The Lancet Global Health ( IF 19.9 ) Pub Date : 2022-07-12 , DOI: 10.1016/s2214-109x(22)00250-9 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 , 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 , 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 , 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 , H.C. Boldt , 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 , Faraja S Chiwanga , Tsengelmaa Chuluunbat , Krzysztof Cieslik , Antony Clark , Ruellyn L Cockcroft , Codruta Comsa , Maria G Correa Llano , Timothy W Corson , Line Couitchere , Kristin E Cowan-Lyn , Monika Csóka , Wantanee Dangboon , Anirban Das , Pranab Das , Sima Das , Jacquelyn M Davanzo , Alan Davidson , Sonia De Francesco , Patrick De Potter , Karina Quintero D , Hakan Demirci , Laurence Desjardins , Rosdali Y Díaz Coronado , Helen Dimaras , Andrew J Dodgshun , Carla R Donato Macedo , Monica D Dragomir , Yi Du , Magritha Du Bruyn , Johannes Du Plessis , Gagan Dudeja , Katrin Eerme , I Wayan Eka Sutyawan , Asmaa El Kettani , Amal M Elbahi , James E Elder , Alaa M Elhaddad , Moawia MA Elhassan , Mahmoud M Elzembely , Connor Ericksen , Vera A Essuman , Ted Grimbert A Evina , Ifeoma R Ezegwui , Zehra Fadoo , Adriana C Fandiño , Mohammad Faranoush , Oluyemi Fasina , Delia DPG Fernández , Ana Fernández-Teijeiro , Allen Foster , Shahar Frenkel , Ligia D Fu , Soad L Fuentes-Alabi , Juan L Garcia , David García Aldana , Henry N Garcia Pacheco , Jennifer A Geel , Fariba Ghassemi , Ana V Girón , Marco A Goenz , Aaron S Gold , Hila Goldberg , Glen A Gole , Nir Gomel , Efren Gonzalez , Graciela Gonzalez Perez , Liudmira González-Rodríguez , Malka Gorfine , Jaime Graells , Pernille A Gregersen , Nathalia DAK Grigorovski , Koffi M Guedenon , D Sanjeeva Gunasekera , Ahmet K Gündüz , Himika Gupta , Sanjiv Gupta , Vineeta Gupta , Theodora Hadjistilianou , Patrick Hamel , Syed A Hamid , Norhafizah Hamzah , Eric D Hansen , J William Harbour , M. Elizabeth Hartnett , Murat Hasanreisoglu , Hassan Muhammad , Sadiq Hassan , Shadab Hassan , Wojciech Hautz , Huda Haydar , Stanislava Hederova , Laila Hessissen , Suradej Hongeng , Diriba F Hordofa , G. Baker Hubbard , Marlies Hummelen , Kristina Husakova , Allawi N Hussein Al-Janabi , Affiong Ibanga , Russo Ida , Vesna R Ilic , Ziyavuddin Islamov , Vivekaraj Jairaj , Teyyeb Janjua , Irfan Jeeva , Xunda Ji , Dong Hyun Jo , Michael M Jones , Theophile B Kabesha Amani , Rolande L Kabore , Swathi Kaliki , Abubakar Kalinaki , Pius Kamsang , Mehmet Kantar , Noa Kapelushnik , Tamar Kardava , Rejin Kebudi , Jonny Keomisy , Tomas Kepak , Petra Ketteler , Zohora J Khan , Hussain A Khaqan , Vikas Khetan , Alireza Khodabande , Zaza Khotenashvili , Jonathan W Kim , Jeong Hun Kim , Hayyam Kiratli , Tero T. Kivela , Artur Klett , Irem Koç , Jess Elio Kosh Komba Palet , Dalia Krivaitiene , Mariana Kruger , Kittisak Kulvichit , Mayasari W Kuntorini , Alice Kyara , Geoffrey C Lam , Scott A Larson , Slobodanka Latinović , Kelly D Laurenti , Yotam Lavy , Alenka Lavric Groznik , Amy A Leverant , Cairui Li , Kaijun Li , Ben Limbu , Chun-Hsiu Liu , BoonLong Quah , Juan P López , Robert M Lukamba , Sandra Luna-Fineman , Delfitri Lutfi , Lesia Lysytsia , Shiran Madgar , George N Magrath , Amita Mahajan , Puja Maitra , Erika Maka , Emil K Makimbetov , Azza Maktabi , Carlos Maldonado , Ashwin Mallipatna , Rebecca Manudhane , Lyazat Manzhuova , Nieves Martín-Begue , Sidra Masud , Ibrahim O Matende , Clarissa CDS Mattosinho , Marchelo Matua , Ismail Mayet , Freddy B Mbumba , John D McKenzie , Azim Mehrvar , Aemero A Mengesha , Vikas Menon , Gary John V Mercado , Marilyn B Mets , Edoardo Midena , Audra Miller , Divyansh KC Mishra , Furahini G Mndeme , Ahmed A Mohamedani , Mona T Mohammad , Annette C Moll , Margarita M Montero , Claude Moreira , Prithvi Mruthyunjaya , Mchikirwa S Msina , Gerald Msukwa , Sangeeta S Mudaliar , Kangwa I M Muma , Francis L Munier , Timothy G Murray , Kareem O Musa , Asma Mushtaq , Anne A Musika , Hamzah Mustak , Tajudeen Mustapha , Okwen M Muyen , Khumo H Myezo , Gita Naidu , Natasha Naidu , Akshay Gopinathan Nair , Sundaram Natarajan , Larisa Naumenko , Paule Aïda Ndoye Roth , Yetty M Nency , Vladimir Neroev , Yvonne Ng , Marina Nikitovic , Elizabeth D Nkanga , Henry E Nkumbe , Marcel N Numbi , Kalle Nummi , Murtuza Nuruddin , Mutale Nyaywa , Chinsisi Nyirenda , Ghislaine Obono-Obiang , Scott CN Oliver , Joaquin Oporto , Miriam Ortega-Hernández , Alexander H Oscar , Diego Ossandon , Halimah Pagarra , Vivian Paintsil , Luisa Paiva , Mahesh Shanmugam Palanivelu , Ruzanna Papyan , Raffaele Parrozzani , Claudia R Pascual Morales , Katherine E Paton , Jacob Pe'er , Jesús Peralta Calvo , Sanja Perić , Chau TM Pham , Remezo Philbert , David A Plager , Pavel Pochop , Rodrigo A. Polania , Vladimir Polyakov , Jimena Ponce , Ali O Qadir , Seema Qayyum , Jiang Qian , David Refaeli , Ardizal Rahman , Purnima Rajkarnikar , Rajesh Ramanjulu , Aparna Ramasubramanian , Marco A Ramirez-Ortiz , Jasmeen K Randhawa , Hoby Lalaina Randrianarisoa , Léa Raobela , Riffat Rashid , M.A. Reddy , Lorna A Renner , David Reynders , Dahiru Ribadu , Petra Ritter-Sovinz , Anna Rogowska , Duangnate Rojanaporn , Livia Romero , Soma R Roy , Raya H Saab , Svetlana Saakyan , Ahmed H Sabhan , Mandeep S Sagoo , Azza MA Said , Rohit Saiju , Beatriz Salas , Sonsoles San Román Pacheco , Gissela L Sánchez , Alma Janeth Sanchez Orozco , Phayvanh Sayalith , Trish A Scanlan , Sabrina Schlüter , Christoph Schwab , Ahad Sedaghat , Rachna Seth , Mariana Sgroi , Ankoor S Shah , Shawkat A Shakoor , Manoj K Sharma , Sadik T Sherief , Carol L Shields , David Sia , Sorath Noorani Siddiqui , Sidi Sidi cheikh , Sónia Silva , Arun D Singh , Usha Singh , Penny Singha , Rita S Sitorus , Alison H Skalet , Hendrian D Soebagjo , Tetyana Sorochynska , Grace Ssali , Andrew W Stacey , Sandra E Staffieri , Erin D Stahl , David M Steinberg , David K Stones , Caron Strahlendorf , Maria Estela Coleoni Suarez , Sadia Sultana , Xiantao Sun , Rosanne Superstein , Eddy Supriyadi , Supawan Surukrattanaskul , Shigenobu Suzuki , Karel Svojgr , Fatoumata Sylla , Gevorg Tamamyan , Deborah Tan , Alketa Tandili , Jing Tang , Fanny F Tarrillo Leiva , Maryam Tashvighi , Bekim Tateshi , Kok Hoi Teh , Edi S Tehuteru , Luiz F Teixeira , Manca Tekavcic Pompe , Abdullah Dahan M Thawaba , Tuyisabe Theophile , Helen Toledano , Doan L Trang , Fousseyni Traoré , Devjyoti Tripathy , Samuray Tuncer , Harba Tyau-Tyau , Ali B Umar , Emel Unal , Ogul E Uner , Steen F Urbak , Tatiana L Ushakova , Rustam H Usmanov , Sandra Valeina , Paola Valente , Milo van Hoefen Wijsard , Jacqueline Karina Vasquez Anchaya , Leon O Vaughan , Nevyana V Veleva-Krasteva , Nishant Verma , Andi A Victor , Maris Viksnins , Edwin G Villacís Chafla , Victor M Villegas , Victoria Vishnevskia-Dai , Keith Waddell , Amina H Wali , Yi-Zhuo Wang , Nutsuchar Wangtiraumnuay , Julie Wetter , Widiarti Widiarti , Matthew W Wilson , Amelia DC Wime , Atchareeya Wiwatwongwana , Damrong Wiwatwongwana , Charlotte Wolley Dod , Emily S Wong , Phanthipha Wongwai , Si-qi Wu , Daoman Xiang , Yishuang Xiao , Bing Xu , Kang Xue , Antonio Yaghy , Jason C Yam , Huasheng Yang , Jenny M Yanga , Muhammad A Yaqub , Vera A Yarovaya , Andrey A Yarovoy , Huijing Ye , Roberto I Yee , Yacoub A Yousef , Putu Yuliawati , Arturo M Zapata López , Ekhtelbenina Zein , Yi Zhang , Katsiaryna Zhilyaeva , Nida Zia , Othman AO Ziko , Marcia Zondervan , Richard Bowman
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.
中文翻译：
全球视网膜母细胞瘤结果研究：对来自 149 个国家的 4064 名患者进行的前瞻性、聚类分析
背景
视网膜母细胞瘤是全世界最常见的眼内癌症。有一些证据表明，不同地区的视网膜母细胞瘤儿童的治疗结果存在重大差异，但这些差异尚未在全球范围内进行评估。我们的目的是报告全球视网膜母细胞瘤儿童的 3 年结局，并调查与生存相关的因素。
方法
我们对 2017 年 1 月 1 日至 2017 年 12 月 31 日期间诊断的未接受治疗的视网膜母细胞瘤患者进行了前瞻性聚类分析，然后进行了 3 年的治疗和随访。患者是从全球 260 个专业治疗中心招募的。从参与中心获得有关主要治疗和附加治疗、随访持续时间、转移、眼球抢救和生存结果的数据。我们使用 Cox 回归模型分析了死亡时间和摘除时间。
发现
该队列包括来自 149 个国家的 4064 名儿童。诊断时的中位年龄为 23·2 个月 (IQR 11·0–36·5)。来自高收入国家的 636 名儿童中有 5 名（0·8%）在诊断时报告了眼外肿瘤扩散（cTNMH 分类的 cT4）；来自中高收入国家的 1027 名儿童中有 55 名（5·4%）；342来自中低收入国家的 1738 名儿童中的 19·7%，以及来自低收入国家的 457 名儿童中的 196 名 (42·9%)。所有儿童均可进行摘除手术，4064 名儿童中有 4014 名（98·8%）可进行静脉化疗。高收入国家儿童的 3 年生存率为 99·5%（95% CI 98·8–100·0），中高收入国家儿童的 3 年生存率为 91·2%（89·5–93·0） -收入国家，来自中低收入国家的儿童为 80·3% (78·3–82·3)，来自低收入国家的儿童为 57·3% (52·1–63·0)。分析显示，与高收入国家相比，生存率较差的独立因素包括居住在低收入国家（风险比 16·67；95% CI 4·76–50·00）、cT4 晚期肿瘤与 cT1 相比（8·98 ; 4·44–18·18），以及3岁以下儿童的诊断年龄较大（每年1·38；1·23–1·56）。对于 3-7 岁的儿童，死亡风险略有下降（斜率变化 p=0·0104）。
解释
这项研究估计涵盖了 2017 年全球所有新视网膜母细胞瘤病例的大约一半，显示儿童的生存存在严重不平等，具体取决于其居住国的国民收入水平。在高收入国家，视网膜母细胞瘤导致的死亡很少见，而在低收入国家，估计 3 年生存率略高于 50%。尽管几乎所有国家都可以提供基本治疗，但低收入国家的早期诊断和治疗是改善生存结果的关键。
资金
伊丽莎白女王钻禧信托基金。
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