Laboratory of Cellular Biophysics, The Rockefeller University, New York, New York, USA.
The Fibrolamellar Registry, New York, New York, USA.
Hepatol Commun. 2022 Dec;6(12):3539-3549. doi: 10.1002/hep4.2105. Epub 2022 Oct 17.
Fibrolamellar hepatocellular carcinoma (FLC) is a rare primary liver cancer that affects primarily adolescents and young adults. It is associated with a poor overall prognosis. There is a need to better define risk factors, but small sample size has limited such studies. An FLC patient registry now provides data sufficient for statistically robust inferences. We leveraged a unique patient community-based FLC registry to analyze the prognostic impact of demographic and clinical characteristics evident at diagnosis. Variables were analyzed using Cox proportional hazards regression to calculate hazard ratios (HRs) and 95% confidence intervals (CIs). In multivariable models of 149 patients (88 females and 61 males), female gender was associated with statistically significant improved survival with HR of 0.52 (95% CI 0.29-0.93). Factors evident at diagnosis that are associated with worse survival included the presence of 10 or more tumors within the liver (HR 7.1; 95% CI 2.4-21.04), and metastases at diagnosis (HR 2.17; 95% CI 1.19-3.94). Positive lymph nodes at diagnosis, despite being found significantly associated with worse survival in a univariate analysis, did not remain significant when adjusted for covariates in a multivariable analysis. We found no statistically significant effect of age at diagnosis nor tumor size at diagnosis on survival. Female gender may confer a favorable prognosis in FLC. Established high-risk prognostic factors that we confirmed in this Registry included the diagnostic presence of numerous intrahepatic tumors, and metastases. This is the first study derived from a FLC patient community-based registry, and highlights how registries of rare tumors can empower patients to meaningfully advance clinical and scientific discoveries.
纤维板层肝细胞癌 (FLC) 是一种罕见的原发性肝癌,主要影响青少年和年轻成年人。它的整体预后较差。需要更好地定义风险因素,但由于样本量小,限制了此类研究。现在有一个 FLC 患者登记处提供了足够的数据,可以进行具有统计学意义的推断。我们利用一个独特的基于患者的 FLC 登记处来分析诊断时明显的人口统计学和临床特征对预后的影响。使用 Cox 比例风险回归分析变量,以计算风险比 (HR) 和 95%置信区间 (CI)。在 149 名患者(88 名女性和 61 名男性)的多变量模型中,女性性别与统计学上显著改善的生存相关,HR 为 0.52(95%CI 0.29-0.93)。诊断时与生存较差相关的因素包括肝脏内存在 10 个或更多肿瘤(HR 7.1;95%CI 2.4-21.04)和诊断时转移(HR 2.17;95%CI 1.19-3.94)。尽管在单变量分析中发现诊断时存在淋巴结阳性与生存较差显著相关,但在多变量分析中调整协变量后并不显著。我们发现诊断时的年龄和肿瘤大小对生存没有统计学上的显著影响。女性性别可能在 FLC 中带来有利的预后。我们在本登记处确认的既定高危预后因素包括诊断时存在大量肝内肿瘤和转移。这是第一项源自 FLC 患者社区登记处的研究,强调了罕见肿瘤登记处如何使患者能够有意义地推进临床和科学发现。