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Incidence of second malignancies in individuals diagnosed with malignant peripheral nerve sheath tumors.

诊断为恶性外周神经鞘瘤的个体中第二恶性肿瘤的发生率。

  • 影响因子:3.10
  • DOI:10.1007/s11060-020-03478-9
  • 作者列表:"Williams LA","Moertel CL","Richardson M","Marcotte EL
  • 发表时间:2020-04-01
Abstract

BACKGROUND:The incidence of malignant peripheral nerve sheath tumors (MPNSTs) is low in the general population, although individuals with Neurofibromatosis Type I (NF1) are particularly susceptible. These tumors generally have a high probability of metastasis. The rate and types of second malignancies (SMNs) after a primary diagnosis of MPNST are not well characterized. We aimed to quantify the rate of SMNs among individuals with a first primary MPNST using population-based data. METHODS:We estimated age-standardized incidence rates (SIRs) for SMNs among 1,579 primary MPNST cases between ages 0-85+ using SEER 18 (2000-2015). We estimated incidence rate ratios (IRRs) and 95% confidence intervals (95% CI) for SMNs in MPNST cases compared with general population rates. We conducted sex-stratified and age-restricted analyses (< 30 years at diagnosis). RESULTS:Seven percent (108/1579) of MPNST cases developed a SMN (SIR of 4635 cases/million). Compared to the general population, MPNST cases were more likely to develop SMNs (IRR: 29.3; 95% CI 23.8-34.8) and had a much higher rate of second MPNSTs (IRR: 15,992.9; 95% CI 9594.5-22,391.3). Aside from a second MPNST, second cancers were frequently diagnosed in the breast, lung, skin, and soft tissue in females and were myeloid and skin malignancies in males. When restricted to < 30 years of age, second MPNSTs were the most common cancers diagnosed. CONCLUSIONS:The rate of SMNs among MPNST cases is tremendously higher than that observed among individuals with other cancers, particularly for second MPNSTs. These findings suggest rates of SMNs may also be higher in NF1 individuals.

摘要

背景: 恶性外周神经鞘瘤 (MPNSTs) 在普通人群中的发病率较低,尽管 I 型神经纤维瘤病 (NF1) 个体尤其易感。这些肿瘤通常有很高的转移概率。初步诊断 MPNST 后第二恶性肿瘤 (SMNs) 的发生率和类型没有得到很好的表征。我们的目的是使用基于人群的数据量化首次原发性 MPNST 个体中 SMNs 的发生率。 方法: 我们使用 SEER 18 (1,579-2000) 估计了 2015 例 0 ~ 85 岁之间的原发性 MPNST 病例中 smn 的年龄标准化发病率 (SIRs)。我们估计了 MPNST 病例中 SMNs 的发病率比 (IRRs) 和 95% 置信区间 (95% CI),并与普通人群率进行了比较。我们进行了性别分层和年龄限制分析 (诊断时 <30 年)。 结果: 7% (108/1579) 的 MPNST 病例发生 SMN (SIR 为 4635 例/百万)。与普通人群相比,MPNST 病例更容易发生 SMNs (IRR: 29.3; 95% CI 23.8-34.8),第二次 MPNST 发生率更高 (IRR: 15,992.9; 95% CI 9594.5-22,391.3)。除第二个 MPNST 外,第二个癌症在女性中经常被诊断为乳腺、肺、皮肤和软组织,在男性中是髓系和皮肤恶性肿瘤。当限制到 <30 岁时,第二 MPNSTs 是最常见的癌症诊断。 结论: MPNST 病例中 SMNs 的发生率远远高于其他癌症患者,尤其是第二 MPNST。这些研究结果表明,NF1 个体中 SMNs 的发生率也可能较高。

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