Princess Máxima Center for Pediatric Oncology, Utrecht, the Netherlands.
Princess Máxima Center for Pediatric Oncology, Utrecht, the Netherlands.
Crit Rev Oncol Hematol. 2020 Dec;156:103140. doi: 10.1016/j.critrevonc.2020.103140. Epub 2020 Oct 23.
Sacrococcygeal teratoma (SCT) is a rare extragonadal germ cell tumour mostly diagnosed during infancy and early childhood. Neonatal SCTs are mostly mature, but can also contain immature and/or malignant components. Recurrence of an SCT alters prognosis, especially when it is malignant, of which its mechanism is not yet fully understood. This study is a review and meta-analysis of the literature on malignant recurrences after an initially mature SCT.
A literature search was performed to identify studies describing children with SCT and presenting specific information on histology of the initial tumour as well as the recurrence. Random effect models for mature recurrence and malignant recurrence after an initially mature SCT were employed to pool study-specific percentages in order to estimate an overall percentage and its associated 95 % confidence intervals (CI). Inverse variance method, which gives more weight to larger studies, was used to pool outcomes for the different studies.
A total of 22 articles, comprising 1516 patients with SCT, were included in the meta-analysis. The pooled proportions of mature and malignant recurrences after mature SCT were 3 % (95 % CI 1-4 %) and 5% (95 % CI 3-6 %), respectively. Fifty-seven (56 %) of a total of 102 recurrences after resection of an initially mature SCT were malignant, mostly yolk sac tumour (YST). Many recurrences occurred within 1-6 years, however some occurred as long as 20 years after initial diagnosis.
A substantial number of recurrences of mature SCT present as a malignant tumour. Overlooking malignant components on initial pathological evaluation and the progression of mature SCT cells to malignant cells may play a role. Treatment of mature SCTs with resection alone requires thorough follow-up of at least 6 years. Future research is needed to determine whether SCTs with malignant microfoci should be treated or followed-up differently from mature or immature SCTs. In addition, the value of serum biomarkers in follow-up after SCT needs to be further evaluated.
骶尾部畸胎瘤（SCT）是一种罕见的性腺外生殖细胞肿瘤，主要在婴儿和幼儿期诊断。新生儿 SCT 大多为成熟型，但也可能包含不成熟和/或恶性成分。SCT 的复发改变了预后，特别是当它是恶性的时，其机制尚未完全了解。本研究是对最初成熟 SCT 后恶性复发的文献进行回顾和荟萃分析。
进行文献检索，以确定描述具有 SCT 并提供初始肿瘤组织学特定信息以及复发情况的研究。采用成熟复发和最初成熟 SCT 后恶性复发的随机效应模型，对研究特异性百分比进行汇总，以估计总体百分比及其相关 95%置信区间（CI）。采用逆方差法对不同研究的结果进行汇总，该方法对较大的研究给予更多权重。
共有 22 篇文章，包含 1516 例 SCT 患者，纳入荟萃分析。最初成熟 SCT 后成熟和恶性复发的汇总比例分别为 3%（95%CI 1-4%）和 5%（95%CI 3-6%）。在总共 102 例切除最初成熟 SCT 后的复发中，有 57 例（56%）为恶性，主要为卵黄囊瘤（YST）。许多复发发生在初始诊断后 1-6 年内，但有些复发发生在初始诊断后长达 20 年。
相当数量的成熟 SCT 复发表现为恶性肿瘤。在初始病理评估中忽略恶性成分以及成熟 SCT 细胞向恶性细胞的进展可能起作用。仅通过切除治疗成熟 SCT 需要至少 6 年的彻底随访。需要进一步研究确定是否应将具有恶性微焦点的 SCT 与成熟或不成熟 SCT 区别对待或随访。此外，还需要进一步评估血清生物标志物在 SCT 后随访中的价值。
