Global, regional, and national burden and changing trends of hepatoblastoma and retinoblastoma from 1990 to 2021, and projections until 2050: findings from the Global Burden of Disease Study 2021.
Using GBD 2021 modeled data, the study reports declining global hepatoblastoma burden but increasing retinoblastoma incidence and prevalence, marked socioeconomic and regional disparities, and projections of rising retinoblastoma burden through 2050.
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Using GBD 2021 modeled data, the study reports declining global hepatoblastoma burden but increasing retinoblastoma incidence and prevalence, marked socioeconomic and regional disparities, and projections of rising retinoblastoma burden through 2050.
Research significance
The evidence supports identifying populations and regions with disproportionate mortality and disability burden; it can be inferred—but is not tested here—that targeted screening, referral capacity, genetic counseling, and resource allocation could improve timely care and outcomes, particularly in low-SDI settings.
Source abstract
BACKGROUND: Hepatoblastoma and retinoblastoma, two common extracranial embryonal tumors, are the most prevalent primary cancer of liver and eye in childhood, respectively. Despite high survival in high-income regions, outcomes remain extremely poor in low-income regions, underscoring global health inequities and the need for evidence-based resource allocation. To address this gap, we explored the global, regional, and national burden of the two diseases from 1990 to 2021 and projected trends until 2050. METHODS: We extracted data on incidence, prevalence, deaths, and disability-adjusted life-years (DALYs) of hepatoblastoma and retinoblastoma by age, sex, region, and country from the Global Burden of Disease (GBD) 2021 database. Age-standardised rates (ASRs) with 95% uncertainty intervals (UIs) and estimated annual percentage changes (EAPCs) were calculated to measure disease burden and trends. Spearman correlation analysis was applied to evaluate the association between the socio-demographic index (SDI) and disease burden. The Bayesian age-period-cohort (BAPC) model was utilized for projections. RESULTS: From 1990 to 2021, ASRs of incidence, prevalence, deaths, and DALYs of hepatoblastoma decreased significantly (EAPCs: -2.03, -1.84, -2.54, -2.53, respectively). For retinoblastoma, the ASRs of incidence and prevalence first increased until 2019 and then decreased, yet exhibited overall upward trends (EAPCs: 1.35 for incidence, 1.36 for prevalence). However, the deaths and DALYs of retinoblastoma went downward. The combined burden of both cancers also declined from 1990 to 2021. Disease burden peaked at age 0-6 days for hepatoblastoma, and at 0-6 days and 2-4 years for retinoblastoma. In low SDI regions, ASRs of deaths and DALYs of retinoblastoma were higher than those of hepatoblastoma. V-shaped associations were seen between SDI and both ASRs of incidence and prevalence of hepatoblastoma (incidence: r=-0.241, prevalence: r=-0.159) and retinoblastoma (incidence: r=0.221, prevalence: r=0.224) from 1990 to 2021. The deaths and DALYs of both cancers declined with increasing SDI. Positive correlation was found between SDI and EAPCs of ASRs of hepatoblastoma and retinoblastoma from 1990 to 2021 (all r>0), except for those of retinoblastoma deaths and DALYs which were negatively correlated with SDI (deaths: r=-0.282, DALYs: r=-0.192). Projections suggested hepatoblastoma burden would decline globally but rise in America, while retinoblastoma incidence and prevalence would increase globally. CONCLUSIONS: Despite decreased global burden of hepatoblastoma, retinoblastoma incidence and prevalence increased. Besides, substantial age-related, regional and socioeconomic disparities existed. Predictions showed decreasing hepatoblastoma burden, but rising retinoblastoma cases. Targeted strategies are needed, especially in low SDI regions, including antenatal examination, early screening, genetic counseling, environmental and nutritional improvements, and international collaboration to strengthen healthcare training, referral networks, and multidisciplinary consultation to reduce disparities and promote children health.