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Global Retinoblastoma Presentation and Analysis by National Income Level Publisher Pubmed



Fabian ID1, 2 ; Abdallah E3 ; Abdullahi SU4 ; Abdulqader RA5 ; Adamou Boubacar S6 ; Ademolapopoola DS7 ; Adio A8 ; Afshar AR9 ; Aggarwal P10 ; Aghaji AE11 ; Ahmad A12 ; Akib MNR13 ; Al Harby L14 ; Al Ani MH15 Show All Authors
Authors
  1. Fabian ID1, 2
  2. Abdallah E3
  3. Abdullahi SU4
  4. Abdulqader RA5
  5. Adamou Boubacar S6
  6. Ademolapopoola DS7
  7. Adio A8
  8. Afshar AR9
  9. Aggarwal P10
  10. Aghaji AE11
  11. Ahmad A12
  12. Akib MNR13
  13. Al Harby L14
  14. Al Ani MH15
  15. Alakbarova A16
  16. Portabella SA17
  17. Albadri SAF18
  18. Alcasabas APA19
  19. Aldahmash SA20
  20. Alejos A21
  21. Alemanyrubio E22
  22. Alfa Bio AI23
  23. Alfonso Carreras Y24
  24. Alhaddad C25
  25. Alhussaini HHY26
  26. Ali AM27
  27. Alia DB28
  28. Aljadiry MF18
  29. Aljumaly U29
  30. Alkatan HM20
  31. Alleriksson C30
  32. Almafrachi AARM31
  33. Almeida AA32
  34. Alsawidi KM33
  35. Alshaheen AASM5
  36. Alshammary EH34
  37. Amiruddin PO35
  38. Antonino R36
  39. Astbury NJ1
  40. Atalay HT37
  41. Atchaneeyasakul LO38
  42. Atsiaya R39
  43. Attaseth T40
  44. Aung TH41
  45. Ayala S42
  46. Baizakova B43
  47. Balaguer J44
  48. Balayeva R16
  49. Balwierz W45
  50. Barranco H44
  51. Bascaran C1
  52. Beck Popovic M46
  53. Benavides R47
  54. Benmiloud S48
  55. Bennani Guebessi N49
  56. Berete RC50
  57. Berry JL51
  58. Bhaduri A52
  59. Bhat S53
  60. Biddulph SJ54
  61. Biewald EM55
  62. Bobrova N56
  63. Boehme M55
  64. Boldt HC57
  65. Bonanomi MTBC58
  66. Bornfeld N55
  67. Bouda GC59
  68. Bouguila H60
  69. Boumedane A61
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  211. Khotenashvili Z169
  212. Kim JW51
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  215. Kivela TT179
  216. Klett A180
  217. Komba Palet JEK181
  218. Krivaitiene D182
  219. Kruger M183
  220. Kulvichit K184
  221. Kuntorini MW35
  222. Kyara A80
  223. Lachmann ES185
  224. Lam CPS186
  225. Lam GC187
  226. Larson SA57
  227. Latinovic S188
  228. Laurenti KD189
  229. Le BHA190
  230. Lecuona K191
  231. Leverant AA192
  232. Li C193
  233. Limbu B194
  234. Long QB195
  235. Lopez JP196
  236. Lukamba RM197
  237. Lumbroso L98
  238. Lunafineman S198
  239. Lutfi D199
  240. Lysytsia L200
  241. Magrath GN163
  242. Mahajan A201
  243. Majeed AR135
  244. Maka E88
  245. Makan M202
  246. Makimbetov EK203
  247. Manda C122
  248. Martin Begue N17
  249. Mason L204
  250. Matende IO39
  251. Materin M175
  252. Mattosinho CCDS137
  253. Matua M205
  254. Mayet I54
  255. Mbumba FB206
  256. Mckenzie JD108, 207
  257. Medinasanson A208
  258. Mehrvar A209
  259. Mengesha AA210
  260. Menon V211
  261. Mercado GJVD19
  262. Mets MB189
  263. Midena E212
  264. Mishra DKC213
  265. Mndeme FG214
  266. Mohamedani AA215
  267. Mohammad MT216
  268. Moll AC217
  269. Montero MM218
  270. Morales RA64
  271. Moreira C219
  272. Mruthyunjaya P154
  273. Msina MS214
  274. Msukwa G122
  275. Mudaliar SS141
  276. Muma KI220
  277. Munier FL221
  278. Murgoi G84
  279. Murray TG128
  280. Musa KO222
  281. Mushtaq A12
  282. Mustak H191
  283. Muyen OM223
  284. Naidu G54
  285. Nair AG224, 225
  286. Naumenko L226
  287. Ndoye Roth PA227
  288. Nency YM228
  289. Neroev V229
  290. Ngo H230
  291. Nieves RM218
  292. Nikitovic M158
  293. Nkanga ED231
  294. Nkumbe H113
  295. Nuruddin M232
  296. Nyaywa M233
  297. Obonoobiang G234
  298. Oguego NC11
  299. Olechowski A82
  300. Oliver SCN235
  301. Oseibonsu P236
  302. Ossandon D237
  303. Paezescamilla MA148
  304. Pagarra H13
  305. Painter SL160
  306. Paintsil V236
  307. Paiva L238
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  309. Palanivelu MS213
  310. Papyan R240
  311. Parrozzani R212
  312. Parulekar M160
  313. Pascual Morales CR151
  314. Paton KE241
  315. Pawinskawasikowska K45
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  318. Peric S242
  319. Pham CTM243
  320. Philbert R244
  321. Plager DA86
  322. Pochop P245
  323. Polania RA246
  324. Polyakov VG247, 248
  325. Pompe MT249
  326. Pons JJ250
  327. Prat D2
  328. Prom V176
  329. Purwanto I251
  330. Qadir AO252
  331. Qayyum S12
  332. Qian J253
  333. Rahman A106
  334. Rahman S9
  335. Rahmat J146
  336. Rajkarnikar P194
  337. Ramanjulu R213
  338. Ramasubramanian A77
  339. Ramirezortiz MA254
  340. Raobela L255
  341. Rashid R256
  342. Reddy MA14
  343. Reich E257
  344. Renner LA258
  345. Reynders D259
  346. Ribadu D260
  347. Riheia MM261
  348. Rittersovinz P262
  349. Rojanaporn D40
  350. Romero L134
  351. Roy SR232
  352. Saab RH263
  353. Saakyan S229
  354. Sabhan AH18
  355. Sagoo MS264
  356. Said AMA265
  357. Saiju R194
  358. Salas B266
  359. San Roman Pacheco S267
  360. Sanchez GL268
  361. Sayalith P269
  362. Scanlan TA80
  363. Schefler AC42
  364. Schoeman J259
  365. Sedaghat A270
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Source: JAMA Oncology Published:2020


Abstract

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. © 2020 American Medical Association. All rights reserved.
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