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Global Retinoblastoma Presentation and Analysis by National Income Level

Global Retinoblastoma Study GroupThe Goldschleger Eye Institute, Sheba Medical Center, Tel Hashomer, Tel Aviv University, Tel Aviv, IsraelIdo Didi FabianThe Goldschleger Eye Institute, Sheba Medical Center, Tel Hashomer, Tel Aviv University, Tel Aviv, IsraelAbdallah ElhassanOphthalmology Department of Rabat, Mohammed V University, Rabat, MoroccoShehu U. AbdullahiAminu Kano Teaching Hospital, Bayero University, Kano, NigeriaRula Ahmed AbdulqaderNational Hospital of Niamey, Niamey, NigerSahadatou Adamou BoubacarNational Hospital of Niamey, Niamey, NigerDupe Ademola-PopoolaDepartment of Ophthalmology, University of Port Harcourt Teaching Hospital, Port Harcourt, NigeriaAdedayo AdioUniversity of California, San FranciscoArmin R. AfsharDepartment of Pediatrics, Banaras Hindu University, Varanasi, IndiaPriyanka AggarwalDepartment of Ophthalmology, College of Medicine, University of Nigeria, Enugu, NigeriaAE AghajiDepartment of Ophthalmology, College of Medicine, University of Nigeria, Enugu, NigeriaAlia AhmadMarliyanti N. R. AkibThe Royal London Hospital, Barts Health NHS Trust, and Moorfields Eye Hospital NHS Foundation Trust, London, United KingdomLamis Al HarbyHawler Medical University, Erbil, IraqMouroge H. Al AniHawler Medical University, Erbil, IraqAygun AlakbarovaZarifa Aliyeva National Center of Ophthalmology, Baku, AzerbaijanS. Alarcón PortabellaPediatric Oncology Unit, Children Welfare Teaching Hospital, College of Medicine, University of Baghdad, Baghdad, IraqSafa A. FarajPhilippine General Hospital, University of the Philippines, Manila, PhilippinesAna Patricia AlcasabasCollege of Medicine, King Saud University, Riyadh, Saudi ArabiaSaad Al-DahmashCollege of Medicine, King Saud University, Riyadh, Saudi ArabiaAmanda AlejosUnidad Nacional de Oncología Pediátrica, Guatemala City, GuatemalaErnesto Alemany-RubioInstituto Cubano de Oftalmología "Ramón Pando Ferrer"Amadou I Alfa BioUniversity of Parakou, Parakou, BeninYvania Alfonso CarrerasSt Damien Pediatric Hospital, Port-au-Prince, HaitiChristiane Al‐HaddadDepartment of Ophthalmology, American University of Beirut Medical Center, Beirut, LebanonHamoud H. Y. Al-HussainiPediatric Oncology Department, National Oncology Center, Sana’a, YemenAmany AliPediatric Oncology Department, South Egypt Cancer Institute, Assiut University, Assiut, EgyptDonjeta B. AliaUniversity Hospital Center Mother Theresa, Tirana, AlbaniaMazin Faisal Al‐JadiryPediatric Oncology Unit, Children Welfare Teaching Hospital, College of Medicine, University of Baghdad, Baghdad, IraqUsama Al‐JumailyImam Hussein Cancer Center, Karbala, IraqHind M. AlkatanSt Erik Eye Hospital, Stockholn, SwedenCharlotta All‐ErikssonSt Erik Eye Hospital, Stockholn, SwedenAli A. R. M. Al-MafrachiIbn Al Haitham Teaching Eye Hospital, Baghdad, IraqArgentino A AlmeidaTripoli Eye Hospital, University of Tripoli, Tripoli, LibyaKhalifa M AlsawidiTripoli Eye Hospital, University of Tripoli, Tripoli, LibyaAthar Abdul Samad Majeed Al-ShaheenOncology Unit, Child's Central Teaching Hospital, Baghdad, IraqEntissar Hadi Al-ShammaryOncology Unit, Child’s Central Teaching Hospital, Baghdad, IraqPrimawita O. AmiruddinBambino Gesù IRCCS Children’s Hospital, Rome, ItalyAntonino RomanzoBambino Gesù IRCCS Children’s Hospital, Rome, ItalyNick AstburyDepartment of Ophthalmology, School of Medicine, Gazi University, Ankara, TurkeyHatice Tuba AtalayDepartment of Ophthalmology, School of Medicine, Gazi University, Ankara, TurkeyL.–O. AtchaneeyasakulLighthouse For Christ Eye Centre, Mombasa, KenyaRose AtsiayaLighthouse For Christ Eye Centre, Mombasa, KenyaTaweevat AttasethUniversity Of Medicine 1 YangonThan AungRetina Consultants of Houston, Houston, TexasSilvia González AyalaRetina Consultants of Houston, Houston, TexasBaglan BaizakovaScientific Center of Pediatrics and Pediatric Surgery, Almaty, KazakhstanJulia BalaguerPediatric Oncology Unit, Hospital Universitario y Politécnico La Fe, Valencia, SpainRuhengiz BalayevaZarifa Aliyeva National Center of Ophthalmology, Baku, AzerbaijanWalentyna BalwierzPediatric Oncology Unit, Hospital Universitario y Politécnico La Fe, Valencia, SpainHonorio BarrancoPediatric Oncology Unit, Hospital Universitario y Politécnico La Fe, Valencia, SpainCovadonga BascaránInternational Centre for Eye Health, London School of Hygiene & Tropical Medicine, London, United KingdomMaja PopovićHospital Nacional de Niños Dr Carlos Sáenz Herrera, San Jose, Costa RicaRaquel BenavidesHospital Nacional de Niños Dr Carlos Sáenz Herrera, San Jose, Costa RicaSarra BenmiloudDepartment of Pediatric Oncology, University Hassan II Fès, Fez, MoroccoNissrine Bennani GuebessiOphthalmologic Department of the Teaching Hospital of Treichville, Abidjan, Côte d’IvoireR. BérétéChildren's Hospital Los Angeles, Keck School of Medicine, University of Southern California, Los AngelesJesse L. BerryChildren’s Hospital Los Angeles, Keck School of Medicine, University of Southern California, Los AngelesAnirban BhaduriThe Calcutta Medical Research Institute, Kolkata, IndiaSunil BhatDepartment of Pediatric Hematology and Oncology, Narayana Health City, Bangalore, IndiaS J BiddulphUniversity of the Witwatersrand, Johannesburg, South AfricaEva BiewaldDepartment of Ophthalmology, Essen University Hospital, University Duisburg-Essen, Essen, GermanyNadia BobrovaDepartment of Ophthalmology, Essen University Hospital, University Duisburg-Essen, Essen, GermanyMarianna BoehmeDepartment of Ophthalmology, Essen University Hospital, University Duisburg-Essen, Essen, GermanyH. Culver BoldtHospital das Clínicas da FMUSP, São Paulo, BrazilMaria Teresa B.C. BonanomiDepartment of Ophthalmology, Essen University Hospital, University Duisburg-Essen, Essen, GermanyNorbert BornfeldDepartment of Ophthalmology, Essen University Hospital, University Duisburg-Essen, Essen, GermanyGabrielle Chantal BoudaInstitut Hédi Raïs d’Ophtalmologie, Faculté de Médecine, Université Tunis El Manar, Tunis, TunisiaH. BouguilaEtablissement Hospitalière Spécialise Emir Abdelkader CEA Service d’Oncologie Pédiatrique, Oran, AlgeriaAmaria BoumedaneEtablissement Hospitalière Spécialise Emir Abdelkader CEA Service d’Oncologie Pédiatrique, Oran, AlgeriaRachel C. BrennanSolid Tumor Division, Department of Oncology, St. Jude Children’s Research Hospital, Memphis, TennesseeBénédicte G. BrichardSiriraj Hospital, Mahidol University, Bangkok, ThailandJassada BuaboonnamSiriraj Hospital, Mahidol University, Bangkok, ThailandPatricia Calderón-SoteloHospital Infantil Manuel de Jesús Rivera, Managua, NicaraguaDoris A. Calle JaraHospital del Niño Dr. Francisco De Icaza Bustamante, Guayaquil, EcuadorJayne E CamugliaDepartment of Ophthalmology, Queensland Children's Hospital, Brisbane, Queensland, AustraliaMiriam R. CanoSalud Ocular, Ministerio de Salud Publica, Asuncion, ParaguayMichael CapraOur Lady's HospitalNathalie CassouxInstitut CurieGuilherme CastelaCentro Hospital Universitário de Coimbra, University of Coimbra, Coimbra, PortugalLuis CastilloHospital Pereira Rossell, Montevideo, UruguayJaume Catalá‐MoraNationalScientific and Technical Research Council, CONICET, Buenos Aires, ArgentinaGuillermo ChantadaPaediatric Ophthalmology Department, Mayo Hospital and College of Allied Visual Sciences, King Edward Medical University, Lahore, PakistanShabana ChaudhryDepartment of Ophthalmic Plastic Surgery, Orbit and Ocular Oncology, PBMA's H. V. Desai Eye Hospital, Pune, Maharashtra, IndiaSonal S. ChauguleDepartment of Ophthalmic Plastic Surgery, Orbit and Ocular Oncology, PBMA’s H. V. Desai Eye Hospital, Pune, Maharashtra, IndiaArgudit ChauhanOcular Oncology Service, Dr Rajendra Prasad Centre for Ophthalmic Sciences, All India Institute of Medical Sciences, New Delhi, IndiaBhavna ChawlaOcular Oncology Service, Dr Rajendra Prasad Centre for Ophthalmic Sciences, All India Institute of Medical Sciences, New Delhi, IndiaVioleta ChernodrinskaEye Clinic, Department of Ophthalmology, University Hospital Alexandrovska, Medical University, Sofia, Sofia, BulgariaFaraja ChiwangaMuhimbili National Hospital, Dar es Salaam, TanzaniaTsengelmaa ChuluunbatDepartment of Ophthalmology, The Children's Memorial Health Institute, Warsaw, PolandKrzysztof CieślikDepartment of Ophthalmology, The Children’s Memorial Health Institute, Warsaw, PolandRuellyn L. CockcroftInstitute of Oncology, Prof. Dr Al. Trestioreanu, Bucharest, RomaniaCodruta ComsaUniversity of CincinnatiZélia M. CorrêaUniversity of CincinnatiMaria G. Correa LlanoHospital Sant Joan de Déu, Barcelona, SpainTimothy W. CorsonBustamante Hospital for Children, Kingston, JamaicaKristin E. Cowan-LynSemmelweis University, Budapest, HungaryMonika CsókaDepartment of Ophthalmology, Xinhua Hospital, Shanghai Jiao Tong University School of Medicine, Shanghai, ChinaXuehao CuiDepartment of Ophthalmology, Xinhua Hospital, Shanghai Jiao Tong University School of Medicine, Shanghai, ChinaIsac V. Da GamaQuelimane Central Hospital, Quelimane, Mozambique

JAMA Oncologyjournal2020en

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Annotatsiya

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- and 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.

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Mavzular

Ocular Oncology and Treatments Cancer, Hypoxia, and Metabolism Neuroblastoma Research and Treatments

Identifikatorlar

DOI: 10.1001/jamaoncol.2019.6716

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