Jia Miao, He Enyang, Sun Wenjing, Cui Hualei, Zhao Hailan, Zhao Wei, Liu Wei, Guo Zhenhua, Wang Yi, Feng Wei
Department of Ultrasound, Guizhou Hospital of Shanghai Children's Medical Center, Shanghai Jiao Tong University School of Medicine, Guizhou Provincial People's Hospital, Guiyang, 550000, China.
Tianjin Children's Hospital (Tianjin University Children's Hospital), Tianjin, 300134, China.
BMC Cancer. 2025 Jul 5;25(1):1148. doi: 10.1186/s12885-025-14566-2.
Hepatoblastoma (HB), the most common pediatric primary liver cancer, accounts for 80% of childhood liver malignancies but only 1% of all pediatric cancers. Approximately 90% of cases occur in children under five years old, with 20-40% of patients presenting with distant metastases at diagnosis. Surgical resection is critical, but only 50-60% of tumors are completely resectable initially; unresectable or highly invasive cases require liver transplantation, highlighting the disease's severe clinical and economic burden. Despite regional disparities in incidence and mortality, the global burden of HB remains underquantified, with limited understanding of its correlation with socioeconomic factors., This study aims to comprehensively assess the disease burden of HB and its correlation with socioeconomic factors, identify high-burden regions, and provide evidence to optimize resource allocation and reduce disease burden.
Using data from the global burden of disease (GBD) 2021 Study, we analyzed HB incidence, mortality, prevalence, and disability-adjusted life years (DALYs) across 204 countries/regions from 1990 to 2021. Linear regression was employed to calculate the estimated annual percentage change (EAPC) to assess time trends, while joinpoint regression calculate the annual percentage change (APC) and average annual percentage change (AAPC) to identified inflection points in trends. Subgroup analyses were conducted by time-varying socio-demographic index (SDI), geographic region, and age.
From 1990 to 2021, global HB incidence rate decreased by 60.11%, with mortality and DALYs rates showing parallel decreases. (EAPC=-0.53 for both). However, high-SDI region exhibited increasing incidence (EAPC = 0.21), driven by Australasia (EAPC = 0.81) and high-income North America (EAPC = 0.73), In 2021, Low-SDI regions, particularly West Sub-Saharan Africa, ranked first globally in all assessed HB burden indicators. China had the highest case count, and India had the most deaths. Mali had the highest incidence and mortality rates, whereas Estonia had the lowest. Joinpoint regression analysis revealed a post-2004 slowing of the global decline in age-standardized HB incidence and prevalence, and post-2006 slowing of the global decline in mortality rates. Concurrently, high-SDI regions exhibited persistent upward trends (AAPC = 0.75), particularly in high-income North America (AAPC = 2.23), Australasia (AAPC = 2.02), and Western Europe (AAPC = 0.73), where incidence rates have risen consistently since the early 2000s. These trends contrast with sustained declines in regions like Central Sub-Saharan Africa.
HB remains a significant global health issue. Over the past 32 years, the global burden of HB has generally declined, but the rate of decline has slowed, with stark disparities between socioeconomic regions. Global cooperation is essential in addressing the challenges posed by HB. The rise in high-SDI areas highlights the need to distinguish diagnostic improvements from true incidence increases. The burden is particularly heavy in low-middle-SDI regions and certain geographical areas. Priorities include enhancing access to pediatric oncology care in low-resource settings, optimizing prenatal/postnatal screening, and addressing socioeconomic drivers of inequity to reduce HB's burden globally.
肝母细胞瘤(HB)是最常见的儿童原发性肝癌,占儿童肝脏恶性肿瘤的80%,但仅占所有儿童癌症的1%。约90%的病例发生在5岁以下儿童,20%-40%的患者在诊断时出现远处转移。手术切除至关重要,但最初只有50%-60%的肿瘤可完全切除;不可切除或高侵袭性病例需要肝移植,这凸显了该疾病严重的临床和经济负担。尽管发病率和死亡率存在地区差异,但全球HB负担仍未得到充分量化,对其与社会经济因素的相关性了解有限。本研究旨在全面评估HB的疾病负担及其与社会经济因素的相关性,确定高负担地区,并为优化资源分配和减轻疾病负担提供依据。
利用全球疾病负担(GBD)2021研究的数据,我们分析了1990年至2021年期间204个国家/地区的HB发病率、死亡率、患病率和伤残调整生命年(DALYs)。采用线性回归计算估计年度百分比变化(EAPC)以评估时间趋势,同时采用连接点回归计算年度百分比变化(APC)和平均年度百分比变化(AAPC)以确定趋势中的拐点。按随时间变化的社会人口指数(SDI)、地理区域和年龄进行亚组分析。
1990年至2021年,全球HB发病率下降了60.11%,死亡率和DALYs率也呈平行下降(两者的EAPC均为-0.53)。然而,高SDI地区的发病率呈上升趋势(EAPC = 0.21),这是由澳大拉西亚(EAPC = 0.81)和高收入的北美地区(EAPC = 0.73)推动的。2021年,低SDI地区,特别是撒哈拉以南非洲西部地区,在所有评估的HB负担指标中全球排名第一。中国的病例数最多,印度的死亡人数最多。马里的发病率和死亡率最高,而爱沙尼亚的发病率和死亡率最低。连接点回归分析显示,2004年后全球年龄标准化HB发病率和患病率下降速度放缓,2006年后全球死亡率下降速度放缓。与此同时,高SDI地区呈现持续上升趋势(AAPC = 0.75),特别是在高收入的北美地区(AAPC = 2.23)、澳大拉西亚(AAPC = 2.02)和西欧(AAPC = 0.73),自21世纪初以来发病率持续上升。这些趋势与撒哈拉以南非洲中部等地区的持续下降形成对比。
HB仍然是一个重大的全球健康问题。在过去32年中,全球HB负担总体上有所下降,但下降速度放缓,社会经济地区之间存在明显差异。全球合作对于应对HB带来的挑战至关重要。高SDI地区发病率的上升凸显了区分诊断改善与实际发病率增加的必要性。中低SDI地区和某些地理区域的负担尤其沉重。优先事项包括在资源匮乏地区增加儿童肿瘤护理的可及性,优化产前/产后筛查,以及解决不平等的社会经济驱动因素,以减轻全球HB的负担。