Subsequent malignant neoplasms in the pediatric age in retinoblastoma survivors in Argentina.

Publisher: John Wiley Country of Publication: United States NLM ID: 101186624 Publication Model: Print-Electronic Cited Medium: Internet ISSN: 1545-5017 (Electronic) Linking ISSN: 15455009 NLM ISO Abbreviation: Pediatr Blood Cancer Subsets: MEDLINE

Original Publication: Hoboken, N.J. : John Wiley, c 2004-

Bone Neoplasms*/complications ; Breast Neoplasms*/epidemiology ; Central Nervous System Neoplasms*/complications ; Leukemia*/complications ; Neoplasms*/complications ; Neoplasms, Second Primary*/diagnosis ; Neoplasms, Second Primary*/epidemiology ; Neoplasms, Second Primary*/etiology ; Retinal Neoplasms*/complications ; Retinal Neoplasms*/epidemiology ; Retinal Neoplasms*/therapy ; Retinoblastoma*/complications ; Retinoblastoma*/epidemiology ; Retinoblastoma*/therapy ; Sarcoma*/epidemiology ; Sarcoma*/etiology ; Sarcoma*/therapy ; Sarcoma, Ewing*/complications ; Skin Neoplasms*/complications ; Soft Tissue Neoplasms*/complications; Adolescent ; Argentina/epidemiology ; Child ; Female ; Humans ; Incidence ; Risk Assessment ; Survivors

Background: Retinoblastoma survivors in low- and middle-income countries are exposed to high-intensity treatments that potentially place them at higher risk of early subsequent malignant neoplasms (SMNs).
Methods: We followed 714 (403 [56.4%] nonhereditary and 311 [43.5%] hereditary) retinoblastoma survivors diagnosed from August 1987 to December 2016, up to the age of 16 years. We quantified risk of SMNs with cumulative incidence (CI) and standardized incidence ratios (SIR) analysis. Multivariate regression Cox model was used to determine the association of treatments and risk of SMNs.
Results: Median follow-up was of 9 years (range: 0.18-16.9) and 24 survivors (3.36%) developed 25 SMNs (n = 22 hereditary, n = 2 nonhereditary). SMNs included sarcomas (osteosarcomas, Ewing sarcomas, rhabdomyosarcomas; n = 12), leukemias (n = 5), and central nervous system tumors (CNS; n = 3). All cases of acute myeloid leukemia (AML) and most of Ewing sarcomas occurred within 5 years of retinoblastoma diagnosis. The type of SMN was the main indicator of mortality (five of five patients with leukemias, six of 12 with sarcomas, and zero of three with CNS tumors died). Compared to the general population, radiation increased the risk of Ewing sarcoma in hereditary survivors by 700-fold (95% CI = 252-2422.6) and chemotherapy increased the risk of AML by 140-fold (95% CI = 45.3-436). The CI of SMNs for hereditary survivors was 13.7% (95% CI = 8.4-22.1) at 15 years.
Conclusion: Retinoblastoma survivors from Argentina are at higher risk of developing SMNs early in life compared to the general Argentinean population, especially those treated with radiation plus chemotherapy. AML and Ewing sarcoma presented within 5 years of retinoblastoma diagnosis are associated with chemotherapy and radiation exposure.
(© 2022 Wiley Periodicals LLC.)

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Keywords: cumulative incidence; low- and middle-income countries; pediatric childhood survivors; retinoblastoma; subsequent malignant neoplasm

Date Created: 20220422 Date Completed: 20220627 Latest Revision: 20220714

20240104

10.1002/pbc.29710

35451226