目的:我们报告了一例疑似复发性颅内表皮样囊肿(EDC)的独特病例,该病例在初次切除后大约25年的病理上被发现恶性转化为鳞状细胞癌(SCC)。此外,我们进行了系统评价,包括94项报告颅内EDC向SCC转化的研究.
方法:94项研究纳入我们的系统评价。PubMed,Scopus,CochraneCentral,和EMBASE于2020年4月检索了有关EDC内组织学证实的SCC的研究。Kaplan-Meier估计用于估计事件发生时间,包括生存时间,和对数秩检验用于检验显著性。所有分析均使用STATA14.1(StataCorp,学院站,德克萨斯州);测试是双面的,并且使用0.05的α阈值定义统计学显著性。
结果:总的中位转化时间为60个月(95%CI,12-96)。无手术组的转化时间明显缩短(10个月,95%CI未定义)与其他两组(60个月,95%CI,仅手术12-72,70个月,手术+辅助治疗组95%CI,9-180,两者P<0.01)。手术+辅助治疗组的总生存期(OS)显著延长(13个月,95%CI,9-24)与其他两组(3个月,95%CI,仅手术1-7,6个月,95%CI,无手术组1-12,两者P<0.01)。
结论:我们报告了一例颅内EDC向SCC的迟发性恶性转化的罕见病例,发生在初次切除后近25年。与仅手术和手术+辅助治疗组相比,非手术组的转化时间在统计学上明显缩短。手术+辅助治疗组的OS显著高于仅手术和不手术组。
OBJECTIVE: We report a unique
case of a suspected recurrent intracranial epidermoid cyst (EDC) that was found on pathology to have undergone malignant transformation to squamous cell carcinoma (SCC) approximately 25 years after initial resection. Additionally, we performed a systematic review including 94 studies reporting intracranial EDC to SCC transformation.
METHODS: Ninety-four studies were included in our systematic review. PubMed, Scopus, Cochrane Central, and EMBASE were searched in April 2020 for studies regarding histologically confirmed SCC arising within an EDC. Kaplan-Meier estimations were used to estimate time to event including survival, and log rank tests were used to test for significance. All analyses were conducted using STATA 14.1 (StataCorp, College Station, Texas, USA); tests were two-sided, and statistical significance was defined using the alpha threshold of 0.05.
RESULTS: The overall median time to transformation was 60 months (95% confidence interval {CI}, 12-96). Transformation time was significantly shorter in the no surgery group (10 months, 95% CI undefined) versus the other 2 groups (60 months, 95% CI, 12-72 in surgery only and 70 months, 95% CI, 9-180 in surgery + adjuvant therapy group, both P < 0.01). Overall survival was significantly longer in the surgery + adjuvant therapy group (13 months, 95% CI, 9-24) versus the other 2 groups (3 months, 95% CI, 1-7 in surgery only and 6 months, 95% CI, 1-12 in the no surgery group, both P < 0.01).
CONCLUSIONS: We report a rare
case of delayed malignant transformation of an intracranial EDC to SCC, occurring nearly 25 years after initial resection. Transformation time in the no-surgery group was statistically significantly shorter as compared to the surgery only and surgery + adjuvant therapy groups. Overall survival was statistically significantly higher in the surgery + adjuvant therapy group as compared to the surgery only and no surgery groups.