENKO Larisa ZHILYAEVA Katsiaryna BARTOSZEK Paulina BRICHARD Benedicte G PATRICK De Potter BIO Amadou I Alfa SALAS Beatriz ESTELA Coleoni Suarez Maria MBUMBA Freddy B BONANOMI Maria Teresa B C DONATO MACEDO Carla R. GRIGOROVSKI Nathalia D A K MATTOSINHO Clarissa C D S TEIXEIRA Luiz F OSCAR Alexander H VELEVA-KRASTEVA Nevyana V BOUDA Gabrielle C KABORE Rolande L PHILBERT Remezo EVINA Ted Grimbert A NKUMBE Henry E KAMSANG Pius MUYEN Okwen M DIMARAS Helen MALLIPATNA Ashwin HAMEL Patrick SUPERSTEIN Rosanne PATON Katherine E STRAHLENDORF Caron PALET Jess Elio Kosh Komba TYAU-TYAU Harba CAVIERES Isabel LOPEZ Juan P OPORTO Joaquin OSSANDON Diego CHEN Wensi XIANG Daoman DU Yi LI Kaijun JI Xunda TANG Jing LI Cairui XU Bing QIAN Jiang XUE Kang SUN Xiantao WANG Yi-Zhuo ZHANG Yi WU Si-qi XIAO Yishuang YANG Huasheng YE Huijing POLANIA Rodrigo A BERETE Rokia C COUITCHERE Line PERIC Sanja ALEMANY-RUBIO Ernesto GONZALEZ-RODRIGUEZ Liudmira AUTRATA Rudolf KEPÁK Tomáš POCHOP Pavel SVOJGR Karel GREGERSEN Pernille A URBAK Steen F MONTERO Margarita M BUDIONGO Aleine YANGA Jenny M AMANI Theophile B Kabesha LUKAMBA Robert M NUMBI Marcel N CALLE JARA Doris A VILLACIS CHAFLA Edwin G SANCHEZ Gissela L ABOUELNAGA Sherif AFIFI Mahmoud A ELHADDAD Alaa M ALI Amany M ELZEMBELY M. SAID Azza M A ZIKO Othman A O FUENTES-ALABI Soad L GOENZ Marco A EERME Katrin KLETT Artur HORDOFA Diriba F MENGESHA Aemero A SHERIEF Sadik T KIVELA Tero T NUMMI Kalle CASSOUX Nathalie DESJARDINS Laurence OBONO-OBIANG Ghislaine KARDAVA Tamar KHOTENASHVILI Zaza BECHRAKIS Nikolaos E BIEWALD Eva M SCHLUETER Sabrina KETTELER Petra AMANKWAA-FREMPONG Doreen ESSUMAN Vera A PAINTSIL Vivian RENNER Lorna A ALEJOS Amanda GIRON Ana V CARRERAS Yvania Alfonso FU Ligia D MALDONADO Carlos WONG Emily S YAM Jason C CSOKA Monika MAKA Erika AGGARWAL Priyanka GUPTA Vineeta BHADURI Anirban BHATTACHARYYA Arpita DAS Anirban CHAWLA Bhavna DAS Pranab DAS Sima GUPTA Himika GUPTA Sanjiv VERMA Nishant KALIKI Swathi KHETAN Vikas MAITRA Puja MAHAJAN Amita MENON Vikas MISHRA Divyansh K C PALANIVELU Mahesh Shanmugam RAMANJULU Rajesh MUDALIAR Sangeeta S NAIR Akshay Gopinathan NATARAJAN Sundaram SETH Rachna SINGH Usha BHAT Sunil DUDEJA Gagan TRIPATHY Devjyoti AKIB Indonesia Marliyanti N R PAGARRA Halimah AMIRUDDIN Primawita O KUNTORINI Mayasari W ARMYTASARI Inggar SUPRIYADI Eddy SUTYAWAN I Wayan Eka YULIAWATI Putu LUTFI Delfitri SOEBAGJO Hendrian D RAHMAN Ardizal SITORUS Rita S VICTOR Andi A TEHUTERU Edi S WIDIARTI Widiarti NENCY Yetty M FARANOUSH Mohammad MEHRVAR Azim TASHVIGHI Maryam SEDAGHAT Ahad GHASSEMI Fariba KHODABANDE Alireza ABDULQADER Rula A AL-SHAHEEN Athar A S M AL ANI MOUROGE H. HAYDAR Huda AL-BADRI Safaa A F AL-JADIRY Mazin F SABHAN Ahmed H AL-JUMAILY Usama AL-MAFRACHI Ali A R M AL-SHAMMARY Entissar H AL-JANABI Allawi N Hussein QADIR Ali O CAPRA Michael BLUM Sharon GOMEL Nir FABIAN Ido Didi GOLDBERG Hila KAPELUSHNIK Noa MADGAR Shiran VISHNEVSKIA-DAI Victoria FRENKEL Shahar PEER Jacob GORFINE Malka REFAELI David STEINBERG David M LAVY Yotam TOLEDANO Helen CASPI Shani SONIA De Francesco HADJISTILIANOU Theodora IDA Russo VALENTE Paola MIDENA Edoardo PARROZZANI Raffaele COWAN-LYN Kristin E VAUGHAN Leon O SUZUKI Shigenobu MOHAMMAD Mona T YOUSEF Yacoub A MANZHUOVA Lyazat ATSIAYA Rose MATENDE Ibrahim O BEGIMKULOVA Ainura S MAKIMBETOV Emil K KEOMISY Jonny SAYALITH Phayvanh VALEINA Sandra VIKSNINS Maris AL-HADDAD Christiane E SAAB Raya H ALSAWIDI Khalifa M ELBAHI Amal M KRIVAITIENE Dalia TATESHI Bekim RANDRIANARISOA Hoby Lalaina RAOBELA Lea MSUKWA Gerald NYIRENDA Chinsisi HAMZAH Norhafizah TEH Kok Hoi SYLLA Fatoumata TRAORE Fousseyni CHEIKH Sidi Sidi ZEIN Ekhtelbenina PEREZ Graciela Gonzalez JANETH Sanchez Orozco Alma ORTEGA-HERNANDEZ Miriam RAMIREZ-ORTIZ Marco A CHULUUNBAT Tsengelmaa ABDALLAH Elhassan BENMILOUD Sarra ASMAA El Kettani HESSISSEN Laila ALMEIDA Argentino A LIMBU Ben RAJKARNIKAR Purnima SAIJU Rohit MOLL Annette C WIJSARD Milo van Hoefen COCKCROFT Ruellyn L NG Yvonne DODGSHUN Andrew J CALDERON-SOTELO Patricia ABDULLAHI Shehu U HASSAN Sadiq UMAR Ali B ABDULRAHAMAN Aminatu A WALI Amina H ADEMOLA-POPOOLA Dupe S ADIO Adedayo AGHAJI Ada E EZEGWUI Ifeoma R AKINSETE Adeseye MUSA Kareem O FASINA Oluyemi IBANGA Affiong NKANGA Elizabeth D MUSTAPHA Tajudeen RIBADU Dahiru HUMMELEN Marlies AHMAD Alia MUSHTAQ Asma QAYYUM Seema CHAUDHRY Shabana FADOO Zehra JEEVA Irfan MASUD Sidra HAMID Syed A ZIA Nida HASSAN Shadab SIDDIQUI Sorath Noorani JANJUA Teyyeb YAQUB Muhammad A KHAQAN Hussain A KARINA Quintero D YEE Roberto I JAIRAJ Vivekaraj CANO Miriam R FERNANDEZ Delia D P G DIAZ CORONADO Rosdali Y ZAPATA LOPEZ Arturo M GARCIA Juan L PONCE Jimena GARCIA PACHECO Henry N PASCUAL MORALES Claudia R KARINA Vasquez Anchaya Jacqueline TARRILLO LEIVA Fanny F ALCASABAS Ana Patricia A MERCADO Gary John V CIESLIK Krzysztof HAUTZ Wojciech ROGOWSKA Anna CASTELA Guilherme SILVA Sonia JO Dong Hyun KIM Jeong Hun COMSA Codruta DRAGOMIR Monica D NEROEV Vladimir SAAKYAN Svetlana POLYAKOV Vladimir USHAKOVA Tatiana L YAROVAYA Vera A YAROVOY Andrey A THEOPHILE Tuyisabe SALEH Al Mesfer MAKTABI Azza AL-DAHMASH Saad A ALKATAN Hind M MOREIRA Claude ROTH Paule Aida Ndoye ILIC Vesna R NIKITOVIC Marina LATINOVIC Slobodanka QUAH BoonLong TAN Deborah HEDEROVA Stanislava HUSAKOVA Kristina GROZNIK Alenka Lavric POMPE Manca Tekavcic DAVIDSON Alan MAGRITHA Du Bruyn JOHANNES Du Plessis STONES David K GEEL Jennifer A MYEZO Khumo H KRUGER Mariana MAYET Ismail NAIDU Gita NAIDU Natasha MUSTAK Hamzah REYNDERS David WETTER Julie SILVIA Alarcon Portabella MARTIN-BEGUE Nieves CHARLOTTE Wolley Dod BALAGUER Julia BARRANCO Honorio CATALA-MORA Jaume CORREA LLANO Maria G FERNANDEZ-TEIJEIRO Ana DAVID Garcia Aldana JESUS Peralta Calvo SONSOLES San Roman Pacheco GUNASEKERA D Sanjeeva ELHASSAN Moawia M A MOHAMEDANI Ahmed A ALL-ERIKSSON Charlotta BARTUMA Katarina POPOVIC Maja Beck MUNIER Francis L LIU Chun-Hsiu CHIWANGA Faraja S KYARA Alice MNDEME Furahini G MSINA Mchikirwa S SCANLAN Trish A ATCHANEEYASAKUL La-ongsri BUABOONNAM Jassada DANGBOON Wantanee SINGHA Penny HONGENG Suradej KULVICHIT Kittisak ROJANAPORN Duangnate SURUKRATTANASKUL Supawan WANGTIRAUMNUAY Nutsuchar WIWATWONGWANA Atchareeya WIWATWONGWANA Damrong WONGWAI Phanthipha SHARMA Manoj K GUEDENON Koffi M BOUGUILA Hedi ATALAY Hatice T HASANREISOGLU Murat ATASEVEN Eda KANTAR Mehmet GUNDUZ Ahmet K KEBUDI Rejin KIRATLI Hayyam KOC Irem TUNCER Samuray UNAL Emel KALINAKI Abubakar MATUA Marchelo WADDELL Keith MUSIKA Anne A SSALI Grace LAMIS Al Harby REDDY M Ashwin ASTBURY Nicholas J BASCARAN Covadonga BOWMAN Richard BURTON Matthew J FOSTER Allen ZONDERVAN Marcia SAGOO Mandeep S BOBROVA Nadia SOROCHYNSKA Tetyana LYSYTSIA Lesia CASTILLO Luis AFSHAR Armin R BERRY Jesse L KIM Jonathan W RANDHAWA Jasmeen K BINKLEY Elaine BOLDT H C LARSON Scott A BRENNAN Rachel C CHANDRAMOHAN Arthika STACEY Andrew W CORSON Timothy W PLAGER David A DAVANZO Jacquelyn M SINGH Arun D DEMIRCI Hakan ERICKSEN Connor MAGRATH George N GOLD Aaron S MURRAY Timothy G GONZALEZ Efren SHAH Ankoor S HANSEN Eric D HARTNETT M Elizabeth HARBOUR J William HUBBARD G Baker UNER Ogul E LAURENTI Kelly D METS Marilyn B LEVERANT Amy A RAMASUBRAMANIAN Aparna LUNA-FINEMAN Sandra MILLER Audra SKALET Alison H MRUTHYUNJAYA Prithvi HASSAN Muhammad OLIVER Scott Cn SHIELDS Carol L YAGHY Antonio STAHL Erin D WILSON Matthew W VILLEGAS Victor M ISLAMOV Ziyavuddin USMANOV Rustam H GRAELLS Jaime ROMERO Livia PHAM Chau T M TRANG Doan L AL-HUSSAINI Hamoud Hy THAWABA Abdullah Dahan M MUMA Kangwa I M NYAYWA Mutale
Year of publication	2022
Type	Article in Periodical
Magazine / Source	LANCET GLOBAL HEALTH
MU Faculty or unit	Faculty of Medicine
Citation
Web	https://www.sciencedirect.com/science/article/pii/S2214109X22002509?via%3Dihub
Doi	http://dx.doi.org/10.1016/S2214-109X(22)00250-9
Keywords	Retinoblastoma
Description	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. Enudeation 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% (524-63-0) 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.