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成人脉络丛乳头状瘤大体全切除的相对患者获益。

The relative patient benefit of gross total resection in adult choroid plexus papillomas.

机构信息

Department of Neurological Surgery, University of California, San Francisco, San Francisco, CA 94143-0112, USA.

出版信息

J Clin Neurosci. 2013 Jun;20(6):808-12. doi: 10.1016/j.jocn.2012.08.003. Epub 2013 Apr 25.

DOI:10.1016/j.jocn.2012.08.003
PMID:23623658
Abstract

Choroid plexus papillomas are rare neuroepithelial tumors found mainly in children. Although well studied in the pediatric population, there is a paucity of literature focusing specifically on adults. We sought to assess the relative advantage of gross total resection (GTR) and further characterize the natural history of this disease in adults. A comprehensive PubMed search was performed to identify adults who underwent surgical resection for choroid plexus papillomas with clearly reported age, tumor location, and extent of resection. Kaplan-Meier analysis was used to assess progression-free survival (PFS) and overall survival (OS). Multivariate analysis was performed using Cox proportional hazards models. A total of 193 patients were identified with a mean age of 39.9 ± 1.1 years. GTR was achieved in 72% of patients with subtotal resection (STR) in 28%. GTR was associated with a significant increase in both PFS (p = 0.015) and OS (p = 0.004) compared to STR. In a multivariate Cox proportional hazards model we found that only GTR was associated with recurrence (hazard ratio [HR] = 0.47, 95% confidence interval [CI] 0.25-0.90), while both age (HR = 1.03, 95% CI 1.00-1.05) and GTR (HR = 0.36, 95% CI 0.17-0.78) were associated with OS. Interestingly, our observed recurrence and death rates were higher than those in previously published studies. These findings demonstrate the benefit of GTR for the treatment of choroid plexus papillomas in adults. Our analysis suggests that these lesions are not as indolent as previously thought and while GTR is preferred, it is not always curative.

摘要

脉络丛乳头状瘤是一种罕见的神经上皮肿瘤,主要发生在儿童中。虽然在儿科人群中得到了很好的研究,但针对成人的专门文献相对较少。我们旨在评估大体全切除(GTR)的相对优势,并进一步描述这种疾病在成人中的自然史。我们进行了全面的 PubMed 检索,以确定接受手术切除脉络丛乳头状瘤的成年人,这些患者的年龄、肿瘤位置和切除范围均有明确报告。采用 Kaplan-Meier 分析评估无进展生存期(PFS)和总生存期(OS)。使用 Cox 比例风险模型进行多变量分析。共确定了 193 例患者,平均年龄为 39.9±1.1 岁。72%的患者行 GTR,28%的患者行次全切除(STR)。与 STR 相比,GTR 显著提高了 PFS(p=0.015)和 OS(p=0.004)。在多变量 Cox 比例风险模型中,我们发现仅 GTR 与复发相关(风险比 [HR] = 0.47,95%置信区间 [CI] 0.25-0.90),而年龄(HR = 1.03,95% CI 1.00-1.05)和 GTR(HR = 0.36,95% CI 0.17-0.78)均与 OS 相关。有趣的是,我们观察到的复发率和死亡率高于以前发表的研究。这些发现表明 GTR 对成人脉络丛乳头状瘤的治疗有益。我们的分析表明,这些病变并不像以前认为的那样惰性,虽然 GTR 是首选,但它并不总是能治愈。

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引用本文的文献

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Choroid plexus tumors in adults: a retrospective mono-institutional study.成人脉络丛肿瘤:一项单中心回顾性研究。
Neurol Sci. 2025 Apr;46(4):1859-1866. doi: 10.1007/s10072-024-07894-x. Epub 2024 Dec 2.
2
The composition of choroid plexus tumor research: a bibliometric analysis of the 100 most impactful studies to date.脉络丛肿瘤研究构成:迄今 100 项最具影响力研究的文献计量分析。
Childs Nerv Syst. 2024 Feb;40(2):453-462. doi: 10.1007/s00381-023-06157-y. Epub 2023 Sep 20.
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Other Less Prevalent Tumors of the Central Nervous System.
中枢神经系统其他不常见肿瘤。
Adv Exp Med Biol. 2023;1405:607-643. doi: 10.1007/978-3-031-23705-8_24.
4
Spinal drop of atypical choroid plexus papilloma in an adult patient: A case report and literature review.一名成年患者非典型脉络丛乳头状瘤的脊髓播散:病例报告及文献综述
Surg Neurol Int. 2020 Nov 25;11:409. doi: 10.25259/SNI_649_2020. eCollection 2020.
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The genetic landscape of choroid plexus tumors in children and adults.儿童和成人脉络丛肿瘤的基因图谱。
Neuro Oncol. 2021 Apr 12;23(4):650-660. doi: 10.1093/neuonc/noaa267.
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Epigenetics impacts upon prognosis and clinical management of choroid plexus tumors.表观遗传学影响脉络丛肿瘤的预后和临床管理。
J Neurooncol. 2020 May;148(1):39-45. doi: 10.1007/s11060-020-03509-5. Epub 2020 Apr 28.
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Choroid plexus papilloma presenting as an occipital mass with neck pain: Case report.表现为枕部肿块伴颈部疼痛的脉络丛乳头状瘤:病例报告
Medicine (Baltimore). 2018 Sep;97(38):e12466. doi: 10.1097/MD.0000000000012466.
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Methylation profiling of choroid plexus tumors reveals 3 clinically distinct subgroups.脉络丛肿瘤的甲基化谱分析揭示了3个临床不同的亚组。
Neuro Oncol. 2016 Jun;18(6):790-6. doi: 10.1093/neuonc/nov322. Epub 2016 Jan 28.