Long-term risk of subsequent cancer incidence among hereditary and nonhereditary retinoblastoma survivors.
Long-term risk of subsequent cancer incidence among hereditary and nonhereditary retinoblastoma survivors.
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遗传性和非遗传性视网膜母细胞瘤幸存者发生癌症的长期风险。
DOI：
10.1038/s41416-020-01248-y
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发表时间：
2021-03
影响因子：
8.8
通讯作者：
Morton LM
中科院分区：
文献类型：
--
作者：
Schonfeld SJ;Kleinerman RA;Abramson DH;Seddon JM;Tucker MA;Morton LM
遗传性视网膜母细胞瘤后肉瘤和黑色素瘤的风险增加是众所周知的，而对上皮性恶性肿瘤（SMNs）和多发性（≥2）SMNs的风险知之甚少。利用长期随访和详细的组织学信息，我们量化了1128名遗传性和924名非遗传性视网膜母细胞瘤幸存者（1914-2006年诊断，随访至2016年）的SMN事件风险。标准化发病率（SIRs）比较了视网膜母细胞瘤后与一般人群的癌症风险。我们估计了考虑竞争死亡风险的累积发病率。遗传幸存者的SMN风险显著增加（N = 239; SIR = 11.9； 95%可信区间[CI] 10.4-13.5），肉瘤、鼻腔肿瘤和松果体母细胞瘤的SIRs为80倍。黑色素瘤、中枢神经系统、口腔和乳房SMNs （SIRs = 3.1-17）的风险也显著增加，但子宫、肾脏、肺、膀胱、胰腺或其他类型的SMNs风险没有显著增加。遗传性视网膜母细胞瘤后50年的累积发病率，第一次SMN为33.1% (95% CI 29.0-37.2)，第二次SMN为6.0% （95% CI 3.8-8.2）。非遗传性视网膜母细胞瘤后SMN风险未增加（N = 25; SIR = 0.8; 95% CI 0.5-1.2）。除了遗传性视网膜母细胞瘤后肉瘤和黑色素瘤的风险外，我们还证明了比以前认为的更有限数量的上皮恶性肿瘤的风险增加。累积发病率估计强调遗传性视网膜母细胞瘤后SMN的长期负担。
Increased sarcoma and melanoma risks after hereditary retinoblastoma are well established, whereas less is known about epithelial subsequent malignant neoplasms (SMNs) and risks for multiple (≥2) SMNs. Leveraging long-term follow-up and detailed histologic information, we quantified incident SMN risk among 1128 hereditary and 924 nonhereditary retinoblastoma survivors (diagnosed 1914–2006; follow-up through 2016). Standardised incidence ratios (SIRs) compared cancer risk after retinoblastoma relative to the general population. We estimated cumulative incidence accounting for competing risk of death. Hereditary survivors had statistically significantly increased SMN risk (N = 239; SIR = 11.9; 95% confidence interval [CI] 10.4–13.5), with SIRs >80-fold for sarcomas, nasal cavity tumours and pineoblastoma. Significantly increased risks were also observed for melanoma and central nervous system, oral cavity and breast SMNs (SIRs = 3.1–17), but not the uterus, kidney, lung, bladder, pancreas or other types. Cumulative incidence 50 years following hereditary retinoblastoma was 33.1% (95% CI 29.0–37.2) for a first SMN and 6.0% (95% CI 3.8–8.2) for a second SMN. SMN risk was not increased after nonhereditary retinoblastoma (N = 25; SIR = 0.8; 95% CI 0.5–1.2). Beyond the established sarcoma and melanoma risks after hereditary retinoblastoma, we demonstrate increased risk for a more limited number of epithelial malignancies than previously suggested. Cumulative incidence estimates emphasise long-term SMN burden after hereditary retinoblastoma.
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影响因子：
4.7
作者：
Francis, Jasmine H.;Kleinerman, Ruth A.;Abramson, David H.
通讯作者：
Abramson, David H.
影响因子：
45.3
作者：
Kleinerman, Ruth A.;Schonfeld, Sara J.;Morton, Lindsay M.
通讯作者：
Morton, Lindsay M.
影响因子：
8.8
作者：
MacCarthy A;Bayne AM;Brownbill PA;Bunch KJ;Diggens NL;Draper GJ;Hawkins MM;Jenkinson HC;Kingston JE;Stiller CA;Vincent TJ;Murphy MF
通讯作者：
Murphy MF
影响因子：
10.3
作者：
Fletcher, O;Easton, D;Peto, J
通讯作者：
Peto, J
影响因子：
45.3
作者：
Kivelä, T
通讯作者：
Kivelä, T