Publication:
Global Retinoblastoma Presentation and Analysis by National Income Level

Issued Date

2020-01-01

Resource Type

Article

ISSN

23742445
23742437

DOI

10.1001/jamaoncol.2019.6716

Other identifier(s)

2-s2.0-85081545321

Rights

Mahidol University

Rights Holder(s)

SCOPUS

Bibliographic Citation

JAMA Oncology. (2020)

Suggested Citation

Ido Didi Fabian, Elhassan Abdallah, Shehu U. Abdullahi, Rula A. Abdulqader, Sahadatou Adamou Boubacar, Dupe S. Ademola-Popoola, Adedayo Adio, Armin R. Afshar, Priyanka Aggarwal, Ada E. Aghaji, Alia Ahmad, Marliyanti N.R. Akib, Lamis Al Harby, Mouroge H. Al Ani, Aygun Alakbarova, Silvia Alarcón Portabella, Safaa A.F. Al-Badri, Ana Patricia A. Alcasabas, Saad A. Al-Dahmash, Amanda Alejos, Ernesto Alemany-Rubio, Amadou I. Alfa Bio, Yvania Alfonso Carreras, Christiane Al-Haddad, Hamoud H.Y. Al-Hussaini, Amany M. Ali, Donjeta B. Alia, Mazin F. Al-Jadiry, Usama Al-Jumaly, Hind M. Alkatan, Charlotta All-Eriksson, Ali A.R.M. Al-Mafrachi, Argentino A. Almeida, Khalifa M. Alsawidi, Athar A.S.M. Al-Shaheen, Entissar H. Al-Shammary, Primawita O. Amiruddin, Romanzo Antonino, Nicholas J. Astbury, Hatice T. Atalay, La Ongsri Atchaneeyasakul, Rose Atsiaya, Taweevat Attaseth, Than H. Aung, Silvia Ayala, Baglan Baizakova, Julia Balaguer, Ruhengiz Balayeva, Walentyna Balwierz, Honorio Barranco, Covadonga Bascaran, Maja Beck Popovic, Raquel Benavides, Sarra Benmiloud, Nissrine Bennani Guebessi, Rokia C. Berete, Jesse L. Berry, Anirban Bhaduri, Sunil Bhat, Shelley J. Biddulph, Eva M. Biewald, Nadia Bobrova, Marianna Boehme, H. C. Boldt, Maria Teresa B.C. Bonanomi, Norbert Bornfeld, Gabrielle C. Bouda, Hédi Bouguila, Amaria Boumedane, Rachel C. Brennan, Bénédicte G. Brichard, Jassada Buaboonnam, Patricia Calderón-Sotelo, Doris A. Calle Jara, Jayne E. Camuglia, Miriam R. Cano, Michael Capra, Nathalie Cassoux, Guilherme Castela, Luis Castillo, Jaume Català-Mora, Guillermo L. Chantada, Shabana Chaudhry, Sonal S. Chaugule, Argudit Chauhan, Bhavna Chawla, Violeta S. Chernodrinska, Faraja S. Chiwanga, Tsengelmaa Chuluunbat, Krzysztof Cieslik, Ruellyn L. Cockcroft, Codruta Comsa, Zelia M. Correa, Maria G. Correa Llano, Timothy W. Corson, Kristin E. Cowan-Lyn, Monika Csóka, Xuehao Cui, Isac V. Da Gama, Wantanee Dangboon Global Retinoblastoma Presentation and Analysis by National Income Level. JAMA Oncology. (2020). doi:10.1001/jamaoncol.2019.6716 Retrieved from: https://repository.li.mahidol.ac.th/handle/20.500.14594/53618

Research Projects

Organizational Units

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Thesis

Title

Global Retinoblastoma Presentation and Analysis by National Income Level

Author(s)

Ido Didi Fabian
 Elhassan Abdallah
 Shehu U. Abdullahi
 Rula A. Abdulqader
 Sahadatou Adamou Boubacar
 Dupe S. Ademola-Popoola
 Adedayo Adio
 Armin R. Afshar
 Priyanka Aggarwal
 Ada E. Aghaji
 Alia Ahmad
 Marliyanti N.R. Akib
 Lamis Al Harby
 Mouroge H. Al Ani
 Aygun Alakbarova
 Silvia Alarcón Portabella
 Safaa A.F. Al-Badri
 Ana Patricia A. Alcasabas
 Saad A. Al-Dahmash
 Amanda Alejos
 Ernesto Alemany-Rubio
 Amadou I. Alfa Bio
 Yvania Alfonso Carreras
 Christiane Al-Haddad
 Hamoud H.Y. Al-Hussaini
 Amany M. Ali
 Donjeta B. Alia
 Mazin F. Al-Jadiry
 Usama Al-Jumaly
 Hind M. Alkatan
 Charlotta All-Eriksson
 Ali A.R.M. Al-Mafrachi
 Argentino A. Almeida
 Khalifa M. Alsawidi
 Athar A.S.M. Al-Shaheen
 Entissar H. Al-Shammary
 Primawita O. Amiruddin
 Romanzo Antonino
 Nicholas J. Astbury
 Hatice T. Atalay
 La Ongsri Atchaneeyasakul
 Rose Atsiaya
 Taweevat Attaseth
 Than H. Aung
 Silvia Ayala
 Baglan Baizakova
 Julia Balaguer
 Ruhengiz Balayeva
 Walentyna Balwierz
 Honorio Barranco
 Covadonga Bascaran
 Maja Beck Popovic
 Raquel Benavides
 Sarra Benmiloud
 Nissrine Bennani Guebessi
 Rokia C. Berete
 Jesse L. Berry
 Anirban Bhaduri
 Sunil Bhat
 Shelley J. Biddulph
 Eva M. Biewald
 Nadia Bobrova
 Marianna Boehme
 H. C. Boldt
 Maria Teresa B.C. Bonanomi
 Norbert Bornfeld
 Gabrielle C. Bouda
 Hédi Bouguila
 Amaria Boumedane
 Rachel C. Brennan
 Bénédicte G. Brichard
 Jassada Buaboonnam
 Patricia Calderón-Sotelo
 Doris A. Calle Jara
 Jayne E. Camuglia
 Miriam R. Cano
 Michael Capra
 Nathalie Cassoux
 Guilherme Castela
 Luis Castillo
 Jaume Català-Mora
 Guillermo L. Chantada
 Shabana Chaudhry
 Sonal S. Chaugule
 Argudit Chauhan
 Bhavna Chawla
 Violeta S. Chernodrinska
 Faraja S. Chiwanga
 Tsengelmaa Chuluunbat
 Krzysztof Cieslik
 Ruellyn L. Cockcroft
 Codruta Comsa
 Zelia M. Correa
 Maria G. Correa Llano
 Timothy W. Corson
 Kristin E. Cowan-Lyn
 Monika Csóka
 Xuehao Cui
 Isac V. Da Gama
 Wantanee Dangboon

Other Contributor(s)

Hawler Medical University
 Dr. Rajendra Prasad Centre for Ophthalmic Sciences
 Filatov Institute of Eye Diseases and Tissue Therapy
 Instytut "Pomnik - Centrum Zdrowia Dziecka"
 University of Tripoli
 University Hospital Centre Tirana
 Centre Hospitalier Universitaire Yalgado Ouedraogo
 Hospital Nacional de Niños Dr. Carlos Sáenz Herrera
 Centro Hospitalario Pereira Rossell
 Muhimbili National Hospital
 University of the Philippines Manila
 Baghdad Teaching Hospital
 University of Baghdad
 Children's Hospital Lahore
 Université de Tunis El Manar, Institut Hedi Raies d'ophtalmologie de Tunis
 Calcutta Medical Research Institute
 American University of Beirut Medical Center
 King Edward Medical University Lahore
 Hospital Universitari i Politècnic La Fe
 London School of Hygiene & Tropical Medicine
 University of Port Harcourt
 University of Nigeria
 Shanghai Jiao Tong University
 Semmelweis Egyetem
 Indiana University-Purdue University Indianapolis
 University of Iowa
 University of California, San Francisco
 Centre Hospitalier Universitaire Vaudois
 Uniwersytet Jagielloński Collegium Medicum
 University of Louisville
 Starship Children's Health
 Our Lady's Hospital for Sick Children
 Centre Hospitalier Universitaire Ibn-Rochd
 St. Jude Children's Research Hospital
 Centro Hospitalar e Universitário de Coimbra
 IRCCS Ospedale Pediatrico Bambino Gesù
 University of Cincinnati College of Medicine
 Moorfields Eye Hospital NHS Foundation Trust
 University of Witwatersrand
 Faculty of Medicine, Ramathibodi Hospital, Mahidol University
 Centre Hospitalier Universitaire Hassan II
 Keck School of Medicine of USC
 Gazi Üniversitesi
 King Saud University
 Cliniques Universitaires Saint-Luc
 Hospital Universitari Vall d'Hebron
 Institutul Oncologic Prof. Dr. Alexandru Trestioreanu Bucuresti
 University Hospital Alexandrovska
 Faculty of Medicine, Siriraj Hospital, Mahidol University
 University of Ilorin
 Banaras Hindu University
 Universidade de Sao Paulo - USP
 Universitäts Klinikum Essen und Medizinische Fakultät
 Prince of Songkla University
 Tel Aviv University
 Consejo Nacional de Investigaciones Científicas y Técnicas
 Bayero University
 Fundacion Hospital de Pediatria Professor Dr. Juan P. Garrahan
 Assiut University
 Imam Hussein Cancer Center
 Etablissement Hospitaliere Specialise Emir Abdelkader CEA Service d'Oncologie Pediatrique
 Scientific Center of Pediatrics and Pediatric Surgery
 Mohammed v University
 Université Paris Descartes
 Queensland Children's Hospital
 Basra Specialty Hospital
 National Center for Maternal and Child Health
 University of Medicine 1
 Central Hospital Quelimane
 Ibn Al-Haitham Teaching Eye Hospital
 University of Parakou
 PBMA's H. V. Desai Eye Hospital
 St. Damien Pediatric Hospital
 Teaching Hospital of Treichville
 National Centre of Ophthalmology Named after Acad. Zarifa Aliyeva
 Beira Central Hospital
 Instituto Cubano de Oftalmología Ramón Pando Ferrer
 Hospital Sant Joan de Déu
 Ministerio de Salud Pública
 St Erik Eye Hospital
 National Oncology Centre
 Hospital del Niño Dr. Francisco de Icaza Bustamante
 Retina Consultants of Houston
 Unidad Nacional de Oncología Pediátrica
 Lighthouse for Christ Eye Centre
 Cicendo Eye Hospital
 National Hospital of Niamey
 Bustamante Hospital for Children
 Hospital Infantil Manuel de Jesus Rivera

Abstract

© 2020 Cambridge University Press. All rights reserved. Importance: Early diagnosis of retinoblastoma, the most common intraocular cancer, can save both a child's life and vision. However, anecdotal evidence suggests that many children across the world are diagnosed late. To our knowledge, the clinical presentation of retinoblastoma has never been assessed on a global scale. Objectives: To report the retinoblastoma stage at diagnosis in patients across the world during a single year, to investigate associations between clinical variables and national income level, and to investigate risk factors for advanced disease at diagnosis. Design, Setting, and Participants: A total of 278 retinoblastoma treatment centers were recruited from June 2017 through December 2018 to participate in a cross-sectional analysis of treatment-naive patients with retinoblastoma who were diagnosed in 2017. Main Outcomes and Measures: Age at presentation, proportion of familial history of retinoblastoma, and tumor stage and metastasis. Results: The cohort included 4351 new patients from 153 countries; the median age at diagnosis was 30.5 (interquartile range, 18.3-45.9) months, and 1976 patients (45.4%) were female. Most patients (n = 3685 [84.7%]) were from low- A nd middle-income countries (LMICs). Globally, the most common indication for referral was leukocoria (n = 2638 [62.8%]), followed by strabismus (n = 429 [10.2%]) and proptosis (n = 309 [7.4%]). Patients from high-income countries (HICs) were diagnosed at a median age of 14.1 months, with 656 of 666 (98.5%) patients having intraocular retinoblastoma and 2 (0.3%) having metastasis. Patients from low-income countries were diagnosed at a median age of 30.5 months, with 256 of 521 (49.1%) having extraocular retinoblastoma and 94 of 498 (18.9%) having metastasis. Lower national income level was associated with older presentation age, higher proportion of locally advanced disease and distant metastasis, and smaller proportion of familial history of retinoblastoma. Advanced disease at diagnosis was more common in LMICs even after adjusting for age (odds ratio for low-income countries vs upper-middle-income countries and HICs, 17.92 [95% CI, 12.94-24.80], and for lower-middle-income countries vs upper-middle-income countries and HICs, 5.74 [95% CI, 4.30-7.68]). Conclusions and Relevance: This study is estimated to have included more than half of all new retinoblastoma cases worldwide in 2017. Children from LMICs, where the main global retinoblastoma burden lies, presented at an older age with more advanced disease and demonstrated a smaller proportion of familial history of retinoblastoma, likely because many do not reach a childbearing age. Given that retinoblastoma is curable, these data are concerning and mandate intervention at national and international levels. Further studies are needed to investigate factors, other than age at presentation, that may be associated with advanced disease in LMICs.

Keyword(s)

Biochemistry, Genetics and Molecular Biology
 Medicine

Availability

URI

https://repository.li.mahidol.ac.th/handle/20.500.14594/53618

Collections

Scopus 2020